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Archive for the ‘Diagnostic Immunology’ Category

The History of Hematology and Related Sciences

Curator: Larry H. Bernstein, MD, FCAP

 

The History of Hematology and Related Sciences: A Historical Review of Hematological Diagnosis from 1880 -1980

 

Blood Description: The Analysis of Blood Elements a Window into Diseases

Diagnosing bacterial infection (BI) remains a challenge for the attending physician. An ex vivo infection model based on human fixed polymorphonuclear neutrophils (PMNs) gives an autofluorescence signal that differs significantly between stimulated and unstimulated cells. We took advantage of this property for use in an in vivo pneumonia mouse model and in patients hospitalized with bacterial pneumonia. A 2-fold decrease was observed in autofluorescence intensity for cytospined PMNs from broncho-alveolar lavage (BAL) in the pneumonia mouse model and a 2.7-fold decrease was observed in patients with pneumonia when compared with control mice or patients without pneumonia, respectively. This optical method provided an autofluorescence mean intensity cut-off, allowing for easy diagnosis of BI. Originally set up on a confocal microscope, the assay was also effective using a standard epifluorescence microscope. Assessing the autofluorescence of PMNs provides a fast, simple, cheap and reliable method optimizing the efficiency and the time needed for early diagnosis of severe infections. Rationalized therapeutic decisions supported by the results from this method can improve the outcome of patients suspected of having an infection.

Monsel A, Le´cart S, Roquilly A, Broquet A, Jacqueline C, et al. (2014) Analysis of Autofluorescence in Polymorphonuclear Neutrophils: A New Tool for Early Infection Diagnosis. PLoS ONE 9(3): e92564.
http://dx.doi.org:/10.1371/journal.pone.0092564

This study was designed to validate or refute the reliability of total lymphocyte count (TLC) and other hematological parameters as a substitute for CD4 cell counts. Participants consisted of two groups, including 416 antiretroviral naive (G1) and 328 antiretroviral experienced (G2) patients. CD4+ T cell counts were performed using a Cyflow machine. Hematological parameters were analyzed using a hematology analyzer. The median ± SEM CD4 count (range) of participants in G1 was 199 ± 10.9 (5–1840 cells/μL) and the median ± SEM TLC (range) was 1. 61 ± 0.05 (0.07–6.63 × 103/μL). The corresponding values among G2 were 421 ± 15.8 (13–1801) and 2.13 ± 0.04 (0.06–5.58), respectively. Using a threshold value of 1.2 × 103/μL for TLC alone, the sensitivity of G1 was 88.4% (specificity (SP) 67.4%, the positive predictive value (PPV) 53.5% and negative predictive value (NPV) of 93.2% for CD4 , 200 cells/μL, the sensitivity for G2 was 83.3%, SP 85.3%, PPV 23.8%, and NPV of 93.2%. Using multiple parameters, including TLC , 1.2 × 103/μL, hemoglobin , 10 g/dL, and platelets , 150 × 103/L, the sensitivity increased to 96.0% (SP, 82.7%; PPV, 80%; NPV, 96.7%) among G1, while no change was observed in the G2 cohort. TLC , 1.2 × 103/μL alone is an insensitive predictor of CD4 count of , 200 cells/μL. Incorporating hemoglobin , 10 g/dL, and platelets , 150 × 103/L enhances the ability of TLC , 1.2 × 103/μL to predict CD4 count , 200 cells/μL among the antiretroviral-naïve cohort. We recommend the use of multiple, inexpensively measured hematological parameters in the form of an algorithm for predicting CD4 count level.

Evaluating Total Lymphocyte Counts and Other Hematological Parameters as a Substitute for CD4 Counts in the Management of HIV Patients in Northeastern Nigeria. BA Denue, AU Abja, IM Kida, AH Gabdo, AA Bukar and CB Akawu.
Retrovirology: Research and Treatment 2013:5 9–16 http://dx.doi.org:/10.4137/RRT.S11562

Sepsis is a syndrome that results in high morbidity and mortality. We investigated the delta neutrophil index (DN) as a predictive marker of early mortality in patients with gram-negative bacteremia. Retrospective study. The DN was measured at onset of bacteremia and 24 hours and 72 hours later. The DN was calculated using an automatic hematology analyzer. Factors associated with 10-day mortality were assessed using logistic regression. A total of 172 patients with gram-negative bacteremia were included in the analysis; of these, 17 patients died within 10 days of bacteremia onset. In multivariate analysis, Sequental organ failure assessment scores (odds ratio [OR]: 2.24, 95% confidence interval [CI]: 1.31 to 3.84; P = 0.003), DN-day 1 ≥ 7.6% (OR: 305.18, 95% CI: 1.73 to 53983.52; P = 0.030) and DN-day 3 ≥ DN day 1 (OR: 77.77, 95% CI: 1.90 to 3188.05; P = 0.022) were independent factors associated with early mortality in gram-negative bacteremia. Of four multivariate models developed and tested using various factors, the model using both DN-day 1 ≥ 7.6% and DN-day 3 ≥ DN-day 1 was most predictive early mortality. DN may be a useful marker of early mortality in patients with gram-negative bacteremia. We found both DN-day 1 and DN trend to be significantly associated with early mortality.

Delta Neutrophil Index as a Prognostic Marker of Early Mortality in Gram Negative Bacteremia. HW Kim, JH Yoon, SJ Jin, SB Kim, NS Ku, SJ Jeong,
et al. Infect Chemother 2014;46(2):94-102. pISSN 2093-2340·eISSN 2092-6448
http://dx.doi.org/10.3947/ic.2014.46.2.94
Various indices derived from red blood cell (RBC) parameters have been described for distinguishing thalassemia and iron deficiency. We studied the microcytic to hypochromic RBC ratio as a discriminant index in microcytic anemia and compared it to traditional indices in a learning set and confirmed our findings in a validation set. The learning set comprised samples from 371 patients with microcytic anemia mean cell volume (MCV < 80 fL), which were measured on a CELL-DYN Sapphire analyzer and various discriminant functions calculated. Optimal cutoff values were established using ROC analysis. These values were used in the validation set of 338 patients. In the learning set, a microcytic to hypochromic RBC ratio >6.4 was strongly indicative of thalassemia (area under the curve 0.948). Green-King and England-Fraser indices showed comparable area under the ROC curve. However, the microcytic to hypochromic ratio had the highest sensitivity (0.964). In the validation set, 91.1% of microcytic patients were correctly classified using the M/H ratio. Overall, the microcytic to hypochromic ratio as measured in CELL-DYN Sapphire performed equally well as the Green-King index in identifying thalassemia carriers, but with higher sensitivity, making it a quick and inexpensive screening tool.
Differential diagnosis of microcytic anemia: the role of microcytic and hypochromic erythrocytes. E. Urrechaga, J.J.M.L. Hoffmann, S. Izquierdo, J.F. Escanero. Intl Jf Lab Hematology Aug 2014. http://dx.doi.org:/10.1111/ijlh.12290

Achievement of complete response (CR) to therapy in chronic lymphocytic leukemia (CLL) has become a feasible goal, directly correlating with prolonged survival. It has been established that the classic definition of CR actually encompasses a variety of disease loads, and more sensitive multiparameter flow cytometry [and polymerase chain reaction methods] can detect the disease burden with a much higher sensitivity. Detection of malignant cells with a sensitivity of 1 tumor cell in 10,000 cells (10–4), using the above-mentioned sophisticated techniques, is the current cutoff for minimal residual disease (MRD). Tumor burdens lower than 10–4 are defined as MRD-negative. Several studies in CLL have determined the achievement of MRD negativity as an independent favorable prognostic factor, leading to prolonged disease-free and overall survival, regardless of the treatment protocol or the presence of other pre-existing prognostic indicators. Minimal residual disease evaluation using flow cytometry is a sensitive and applicable approach which is expected to become an integral part of future prospective trials in CLL designed to assess the role of MRD surveillance in treatment tailoring.

Minimal Residual Disease Surveillance in Chronic Lymphocytic Leukemia by Fluorescence-Activated Cell Sorting. S Ringelstein-Harlev, R Fineman.
Rambam Maimonides Med J. Oct 2014   5 (4)  e0027. http://dx.doi.org:/10.5041/RMMJ.10161

Natural Killer cells (CD3-CD16+CD56+) are a major players in innate immunity, both as direct cytotoxic effectors as well as regulators for other innate immunity cell types. We have shown that, using the FlowCellect™ human NK cell characterization kit, one can achieve accurate phenotyping on a variety of sample types, including whole blood samples. Using the same kit to perform an NK cell cytotoxicity test, we demonstrate that unbound K562 target cells can be clearly distinguished from those that have been engaged by CD56+ NK cells, and each of these populations can be further investigated for viability using the eFluor 660® dye.

Analysis of NK cell subpopulations in whole blood

Analysis of NK cell subpopulations in whole blood

Analysis of NK cell subpopulations in whole blood

A

Proportion of K562 target cells bound to NK cells

Proportion of K562 target cells bound to NK cells

In a 5:1 effector cell:target cell population, 8% of the K562 cells were bound to NK cells (Figure 3B). 84% of the bound K562 cells were viable (Figure 3C) stained with fixable viability dye), while 96% of the unbound K562 cells were viable (Figure 3D). (B,C,D not shown)

Characterization of Natural Killer Cells Using Flow Cytometry.
EMD Millipore is a division of Merck KGaA, Darmstadt, Germany.

Red blood cell distribution width (RDW) is increased in liver disease. Its clinical significance, however, remains largely unknown. The aim of this study was to identify whether RDW was a prognostic index for liver disease. Retrospective: 33 patients with non-cirrhotic HBV chronic hepatitis, 125 patients with liver cirrhosis after HBV infection, 81 newly diagnosed primary epatocellular carcinoma (pHCC) patients, 17 alcoholic liver cirrhosis patients and 42 patients with primary biliary cirrhosis (PBC). Sixty-six healthy individuals represented the control cohort. The relationship between RDW on admission and clinical features: The association between RDW and hospitalization outcome was estimated by receiver operating curve (ROC) analysis and a multivariable logistic regression model. Increased RDW was observed in liver disease patients. RDW was positively correlated with serum bilirubin and creatinine levels, prothrombin time, and negatively correlated with platelet counts and serum albumin concentration. A subgroup analysis, considering the different etiologies, revealed similar findings. Among the patients with liver cirrhosis, RDW increased with worsening of Child-Pugh grade. In patients with PBC, RDW positively correlated with Mayo risk score. Increased RDW was associated with worse hospital outcome, as shown by the AUC [95% confidence interval (CI)] of 0.76 (0.67 – 0.84). RDW above 15.15% was independently associated with poor hospital outcome after adjustment for serum bilirubin, platelet count, prothrombin time, albumin and age, with the odds ratio (95% CI) of 13.29 (1.67 – 105.68). RDW is a potential prognostic index for liver disease.

Red blood cell distribution width is a potential prognostic index for liver disease
Z Hua , Y Suna , Q Wanga , Z Han , Y Huang , X Liu , C Ding, et al.
Clin Chem Lab Med 2013; 51(7):1403–1408.
http://dx.doi.org:/10.1515/cclm-2012-0704

Blood Plasma and Red Blood Cells

Whole blood consists of red and white blood cells, as well as platelets suspended in a liquid referred to as blood plasma. According to the American Red Cross, plasma is 92% water and makes up 55% of blood volume. The permeability of blood plasma is equal to 1.

Red blood cells make up slightly lower blood volume than blood plasma — about 45% of whole blood. As you probably already know, these types of blood cells contain hemoglobin, which in turn consists of iron that helps transport oxygen throughout the body. The permeability of red blood cells is slightly less than 1,
(1 – 3.9e-6). Or to put it in words, red blood cell particles are diamagnetic.

Due to their magnetic properties, red blood cells may be separated from the plasma via a magnetophoretic approach. If the blood were to be in a channel subject to a magnetophoretic force, we could control where the red blood cells and the plasma go within the channels. In other words, because the red blood cells have different permeability, they can be separated from the flow channel. However, such methodology is beyond the year 1980.

Timeline of Major Hematology Landmarks

1877 Paul Ehrlich develops techniques to stain blood cells to improve microscopic visualization.

1897 The Diseases of Infancy and Childhood contains a 20-page chapter on diseases of the blood and is the first American pediatric medical textbook to provide significant hematologic information.

1821–1902 Rudolph Virchow, during a long and illustrious career, demonstrates the importance of fibrin in the blood coagulation process, coins the terms embolism and thrombosis, identifies the disease leukemia, and theorizes that leukocytes are made in response to inflammation.

1901 Karl Landsteiner and colleagues identify blood groups of A, B, AB, and O.

1907 Ludvig Hektoen suggests that the safety of transfusion might be improved by crossmatching blood between donors and patients to exclude incompatible mixtures. Reuben Ottenberg performs the first blood transfusion using blood typing and crossmatching in New York. Ottenberg also observes the Mendelian inheritance of blood groups and recognizes the “universal” utility of group O donors.

1910 The first clinical description of sickle cell published in medical literature.

1914 Sodium citrate is found to prevent blood from clotting, allowing blood to be stored between collection and transfusion.

1924 Pediatrics is the first comprehensive American publication on pediatric hematology.

1925 Alfred P. Hart performs the first exchange transfusion.

1925 Thomas Cooley describes a Mediterranean hematologic syndrome of anemia, erythroblastosis, skeletal disorders, and splenomegaly that is later called Cooley’s anemia and now thalassemia.

1936 Chicago’s Cook County Hospital establishes the first true “blood bank” in the United States.

1938 Dr. Louis Diamond (known as the “father of American pediatric hematology”) along with Dr. Kenneth Blackfan describes the anemia still known as Diamond-Blackfan anemia.

1941 The Atlas of the Blood of Children is published by Blackfan, Diamond, and Leister.

1945 Coombs, Mourant, and Race describe the use of antihuman globulin (later known as the “Coombs Test”) to identify “incomplete” antibodies.

1954 The blood product cryoprecipitate is developed to treat bleeds in people with hemophilia.

1950s The “butterfly” needle and intercath are developed, making IV access easier and safer.

1961 The role of platelet concentrates in reducing mortality from hemorrhage in cancer patients is recognized.

1962 The first antihemophilic factor concentrate to treat coagulation disorders in hemophilia patients is developed through fractionation.

1969 S. Murphy and F. Gardner demonstrate the feasibility of storing platelets at room temperature, revolutionizing platelet transfusion therapy.

1971 Hepatitis B surface antigen testing of blood begins in the United States.

1972 Apheresis is used to extract one cellular component, returning the rest of the blood to the donor.

1974 Hematology of Infancy and Childhood is published by Nathan and Oski.

As I write today my hospital celebrates its 150th anniversary. Great Ormond Street Children’s Hospital was founded on 14 February 1852 by the visionary Dr Charles West followed his belief that hospital care allied to research in children’s diseases would reduce child mortality from above 50% by the age of 15 years. It is foolish to believe that we can progress in medicine without a knowledge of the past and that much of life is based upon experience. When putting together a series of articles on the history of haematology, initially published in BJH, this was the main raison d’être, along with the belief that the practice of medicine has become increasingly serious but should also be fun and interesting and even occasionally uplifting to the spirit.

The central problem of any survey of the history of haematology is usually the question of balance. Achieving a degree of balance among themes and topics that will be satisfactory to practicing haematologists/physicians with an interest in blood diseases is essentially impossible. Our preference has been for themes of general interest rather than those of a purely scientific view into a field that has led the way in understanding the molecular basis of human disease.

  1. M. Hann, London, 2002; O. P. Smith, Dublin, 2002.

Origins of the Discipline `Neonatal Haematology’, 1925-75

In every modern neonatal intensive care unit (NICU), haematological problems are encountered daily. Many of these problems involve varieties of anaemia, neutropenia or thrombocytopenia that are unique to NICU patients. A characteristic aspect of these unique problems is that, if the neonate survives, the haematological problem will remit and will not recur later in life, nor will it evolve into a chronic illness (although the problem might occur in a future newborn sibling). This characteristic comes about because the common haematological problems of NICU patients are not genetic defects but are environmental stresses (such as infection, alloimmunization or a variety of maternal illnesses) that are imposed on a developmentally immature haematopoietic system.

In the USA, and in some parts of Europe, the unique haematological problems that occur among NICU patients are diagnosed and treated by neonatologists, not by paediatric haematologists. Although these haematological conditions were generally first described by haematologists, the conditions occur, obviously, in neonates. Thus, the neonatologist, who is familiar with intensive care management of neonates, has also become familiar with the diagnosis and management of the neonate’s common haematological disorders. A growing number of neonatologists have sought specific additional training in haematology, with the goals of discovering the mechanisms underlying the unique haematological problems of NICU patients and improving the management and outcome of the patients who have these conditions. These physicians have remained as neonatologists and they do not practice paediatric haematology, although their research contributions certainly come under the purview of haematology, or more precisely under the discipline of `neonatal haematology’. In many places in Europe, it is the haematologists rather than the neonatologists who have an academic and clinical interest in neonatal haematology.

The roots of the discipline of neonatal haematology can be traced to the early application of haematological methods to animal and human embryos and fetuses, such as found in the reports of Maximow (1924) and Wintrobe & Schumacker (1936). The clinical underpinnings of this discipline include reports of anaemia (Fikelstein, 1911) and jaundice (Blomfeld, 1901; YlppoÈ, 1913) among neonates.

Before the 1930s, very few studies and very few published clinical case reports originated from premature nurseries. Such nurseries had dubious beginnings, which were criticized by some physicians as more resembling circus exhibitions than medical care wards (Bonar, 1932). These units generally had mortality rates greatly exceeding 50% on the day of admission, with the majority of the first-day survivors having late deaths or serious long-term morbidity.

It was not until publication of the review of premature nursery care at the Children’s Hospital of Michigan, in 1932, that it was clear that some units had instituted systematic attempts to monitor and improve outcomes. A special care nursery had been established at the Children’s Hospital in 1926 and, in 1932, Drs Marsh Poole and Thomas Cooley reported their experience in that unit (Poole & Cooley, 1932). The report included  incubator design with temperature and humidity control, growth curves of patients on various feeding practices, mortality statistics and attempts to determine causes of death.

At the time premature nursery care was beginning to merit academic credentials, reports were published of haematological problems that were unique to the neonate. These papers included the seminal publication on erythroblastosis fetalis by Drs Diamond (Fig 1), Blackfan and Baty (Diamond et al, 1932), and the report of sepsis neonatorum at the Yale New Haven Hospital by Ethyl C. Dunham (Fig 2) (Dunham,

1933).

The first major textbook devoted to clinical haematology, as well as the first textbook of neonatology, contained very little information about what are today’s common NICU haematological problems. For instance, in the first edition of Clinical Hematology by Dr Maxwell M. Wintrobe (Fig 3), of the Johns Hopkins University Hospital (Wintrobe, 1942), several topics related to paediatric haematology were reviewed, but discussions of the haematological problems of neonates were limited to three – erythroblastosis fetalis, haemorrhagic disease of the newborn and the `anaemia of prematurity’. Similarly, Premature Infants: A Manual for

Physicians, the original neonatology textbook, published in 1948 by Dr Ethyl C. Dunham (Fig 2; Dunham, 1948), had only a few pages devoted to haematological problems – the same three discussed by Dr Wintrobe. Also, the classic neonatology text book, `The Physiology of the Newborn Infant’, published in 1945 by Dr Clement A. Smith, contained almost no discussion of haematological problems (Smith, 1945). hrombocytopenia, which is now diagnosed among 25-30% of NICU patients, and neutropenia, now diagnosed in 8-10% of NICU patients, were not mentioned.

The first article published in Paediatrics (1948) dealing with a neonatal haematological problem was in volume two, in which Dr Diamond detailed his technique for performing a replacement transfusion (which later became known as an `exchange’ transfusion) as a treatment for erythroblastosis fetalis (Diamond, 1949). The second paper published by Paediatrics containing aspects of neonatal haematology was 1 year later, when Sliverman & Homan (1949) described leucopenia among neonates with sepsis. Most of the 25 infants they described, who were treated at Babies Hospital in New York over an 11-year period, had `late-onset’ sepsis, beginning after 3 days of life. They reported 14 neonates with Escherichia coli sepsis and four with streptococcal or staphylococcal sepsis, and observed that leucopenia occurred occasionally among these patients but was uncommon. (Indeed, today neutropenia remains uncommon in `late-onset’ sepsis, but common in congenital or `early onset’ sepsis.)

Louis K. Diamond, MD, at Children's Hospital, Boston,

Louis K. Diamond, MD, at Children’s Hospital, Boston,

Louis K. Diamond, MD, at Children’s Hospital, Boston, MA. , date unknown (obtained with the kind assistance of Charles F. Simmons, MD, Harvard University).

Diagnosing neutropenia, anaemia or thrombocytopenia in a neonate obviously requires knowledge of the expected normal range for neutrophil concentration, haematocrit and platelet concentration in the appropriate reference population. Early contributions to neonatal haematology included the publications of these reference ranges. The landmark studies included the range of blood leucocyte and neutrophil concentrations in neonates published in 1935 by Dr Katsuji Kato from the Department of Paediatrics at the University of Chicago (Kato, 1935). He tabulated the leucocyte concentrations and differential counts of 1081 children, ranging from birth to 15 years of age. A striking finding of his report (Fig 4) was the very high neutrophil counts during the first hours and days of life. Blood neutrophil concentrations among neonates with infections were published during the early and mid-1970s by Dr Marietta Xanthou (Fig 5) at the Hammersmith Hospital in London (Xanthou, 1970, 1972), and by Drs Barbara Manroe and Charles Rosenfeld (Fig 6) at the University of Texas Southwestern Medical Center in Dallas, Texas (Manroe et al, 1977).

Normal values for haemoglobin, haematocrit, erythrocyte indices and leucocyte concentrations were refined by DeMarsh et al (1942, 1948), and in a series of publications in the early 1950s in Archives of Diseases of Children by Gairdner et al (1952a, b). These were followed by observations on human fetal haematopoiesis by Thomas and Yoffey in the British Journal of Haematology (Thomas & Yoffey, 1962, 1964), and by the work on blood volume during the 1960s (Usher et al, 1963, Usher & Lind, 1965; Yao et al, 1967, 1968). Normal ranges for blood platelet counts in ill and well preterm and term infants were published in the early 1970s (Sell et al, 1973; Corrigan, 1974).

The first publication addressing the problem of neutropenia accompanying fatal early onset bacterial sepsis was that of Tygstrup et al (1968). This was a report of a near-term male with congenital Listeria sepsis who lived for only 4 h. The platelet count was 80*109/l and the leucocyte count was 13´7*109/l, but no granulocytes were observed on the differential count, which consisted of 84% lymphocytes, 8% monocytes and 8% leucocyte precursors. A sternal marrow aspirate was taken of the infant shortly before death that revealed myeloblasts, promyelocytes and myelocytes, but no band or segmented neutrophils.

An important advance in understanding the blood neutrophil count during neonatal sepsis occurred with the back-to-back papers in Archives of Diseases of Childhood in 1972 by Dr Marietta Xanthou of Hammersmith Hospital, London (Xanthou, 1972), and Drs Gregory and Hey of Babies’ Hospital, Newcastle upon Tyne (Gregory & Hey, 1972). Both papers reported that neonates who had life threatening (or indeed fatal) infections became neutropenic prior to death. Dr Xanthou reported 35 ill preterm and term babies within their first 28 d of life. Twenty-four were ill but not infected, and these had normal blood neutrophil concentrations and morphology. However, among the 11 who were ill with a bacterial infection, neutrophilia was observed in the survivors, but neutropenia, a `left shift’, and toxic granulation were observed in the non-survivors. Consistent with this observation, Gregory and Hey reported three neonates who died with overwhelming bacterial sepsis and noted that all had profound neutropenia. Neutrophilia was common among the survivors and neutropenia, a “left shift’, and specific neutrophil morphological changes were seen among those who subsequently died.

A pivotal publication that launched the search for mechanistic information and successful treatments was that of Dr Barbara Manroe, a fellow in Neonatal Medicine, and her mentor Dr Charles Rosenfeld (Fig 6) from the University of Texas, South-western, Parkland Hospital in Dallas, Texas (Manroe et al, 1977). They evaluated 45 neonates who had culture-proven group B streptococcal infection and found that 39 had abnormal leucocyte counts: 25 neutrophilia and 14 neutropenia, and that 41 had a `left shift’. This paper was the first to quantify the `left shift’ using a method that has since become popular in neonatology – the ratio of immature neutrophils to total neutrophils on the differential cell count.

From these beginning, hundreds of studies using experimental models and clinical observations and trials were published, detailing the kinetic and molecular mechanisms accounting for this common variety of neutropenia. Marked improvements in the survival of neonates with this condition have come about through combined efforts, including early maternal screening for GBS carriage, early anti-microbial administration to ill neonates, non-specific antibody administration and a variety of measures to improve supportive care of neonates with early onset sepsis.

In the early 1930s, Dr Helen Mackay worked as a paediatrician in Mother’s Hospital, a maternity hospital located in the north-east section of London. Acting on the observation of Lichtenstein (1921) that infants of subnormal birth weight regularly became anaemic in the first months of life, she measured and reported serial heel-stick haemoglobin levels on 150 infants during their first 6 months. Thirty-nine of these infants weighed under five pounds at birth (six were under four pounds), 52 weighed five to six pounds, and 59 weighed six pounds and upwards. She showed that babies of the lightest birth weights had the most rapid fall in haemoglobin and that these fell to lower levels than those of babies of heavier birth weight (MacKay et al, 1935). Figure 7 contrasts this fall in babies weighing `3-4 lbs odd at birth’ with those weighing `5 lbs odd at birth’.

Her attempts to prevent the anaemia of prematurity failed,  but her work constituted the first clear definition of the `anaemia of prematurity’ and showed that iron administration did not prevent this condition. In the early 1950s, Douglas Gairdner, John Marks and Janet D. Roscoe, of the Department of Pathology of Cambridge Maternity Hospital, published pioneering studies in blood formation in infancy (Gairdner et al, 1952a, b). Studying 105 blood samples and 102 bone marrow samples, they concluded that `erythropoiesis ceases when the oxygen saturation just after birth increases from about 65% in the umbilical vein to .95% just after birth’. Publications by Dr Irving Schulman, in the mid- to late 1950s, defined three phases of the anaemia of prematurity and provided a mechanistic explanation for the anaemia (Schulman & Smith, 1954; Schulman, 1959). His work illustrated that the early and intermediate phases of this anaemia occur in the face of relative iron excess and are unaffected by prophylactic iron administration.

Haemoglobin levels during the first 25 weeks of life among

Haemoglobin levels during the first 25 weeks of life among

Haemoglobin levels during the first 25 weeks of life among neonates in London [by permission; Archives Diseases of Children, (MacKay, 1935)].

In 1963, Dr Sverre Halvorsen of the Department of Paediatrics at Rikshospatalet in Oslo, Norway (Fig 9), provided an underlying explanation for the observations made by MacKay, Gairdner and Schulman (Halvorson, 1963). He observed that, compared with the blood of healthy adults, umbilical cord blood of healthy neonates had a high erythropoietin concentration, but the concentration was considerably higher in the plasma of severely erythroblastotic, anaemic infants. Among the healthy infants, erythropoietin levels fell to unmeasurably low concentrations after delivery, but levels remained elevated in hypoxic and cyanotic infants. Dr Per Haavardsholm Finne, also of the Children’s Department, Paediatric Research Institute and Department of Obstetrics and Gynaecology at Rikshospitalet in Oslo, observed high oncentrations of erythropoietin in the amniotic fluid and the umbilical cord blood after fetal hypoxia (Finne, 1964, 1967).

In subsequent studies, Dr Halvorsen observed lower plasma erythropoietin concentrations in the cord blood of preterm infants at delivery than in term neonates at delivery (Halvorsen & Finne, 1968). These observations supported the concept of Gairdner et al (1952a, b) that the postnatal fall in erythropoiesis (the `physiologic anaemia’ of neonates) is as a result of an increase in oxygen delivery to tissues following birth and is mediated by a fall in circulating erythropoietin concentration. The observations gave rise to the postulate that the `anaemia of prematurity’ was an exaggeration of this physiological anaemia and involved a limitation of preterm infants to appropriately increase erythropoietin production.

Many landmark reports of haematological findings of neonates that were published between 1925 and 1975 were not detailed in this review because they were outside the restricted topics selected.

Robert D. Christensen, MD, Gainesville, FL
Brit J Haem 2001; 113: 853-860

Towards Molecular Medicine; Reminiscences of the Haemoglobin Field

When historians of medicine in the twentieth century start to piece together the complex web of events that led from a change of emphasis of medical research from studies of patients and their organs to disease at the levels of cells and molecules they will undoubtedly have their attention drawn to the haemoglobin field, particularly the years that followed Linus Pauling’s seminal paper in 1949 which described sickle-cell anaemia as a `molecular disease’. These are personal reminiscences of some of the highlights of those exciting times, and of those who made them happen.

One of my first patients serving the RAMC was a Nepalese Ghurka child who was kept alive from the first few months of life with regular blood transfusion without a diagnosis. Henry Kunkel published a paper which described how, using electrophoresis in slabs of starch, he had found a minor component of human haemoglobin (Hb), Hb A2, the proportion of which was elevated in some carriers of thalassaemia. After several weeks spent knee deep in potato starch, we found that the Ghurka child’s parents had increased Hb A2 levels and, hence, that she was likely to be homozygous for thalassaemia. I was hauled up before the Director General of Medical Services for the Far East Land Forces and told that I could be court marshalled for not getting permission from the War House (Office) to publish information about military personnel. `And, in any case’, he added, `it is bad form to tell the world that one of our pukka regiments has bad genes; don’t do it again’.

Just before the end of my National Service I arranged to go to Johns Hopkins Hospital in Baltimore to train in genetics and haematology. I was told that I was wasting my time working on haemoglobin because there was `nothing left to do’. `Start exploring red cell enzymes’, he suggested. On arriving in Baltimore in 1960 it turned out that human genetics, and the haemoglobin field in particular, were bubbling with excitement and potential. The only lessons for those contemplating careers in medical research from this chapter of academic and military gaffs are that, regardless of the working conditions, when there are sick people there are always interesting research questions to be asked.

The excitement of the haemoglobin field in 1960 reflected the chance amalgamation of several disciplines in the 1950s, particularly X-ray crystallography, protein chemistry, human genetics and haematology.

From the early 1930s the structure of proteins became one of the central problems of biochemistry. At that time, the only way of tackling this problem was by X-ray crystallography. In 1937 Felix Haurowitz suggested to Max Perutz (Fig 1) that an X-ray study of haemoglobin might be a good subject for his doctoral thesis. He was given some large crystals of horse methaemoglobin which gave excellent Xray diffraction patterns.

Max Perutz

Max Perutz

However, there was a major snag; an X-ray diffraction pattern provided only half the information required to solve the structure of a protein, that is the amplitudes of diffracted rays, while the other half, their phases, could not be determined. But in 1953, they discovered that it could be solved in two dimensions by comparison of the diffraction patterns of a crystal of native haemoglobin with that of haemoglobin reacted with mecuribenzoate, which combines with its two reactive sulphydryl groups. In short, to solve the structure in three dimensions required the comparison of the diffraction patterns of at least three crystals, one native and two with heavy atoms combined with different sites on the haemoglobin molecule. In 1959 this approach yielded the first three-dimensional model of haemoglobin, at 5´5 AÊ resolution.

Protein chemistry evolved side-by-side with X-ray crystallography during the 1950s. In 1951 Fred Sanger solved the structure of insulin, a remarkable tour de force which showed that proteins have unique chemical structures and amino acid sequences. Sanger had perfected methods for fractionation and characterization of small peptides by paper chromatography or electrophoresis. In 1956 Vernon Ingram (Fig 2), who, like Max Perutz, was a refugee from Germany, was set the task of studying the structure of haemoglobin from patients with sickle-cell anaemia. Ingram separated the peptides produced after globin had been hydrolysed with the enzyme trypsin, which cuts only at lysine and arginine residues. Although these amino acids accounted for 60 residues per mol of haemoglobin, only 30 tryptic peptides were obtained, indicating that haemoglobin consists of two identical half molecules. Re-examination of the amino-terminal sequences of haemoglobin by groups in the United States and Germany showed 2 mols of valine ± leucine and 2 mols of valine ± histidine ± leucine per mol of globin. These findings, which were in perfect agreement with the X-ray crystallographic results, suggested that haemoglobin is a tetramer composed of two pairs of unlike peptide chains, which were called α and β.

A seminal advance, and one which was to mark the beginning of molecular medicine, was the chance result of an overnight conversation on a train journey between Denver and Chicago. Linus Pauling, the protein chemist, and William Castle (Fig 3), one of the founding fathers of experimental haematology, were returning from a meeting in Denver and Castle mentioned to Pauling that he and his colleagues had noticed that when red cells from patients with sickle-cell anaemia are deoxygenated and sickle they show birefringence in polarized light.

Five generations of Boston haematology. Seated is William Castle. Standing (left to right) are Stuart Orkin, David Nathan and Alan Michelson. The picture on the left is of Dean David Edsall of Harvard Medical School who established the Thorndyke Laboratory at the Boston City Hospital. He was succeeded by Dean Peabody, who recruited both George Minot, who won the Nobel Prize for his work on pernicious anaemia, and William Castle, who should have also received it.

Pauling guessed that this might reflect a structural difference between normal and sickle-cell haemoglobin which could be detected by a change in charge. He gave this problem to one of his postdoctoral students, a young medical graduate called Harvey Itano. At that time they knew that a Swede, Arne Tiselius, had invented a machine for separating proteins according to their charge by electrophoresis. As there was no machine of this kind in Pauling’s laboratory, Itano and his colleagues set to and built one. Eventually they found that the haemoglobin of patients with sickle-cell anaemia behaves differently to that of normal people in an electric field, indicating that it must have a different amino acid composition. Even better, the haemoglobin of sickle-cell carriers was a mixture of both types of haemoglobin. This work was published in Science in 1949, under the title `Sickle-cell anaemia: a molecular disease’.

Perutz and Crick suggested to Ingram that he should apply Sanger’s techniques of peptide analysis to see if he could find any difference between normal and sickle cell haemoglobin. After digesting haemoglobin with trypsin, Ingram separated the peptides by electrophoresis and chromatography in two dimensions to produce what he later called `fingerprints’. He recalls that his first efforts looked like a watercolour that had been left out in the rain. But gradually things improved and he was able to show that the fingerprints of Hbs A and S were identical except for the position of one peptide. Using a method that had been developed a few years earlier by Pehr Edman, which allowed a peptide to be degraded one amino acid at a time in a stepwise fashion, Ingram found that this difference was due to the substitution of valine for glutamic acid at position 6 in the β chain of Hb S.

As well as demonstrating how a crippling disease can result from only a single amino acid difference in the haemoglobin molecule, this beautiful work had broader implications for molecular genetics. Although nothing was known about the nature of the genetic code at the time, the findings were compatible with the notion that the primary product of the β-globin gene is a peptide chain, a further development of the one-gene-one-enzyme concept, suggested earlier by Beadle and Tatum from their studies of Neurospora, and a prelude to the later studies of Yanofsky on Escherichia coli, which were to confirm this principle.

With the advent of simple filter paper electrophoresis, haemoglobin analysis became the province of clinical research laboratories during the 1950s and `new’ abnormal haemoglobins appeared almost by the week. Although many scientists were involved it was Hermann Lehmann (Fig 4) who became the father figure. Like Handel, Hermann was born in Halle and, also like the composer, made his home in Great Britain. He came to England as a refugee and at the beginning of the Second World War had a short period of internment as a `friendly alien’ at Huyton, close to Liverpool, an experience shared with many others, including Max Perutz. He travelled widely during his later war service in the RAMC and developed a wide international network which enabled him to discover 81 haemoglobin variants during his career.

Harvey Itano and Elizabeth Robinson showed that Hb Hopkins 2 is an a chain variant. Hence, it was now clear that there must be at least two unlinked loci involved in regulating haemoglobin production, a and b. The discovery of the λ and δ chains of Hbs F and A2, respectively, meant that there must be at least four loci involved. Subsequent family studies and analyses of unusual variants resulting from the production of δβ or λβ fusion chains led to the ordering of the non-α globin genes.

It had been known for some years that children with severe forms of thalassaemia might have persistent production of HbF and it was found later that some carriers might have elevated levels of Hb A2. The seminal observation in favour of this notion came from the study of patients who had inherited the sickle-cell gene from one parent and thalassaemia from the other. Sickle-cell thalassaemia was first described by Ezio Silvestroni and his wife Ida Bianco in 1946, although at the time they could not have known the full significance of their finding.  Phillip Sturgeon and his colleagues in the USA found that the pattern of haemoglobin production in patients with sickle-cell thalassaemia is quite different to that of heterozygotes for the sickle-cell gene; the effect of the thalassaemia gene is to reduce the amount of Hb A to below that of Hb S, i.e. exactly the  opposite to the ratio observed in sickle-cell carriers. As it was known that the sickle-cell mutation occurs in the β globin gene, it could be inferred that the action of the thalassaemia gene was to reduce the amount of β globin production from the normal allele. Indeed, from the few family studies available in 1960 there was a hint that this form of thalassaemia might be an allele of the β globin gene. Another major observation that was made in the mid-50 s was the association of unusual tetramer haemoglobins, β4 (Hb H) and λ4 (Hb Bart’s), with a thalassaemia phenotype. In 1959 Vernon Ingram and Tony Stretton proposed in a seminal article that there are two major classes, α and β, just as there are two major types of structural haemoglobin variants. They extended the ideas of Linus Pauling and Harvey Itano, who had suggested that defective globin synthesis in thalassaemia might be due to `silent’ mutations of the β globin genes, and postulated that the defects might lie outside the structural gene in the area of DNA in the connecting unit. work on the interactions of thalassaemia and haemoglobin variants in the late 1950s had moved the field to a considerably higher level of understanding than is apparent in the earlier papers of Pauling and Itano. In any case, in their paper Ingram and Stretton generously acknowledged the ideas of other workers, including Lehmann, Gerald, Neel and Ceppellini, that had allowed them to develop their conceptual framework of the general nature of thalassaemia. This interpretation of events, and the input of scientists from many different disciplines into these concepts, is supported by the published discussions of several conferences on haemoglobin held in the late 1950s.

Historical Review. Towards Molecular Medicine; Reminiscences of the Haemoglobin Field. D. J. Weatherall, Weatherall Institute of Molecular Medicine, University of Oxford. Brit J  Haem 115:729-738.

The Emerging Understanding of Sickle Cell Disease

The first indisputable case of sickle cell disease in the literature was described in a dental student studying in Chicago between 1904 and 1907 (Herrick, 1910). Coming from the north of the island of Grenada in the eastern Caribbean, he was first admitted to the Presbyterian Hospital, Chicago, in late December 1904 and a blood test showed the features characteristic of homozygous sickle cell (SS) disease. It was a happy coincidence that he was under the care of Dr James Herrick (Fig 1) and his intern Dr Ernest Irons because both had an interest in laboratory investigation and Herrick had previously presented a paper on the value of blood examination in reaching a diagnosis (Herrick, 1904-05). The resulting blood test report by Dr Irons described and contained drawings of the abnormal red cells (Fig 2) and the photomicrographs, showing irreversibly sickled cells.

People with positive sickle tests were divided into asymptomatic cases, `latent sicklers’, and those with features of the disease, `active sicklers’, and it was Dr Lemuel Diggs of Memphis who first clearly distinguished symptomatic cases called sickle cell anaemia from the latent asymptomatic cases which were termed the sickle cell trait (Diggs et al, 1933).

Prospective data collection in 29 cases of the disease showed sickling in all 42 parents tested (Neel, 1949), providing strong support for the theory of homozygous inheritance. A Colonial Medical Officer working in Northern Rhodesia (Beet, 1949) reached similar conclusions at the same time with a study of one large family (the Kapokoso-Chuni pedigree). The implication that sickle cell anaemia should occur in all communities in which the sickle cell trait was common and that its frequency would be determined by the prevalence of the trait did not appear to fit the observations from Africa. Despite a sickle cell trait prevalence of 27% in Angola, Texeira (1944) noted the active form of the disease to be `extremely rare’ and similar observations were made from East Africa. Lehmann and Raper (1949, 1956) found a positive sickling test in 45% of one community, from which homozygous inheritance would have predicted that nearly 10% of children had SS disease, yet not a single case was found. The discrepancy led to a hypothesis that some factor inherited from non-black ancestors in America might be necessary for expression of the disease (Raper, 1950).

The explanation for this apparent discrepancy gradually emerged. Working with the Jaluo tribe in Kenya, Foy et al (1951) found five cases of sickle cell anaemia among very young children and suggested that cases might be dying at an age before those sampled in surveys. A similar hypothesis was advanced by Jelliffe (1952) and was supported by data from the then Belgian Congo (Lambotte-Legrand Lambotte-Legrand, 1951, Lambotte-Legrand, 1952, Vandepitte, 1952). Although most cases were consistent with the concept of homozygous inheritance, exceptions continued to occur. Patients with a non-sickling parent of Mediterranean ancestry were later recognized to have sickle cell-β thalassaemia (Powell et al, 1950; Silvestroni & Bianco, 1952; Sturgeon et al, 1952; Neel et al, 1953a), a condition also widespread in African and Indian subjects that presents a variable syndrome depending on the molecular basis of the β thalassaemia mutation and the amount of HbA produced.

Phenotypically, there are two major groups in subjects of African origin, sickle cell-β+ thalassaemia manifesting 20-30% HbA and mutations at 229(A,G) or 288(C,T), and sickle cell-β0 thalassaemia with no HbA and mutations at IVS2-849(A,G) or IVS2-1(G,A). In Indian subjects, a more severe β thalassaemia mutation IVS1-5(G,C) results in a sickle cell-β+ thalassaemia condition with 3-5% HbA and a relatively severe clinical course.

Other double heterozygote conditions causing sickle cell disease include sickle cell-haemoglobin C (SC) disease, (Kaplan et al, 1951; Neel et al, 1953b), sickle cellhaemoglobin O Arab (Ramot et al, 1960), sickle cellhaemoglobin Lepore Boston (Stammatoyannopoulos & Fessas, 1963) and sickle cell-haemoglobin D Punjab (Cooke & Mack, 1934). The latter condition was first described in siblings in 1934, who were reinvestigated for confirmation of HbD (Itano, 1951), the clinical features reported (Sturgeon et al, 1955) and who were finally identified as HbD Punjab (Babin et al, 1964), representing a remarkable example of longitudinal observation and investigation in the same family over 30 years.

The maintenance of high frequencies of the sickle cell trait in the presence of almost obligatory losses of homozygotes in Equatorial Africa implied that there was either a very high frequency of HbS arizing by fresh mutations or that the sickle cell trait conveyed a survival advantage in the African environment. There followed a remarkable period in the 1950s when three prominent scientists were each addressing this problem in East Africa, Dr Alan Raper and Dr Hermann Lehmann in Uganda and Dr Anthony Allison in Kenya. It was quickly calculated that mutation rates were far too low to balance the loss of HbS genes from deaths of homozygotes (Allison, 1954a). An increased fertility of heterozygotes was proposed (Foy et al, 1954; Allison, 1956a) but never convincingly demonstrated. Raper (1949) was the first to suggest that the sickle cell trait might have a survival advantage against some adverse condition in the tropics and Mackey & Vivarelli (1952) suggested that this factor might be malaria. The close geographical association between the distribution of malaria and the sickle cell gene supported this concept (Allison, 1954b) and led to an exciting period in the history of research in sickle cell disease.

The first observations on malaria and the sickle cell trait were from Northern Rhodesia where Beet (1946, 1947) noted that malarial parasites were less frequent in blood films from subjects with the sickle cell trait. Allison (1954c) drew attention to this association, concluding that persons with the sickle cell trait developed malaria less frequently and less severely than those without the trait. This communication marked the beginning of a considerable controversy.Two studies failed to document differences in parasite densities between `sicklers’ and `non-sicklers’ (Moore et al, 1954; Archibald & Bruce-Chwatt, 1955) and Beutler et al (1955) were unable to reproduce the inoculation experiments of Allison (1954c). Raper (1955) speculated that some feature of Allison’s observations had accentuated a difference of lesser magnitude and postulated that the sickle cell trait might inhibit the establishment of malaria in non-immune subjects. The conflicting results in these and other studies appear to have occurred because the protective effect of the sickle cell trait was overshadowed by the role of acquired immunity. Examination of young children before the development of acquired immunity confirmed both lower parasite rates and densities in children with the sickle cell trait (Colbourne & Edington, 1956; Edington & Laing, 1957; Gilles et al, 1967) and it is now generally accepted that the sickle cell trait confers some protection against falciparum malaria during a critical period of early childhood between the loss of passively acquired immunity and the development of active immunity (Allison, 1957; Rucknagel & Neel, 1961; Motulsky, 1964). The mechanism of such an effect is still debated, although possible factors include selective sickling of parasitized red cells (Miller et al, 1956; Luzzatto et al, 1970) resulting in their more effective removal by the reticulo-endothelial system, inhibition of parasite growth by the greater potassium loss and low pH of sickled red cells (Friedman et al, 1979), and greater endothelial adherence of parasitized red cells (Kaul et al, 1994).

The occurrence of the sickle cell mutation and the survival advantage conferred by malaria together determine the primary distribution of the sickle cell gene. Equatorial Africa is highly malarial and the sickle cell mutation appears to have arisen independently on at least three and probably four separate occasions in the African continent, and the mutations were subsequently named after the areas where they were first described and designated the Senegal, Benin, Bantu and Cameroon haplotypes of the disease (Kulozik et al, 1986; Chebloune et al, 1988; Lapoumeroulie et al, 1992). The disease seen in North and South America, the Caribbean and the UK is predominantly of African origin and mostly of the Benin haplotype, although the Bantu is proportionately more frequent in Brazil (Zago et al, 1992). It is therefore easy to understand the common misconception held in these areas that the disease is of African origin.

However, the sickle cell gene is widespread around the Mediterranean, occurring in Sicily, southern Italy, northern Greece and the south coast of Turkey, although these are all of the Benin haplotype and so, ultimately, of African origin. In the Eastern province of Saudi Arabia and in central India, there is a separate independent occurrence of the HbS gene, the Asian haplotype. The Shiite population of the Eastern Province traditionally marry first cousins, tending to increase the prevalence of SS disease above that expected from the gene frequency (Al-Awamy et al, 1984). Furthermore, extensive surveys performed by the Anthropological Survey of India estimate an average sickle cell trait frequency of 15% across the states of Orissa, Madhya Pradesh and Masharastra which, with the estimated population of 300 million people, implies that there may be more cases of sickle cell disease born in India than in Africa. The Asian haplotype of sickle cell disease is generally associated with very high frequencies of alpha thalassaemia and high levels of fetal haemoglobin, both factors believed to ameliorate the severity of the disease.

The promotion of sickling by low oxygen tension and acid conditions was first recognized by Hahn & Gillespie (1927) and further investigated by others (Lange et al, 1951; Allison, 1956b; Harris et al, 1956). The morphological and some functional characteristics of irreversibly sickled cells were described (Diggs & Bibb, 1939; Shen et al, 1949), but the essential features of the polymerization of reduced HbS molecules had to await the developments of electron microscopy (Murayama, 1966; Dobler & Bertles, 1968; Bertles & Dobler, 1969; White & Heagan, 1970) and Xray diffraction (Perutz & Mitchison, 1950; Perutz et al, 1951). The early observations on the inducement of sickling by hypoxia led to the first diagnostic tests utilizing sealed chambers in which oxygen was removed by white cells (Emmel, 1917), reducing agents such as sodium metabisulphite (Daland & Castle, 1948) or bacteria such as Escherichia coli (Raper, 1969). These slide sickling tests are very reliable with careful sealing and the use of positive controls, but require a microscope and some expertise in its use. An alternative method of detecting HbS utilizes its relative insolubility in hypermolar phosphate buffers (Huntsman et al, 1970), known as the solubility test. Both the slide sickle test and the solubility test detect the presence of HbS, but fail to make the vital distinction between the sickle cell trait and forms of sickle cell disease. This requires the process of haemoglobin electrophoresis, which detects the abnormal mobility of HbS, HbC and many other abnormal haemoglobins within an electric field.

The contributions of several workers on the determinants of sickling (Daland & Castle, 1948), birefringence of deoxygenated sickled cells (Sherman, 1940) the lesser degree of sickling in very young children which implied that it was a feature of adult haemoglobin (Watson, 1948) led Pauling to perform Tiselius moving boundary electrophoresis on haemoglobin solutions from subjects with sickle cell anaemia and the sickle cell trait. The demonstration of electrophoretic and, hence, implied chemical differences between normal, sickle cell trait and sickle cell disease led to the proposal that it was a molecular disease (Pauling et al, 1949). The chance encounter between Castle and Pauling who shared a train compartment returning from a meeting in Denver in 1945, its background and implications, has passed into the folklore of medical research (Conley, 1980; Feldman & Tauber, 1997).

The nature of this difference was soon elucidated. The haem groups appeared identical, suggesting that the difference resided in the globin, but early chemical analyses revealed no distinctive differences (Schroeder et al, 1950; Huisman et al, 1955). Analyses of terminal amino acids also failed to reveal differences, although an excess of valine in HbS was noted but considered an experimental error (Havinga, 1953). The development of more sensitive methods of fingerprinting combining high voltage electrophoresis and chromatography allowed the identification of the essential difference between HbA and HbS. This method enabled the separation of constituent peptides and demonstrated that a peptide in HbS was more positively charged than in HbA (Ingram, 1956). This peptide was found to contain less glutamic acid and more valine, suggesting that valine had replaced glutamic acid (Ingram, 1957). The sequence of this peptide was shown to be Val-His-Leu-Thr-Pro-Val-Glu-Lys in HbS instead of the Val-His-Leu-Thr-Pro-Glu-Glu-Lys in HbA (Hunt & Ingram, 1958), a sequence which was subsequently identified as the amino-terminus of the b chain (Hunt & Ingram, 1959). This amino acid substitution was consistent with the genetic code and was subsequently found to be attributable to the nucleotide change from GAG to GTG (Marotta et al, 1977).

Haemolysis and anaemia. The presence of anaemia and jaundice in the first four cases suggested accelerated haemolysis, which was supported by elevated reticulocyte counts (Sydenstricker et al, 1923) and expansion of the bone marrow (Sydenstricker et al, 1923; Graham, 1924). The bone changes of medullary expansion and cortical thinning were noted in early radiological reports (Vogt & Diamond, 1930; LeWald, 1932; Grinnan, 1935). Drawing on a comparison of sickle cell disease and hereditary spherocytosis, Sydenstricker (1924) introduced the term `haemolytic crisis’ that has persisted in the literature to this day, despite the lack of evidence for such an entity in sickle cell disease. The increased requirements of folic acid and the consequence of a deficiency leading to megaloblastic change was not noted until much later (Zuelzer & Rutzky, 1953; Jonsson et al, 1959; MacIver & Went, 1960).

The haemoglobin level in SS disease of African origin is typically between 6 and 9 g/dl and is well tolerated, partly because of a marked shift in the oxygen dissociation curve (Scriver & Waugh, 1930; Seakins et al, 1973) so that HbS within the red cell behaves with a low oxygen affinity. This explains why patients at their steady state haemoglobin levels rarely show classic symptoms of anaemia and fail to benefit clinically from blood transfusions intended to improve oxygen delivery.

Graham R. Serjeant
Sickle Cell Trust, Kingston, Jamaica
Brit J Haem 2001; 112: 3-18

The Immune Haemolytic Anaemias

The growth in knowledge of the scientific basis of haemolytic anaemias, which have been a main interest of the author, has been remarkable, as have consequent advances in the practice of medicine since the mid-1930s. At that time, the cause and mechanism of important disorders such as the acquired antibody determined (immune) haemolytic anaemias, haemolytic disease of the newborn, hereditary spherocytosis and paroxysmal nocturnal haemoglobinuria were unknown or but partially understood.

According to Crosby (1952), William Hunter of London, in an article on pernicious anaemia published in 1888, was the first to use the term `haemolytic’ to denote an anaemia caused by excessive blood destruction. By the turn of the century, the term was being widely used in clinical literature. Peyton Rous, in his comprehensive review `Destruction of the red blood corpuscles in health and disease’ (Rous, 1923), concluded that the generally held view in the early 1930s was that about one-fifteenth of the erythrocyte mass was destroyed daily. Rous was aware of the pioneer work of Winifred Ashby (1919), who, by following the survival of serologically distinct but compatible transfused erythrocytes, had found that normal erythrocytes might live for up to 100 d in the recipients’ circulation. Subsequent work using radioactive chromium (51Cr) as an erythrocyte label, showed that Ashby’s data and conclusions were in fact correct, i.e. that normal erythrocytes in health circulate in the peripheral blood for approximately 110 d. Erythrocyte labelling with 51Cr also had a further advantage over the Ashby method in addition to enabling the life-span of the patients’ erythrocytes to be assessed in the circulation by surface counting, to detect and measure the accumulation of radioactivity in the spleen and liver, and thereby assess the organs’ role in haemolysis

In the first decade of the twentieth century Widal et al (1908a) and Le Gendre & Brulea (1909) reported that autohaemoagglutination was a striking finding in some cases of icteare heamolytique acquis, and also Chauffard & Trosier (1908) and Chauffard & Vincent (1909) had described the presence of haemolysins in the serum of patients suffering from intense haemolysis. The conclusion was that abnormal immune processes, i.e. the development of auto-antibodies damaging the patients’ own erythrocytes, might play a part in the genesis of some cases of acquired haemolytic anaemia. This was indeed antedated by the classic observations of Donath & Landsteiner (1904) and Eason (1906) on the mechanism of haemolysis in paroxysmal cold haemoglobinuria.

That blood might auto-agglutinate when chilled had been described by Landsteiner (1903) and that an unusual degree of the phenomenon might complicate some types of respiratory disease was reported by Clough & Richter (1918) and later by Wheeler et al (1939). A few years later Peterson et al (1943) and Horstmann & Tatlock (1943) reported that cold auto-agglutinins at high titres were frequently found in the serum of patients who had suffered from the then so called primary atypical pneumonia.

Stats & Wasserman’s (1943) review on cold haemagglutination was a valuable contribution to contemporary knowledge. They listed in a table as many as 94 references to papers published between 1890 and 1943 in which cold haemagglutination had been described. In 32 of the papers the patients referred to had suffered from increased haemolysis

Recognition that cold auto-antibodies played an important role in the pathogenesis of some cases of haemolytic anaemia led to the concept that auto-immune haemolytic anaemia (AIMA) might usefully be classified into warm antibody or cold-antibody types, according to whether the patient is forming (warm) antibodies which react (perhaps optimally) at body temperature or (cold) antibodies which react strongly at low temperatures (e.g. 48C) but progressively less well as the temperature is raised and are perhaps inactive at 37oC. The clinical syndrome suffered by the patient would depend not only on the amount of antibody produced but also on its temperature requirement. Another important advance in understanding has been the realization that both types of AIHA could develop in association with a wide range of underlying disorders (secondary AIHA) as well as `idiopathically’, i.e. for no obvious cause (primary AIHA). The author’s own experience was summarized in a review (Dacie & Worlledge, 1969): 99 out of 210 cases of warm AIHA were judged to be secondary as were 39 out of 85 cases of cold AIHA. Petz & Garratty (1980), summarized the data from six centres: 55% out of a total of 656 cases had been reported as secondary. They listed the disorders with which warm antibody AIHA had been associated as chronic lymphocytic leukaemia, Hodgkin’s disease, non-Hodgkin’s lymphomas, thymomas, multiple myeloma, Waldenstrom’s macroglobulinaemia, systemic lupus erythematosus, scleroderma, rheumatoid arthritis, infectious disease/ childhood viral disorders, hypogammaglobulinaemia, dysglobulinaemias, other immune deficiency syndromes, and ulcerative colitis.

Conley (1981), in an interesting review of warm-antibody AIHA patients seen at the Johns Hopkins Hospital, emphasized how important it was to carry out a careful enquiry into the patient’s past history and also to undertake a prolonged follow-up. He stated that a retrospective review of 33 patients whose illnesses in the past have been designated `idiopathic” had revealed an associated immunologically related disorder in 19 of them. An additional three patients had developed a lymphoma 2±10 years after they had developed AIHA. As already referred to, warm-antibody AIHA is now known to complicate a wide range of underlying diseases, particularly malignant lymphoproliferative disorders, other auto-immune disorders and immune deficiency syndromes. What proportion of patients suffering from a lymphoproliferative disorder develop AIHA is an interesting question. Duehrsen et al (1987) stated that this had occurred in 12 out of 637 patients. Early data on the incidence of a positive DAT in SLE were provided by Harvey et al (1954) – in six out of 34 patients tested the DAT had been positive. Later, Mongan et al (1967), who had studied a large number of patients suffering from a variety of connective tissue disorders, reported that the DAT had been positive in 15 out of 23 patients with SLE, none of whom, however, had suffered from overt haemolytic anaemia. It has also been realized since the 1960s that warm-antibody AIHA may develop in patients suffering from a variety of immune deficiency syndromes, both congenital and acquired.

It was in the mid-1960s that it was realized that, in a significant proportion of patients thought to have `idiopathic’ warm-antibody AIHA, the development of the causal auto-antibodies had been triggered in some way by a drug the patient was taking. The first drug implicated was the antihypertensive drug a-methyldopa (Aldomet) (Carstairs et al, 1966a,b). Following the finding that treating hypertensive patients with a-methyldopa led to the formation of anti-erythrocyte auto-antibodies in a significant percentage of patients, renewed interest was taken in the possibility that other drugs might have the same effect. Two main hypotheses have been advanced in relation to how certain drugs in some patients appear to have caused the development of anti-erythrocyte auto-antibodies. One hypothesis was that the drug or its metabolites act on the immune system so as to impair immune tolerance; the other was that the drug affects antigens at the erythrocyte surface in such a way that a normally active immune system responds by developing anti-erythrocyte antibodies. Clearly, too, the patient’s individuality must be an important factor, for only a proportion of patients receiving the same dosage of the offending drug for the same period of time develop a positive DAT and only a small percentage develop overt AIHA.

An interesting development in the history of the immune haemolytic anaemias was the realization in the mid-1950s that, rather rarely, haemolysis was brought about by the patient developing antibodies that were directed against a drug the patient had been taking and that the erythrocytes were in some way secondarily involved. The first drug to be implicated was Fuadin (stibophen), which had been used to treat a patient with schistosomiasis (Harris, 1954, 1956). The patient’s serum contained an antibody that agglutinated his own or normal erythrocytes and/or sensitized them to agglutination by antiglobulin sera; however, this occurred only in the presence of the drug.

In the late 1940s, several accounts of patients with AIHA who had persistently low platelet counts were published, e.g. Fisher (1947) and Evans & Duane (1949); and it was suggested that the patients might have been forming autoantibodies directed against platelets. This concept was further developed by Evans et al (1951). Eight out of their 18 patients with AIHA were thrombocytopenic; four had clinically obvious purpura. Evans et al (1951) suggested that there exists `a spectrum-like relationship between acquired haemolytic anaemia and thrombocytopenic purpura’; also that `on the one hand, acquired haemolytic anaemia with sensitization of the red cells is often accompanied with thrombocytopenia, while, on the other hand, primary thrombocytopenic purpura is frequently accompanied with red cell sensitization with or without haemolytic anaemia’. Many further case reports of AIHA accompanied by severe thrombocytopenia have since been published

There are two features in the blood film of a patient with an acquired haemolytic anaemia which indicate that he or she is suffering from AIHA; one is auto-agglutination, the other is erythrophagocytosis. Spherocytosis, although often present to a marked degree, is of course found in other types of haemolytic anaemia.

The pioneer French observations on auto-agglutination already referred to were generally overlooked until the late 1930s, and serological studies seem seldom to have been undertaken until the publication of Dameshek & Schwartz’s (1938b) report in which they described the presence of `haemolysins’ in cases of acute apparently acquired haemolytic anaemia. Dameshek & Schwartz (1940) summarized contemporary knowledge in an extensive review. They concluded that it was not improbable that haemolysins of various types and `dosages’ were in fact responsible for many cases of human haemolytic anaemias, including congenital haemolytic anaemia, which they suggested might be caused by the `more or less continued action of an haemolysin’.

Six years were to pass before the concept that an abnormal immune mechanism played a decisive role in some cases of acquired haemolytic anaemia was clearly demonstrated by Boorman et al (1946), who reported that the erythrocytes of five patients with acquired acholuric jaundice had been agglutinated by an antiglobulin serum, i.e. that the newly described antiglobulin reaction or Coombs test (Coombs et al, 1945) was positive, while the test had been negative in 28 patients suffering from congenital acholuric jaundice. This work aroused great interest and was soon confirmed.

Until the 1950s, the auto-antibodies responsible for AIHA were generally concluded to be `non-specific’. According to Wiener et al (1953), `Red cell auto-antibodies react not only with the individual’s own red cells but also with the erythrocytes of all other human beings. The substances on the red blood cell envelope with which the auto-antibodies combine are agglutinogens like the ABO, MN and RhHr systems, except that, in the former case, the blood factors with which the auto-antibodies react are not type specific but are shared by all human beings.’ They suggested that the auto-antibodies might be directed to the `nucleus of the RhHr substance’. Earlier work had, however, indicated that the sensitivity of normal group-compatible erythrocytes to a patient’s auto-antibody might vary considerably (Denys & van den Broucke, 1947; Kuhns & Wagley, 1949). That auto-antibodies might have a clearly defined Rh specificity, e.g. anti-e, was described by Race & Sanger (1954) in the second edition of their book. Referring to Wiener et al (1953), they wrote: `This beautifully clear investigation made the present authors realize that a curious result obtained by one of them (Ruth Sanger) in 1953 in Australia had after all been true; the serum of a man who had died of a haemolytic anaemia 3000 miles away contained anti-e; his cells were clearly CDe-cde’. A similar finding, i.e. an auto-anti-e, was described by Weiner et al (1953).

A further development in the unravelling of a complicated story was the realization that some of the antibodies which appeared to be specific were reacting with more basic antigens, although showing a preference for specific antigens, i.e. some specific auto-antibodies appeared to be less specific than their allo-antibody counterparts. Moreover, some antibodies, reacting with specific antigens, have been shown to be partially or completely absorbable by antigen negative cells.

Many apparently `non-specific’ antidl antibodies have been shown to be not strictly `nonspecific’ but to react with antigens of very high frequency, e.g. to be anti-Wrb, anti-Ena, anti-LW or anti-U. Issitt et al (1980)) listed six additional very common antigens that had been identified as targets for anti-dl auto-antibodies, i.e. Hr, Hro, Rh34, Rh29, Kpb and K13.

In relation to human acquired haemolytic anaemia, the discovery in the late 1940s and 1950s that many cases were apparently brought about by the development of damaging anti-erythrocyte antibodies led to intense interest and speculation into the why and how of auto-antibody formation. Of seminal importance at the time were the experiments and theoretical arguments of Burnet (Burnet & Fenner, 1949; Burnet, 1957, 1959, 1972) and the studies on transplantation immunity of Medawar (Billingham et al, 1953; Medawar, 1961). Of particular interest, too, was the report by Bielschowsky et al (1959) of the occurrence of AIHA in an inbred strain of mice – the NZB/BL strain. Remarkably, by the time the mice were 9-months-old the DAT was positive in almost every mouse. Burnet (1963) referred to the gift of the mice to the Walter and Eliza Hall Institute of Medical Research, Melbourne as `the finest gift the Institute has ever received’.

Exactly how is it that auto-antibodies reacting with an erythrocyte surface antigen result in the cell’s premature destruction? The possible role of auto-agglutination in bringing about haemolysis was emphasized by Castle and colleagues as the result of a series of studies carried out in the 1940s and 1950s. As summarized by Castle et al (1950), an antibody which appears to be incapable of causing `lysis in vitro might bring about the following sequence of events in vivo. (1) Red cell agglutination in the peripheral blood; (2) red cell sequestration and separation from plasma in tissue capillaries; (3) ischaemic injury of tissue cells with release of substances that increase the osmotic and mechanical fragilities of red cells locally; (4) local osmotic lysis of red cells or subsequent escape of mechanically fragile red cells into the blood stream where the traumatic motion of the circulation causes their destruction’.

We can expect, as the years pass, that more and more will be known as to the intricate mechanisms that bring about self-tolerance and the mechanisms underlying the occurrence of auto-immune disorders in general, including the role of infectious agents, drugs and genetic factors. Patients with immune haemolytic anaemias can be expected to benefit from the new knowledge; for in parallel with a better understanding as to how immune self-tolerance breaks down will hopefully be the development of more effective drugs and therapies aimed at controlling the breakdown.

The Immune Haemolytic Anaemias: A Century of Exciting Progress in Understanding.  Sir John Dacie, Emeritus Professor of Haematology.
Brit J Haem 2001; 114: 770-785.

A History of Pernicious Anaemia

This is a review of the ideas and observations that have led to our current understanding of pernicious anaemia (PA). PA is a megaloblastic anaemia (MA) due to atrophy of the mucosa of the body of the stomach which, in turn, is brought about by autoimmune factors.

A case report by Osler & Gardner (1877) in Montreal could be that of PA. This anaemic patient had numbness of the fingers, hands and forearms; the red blood cells were large; at autopsy the gastric mucosa appeared atrophic and the marrow had large numbers of erythroblasts with finely granular nuclei. The increased marrow cellularity had also been noted by Cohnheim (1876).

Ehrlich (1880) (Fig 1) distinguished between cells he termed megaloblasts present in the blood in PA from normoblasts present in anaemia as a result of blood loss. Not only were large red blood cells noted in PA, but irregular red cells, ? poikilocytes, were reported in wet blood preparations by Quincke (1877). Megaloblasts in the marrow during life were first noted by Zadek (1921). Hypersegmented neutrophils in peripheral blood in PA were described by Naegeli (1923) and came to be widely recognized after Cooke’s study (Cooke, 1927). The giant metamyelocytes in the marrow were described by Tempka & Braun (1932).

Paul Ehrlich

Paul Ehrlich

Fig 1. Paul Ehrlich (Wellcome Institute Library, London).

The association between PA and spinal cord lesions was described by Lichtheim (1887) and a full account was published by Russell et al (1900), who coined the term `subacute combined degeneration of the spinal cord’ (SCDC) although they were not convinced of its relation to PA. Arthur Hurst at Guy’s Hospital, London, confirmed the association of the neuropathy with PA and added, too, the association of loss of hydrochloric acid in the gastric juice (Hurst & Bell, 1922). Cabot (1908) found that numbness and tingling of the extremities were present in almost all of his 1200 patients and 10% had ataxia. William Hunter (1901) noted the prevalence of a sore tongue in PA, which was present in 40% of Cabot’s series.

In 1934, the Nobel Prize in medicine and physiology was awarded to Whipple, Minot and Murphy. Was there ever an award more deserved? They saved the lives of their patients and pointed the way forward for further research. What was there in liver that was lacking in patients with PA? The effect of liver in restoring the anaemia in Whipple’s iron-deficient dogs was by supplying iron which is  abundant in liver.

Liver given by mouth also provides Cbl and folic acid. But patients with PA cannot absorb Cbl, although some 1% of an oral dose can cross the intestinal mucosa by passive diffusion; this, presumably, is what happened when large amounts of liver were eaten. Beef liver contains about 110 mg of Cbl per 100 g and about 140 mg of folate per 100 g. Cbl is stable and generally resistant to heat; folate is labile unless preserved with reducing agents. The daily requirement of Cbl by man is l-2 mg. The liver diet, if consumed, had enough of these haematinics to provide a response in most MAs.

George Richard Minot

George Richard Minot

George Richard Minot (Wellcome Institute Library, London).

The availability of liver extracts brought about interest in the nature of the haematological response. An optimal response required a peak rise of reticulocytes 5±7 d after the injection of liver extract and the height of the peak was greatest in those with severe anaemia; the flood of reticulocytes was as a result of a synchronous maturation of a vast number of megaloblasts into red cells. There is a steady rise in the red cell count to reach 3 x 1012/l in the 3rd week (Minot & Castle, 1935). Many liver extracts did not have enough antianaemic factor to achieve this and some assayed by the author had only 1-2 mg of Cbl.  It took another 22 years for a pure antianaemic factor to be isolated, although, admittedly, the Second World War intervened; in 1948, an American group led by Karl Folkers and an English group led by E. Lester-Smith published, within weeks of each other, the isolation of a red crystalline substance termed vitamin B12 and subsequently renamed cobalamin.

The structure of this red crystalline compound was studied by the nature of its degradation products and by X-ray crystallography. It soon became apparent that there was a cobalt atom at the heart of the structure and this heavy atom was of great aid to the crystallographers, so much so that, with additional information from the chemists, they were the first to come up with the complete structure. To quote Dorothy Hodgkin: `To be able to write down a chemical structure very largely from purely crystallographic evidence on the arrangement of atoms in space – and the chemical structure of a quite formidably large molecule at that – is for any crystallographer, something of a dream-like situation’. As Lester-Smith (1965) pointed out, it also required some 10 million calculations. In 1964, Dorothy Hodgkin was awarded the Nobel Prize for chemistry.

Barker et al (1958) published an account of the metabolism of glutamate by a Clostridium. The glutamate underwent an isomerization and an orange-coloured co-enzyme was involved that turned out to be Cbl with a deoxyadenosyl group attached to the cobalt.

This Cbl co-enzyme, deoxyadenosylCbl, is the major form of Cbl in tissues; it is also extremely sensitive to light, being changed rapidly to hydroxoCbl. DeoxyadenosylCbl is concerned with the metabolism of methylmalonic acid in man (Flavin & Ochoa, 1957). The other functional form of Cbl is methylCbl involved in conversion of homocysteine to methionine (Sakami & Welch, 1950). Both these pathways are impaired in PA in relapse.

Cbl consists of a ring of four pyrrole units very similar to that present in haem. These, however, have the cobalt atom in the centre instead of iron and the ring is called the corrin nucleus. The cobalamins have a further structure, a base, termed benzimidazole, set at right angles to the corrin nucleus and this may have a link to the cobalt atom (base on position).

By the time Cbl had been isolated from liver it was already known that it was also present in fermentation flasks growing bacteria such as streptomyces species. Other organisms gave higher yields so that kilogram quantities of pure Cbl were obtained; these sources have replaced liver in the production of Cbl. By adding radioactive form of cobalt to the fermentation flasks instead of ordinary cobalt, labelled Cbl became available (Chaiet et al, 1950). The importance of labelled Cbl is that it made it possible to carry out Cbl absorption tests in patients, to design isotope dilution assays for serum Cbl, to design ways of assaying intrinsic factor (IF), to detect antibodies to IF and even to measure glomerular filtratration rate, as free Cbl is excreted by the glomerulus without any reabsorption by the renal tubules.

William Castle at the Thorndike Memorial Laboratory, Boston City Hospital, devised experiments to explore the relationship between gastric juice, the anti-anaemic factor that Castle assumed, correctly, was also present in beef, and the response in PA. The question Castle asked was `Was it possible that the stomach of the normal person could derive something from ordinary food that for him was equivalent to eating liver?’.

The experiment in untreated patients with PA consisted of two consecutive periods of 10 d or more during which daily reticulocyte counts were made. During the first period of 10 d, the PA patient received 200 g of lean beef muscle (steak) each day. There was no reticulocyte response. During the second period, the contents of the stomach of a healthy man were recovered 1 h after the ingestion of 300 g of steak; about 100 g could not be recovered. The gastric contents were incubated for a few hours until liquefied and then given to the PA patient through a tube. This was done daily. On day 6 there was a rise in reticulocytes reaching a peak on day 10, followed by a rise in the red cell count. The response was similar to that obtained with large amounts of oral liver.

Thus, Castle concluded that a reaction was taking place between an unknown intrinsic factor (IF) in the gastric juice and an unknown extrinsic factor in beef muscle. Whereas Minot & Murphy (1926) found that 200-300 g of liver daily was needed to get a response in PA, 10 g liver was adequate when incubated with 10-20 ml normal gastric juice (Reiman & Fritsch, 1934). Castle’s extrinsic factor is the same as the anti-anaemic factor that is Cbl, and IF is needed for its absorption. Presumably the gastric juice in PA lacks IF.

The elegant studies of Hoedemaeker et al (1964) in Holland using autoradiography of frozen sections of human stomach incubated with [57Co]-Cbl showed that IF was produced in the gastric parietal cell. The binding of Cbl to

the parietal cell was abolished by first incubating the section with a serum containing antibodies to IF. The parietal cell in man is thus the source of both hydrochloric acid and IF. The parietal cell is the only source of IF in man as a total gastrectomy is invariably followed by a MA due to Cbl deficiency. IF is a glycoprotein with a molecular weight of 45 000.

Assay of protein fractions of serum after electrophoresis showed that endogenous Cbl is in the position of α-1 globulin. Chromatography of serum after addition of [57Co]-Cbl on Sephadex G-200 showed that Cbl was attached to two proteins, one eluting before the albumin termed transcobalamin I (TCI) and the other after the albumin termed transcobalamin II (TCII). Charles Hall showed that, when labelled Cbl given by mouth is absorbed, it first appears in the position of TCII and later in the position of TCI as well (Hall and Finkler, l965). They concluded that TCII is the prime Cbl transport protein carrying Cbl from the gut into the blood and then to the liver from where it is redistributed by both new TCII as well as TCI. Congenital absence of a functional TCII causes a severe MA in the first few months of life owing to an inability to transport Cbl. Most of the Cbl in serum is on TCI because it has a relatively long half-life of 9±10 d, whereas the half-life of TCII is about 1.5 h. Thus, in assaying the serum Cbl level, it is mainly TCI-Cbl that is being assayed.

With the availability of labelled Cbl, Cbl absorption tests began to be widely used in the 1950s. The commonest method was the urinary excretion test described by Schilling (1953). Here, an oral dose of radioactive Cbl is followed by an injection of 1000 mg of cyano-Cbl. The free cyano-Cbl is largely excreted into the urine over the next 24 h and carries with it about one third of the absorbed labelled Cbl.

Parietal cell antibodies (Taylor et al, 1962) are present in serum in 76-93% of different series of PAs and in the serum of 36% of the relatives of PA patients. The antibody is present in sera from 32% of patients with myxoedema, 28% of patients with Graves’ disease, 20% of relatives of thyroid patients and 23% of patients with Addison’s disease. Parietal cell antibodies are found in between 2-16% of controls, the high 16% figure being in elderly women. There is a higher frequency of PA in women, the female to male ratio being 1.7 to 1.0. The parietal cell antibody is probably important in the production of gastric atrophy. Thyroid antibodies are present in sera from 55% of PAs, in sera from 50% of PA relatives, in 87% of sera from myxoedema patients, in 53% of sera in Graves’ disease and in 46% of relatives of patients with thyroid disease.

There is a high frequency of PA among those disorders that have antibodies against the target organ. Thus, among 286 patients with myxoedema, 9.0% also had PA (Chanarin, 1979), as compared with a frequency of PA of about 1 per 1000 (0.01%) in the general population. Of 102 consecutive patients with vitiligo,
eight also had PA.

Patients with acquired hypogammaglobulinaemia are unable to make humoral antibodies; nevertheless, one third have PA as well. This cannot be as a result of action of IF antibodies and must be because of specific cell-mediated immunity. Tai & McGuigan (1969) demonstrated lymphocyte transformation in the presence of IF in six out of 16 PA patients and Chanarin & James (1974) found 10 out of 51 tests were positive.

Twenty-five patients with PA were tested for the presence of humoral IF antibody in serum and gastric juice and for cell-mediated immunity against IF. All but one gave positive results in one or more tests. It was concluded that these findings establish the autoimmune nature of PA and that the immunity is not merely an interesting byproduct.

Patients with PA treated with steroids show a reversal of the abnormal findings characterizing the disease. If they are still megaloblastic, the anaemia will respond in the first instance (Doig et al, 1957), but in the longer term Cbl neuropathy may be precipitated. The absorption of Cbl improves and may become `normal’ (Frost & Goldwein, 1958). There is a return of IF in the gastric juice (Kristensen and Friis, 1960) and a decline in the amount of IF antibody in serum (Taylor, 1959). In some patients there is return of acid in the gastric juice. Gastric biopsy shows a return of parietal and chief cells (Ardeman & Chanarin, 1965b; Jeffries, 1965). All this is as a result of suppression of cell-mediated immunity against the parietal cell and against IF. Withdrawal of steroids leads to a slow return to the status quo.

The author has dipped freely into the two volumes by the late M. M. Wintrobe. These are: Wintrobe, M.M. (1985) Hematology, the Blossoming of a Science. Lea & Febinge

A History of Pernicious Anaemia
I. Chanarin, Richmond, Surrey
Brit J Haem 111: 407-415
History of Folic Acid

1928 Lucy Wills studied macrocytic anaemia in pregnancy in Bombay, India

1932 Janet Vaughn studied macrocytic anemia associated with coeliac disease and idiopathic steatorrhea (1932) showed a response to marmite

1941 Folic acid extracted from spinach and is a growth factor for S. Faecalis

1941 pteroylglutamic acid synthesized at Amer Cyanamide – Pteridine ring, paraminobenzoic acid, glutamine –  PGA differed from natural compound in some respects

1945 PGA resolved the macrocytic anemia, but not the neuropathy

1979 Stokstad and associates at Berkeley obtained the first purified mammalian enzymes involved in synthesis

Folate antagonists inhibit tumor growth (Hitchings and Elion)(Nobel)

  • Misincorporation of uracil instead of thymine into DNA

Sidney Farber introduced Aminopterine and also Methotrexate for treatment of childhood lymphoblastic leukemia

  • MTX inhibits DHFR enzyme (dihydrofolate reductase) necessary for THF

Wellcome introduces trimethoprim (antibacterial), and also pyramethoprime (antimalarial)

Homocysteine isolated by Du Vineaud, but it was not noticed

Finkelstein and Mudd demonstrated the importance of remethylation for tHy and worked out the transsulfuration pathway

  1. Function of methyl THF is remethylation of homocysteine
  2. Synthesized by MTHFR
Metabolism of folate

Metabolism of folate

Metabolism of folate

Allosterically regulated by S-adenosyl methionine (Stokstad)

MTHF also inhibits glycine methyl transferase controlling excess SAM – transmethylation

JD Finkelstein

JD Finkelstein

James D Finkelstein

  • Homocysteinuria – mental retardation, skeletal malformation, thromboembolic disease; deficiency of cystathionine synthase (controls trans-sulfuration)
  • NTDs – pregnancy
  • Hyperhomocysteinemia and VD

AD Hoffbrand and DG Weir
Brit J Haem 2001; 113: 579-589

The History of Haemophilia in the Royal Families of Europe Queen Victoria.

On 17 July 1998 a historic ceremony of mourning and commemoration took place in the ancestral church of the Peter and Paul Fortress in St Petersburg. President Boris Yeltsin, in a dramatic eleventh-hour change of heart, decided to represent his country when the bones of the last emperor, Tsar Nicholas II, and his family were laid to rest 80 years to the day after their assassination in Yekaterinberg (Binyon, 1998). He described it as ‘ironic that the Orthodox Church, for so long the bedrock of the people’s faith, should find it difficult to give this blessing the country had expected’. ‘I have studied the results of DNA testing carried out in England and abroad and am convinced that the remains are those of the Tsar and his family’ (The Times, 1998a). Unfortunately, politicians and the hierarchy of the Russian Orthodox Church had argued about what to do with the bones previously stored in plastic bags in a provincial city mortuary. Politics, ecclesiastical intrigue, secular ambition, and emotions had fuelled the debate. Yeltsin and the Church wanted to honour a man many consider to be a saint, but many of the older generation are opposed to the rehabilitation of a family which symbolizes the old autocracy.

Our story starts, almost inevitably, with Queen Victoria of England who had nine children by Albert, Prince of Saxe-Coburg-Gotha. Victoria was certainly an obligate carrier for haemophilia as over 20 individuals subsequently inherited the condition (Figs 1 and 2). Princess Alice (1843–78) was Victoria’s third child and second daughter. Having married the Duke of Hesse at an early age, Alice went on to have seven children, one of whom, Frederick (‘Frittie’) was a haemophiliac who died at the age of 3 following a fall from a window.

Prince Leopold with Sir William Jenner at Balmoral in 1877

Prince Leopold with Sir William Jenner at Balmoral in 1877

Prince Leopold with Sir William Jenner at Balmoral in 1877. (Hulton Deutsch Collection Ltd.)

Alexandra was the sixth child and was only 6 years old when her mother and youngest sister died. ‘Sunny’, as she became known, was a favourite of Queen Victoria, who as far as possible directed her upbringing from across the channel: Alexandra (Alix) was forced to eat her baked apples and rice pudding with the same regularity as her English cousins. Alix visited her older sister Elizabeth (Ella) on her marriage to Grand Duke Serge and met Tsarevich Nicholas for the first time: she was 12 and not impressed. Five years later they met again and Alix fell in love, but by now she had been confirmed in the Lutheran Church and religion became the solemn core of her life.

Victoria had other aspirations for Alix. She hoped that she would marry her grandson Albert Victor (The Duke of Clarence) and the eldest son of the Prince of Wales (later Edward VII). The Duke was an unimpressive young man who was somewhat deaf and had limited intellectual abilities. If this arrangement had proceeded then Alix’s haemophilia carrier status would have been introduced into the British Royal Family and the possibility of a British monarch with haemophilia might have become a reality; however, the Duke died in 1892.

Nicholas and Alexandra. Alix and Nicholas were married in 1894 one week after the death of Nicholas’s father (Alexander III). In the same way that Victoria, with her personal aspirations of a marriage between Alix and the Duke of Clarence, had not considered the possibility of haemophilia, neither did the St Petersburg hierarchy consider a marriage to Nicholas undesirable. Haemophilia was already well recognized in Victoria’s descendants. Her youngest son, Leopold, had already died, as had Frittie her grandson. The inheritance of haemophilia had been known for some time since its description by John Conrad Otto (Otto, 1803). However, it was as late as 1913 before the first royal marriage was declined because of the risk of haemophilia, when the Queen of Rumania decided against an association between her son, Crown Prince Ferdinand, and Olga, the eldest daughter of Nicholas and Alexandra. The Queen of Rumania was herself a granddaughter of Queen Victoria and therefore a potential haemophilia carrier!

Alix was received into the Russian Orthodox Church, taking the name of Alexandra Fedorova. The first duty of a Tsarina was to maintain the dynasty and produce a male heir, but between 1895 and 1901 Alix produced four princesses, Olga, Tatiana, Maria and Anastasia. Failure to produce a son made Alix increasingly neurotic and she had at least one false pregnancy. However, in early 1904 she was definitely pregnant.

For a month or so all seemed well with little Alexis, but it was then noticed that the Tsarevitch was bleeding excessively from the umbilicus (a relatively uncommon feature of haemophilia). At first the diagnosis was not admitted by the parents, but eventually the truth had to be faced although even then only by the doctors and immediate family. Alix was grief stricken: ‘she hardly knew a day’s happiness after she realized her boy’s fate’. As a newly diagnosed haemophilia carrier she dwelt morbidly on the fact that she had transmitted the disease. These feelings are well known to some haemophiliac mothers but the situation was different in Russia in the early twentieth century. The people regarded any defect as divine intervention. The Tsar, as head of the Church and leader of the people, must be free of any physical defect, so the Tsarevich’s haemophilia was concealed. The family retreated into greater isolation and were increasingly dominated by the young heir’s affliction (Fig 3).

Up to a third of haemophiliac males do not have a family history of the condition. This is usually thought to be the result of a relatively high mutation rate occurring in either affected males or female carriers. None of Queen Victoria’s ancestors, for many generations, showed any evidence of haemophilia. Victoria was therefore either a victim of a mutation, or the Duke of Kent was not her father.The mutation is unlikely to have been in her mother, Victoire, who had a son and daughter by her first marriage, and there is no sign of haemophilia in their numerous descendants.

Victoire was under considerable pressure to produce an heir. The year before Victoria was born, Princess Charlotte, the only close heir to the throne, had died and the Duke of Kent had somewhat reluctantly agreed to marry Victoire with the aim of producing an heir. The postulate that the Queen’s gardener had a limp has not been substantiated!

The Duke of Kent had no evidence of haemophilia (he was 51 when Victoria was born) but did inherit another condition from his father (George III): porphyria. While a young man in Gibralter he suffered bilious attacks which were recognized as being similar to his father’s complaint.

Had Queen Victoria carried the gene for porphyria we might expect that she would have at least as many descendants with this condition as had haemophilia. Until recently only two possible cases of porphyria have been suggested amongst Victoria’s descendants: Kaiser Wilhelm’s sister and niece (MacAlpine & Hunter, 1969), but they could have inherited it from their Hohenzollern ancestor, Frederick the Great. A recent television programme (Secret History, 1998) claims to have identified two more cases in Victoria’s descendants, Princess Victoria, the Queen’s eldest daughter, and Prince William of Gloucester, nephew of George V. If these two cases are correct then they would tend to confirm that Victoria was indeed the daughter of the Duke of Kent, but the apparent lack of more cases in Victoria’s extended family is difficult to understand. The gene for acute intermittent porphyria has been isolated on chromosome 11. There is still plenty of scope for further genetic analysis on the European Royal Families!

We can only speculate as to the impact on European events over the last 150 years if the marriages within the Royal houses had been different. What is evident is the dramatic effect of haemophilia on the Royal Princes and their families.

Empress Alexandra at the Tsarevich’s bedside during a haemophiliac crisis

Empress Alexandra at the Tsarevich’s bedside during a haemophiliac crisis

Empress Alexandra at the Tsarevich’s bedside during a haemophiliac crisis in 1912. (Radio Times Hulton Picture Library.)

Richard F. Stevens
Royal Manchester Children’s Hospital
Brit J Haem 1999, 105, 25–32

`The longer you can look back ± the further you can look forward’: Winston Churchill in an address to The Royal College of Physicians, London 1944. At the time that Churchill was speaking in 1944, leukaemia was a fatal disease that had been identified 100 years before. The disease was described as the dreaded leukaemias, sinister and poorly understood.

Thomas Hodgkin chose a career in medicine and enrolled as a pupil at Guy’s Hospital in London. Being a Quaker, however, he could not enter the English universities of Oxford and Cambridge and decided to follow the medical courses at Edinburgh. At that times, Aristotelian and Hippocratic medicine were greatly influencing British physicians. Hodgkin, still a medical student, wrote a paper `On the Uses of the Spleen’ where he reported his beliefs on the purposes of the spleen: to regulate fluid volume, clean impurities from the body, supply expandability to the portal system. The subject was a presage of the disease that bears his name.

Hodgkin interrupted his studies at Edinburgh to spend a year in Paris where he met many people who had a great influence in his life and future activities. Among them, were Laennec (Hodgkin played an important role in bringing the stethoscope to Great Britain); Baron von Humboldt who introduced Hodgkin to the field of anthropology; Baron Cuvier, a distinguished anatomist and palaeontologist; and Thomas A. Bowditch, whose expeditions to Africa had a great impact on Hodgkin’s future activities.

In 1825, Thomas Hodgkin returned to London to join the staff at Guy’s Hospital, and in 1826 he was made `Inspector of the Dead’ and `Curator of the Museum of Morbid Anatomy’. In developing the museum he had accumulated, by 1829, over 1600 specimens demonstrating the effects of disease. The correlation of clinical disease to pathological material was quite new: from analyses of pathological specimens Hodgkin was able to describe appendicitis with perforation and peritonitis, the local spread of cancer to draining lymph nodes, noting that the tumour had similar characteristics at both sides, and features of other diseases.

In his historic paper `On Some Morbid Appearances of the Absorbent Glands and Spleen’ (Hodgkin, 1832), he briefly described the clinical histories and gross postmortem findings on six patients from the experience at Guy’s Hospital and included another case sent to him in a detailed drawing by his friend Carswell (Fig 2). In the very first paragraph he wrote: `The morbid alterations of structure which I am about to describe are probably familiar to many practical morbid anatomists, since they can scarcely have failed to have fallen under their observation in the course of cadaveric inspection’. Hodgkin’s studies had convinced him that he was dealing with a primary disease of the absorbent (lymphatic) glands. `This enlargement of the glands appeared to be a primitive affection of those bodies, rather than the result of an irritation propagated to them from some ulcerated surface or other inflamed texture – Unless the word inflammation be allowed to have a more indefinite and loose eaning, this affection – can hardly be attributed to that cause’ was stated on pages 85 and 86 of his 1832 paper. Hodgkin also mentioned that the first reference that he could find to this or similar disease was in fact by Malpighi in 1666.

Wilks (1865) described the disease in detail and, made aware by Bright that the first observations were done by Hodgkin, linked his name permanently to this new entity in a paper entitled `Cases of Enlargement of the Lymphatic Glands and Spleen (or Hodgkin’s Disease) with Remarks’ (Fig 3).

In 1837 Thomas Hodgkin was the outstanding candidate for the position of Assistant Physician at Guy’s Hospital in succession to Thomas Addison who had been promoted to Physician. After 10 years spent as Inspector of the Dead, he had published a great deal, including a two-volume work entitled The Morbid Anatomy of Serous and Mucous Membrane.

Hodgkin, acting in his other capacity, had sent Benjamin Harrison a report on the terrible consequences to native Indians of monopoly trading and on the inhuman treatment they received from officials of the Hudson Bay Company, of which Harrison was the financier. when the opportunity to appoint an Assistant Physician occurred, Harrison exercised an autocratic rule over the hospital and presided at the appointment made by the General Court. Thomas Hodgkin did not get the job and the next day he resigned all his appointments at Guy’s Hospital. Social medicine, medical problems associated with poverty, antislavery, concern for underpriviledged groups such as American Indians and Africans, as well as a strong sense of responsibility defined his life after this separation.

Sternberg (1898) and Reed (1902) are generally credited with the first definitive and thorough descriptions of the histopathology of Hodgkin’s disease. Based on the findings observed in her case series, Dorothy Reed concluded `We believe then, from the descriptions in the literature and the findings in 8 cases examined, that Hodgkin’s disease has a peculiar and typical histological picture and could thus rightly be considered a histopathological disease entity’.

During the successive decades, pathologists began to describe a broader spectrum of histological features. However, it was Jackson and Parker who, in scientific papers and in their well-known book Hodgkin’s Disease and Allied Disorders (Jackson & Parker, 1947), presented the first serious effort at a histopathological classification. They assigned the name `Hodgkin’s granuloma’ to the main body of typical cases. A much more malignant variant, usually characterized by a great abundance of pleomorphic and anaplastic Reed-Sternberg cells and seen in a relativelysmall number of cases was named `Hodgkin’s sarcoma’. A third, similarly infrequent, variant characterized by an extremely slow clinical evolution, a relative paucity of Reed-Sternberg cells and a great abundance of lymphocytes was termed `Hodgkin’s paragranuloma’. It was only approximately 20 years later that Lukes & Butler (1966) reported a characteristic subtype of the heterogeneous `granuloma’ category, to which they assigned the name `nodular sclerosis’. They also proposed a new histopathological classification, still in use to date, with an appreciably greater prognostic relevance and usefulness than the

previous Jackson-Parker classification.

The first human bone marrow transfusion was given to a patient with aplastic anemia in 1939.9 This patient received daily blood transfusions, and an attempt to raise her leukocyte and platelet counts was made using intravenous injection of bone marrow. After World War II and the use of the atomic bomb, researchers tried to find ways to restore the bone marrow function in aplasia caused by radiation exposure. In the 1950s, it was proven in a mouse model that marrow aplasia secondary to radiation can be overcome by syngeneic marrow graft.10 In 1956, Barnes and colleagues published their experiment on two groups of mice with acute leukemia: both groups were irradiated as anti-leukemic therapy and both were salvaged from marrow aplasia by bone marrow transplantation.

The topics of leukemias and lymphomas will not be discussed further in  this discussion.

The related references are:

Leukaemia – A Brief Historical Review from Ancient Times to 1950
British Journal of Haematology, 2001, 112, 282-292

The Story of Chronic Myeloid Leukaemia
British Journal of Haematology, 2000, 110, 2-11

Historical Review of Lymphomas
British Journal of Haematology 2000, 109, 466-476

Historical Review of Hodgkin’s Disease
British Journal of Haematology, 2000, 110, 504-511

Multiple Myeloma: an Odyssey of Discovery
British Journal of Haematology, 2000, 111, 1035-1044

The History of Blood Transfusion
British Journal of Haematology, 2000, 110, 758-767

Hematopoietic Stem Cell Transplantation—50 Years of Evolution and Future Perspectives. Henig I, Zuckerman T.
Rambam Maimonides Med J 2014;5 (4):e0028.
http://dx.doi.org/10.5041/RMMJ.10162

Landmarks in the history of blood transfusion.

1666 Richard Lower (Oxford) conducts experiments involving transfusion of blood from one animal to another

1667 Jean Denis (Paris) transfuses blood from animals to humans

1818 James Blundell (London) is credited with being the first person to transfuse blood from one human to another

1901 Karl Landsteiner (Vienna) discovers ABO blood groups. Awarded Nobel Prize for Medicine in 1930

1908 Alexis Carrel (New York) develops a surgical technique for transfusion, involving anastomosis of vein in the recipient with artery in the donor. Awarded Nobel Prize for Medicine in 1912

1915 Richard Lewinsohn (New York) develops 0.2% sodium citrate as anticoagulant

1921 The first blood donor service in the world was established in London by Percy Oliver

1937 Blood bank established in a Chicago hospital by Bernard Fantus

1940 Landsteiner and Wiener (New York) identify Rhesus antigens in man

1940 Edwin Cohn (Boston) develops a method for fractionation of plasma proteins. The following year, albumin produced by this method was used for the first time to treat victims of the Japanese attack on Pearl Harbour

1945 Antiglobulin test devised by Coombs (Cambridge), which also facilitated identification of several other antigenic systems such as Kell (Coombs et al, 1946), Duffy (Cutbush et al, 1950) and Kidd (Cutbush et al, 1950)

1948 National Blood Transfusion Service (NBTS) established in the UK

1951 Edwin Cohn (Boston) and colleagues develop the first blood cell separator

1964 Judith Pool (Palo Alto, California) develops cryoprecipitate for the treatment of haemophilia

1966 Cyril Clarke (Liverpool) reports the use of anti-Rh antibody to prevent haemolytic disease of the newborn

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Peer Review and Health Care Issues

Larry H. Bernstein, MD, FCAP, Reporter

http://pharmaceuticalintelligence.com/12/1/2014/Peer-Review-and-Health-Care-Issues

(Medscape – Dec 1, 2014)

Peer-reviewed journals retracted 110 papers over the last 2 years. Nature reports the grim details in “Publishing: the peer review scam”.

When a handful of authors were caught reviewing their own

papers, it exposed weaknesses in modern publishing systems.

Editors are trying to plug the holes.

 

The Hill reports that the FDA may lift its ban on blood donations from gay men. The American Red Cross has voiced its support for lifting of the ban.

Advisers for the Food and Drug Administration (FDA) will meet this week to decide whether gay men should be allowed to donate blood, the agency’s biggest step yet toward changing the 30-year-old policy.

If the FDA accepts the recommendation, it would roll back a policy that has been under strong pressure from LGBT advocates and some members of Congress for more than four years.

“We’ve got the ball rolling. I feel like this is a tide-turning vote,” said Ryan James Yezak, an LGBT activist who founded the National Gay Blood Drive and will speak at the meeting. “There’s been a lot of feet dragging and I think they’re realizing it now.”

Groups such as the American Red Cross and America’s Blood Centers also voiced support of the policy change this month, calling the ban “medically and scientifically unwarranted.”

The FDA will use the group’s recommendation to decide whether to change the policy.

“Following deliberations taking into consideration the available evidence, the FDA will issue revised guidance, if appropriate,” FDA spokeswoman Jennifer Rodriguez wrote in a statement.

This reporter has more than 20 years of Blood Bank experience.  The factor in favor of the recommendation is that the HIV 1/2 and other testing is accurate enough to leave the question of donor lifestyle irrelevant.  However, it remains to be seen whether the testing turnaround time is sufficient to prevent the release of units that may be contaminated prior to transfusion, which is problematic for platelets, that have short expirations. In all cases of donor infection, regardless of whether units are released, a finding leads to not releasing the product or to recall.

 

Democrats made a strategic mistake by passing the Affordable Care Act, Sen. Charles Schumer (N.Y.), the third-ranking member of the Senate Democratic leadership, said Tuesday.

Schumer says Democrats “blew the opportunity the American people gave them” in the 2008 elections, a Democratic landslide, by focusing on healthcare reform instead of legislation to boost the middle class.

“After passing the stimulus, Democrats should have continued to propose middle class-oriented programs and built on the partial success of the stimulus,” he said in a speech at the National Press Club.

He said the plight of uninsured Americans caused by “unfair insurance company practices” needed to be addressed, but it wasn’t the change that people wanted when they elected Barack Obama as president.

“Americans were crying out for an end to the recession, for better wages and more jobs; not for changes in their healthcare,” he said.

This reader finds the observation by Senator Schumer very perceptive, regardless of whether the observation in hindsight might have had a different political outcome.  It has been noted that President Obama had a lot on his plate.  Moreover, we have not seen such a poor record of legislation in my lifetime.  There are underlying issues of worldview of elected officials that also contribute to the events.

 

THE PEER-REVIEW SCAM

BY CAT FERGUSON, ADAM MARCUS AND IVAN ORANSKY

N AT U R E |  2 7 N O V  2 0 1 4; VO L 5 1 5 : 480-82.

Most journal editors know how much effort it takes to persuade busy researchers to review a paper. That is why the editor of The Journal of Enzyme Inhibition and Medicinal Chemistry was puzzled by the reviews for manuscripts by one author — Hyung-In Moon, a medicinal-plant researcher then at Dongguk University in Gyeongju, South Korea.

The reviews themselves were not remarkable: mostly favourable, with some suggestions about how to improve the papers. What was unusual was how quickly they were completed — often within 24 hours. The turnaround was a little too fast, and Claudiu Supuran, the journal’s editor-in-chief, started to become suspicious.

In 2012, he confronted Moon, who readily admitted that the reviews had come in so quickly because he had written many of them himself. The deception had not been hard to set up. Supuran’s journal and several others published by Informa Healthcare in London
invite authors to suggest potential reviewers for their papers. So Moon provided names, sometimes of real scientists and sometimes pseudonyms, often with bogus e-mail addresses that would go directly to him or his colleagues. His confession led to the retraction of 28 papers by several Informa journals, and the resignation of an editor.

Moon’s was not an isolated case. In the past 2 years, journals have been forced to retract more than 110 papers in at least 6 instances of peer-review.

PEER-REVIEW RING
Moon’s case is by no means the most spectacular instance of peer-review rigging in recent years. That honour goes to a case that came to light in May 2013, when Ali Nayfeh, then editor-in-chief of the Journal of Vibration and Control, received some troubling news. An author who had submitted a paper to the journal told Nayfeh that he had received e-mails about it from two people claiming to be reviewers. Reviewers do not normally have direct contact with authors, and — strangely — the e-mails came from generic-looking Gmail accounts rather than from the professional institutional accounts that many academics use (see ‘Red flags in review’).
Nayfeh alerted SAGE, the company in Thousand Oaks, California, that publishes the journal. The editors there e-mailed both the Gmail addresses provided by the tipster, and the institutional addresses of the authors whose names had been used, asking for proof of identity and a list of their publications.ew rigging. What all these cases had in common was that researchers exploited vulnerabilities in the publishers’ computerized systems to dupe editors into accepting manuscripts, often by doing their own reviews. The cases involved publishing behemoths Elsevier, Springer, Taylor & Francis, SAGE and Wiley, as well as Informa, at least one of the systems — could make researchers vulnerable to even more serious identity theft. “For a piece of software that’s used by hundreds of thousands of academics worldwide, it really is appalling,” says Mark Dingemanse, a linguist at the Max Planck Institute for Psycholinguistics in Nijmegen, the Netherlands, who has used some of these programs to publish and review papers.

A 14-month investigation that came to involve about 20 people from SAGE’s editorial, legal and production departments. It showed that the Gmail addresses were each linked to accounts with Thomson Reuters’ ScholarOne, a publication-management system used by SAGE and several other publishers, including Informa. Editors were able to track every paper that the person or people behind these accounts had allegedly written or reviewed, says SAGE spokesperson Camille Gamboa. They also checked the wording of reviews, the details of author-nominated reviewers, reference lists and the turnaround time for reviews (in some cases, only a few minutes). This helped the investigators to ferret out further suspicious-looking accounts; they eventually found 130.

SAGE investigators came to realize that authors were both reviewing and citing each other at an anomalous rate. Eventually, 60 articles were found to have evidence of peer-review tampering, involvement in the citation ring or both. “Due to the serious nature of the findings, we wanted to ensure we had researched all avenues as carefully as possible before contacting any of the authors and reviewers,” says Gamboa. When the dust had settled, it turned out that there was one author in the centre of the ring: Peter Chen, an engineer then at the National Pingtung University of Education (NPUE) in Taiwan, who was a co-author on practically all of the papers in question.

PASSWORD LOOPHOLE
Moon and Chen both exploited a feature of ScholarOne’s automated processes. When a reviewer is invited to read a paper, he or she is sent an e-mail with login information. If that communication goes to a fake e-mail account, the recipient can sign into the system under whatever name was initially submitted, with no additional identity verification. Jasper Simons, vice-president of product and market strategy for Thomson Reuters in Charlottesville, Virginia, says that ScholarOne is a respected peer-review system and that it is the responsibility of journals and their editorial teams to invite properly qualified reviewers for their papers.

ScholarOne is not the only publishing system with vulnerabilities. Editorial Manager, built by Aries Systems in North Andover, Massachusetts, is used by many societies and publishers, including Springer and PLOS. The American Association for the Advancement of Science in Washington DC uses a system developed in-house for its journals Science, Science Translational Medicine and Science Signaling, but its open-access offering, Science Advances, uses Editorial Manager. Elsevier, based in Amsterdam, uses a branded version of the same product, called the Elsevier Editorial System.

Usually, editors in the United States and Europe know the scientific community in those regions well enough to catch potential conflicts of interest between authors and reviewers. But Lindsay says that Western editors can find this harder with authors from Asia — “where often none of us knows the suggested reviewers”. In these cases, the journal insists on at least one independent reviewer, identified and invited by the editors.

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Diagnostics Industry and Drug Development in the Genomics Era: Mid 80s to Present

Author and Curator: Larry H Bernstein, MD, FCAP

This article is a Chronological continuation to my article

Milestones in the Evolution of Diagnostics in the US HealthCare System: 1920s to Pre-Genomics

http://pharmaceuticalintelligence.com/2014/11/16/milestones-in-the-evolution-of-diagnostics-in-the-us-healthcare-system-1920s-to-pre-genomics/

Diagnostics Industry and Drug Development in the Genomics Era: Mid 80s to Present

Part 1.1: Companion Diagnostics

http://pharmaceuticalintelligence.com/?s=Companion+Diagnostics

As the life sciences industry adjusts to the insurance-driven environment,

  • specialized medicine is emerging as a critical asset in a development strategy.

Biomarkers and companion diagnostics (CDx) offer companies

  • the ability to target a subset of patients for testing with some potential for greater efficacy
  • based on biomarker selectivity, accelerated regulatory approval and better reimbursement.

Companion diagnostics are not necessarily suitable for developing a product. The decision to embark on a CDx program is a complex one involving a pharmaceutical company’s resources and its clinical goals. Top-performing companies prepare for success by

  • developing consistent criteria and protocols that lead to thoughtful partner selection and follow-through —
  • ensuring a smooth co-development timeline under budget.

Partner selection implies a cooperative development with

  • a diagnostics company having the needed diagnostics selectivity.

Consistent criteria and protocols requires that

  • the test criteria and performance characteristics accurately allow for
  • objective measurement of disease progression or effective treatment.

Criteria to Select the Right Test for a Companion Diagnostics Program

After the company reviews the clinical data for a particular compound and decides to pursue a companion diagnostic, it is time to focus on the test itself. The company should evaluate a potential companion diagnostic test considering the following criteria:

  • Accuracy
  • Reliability
  • Speed
  • Cost

Ideally, the companion diagnostic test for a drug would be

  • both extremely accurate and reliable, while requiring little time and money to develop —

but in reality, these tests can be complicated. A survey was conducted. The four evaluation criteria and the average importance survey respondents assign to them are rated on a scale of 1 to 5. In selecting a companion diagnostics test, all four criteria are expected to earn an average rating of 4.0 or greater, indicating that they are very important  . But

  • the two clinical criteria (accuracy and reliability) are absolutely crucial.
  • accuracy and reliability each earned an average rating of 4.6.

Survey respondents agree — accuracy and reliability are at the heart of a successful companion diagnostic program. These criteria are critically important to the test selection process. The success of an entire companion diagnostic program (and maybe the drug itself)

  • hinges on the test’s ability to accurately and reliably select patients
  • who will respond effectively to a drug.

Without these two characteristics, the test likely

  • will not receive regulatory approval,
  • nor will it be widely used.

The third most important criteria in selecting a companion diagnostic test is speed, earning an average rating of 4.3.

  • It is critical because investigational compounds are on a strict development timeline of their own.

If the companion diagnostic test cannot be developed quickly enough, then the drug might experience delays in

  • regulatory approval or market access and reimbursement.

Because patent protection for a compound is finite, delays can be costly, so most companies would rather spend the extra money to ensure the test is developed quickly. Those additional costs can be recouped if the drug and the companion diagnostic test enter the market sooner than expected.

Cost, earning an average rating of 4.0, ranked last on the list of test selection criteria. Because all, or most, of the companion diagnostic program will be developed outside the pharmaceutical company, cost is a relevant consideration when selecting a test.

Excepted from –  Companion Diagnostics Report
http://cuttingedgeinfo.com/research/clinical-development/companion-diagnostics-biomarkers/

1.2 Refining Processes for the Co-Development of Genome-Based Therapeutics and Companion Diagnostic Tests: Workshop Summary.

Overview Of Co-Development and Companion Diagnostics Policy
http://www.ncbi.nlm.nih.gov/books/NBK195940/

Important Points Highlighted 

  • The draft guidance on companion diagnostics from FDA recommends developing both the test and the drug together, which creates a new opportunity for developers to work and learn together to more clearly define a process and to coordinate a timeline for development.
  • The performance of a companion diagnostic is closely tied to the performance of the associated drug, and this relationship is essential for determining the safety and effectiveness of the products for patient use.
  • The major challenge for co-development is more commercial than regulatory because there are inherent differences in developing tests and drugs, including mismatched markets and resources.
  • Combining the cost of a test and a drug may provide a solution for aligning market differences and accelerating regulatory approval and reimbursement decisions for two different products.

Elizabeth Mansfield, director of the personalized medicine staff in the Office of In Vitro Diagnostics and Radiological Health at FDA, described the historical development of FDA’s policies for companion diagnostics and the main features of the companion diagnostic draft guidance.

The concept of companion diagnostics is not new. Testing for estrogen and progesterone receptor expression has been done since the 1980s (Gustavo Reynoso, CS Wilson Hospital, Binghampton, NY)

  • to determine if a patient would benefit from hormone therapy in treating breast cancer.

FDA approved the use of the drug Herceptin in patients with breast cancer

  • who tested positive for human epidermal growth factor receptor 2 (HER2) in 1998.

This was one of the earliest examples of a co-development companion diagnostic model before there was a formal process in place.  Herceptin was approved for use in patients with metastatic breast cancer, and

  • FDA also approved a test to examine HER2 levels.

One reason for having a test was to decrease risks, Herceptin’s cardiotoxic side effects are now well known. In the case of the drugs Selzentry and Tykerb, which were approved in 2007 and whose use depends on test results, a companion diagnostic policy was not yet in place when they were approved and FDA did not apply the policy retroactively.

Recognizing that tests can be drivers of therapy, FDA began to develop guidance to reflect drug development strategies

  • that account for genetic information.

It held public discussions about

  • pharmacogenomics,

requested voluntary genomic data submissions, and addressed other issues

  • concerning the use of genomic data to guide drug development,

FDA realized that a policy was needed

  • to protect patients while also allowing companies to plan for
  • the development of tests that would support therapeutic approval.

FDA also realized that companies want predictability in the regulatory process.

Companion diagnostics are tests,

  • but the test performance is closely tied to the performance of the associated drug.

Thus, knowledge about the test is essential

  • to understand the safety and effectiveness of the drug.

Tests for the same analyte can differ, sometimes significantly. The technology, cut-off levels, and performance can all vary, and different tests are likely to identify different populations. “It is essential to know all of these parameters

  • before deciding on which test is going to be used,

and if you want to use multiple tests [you will need to know] how these

  • different performance parameters compare and are related to the outcome
  • since test performance actually is a measure of the drug performance.

All of this information is needed to determine

  1. which patients will benefit from the drug and
  2. how to adequately label a drug.

In July 2011 FDA released

  • a draft guidance document for industry and FDA staff on IVD companion devices and
  • held a 90-day comment period.

At the time of the workshop, FDA expected to release the final version of the guidance soon. Mansfield reviewed a few key pieces of the policy. First, the policy defines an IVD companion device as

  • “an in vitro diagnostic device that provides information that is essential
  • for the safe and effective use of a corresponding therapeutic product.”

Such a diagnostic could identify a population

  • for efficacy, for safety, or for other purposes.

The document also differentiates companion diagnostics from

diagnostics used for other purposes.

Thousands of diagnostic tests have been cleared or approved, but

  • only perhaps 15 companion diagnostics had been approved (see Table 2-1) at the time of the workshop.

Understanding Co-Development

In the context of co-development, effectiveness means that a drug or test

  1. is adequate to accomplish a specific purpose,
  2. produces the intended or desired result, and
  3. is actually in operation as opposed to just having the potential for use,

Felix Frueh, entrepreneur-in-residence at Third Rock Ventures stated: For a product to be approved by FDA,

  • effectiveness needs to be demonstrated, so it is primarily a regulatory concern.
  • efficacy refers to the power or capacity to achieve the desired effect under ideal conditions,

It is more of a concern for payers. In a clinical trial inclusion and exclusion criteria create a more idealized situation than would be encountered outside of this environment. Co-development is

  • the “development of a test with a drug to make the use of the drug more effective or safer,”

He said: “It really is a method to make medicine more precise.” Co-development is not

  • an approach to make a clinical trial less costly,
  • nor is it a way to accelerate the time it takes to bring a product to market,
  • and not a biomarker discovery tool,

 “By definition you have to know your marker and you have to know what you’re using it for;

  • only that allows you to create a strategy to align the development of the marker with the drug.”

Next-Generation Sequencing

Some devices are complicated in that they have more than one indication, such as

  1. prostate-specific antigen (PSA) tests for monitoring cancer, which are classified as a Class II device, and
  2. PSA tests for diagnosis, which are classified as a Class III device.

In the case of molecular diagnostics, genomic information will

  • point toward therapies for which no indication in a drug label exists,
  • which is a much bigger regulatory challenge,

Frueh said. “The sheer magnitude of the information that we’ll find

  • on the genetic and molecular level is going to far surpass our capacity to run clinical trials,”

In fact, perhaps clinical trials will not be needed, especially because

  • clinical trials cannot be run for every marker and every condition.

But the same mutation is not always the driving factor for different cancers, said Walter Koch, vice president of global research at Roche Molecular Systems, and a workshop speaker. In contrast to Frueh’s view, Koch said that

  • only through clinical trials can a drug target be validated
  • by showing that the drug produces better outcomes.

Next-generation sequencing will find many variants, but they will not always be targets for a particular disease, he said.

1.3 Gray Sheet – Companion Diagnostics for Breakthrough Drugs Also Getting Swift Attention, FDA Says

http://www.focr.org/9-16-2013-gray-sheet-companion-diagnostics-breakthrough-drugs-also-getting-swift-attention-fda-says

Device center officials cite a host of formal and informal actions available under existing legislative authority to ensure that

  • development and review of companion diagnostics do not hinder
  • the ability of FDA-designated breakthrough therapies from coming to market.

Streamlining coDx Development

Created by FDASIA, the breakthrough therapy designation is aimed at

  • expediting development of drugs and biologics intended to treat serious conditions and
  • for which preliminary clinical evidence indicates the product may demonstrate
  • substantial improvement on a clinically significant endpoint compared to available therapy.

FDA laid out the qualifying criteria, features and submission and response timelines for the breakthrough program in its June draft guidance on expedited pathways (“FDA Expedited Programs Guidance: “Available Therapies” Depends On U.S. Standard Of Care” — “The Pink Sheet,” Jul. 1, 2013).

Drugs that receive the breakthrough designation

  1. also qualify for fast-track status, and
  2. sponsors will receive intensive guidance on an efficient drug development program and
  3. an organizational commitment that involves FDA senior managers.

The program, which is just a year old, has proven immensely popular with sponsors thus far. As of Sept. 6, the Center for Drug Evaluation and Research had received 85 requests for breakthrough designation, granting 27 and denying 35. The Center for Biologics Evaluation and Research had received 10 requests as of Aug. 31, granting none and denying eight. Many of the drugs for which breakthrough designation has been granted include a companion diagnostic, according to agency officials.

1.4 Diagnostics-Drugs Pairings Advance Personalized Medicine

C&N 2012; 90(30): 10-13. http://cen.acs.org/articles/90/i30/Diagnostics-Drugs-Pairings-Advance-Personalized.html

Within two weeks last August, the U.S. Food & Drug Administration approved two new cancer drugs: Roche’s Zelboraf, also called vemurafenib, for metastatic melanoma and Pfizer’s Xalkori, or crizotinib, for non-small-cell lung cancer. What sets those drugs apart from other cancer treatments is

  • the requirement that physicians use a diagnostic test to determine whether each one is right for a patient.

In the year since their approval, the two drugs have become

  • poster children for personalized medicine and
  • for what the industry calls “companion diagnostics.”

FDA defines companion diagnostics as

  • analytical tests that are required for the safe and effective use of a drug.

These assays measure a biomarker such as DNA or a protein

  1. to stratify the patient population and
  2. identify those individuals most likely to benefit or experience side effects from a drug.

On the basis of test results, patients can be

  • included in or excluded from clinical trials and prescriptions.

Such tests become necessary when there’s no other way to identify appropriate patients.

Xalkori and Zelboraf are not the first drugs to use companion diagnostics. That honor goes to Herceptin, Genentech’s drug for HER2 (human epidermal growth factor receptor 2)-positive breast cancer, which was approved in 1998. What was once unusual is now poised to become mainstream. On July 6, FDA approved a companion diagnostic from Qiagen to identify appropriate patients for a new indication for the cancer drug cetuximab, which is comarketed by Eli Lilly & Co. and Bristol-Myers Squibb as Erbitux.

Last summer, FDA released a draft guidance document on companion diagnostics for industry and FDA staff to clarify

  • when such tests will be required for regulatory approval and
  • to outline the regulatory process.

But “we’re not pushing anybody in this direction,” says Elizabeth A. Mansfield, director of personalized medicine in the Office of In Vitro Diagnostic Device Evaluation & Safety in FDA’s Center for Devices & Radiological Health. However, some companies are designing clinical trials this way, she points out, because “they want to be able

  • to select a population that’s going to respond in a robust enough way
  • to convince everyone that their drug actually works.”

Pharmaceutical companies are embracing codevelopment of drugs and diagnostics

  • because that’s where the science is leading them.

“We’ve been treating all tumors alike,” says Richard E. Buller, vice president for translational oncology at Pfizer,

  • “but we have data that says they’re not all alike.”

For example, a receptor

  • might be expressed in one type of breast cancer but not in another.

On the basis of the receptor’s expression, breast cancer can be divided into multiple subtypes, including HER2 positive or estrogen receptor positive, among others. By correctly identifying patients, pharma companies can target these different subtypes with different drugs.

“We’re following the scientific understanding of disease into the clinic,” says Garret M. Hampton, Genentech’s senior director of oncology biomarker development.

1,5 Diagnostics and Biomarkers: Novel Genomics Industry Trends vs Present Market Conditions and Historical Scientific Leaders Memoirs

http://pharmaceuticalintelligence.com/2013/05/15/diagnosis-of-cardiovascular-disease-treatment-and-prevention-current-predicted-cost-of-care-and-the-promise-of-individualized-medicine-using-clinical-decision-support-systems/

Larry H Bernstein, MD, FCAP, Author and Curator

This article has two parts:

  • 1.5.1: Novel Genomics Industry Trends in Diagnostics and Biomarkers vs
    Present Market Transient Conditions

and

  • 2: Historical Scientific Leaders Memoirs

1.5.1: Novel Genomics Industry Trends in Diagnostics and Biomarkers vs
Present Market Transient Conditions

Based on “Forging a path from companion diagnostics to holistic decision
support”, L.E.K.

Executive Insights, 2013;14(12). http://www.LEK.com

The problem of the current format is a “one test/one drug” match, but decision support
may require a combination of validated biomakers obtained on a small biopsy sample
(technically manageable) with confusing results.

While HER2 negative patients are more likely to be pre-menopausal with a more
aggressive tumor than postmenopausal,

  • the HER2 negative designation does not preclude treatment with Herceptin.

So the Herceptin would be given in combination, but

  • with what other drug in a non-candidate?

The point that L.E.K. makes is that

  • providing highly validated biomarkers linked to approved therapies, it is necessary
  • to pursue more holistic decision support tests that interrogate multiple biomarkers
    (panels of companion diagnostic markers) and
  • discovery of signatures for treatments that are also used with a broad range of information,
    such as,
  1. traditional tests,
  2. imaging,
  3. clinical trials,
  4. outcomes data,
  5. EMR data,

Companion diagnostics and their companion therapies is defined here as a method
enabling

  • LIKELY responders to therapies that are specific for patients with specific molecular profile.

The result is that the diagnostics permitted to specific patient types gives access to

  • novel therapies that may otherwise not be approved or reimbursed in other,
    perhaps “similar” patients
  • who lack a matching identification of the key identifier(s) needed to permit the therapy,
  • thus, entailing a poor expected response.

The concept is new because:

(1) The diagnoses may be closely related by classical criteria, but at the same time
they are not alike with respect to efficacy of treatment with a standard therapy.
(2) The companion diagnostics is restricted to dealing with a targeted drug-specific
question without regard to other clinical issues.
(3) The efficacy issue it clarifies is reliant on a deep molecular/metabolic insight
that is not available, except through

emergent genomic/proteomic analysis that has become available and which has
rapidly declining cost to obtain.

  • The limitation example given is HER2 testing for use of Herceptinin therapy for non-candidates (HER2 negative patients).
  • reimbursement and coverage data.

A comprehensive solution of this nature appears to be a distance from realization.
However, is this the direction that will

  • lead to tomorrows treatment decision support approaches?

 Surveying the Decision Support Testing Landscape

As a starting point, L.E.K. characterized the landscape of available tests in the U.S.
that inform treatment decisions

  • compiled from ~50 leading diagnostics companies operating in the U.S.
    between 2004-2011.

L.E.K. identified more than 200 decision support tests that were classified

  • by test purpose, and more specifically,
  1. whether tests inform treatment decisions for a single drug/class
    (e.g., companion diagnostics) vs.
  2. more holistic treatment decisions across multiple drugs/classes
    (i.e., multiagent response tests).

 Treatment Decision Support Tests

Companion Diagnostics
Single drug/class

Predict response/safety or guide dosing of a single drug or class

HercepTest   Dako

  • Determines HER2 protein overexpression
    for Herceptin treatment selection

Multiple drugs/classes

Vysis ALK Break
Apart FISH  Abbott Labs

  • Predicts the NSCLC patient response to Xalkori

Other Decision Support

Provide prognostic and predictive information on the benefit of treatment

Oncotype Dx    Genomic Health, Inc.

  • Predicts both recurrence of breast cancer and
    potential patient benefit to chemotherapy regimens

PML-RARα     Clarient, Inc.

  • Predicts response to all-trans retinoic acid (ATRA)
    and other chemotherapy agents

TRUGENE    Siemens

  • Measures resistence to multiple HIV-1 anti-retroviral agents

Multi-agent Response

Inform targeted therapy class selection by interrogating a panel of biomarkers

Target Now  Caris Life Sciences

  • Examines tumor’s molecular profile to tailor treatment options

Response DX: Lung    Response Genetics, Inc.

  • Examines multiple biomarkers to guide therapeutic treatment
    decisions for NSCLC patients

Source: L.E.K. Analysis

Includes IVD and LDT tests from

  1. top-15 IVD test suppliers,
  2. top-four large reference labs,
  3. top-five AP labs, and
  4. top-20 specialty reference labs.

For descriptive purposes only, may not map to exact regulatory labeling

Most tests are companion diagnostics and other decision support tests

  • that provide guidance on single drug/class therapy decisions.

However, holistic decision support tests (e.g., multi-agent response) are

  • growing the fastest at 56% CAGR.

The emergence of multi-agent response tests suggests diagnostics companies
are already seeing the need

  • to aggregate individual tests (e.g., companion diagnostics)
  • into panels of appropriate markers addressing a given clinical decision need

L.E.K. believes this trend is likely to continue as

  • increasing numbers of  biomarkers become validated for diseases
    and multiplexing tools
  • enabling the aggregation of multiple biomarker interrogations
    into a single test

to become deployed in the clinic.

Personalized Medicine Partnerships

L.E.K. also completed an assessment of publicly available

  • personalized medicine partnership activity from 2009-2011 for
    ~150 leading organizations operating in the U.S.
  • to look at broader decision support trends and
  • emergence of more holistic solutions beyond diagnostic tests.

Survey of partnerships deals was conducted for

  • top-10 academic medical centers research institutions,
  • top-25 biopharma,
  • top-four healthcare IT companies,
  • top-three healthcare imaging companies,
  • top-20 IVD manufacturers,
  • top-20 laboratories,
  • top-10 payers/PBMs,
  • top-15 personalized healthcare companies,
  • top-10 regulatory/guideline entities, and
  • top-20 tools vendors for the period of 01/01/2009 – 12/31/2011.
    Source: Company websites, GenomeWeb, L.E.K. analysis

Across the sample we identified 189 publicly announced partnerships of which

  •  ~65% focused on more traditional areas
    (biomarker discovery, companion diagnostics and targeted therapies).
  •  ~30% included elements geared towards creating more
    holistic decision support models.

Partnerships categorized as holistic decision support by L.E.K. were focused on

  • mining large patient datasets (e.g., from payers or providers),
  • molecular profiling (e.g., deploying next-generation sequencing),
  • creating information technology (IT) infrastructure needed to enable
    holistic decision support models and
  • integrating various datasets to create richer decision support solutions.

Interestingly, holistic decision support partnerships often included stakeholders
outside of biopharma and diagnostics –

  • research tools,
  • payers/PBMs,
  • healthcare IT companies as well as
  • emerging personalized healthcare (PHC) companies
    (e.g., Knome, Foundation Medicine and 23andMe).

This finding suggests that these new stakeholders will be increasingly important
in influencing care decisions going forward.

Holistic Treatment Decision Support

Holistic Decision
Support Focus
Technology Provider Partners
Stakeholder Deploying the Solution
Holistic Decision
Support Activities
Molecular Profiling Life Technologies TGEN/US
Oncology
Sequencing of triple-negative breast  cancer
patients to identify potential treatment strategies
Foundation Medicine Novartis Deployment of cancer genomics analysis  platform to
support Novartis clinical research efforts
Predictive genomics Clarient, Inc.
(GE Healthcare)
Acorn
Research
Biomarker profiling of patients within Acorn’s
network of providers to support clinical research efforts
Genome
Quest
Beth Israel
Deaconess MC
Whole genome analysis and to guide patient
management
Outcomes
Data Mining
AstraZeneca WellPoint Evaluate comparative effectiveness of selected
marketed therapies
23andMe NIH Leverage information linking drug response and
CYP2C9/CYP2C19 variation
Pfizer Medco Leverage patient genotype, phenotype and outcome
for treatment decisions and target therapeutics
Healthcare
IT Infrastructure
IBM WellPoint Deploy IBM’s Watson-based solution to evidence-
based healthcare decision-making support
Oracle Moffitt Cancer Center Deploy Oracle’s informatics platform to store and
manage patient medical information
Data Integration Siemens
Diagnostics
Susquehanna
Health
Integration of imaging and laboratory diagnostics
Cernostics Geisinger
Health
Integration of advanced tissue diagnostics, digital
pathology, annotated biorepository and EMR
to create solutions
next-gen
treatment
solutions
CardioDx GE Healthcare Integration of genomics with imaging data in CVD

Implications

L.E.K. believes the likely debate won’t center on which models and companies
will prevail. It appears that

  • the industry is now moving along the continuum to a truly holistic capability.
  • the mainstay of personalized medicine today will become integrated and
    enhanced by other data.

The companies that succeed will be able to capture vast amounts of information

  • and synthesize it for personalized care.

Holistic models will be powered by increasingly larger datasets and

sophisticated decision-making algorithms.

This will require the participation of an increasingly broad range of participants
to provide the

  • science, technologies, infrastructure and tools necessary for deployment.

There are a number of questions posed by this study, but only some are of
interest to this discussion:

Group A.    Pharmaceuticals and Devices

  • How will holistic decision support impact the landscape ?
    (e.g., treatment /testing algorithms, decision making, clinical trials)

Group B.     Diagnostics and   Decision Support

  • What components will be required to build out holistic solutions?

– Testing technologies

– Information (e.g., associations, outcomes, trial databases, records)

– IT infrastructure for data integration and management, simulation and reporting

  • How can various components be brought together to build seamless holistic
    decision support solutions?

Group C.      Providers and Payers

  • In which areas should models be deployed over time?
  • Where are clinical and economic arguments  most compelling?

2: Historical Scientific Leaders Memoirs – Realtime Clinical Expert Support

Gil David and Larry Bernstein have developed, in consultation with Prof. Ronald
Coifman, in the Yale University Applied Mathematics Program,

A software system that is the equivalent of an intelligent Electronic Health
Records Dashboard that

  • provides empirical medical reference and
  • suggests quantitative diagnostics options.

The current design of the Electronic Medical Record (EMR) is a linear
presentation of portions of the record

  • by services
  • by diagnostic method, and
  • by date, to cite examples.

This allows perusal through a graphical user interface (GUI) that partitions the
information or necessary reports

  • in a workstation entered by keying to icons.

This requires that the medical practitioner finds the

  • history,
  • medications,
  • laboratory reports,
  • cardiac imaging and
  • EKGs, and
  • radiology in different workspaces.

The introduction of a DASHBOARD has allowed a presentation of

  • drug reactions
  • allergies
  • primary and secondary diagnoses, and
  • critical information

about any patient the care giver needing access to the record.

The advantage of this innovation is obvious.  The startup problem is

  1. what information is presented and
  2. how it is displayed,
  3. which is a source of variability and a key to its success.

We are proposing an innovation that supercedes

  • the main design elements of a DASHBOARD and utilizes
  • the conjoined syndromic features of the disparate data elements.

So the important determinant of the success of this endeavor is that

  • it facilitates both the workflow and the decision-making process
    with a reduction of medical error.

Continuing work is in progress in extending the capabilities with model
datasets, and sufficient data because

  • the extraction of data from disparate sources will, in the long run,
    further improve this process.

For instance, the finding of  both ST depression on EKG coincident

  • with an elevated cardiac biomarker (troponin),
  • particularly in the absence of substantially reduced renal function.

The conversion of hematology based data into useful clinical information

  • requires the establishment of problem-solving constructs based on
    the measured data.

The most commonly ordered test used for managing patients worldwide is

  • the hemogram that often incorporates
  • the review of a peripheral smear.

While the hemogram has undergone progressive modification of the measured
features over time

  • the subsequent expansion of the panel of tests has provided a window into
    the cellular changes in the
  1. production
  2. release
  3. or suppression

of the formed elements from the blood-forming organ into the circulation.
In the hemogram one can view

  • data reflecting the characteristics of a broad spectrum of medical conditions.

Progressive modification of the measured features of the hemogram has

  • delineated characteristics expressed as measurements of
  1. size
  2. density, and
  3. concentration,

resulting in many characteristic features of classification. In the diagnosis
of hematological disorders

  • proliferation of marrow precursors
  • domination of a cell line, and
  • features of suppression of hematopoiesis

provide a two dimensional model.  Other dimensions are created by considering

  • the maturity of the circulating cells.

The application of rules-based, automated problem solving should provide a
valid approach to

  • the classification and interpretation of the data used to determine a
    knowledge-based clinical opinion.

The exponential growth of knowledge since the mapping of the human
genome enabled

  • by parallel advances in applied mathematics that have not been a part of
    traditional clinical problem solving.

As the complexity of statistical models has increased

  • the dependencies have become less clear to the individual.

Contemporary statistical modeling has a primary goal of

  • finding an underlying structure in studied data sets.

The development of an evidence-based inference engine that can

  • substantially interpret the data at hand and
  • convert it in real time to a “knowledge-based opinion”
  • could improve clinical decision-making by incorporating
  • multiple complex clinical features as well as duration of
    onset into the model.

An example of a difficult area for clinical problem solving is found in the

  • diagnosis of SIRS and associated sepsis. SIRS
    (and associated sepsis) is a costly diagnosis in hospitalized patients.

Failure to diagnose sepsis in a timely manner creates

  • a potential financial and safety hazard.

The early diagnosis of SIRS/sepsis is made by the application of
defined criteria by the clinician.

  • temperature
  • heart rate
  • respiratory rate and
  • WBC count

The application of those clinical criteria, however, defines the condition
after it has developed and

  • has not provided a reliable method for the early diagnosis of SIRS.

The early diagnosis of SIRS may possibly be enhanced

  • by the measurement of proteomic biomarkers, including
  1. transthyretin
  2. C-reactive protein
  3. procalcitonin
  4. mean arterial pressure

Immature granulocyte (IG) measurement has been proposed as a

  • readily available indicator of the presence of granulocyte precursors (left shift).

The use of such markers, obtained by automated systems

  • in conjunction with innovative statistical modeling, provides
  • a promising approach to enhance workflow and decision making.

Such a system utilizes the conjoined syndromic features of

  • disparate data elements with an anticipated reduction of medical error.

How we frame our expectations is so important that it determines

  • the data we collect to examine the process.

In the absence of data to support an assumed benefit, there is no
proof of validity at whatever cost. This has meaning for

  • hospital operations,
  • for nonhospital laboratory operations,
  • for companies in the diagnostic business, and
  • for planning of health systems.

The problem stated by LL  WEED in “Idols of the Mind” (Dec 13, 2006):
“ a root cause of a major defect in the health care system is that, while  we

  • falsely admire and extol the intellectual powers of highly educated physicians,
  • we do not search for the external aids their minds require”.

HIT use has been

  • focused on information retrieval, leaving
  • the unaided mind burdened with information processing.

We deal with problems in the interpretation of data

  • presented to the physician, and how
  • through better design of the software that presents this data
  • the situation could be improved.

The computer architecture that the physician uses to view the results
is more often than not presented

  • as the designer would prefer, and not as the end-user would like.

In order to optimize the interface for physician,

  • the system would have a “front-to-back” design, with
  • the call up for any patient ideally consisting of a dashboard design
  • that presents the crucial information that the physician
  • would likely act on in an easily accessible manner.

The key point is that each item used has to be closely related to a

  • corresponding criterion needed for a decision.

Feature Extraction.

This further breakdown in the modern era is determined by

  • genetically characteristic gene sequences
  • that are transcribed into what we measure.

Eugene Rypka contributed greatly to clarifying the

  • extraction of features in a series of articles, which
  • set the groundwork for the methods used today in clinical microbiology.

The method he describes is termed S-clustering, and

  • will have a significant bearing on how we can view laboratory data.

He describes S-clustering as extracting features from endogenous data that

  • amplify or maximize structural information to create distinctive classes.

The method classifies by taking the number of features

  • with sufficient variety to map into a theoretic standard.

The mapping is done by

  • a truth table, and each variable is scaled
  • to assign values for each: message choice.

The number of messages and the number of choices forms an N-by N table.
He points out that the message

  • choice in an antibody titer would be converted from 0 + ++ +++ to 0 1 2 3.

Even though there may be a large number of measured values, the variety

  • is reduced by this compression, even though
  •  there is risk of loss of information.

Yet the real issue is how

  • a combination of variables falls into a table with meaningful information.

We are concerned with accurate assignment into

  • uniquely variable groups by information in test relationships.

One determines the effectiveness of each variable by

  • its contribution to information gain in the system.

The reference or null set is the class having no information.
Uncertainty in assigning to a classification is

  • only relieved by providing sufficient information.

The possibility for realizing a good model for approximating

  • the effects of factors supported by data used
  • for inference owes much to the discovery of Kullback-Liebler distance
    or “information”, and
  • Akaike found a simple relationship between K-L information and
    Fisher’s maximized log-likelihood function.

In the last 60 years the application of entropy comparable to

  • the entropy of physics, information, noise, and signal processing,
  • has been fully developed by Shannon, Kullback, and others, and
  • has been integrated with modern statistics,
  • as a result of the seminal work of Akaike, Leo Goodman, Magidson
    and Vermunt, and work by Coifman.

Gil David et al. introduced an AUTOMATED processing of the data available
to the ordering physician and

  • can anticipate an enormous impact in diagnosis and treatment of
    perhaps half of the top 20 most common
  • causes of hospital admission that carry a high cost and morbidity.

For example: anemias (iron deficiency, vitamin B12 and folate deficiency, and
hemolytic anemia or myelodysplastic syndrome); pneumonia; systemic
inflammatory  response syndrome (SIRS) with or without bacteremia; multiple
organ failure and  hemodynamic shock; electrolyte/acid base balance disorders;
acute and chronic  liver disease; acute and chronic renal disease; diabetes
mellitus; protein-energy  malnutrition; acute respiratory distress of the newborn;

acute coronary syndrome; congestive heart failure; disordered bone mineral
metabolism; hemostatic disorders; leukemia and lymphoma; malabsorption
syndromes; and cancer(s) [breast, prostate, colorectal, pancreas, stomach,
liver, esophagus, thyroid, and parathyroid].

Realtime Clinical Expert Support and validation System

We have developed a software system that is the equivalent of an intelligent
Electronic Health Records Dashboard that provides empirical medical reference
and suggests quantitative diagnostics options.

The primary purpose is to

  1. gather medical information,
  2. generate metrics,
  3. analyze them in realtime and
  4. provide a differential diagnosis,
  5. meeting the highest standard of accuracy.

The system builds its unique characterization and provides a list of other patients
that share this unique profile, therefore utilizing the vast aggregated knowledge
(diagnosis, analysis, treatment, etc.) of the medical community. The

  • main mathematical breakthroughs are provided by accurate patient profiling
    and inference methodologies
  • in which anomalous subprofiles are extracted and compared to potentially
    relevant cases.

As the model grows and its knowledge database is extended, the diagnostic and
the prognostic become more accurate and precise. We anticipate that the effect of
implementing this diagnostic amplifier would result in

  • higher physician productivity at a time of great human resource limitations,
  • safer prescribing practices,
  • rapid identification of unusual patients,
  • better assignment of patients to observation, inpatient beds,
    intensive care, or referral to clinic,
  • shortened length of patients ICU and bed days.

The main benefit is a real time assessment as well as diagnostic options based on

  • comparable cases,
  • flags for risk and potential problems

as illustrated in the following case acquired on 04/21/10. The patient was diagnosed by
our system with severe SIRS at a grade of 0.61 .

Hemogram with Severe SIRS

Hemogram with Severe SIRS

The patient was treated for SIRS and the blood tests were repeated during the
following week. The full combined record of our system’s assessment of the patient,
as derived from the further  hematology tests, is illustrated below. The yellow line
shows the diagnosis  that  corresponds  to the first blood test (as also shown in
the image above). The red line shows the next diagnosis that was performed a
week later.

time course

Related article in Pharmaceutical Intelligence:

Rudolph RA, Bernstein LH, Babb J: Information-Induction for the diagnosis of
myocardial infarction. Clin Chem 1988;34:2031-2038.

Bernstein LH, Qamar A, McPherson C, Zarich S, Rudolph R. Diagnosis of
myocardial infarction: integration of serum markers and clinical descriptors using
information theory. Yale J Biol Med 1999; 72: 5-13.

Kaplan L.A.; Chapman J.F.; Bock J.L.; Santa Maria E.; Clejan S.; Huddleston D.J.;
Reed R.G.; Bernstein L.H.; Gillen-Goldstein J. Prediction of Respiratory Distress
Syndrome using the Abbott FLMII amniotic fluid assay. The National Academy of
Clinical Biochemistry (NACB) Fetal Lung Maturity Assessment Project.
Clin Chim Acta 2002; 326(8): 61-68.

Bernstein LH, Qamar A, McPherson C, Zarich S. Evaluating a new graphical
ordinal logit method (GOLDminer) in the diagnosis of myocardial infarction
utilizing clinical features and laboratory data. Yale J Biol Med 1999; 72:259-268.

Bernstein L, Bradley K, Zarich SA. GOLDmineR: Improving models for
classifying patients with chest pain. Yale J Biol Med 2002; 75, pp. 183-198.

Ronald Raphael Coifman and Mladen Victor Wickerhauser. Adapted Waveform
Analysis as a Tool for Modeling, Feature Extraction, and Denoising. Optical
Engineering 1994; 33(7):2170–2174.

Coifman and N. Saito. Constructions of local orthonormal bases for classification
and regression.C. R. Acad. Sci. Paris, 319 Série I:191-196, 1994.

W Ruts, S De Deyne, E Ameel, W Vanpaemel,T Verbeemen, And G Storms.
Dutch norm data for 13 semantic categories and 338 exemplars. Behavior
Research Methods, Instruments,& Computers 2004; 36 (3): 506–515.

De Deyne, S Verheyen, E Ameel, W Vanpaemel, MJ Dry, W Voorspoels,
and G Storms.Exemplar by feature applicability matrices and other Dutch
normative data for semantic concepts.Behavior Research Methods 2008;
40 (4): 1030-1048

Landauer, T. K., Ross, B. H., & Didner, R. S. (1979). Processing visually
presented single words:A reaction time analysis [Technical memorandum].
Murray Hill, NJ: Bell Laboratories..

Weed L. Automation of the problem oriented medical record. NCHSR Research
Digest Series DHEW. 1977;(HRA)77-3177.

Naegele TA. Letter to the Editor. Amer J Crit Care 1993;2(5):433.

The Automated Second Opinion Generator     Larry H Bernstein

http://pharmaceuticalintelligence.com/2012/08/13/the-automated-second
-opinion-generator/

The electronic health record: How far we have travelled and where is journeys end

Larry H Bernstein

http://pharmaceuticalintelligence.com/2012/09/21/the-electronic-health-record-how
-far-we-have-travelled-and-where-is-journeys-end/

The potential contribution of informatics to healthcare is more than currently estimated.

Larry H Bernstein

http://pharmaceuticalintelligence.com/2013/02/18/the-potential-contribution-of
-informatics-to-healthcare-is-more-than-currently-estimated/


Sheila Nirenberg/Cornell and Chethan Pandarinath/Stanford,
“Retinal prosthetic strategy with the capacity to restore normal vision,”
Proc Natl Acad Sci

Cadiovascular–decision-support-systems-for-disease-management-decision-making

Larry H Bernstein

http://pharmaceuticalintelligence.com/2013/05/04/cardiovascular-diseases-decision
-support-systems-for-disease-management-decision-making/?goback=%2Egde
_4346921_member_239739196

Demonstration of a diagnostic clinical laboratory neural network applied to three
laboratory data conditioning problems

Larry H Bernstein

http://pharmaceuticalintelligence.com/2012/08/13/demonstration-of-a-diagnostic
-clinical-laboratory-neural-network-agent-applied-to-three-laboratory-data-
conditioning-problems/

Vinod Khosla: 20 doctor included speculations, musings of a technology optimist
or technology will replace 80 percent of what doctors do

Aviva Lev-Ari

http://pharmaceuticalintelligence.com/2013/05/13/vinod-khosla-20-doctor
-included-speculations-musings-of-a-technology-optimist-or-technology-will
-replace-80-of-what-doctors-do/

Bioengineering of vascular and tissue models

Larry H Bernstein and Aviva Lev-Ari

http://pharmaceuticalintelligence.com/2013/05/05/bioengineering-of-
vascular-and-tissue-models/

The Heart: Vasculature Protection – A Concept-based Pharmacological
Therapy including THYMOSIN Aviva Lev-Ari, PhD, RN 2/28/2013
http://pharmaceuticalintelligence.com/2013/02/28/the-heart-vasculature
-protection-a-concept-based-pharmacological-therapy-including-thymosin/

FDA Pending 510(k) for The Latest Cardiovascular Imaging Technology

Aviva Lev-Ari, PhD, RN 1/28/2013

http://pharmaceuticalintelligence.com/2013/01/28/fda-pending-510k-for-
the-latest-cardiovascular-imaging-technology/

PCI Outcomes, Increased Ischemic Risk associated with Elevated Plasma
Fibrinogen not Platelet Reactivity

Aviva Lev-Ari, PhD, RN 1/10/2013

http://pharmaceuticalintelligence.com/2013/01/10/pci-outcomes-increased-
ischemic-risk-associated-with-elevated-plasma-fibrinogen-not-platelet-reactivity/

The ACUITY-PCI score: Will it Replace Four Established Risk Scores — TIMI,
GRACE, SYNTAX, and Clinical SYNTAX

Aviva Lev-Ari, PhD, RN 1/3/2013

http://pharmaceuticalintelligence.com/2013/01/03/the-acuity-pci-score
-will-it-replace-four-established-risk-scores-timi-grace-syntax-and-clinical-syntax/

Coronary artery disease in symptomatic patients referred for coronary angiography:
Predicted by Serum Protein Profiles

Aviva Lev-Ari, PhD, RN 12/29/2012

http://pharmaceuticalintelligence.com/2012/12/29/coronary-artery-disease-in-
symptomatic-patients-referred-for-coronary-angiography-predicted-by-serum-protein-profiles/

New Definition of MI Unveiled, Fractional Flow Reserve (FFR)CT for Tagging
Ischemia

Aviva Lev-Ari, PhD, RN 8/27/2012

http://pharmaceuticalintelligence.com/2012/08/27/new-definition-of-mi-unveiled-fractional-flow-reserve-ffrct-for-tagging-ischemia/

Related articles


Part 3: Biomarkers in Service of Medical Diagnosis

3.1  Related articles in Leaders in Pharmaceutical Intelligence

Cracking the code of human life: the birth of bioinformatics and computational genomics

Larry H Bernstein

http://pharmaceuticalintelligence.com/2013/02/13/cracking-the-code-of-human-
life-the-birth-of-bioinformatics-and-computational-genomics/

Genetics of conduction disease atrioventricular AV conduction disease block
gene mutations transcription excitability and energy homeostasis

Aviva Lev-Ari

http://pharmaceuticalintelligence.com/2013/04/28/genetics-of-conduction-
disease-atrioventricular-av-conduction-disease-block-gene-mutations-transcription
-excitability-and-energy-homeostasis/

Identification of biomarkers that are related to the actin cytoskeleton

Larry H bernstein

http://pharmaceuticalintelligence.com/2012/12/10/identification-of-biomarkers-
that-are-related-to-the-actin-cytoskeleton/

Regression: A richly textured method for comparison of predictor variables

Larry H Bernstein

http://pharmaceuticalintelligence.com/2012/08/14/regression-a-richly-textured
-method-for-comparison-and-classification-of-predictor-variables/

Diagnostic evaluation od SIRS by immature granulocytes

Larry H Bernstein

http://pharmaceuticalintelligence.com/2012/08/02/diagnostic-evaluation-of
-sirs-by-immature-granulocytes/

Big data in genomic medicine

Larry H Bernstein

http://pharmaceuticalintelligence.com/2012/12/17/big-data-in-genomic-medicine/

Automated inferential diagnosis of SIRS, sepsis, septic shock

Larry H Bernstein

http://pharmaceuticalintelligence.com/2012/08/01/automated-inferential-
diagnosis-of-sirs-sepsis-septic-shock/

http://pharmaceuticalintelligence.com/2012/08/12/1815/

http://pharmaceuticalintelligence.com/2012/08/15/1946/

3.2 Related literature

Journal of Molecular Biomarkers & Diagnosis

http://omicsonline.org/molecular-biomarkers-diagnosis.php

Biomarker is an indicator of a particular disease or physiological
state of an organism.

The Cytoskeleton as Biomarker: Angiosarcoma- Cytoskeleton

Shifalika Tangutoori

Correlation among MDR1, MRP and hTERT Genes Expression Level
and Clinical Response in Colorectal Cancer Patients

Samira shabani, Sara Samanian, Rezvan Mirzaei, Bahar Mahjoubi and
Frouzandeh Mahjoubi

The Utility of Pyrogenic Cytokine (IL-1β, TNF-α and IL-6) Detection In Risk
Stratification of Critically lll Febrile Patients in Emergency Department

Francesco Travaglino, Gerardo Salerno, Veronica Russo, Mariateresa Corsetti,
Rosaria D’Urso, Patrizia Cardelli, Maria Rosaria Torrisi, Vincenzo Visco and
Salvatore Di Somma

Osteochondrosis-Related Gene Expression in Equine Leukocytes Differs
among Affected Joints in Foals

Serteyn D, Piquemal D, Mendoza L, Caudron I, Noguier F, Bruno R,
Sandersen C and Lejeune JP

Evaluation and Identification of Protein Blood Biomarkers for Alzheimer’s
Disease: A Systematic Review and Integrative Analysis

Junseong Park, JiEun Park, Jungsul Lee and Chulhee Choi

Interleukin 23 P 19 Expression in Patients with Ulcerative Colitis and
Its Relation to Disease Severity

Hanan El-Bassat, Lobna Abo Ali, Sahar El Yamany, Hanan Al Shenawy,
Rasha A Al Din and Atef Taha

Significance of Macrophage Migration Inhibitory Factor (MIF) and Anti P 53
Antibodies in Prognosis of Hepatocellular Carcinoma

Lamiss Mohamed Abd Elaziz Sad, Samar Galal Younis and Hala Mohamed Nagi

Prognostic Value of Bone Marker Beta-Crosslaps in Patients with Breast Carcinoma

Nicole Zulauf, Ingo Marzi and Gerhard M Oremek

Targeted Next-generation Sequencing Reveals a Homozygous Nonsense
Mutation in CAPN3 that Causes Limb-girdle Muscular Dystrophy Type 2A
First in Vietnam

Emma Tabe Eko Niba, Van Khanh Tran, Le Anh Tuan-Pham, Dung Chi Vu,
Ngoc Khanh Nguyen, Thinh Huy Tran, Van Thanh Ta, Tomoko Lee, Yasuhiro
Takeshima and Masafumi Matsuo

Aberrant Hypomethylated KRAS and RASGRF2 as a Candidate Biomarker
of Low Level Benzene Hematotoxicity

Jing Yang, Wenlin Bai, Zhongxin Xiao, Yujiao Chen, Li Chen, Lefeng Zhang,
Junxiang Ma, Lin Tian and Ai Gao

The Free Alpha-Hemoglobin: A Promising Biomarker for β-Thalassemia

Uday YH Abdullah, Ahmed GF Al-Attraqchi, Hishamshah M Ibrahim, Zilfalil
Bin Alwi, Atif A Baig, Lekhsan Othman, Noraesah B Mahmud, Rosline B
Hassan, Nor Hedayah A Bakar and Alawiyah B A Abd Rahman

Herceptin

Herceptin

Pathogenic mutation signals

Pathogenic mutation signals

Mechanisms by which a single gene can give rise to multiple gene products

Mechanisms by which a single gene can give rise to multiple gene products

Personalized Medicine

Personalized Medicine

3.1 Honing in on Cancer Biomarkers
http//pharmaceuticalintelligence.com/2013-11-27/larryhbern/Cancer
Biomarkers for Companion Diagnostics

Caitlin Smith    GEN  15 Nov 2013; 33(20)

Introduction and Goals

Some of the newest cancer treatments aim to

  • individualize the therapy to the specific type of cancer and patient.

The large and growing number of different genetic alterations that researchers
observe in cancer cells

  • have made it unfeasible to test for only a handful of targets.
  • clinical testing is moving toward testing for many targets simultaneously.

multiplexed tumor genotyping allows for

  • the simultaneous evaluation of a broad range of common and
    rare tumor alterations,

This expanding the application of targeted therapy

  1. across a greater number of patients who undergo testing, and
  2. directs those patients into the most relevant clinical trials.

Dr. Borger and colleagues are uncovering “molecular signatures of tumors,” or

  • collections of targets present in specific tumor types.

A molecular signature of a tumor is in essence,

  • a map of the abnormalities within a particular tumor that are
  • thought to be critical in driving the disease process,
  • each tumor will have a unique combination of genetic alterations.
  1. the more comprehensive the tumor profiling,
  2. the more detailed the roadmap
  3. we can draw for directing that patient’s care,” Dr. Borger said.

Uncovering the molecular signatures of tumors has another important role —

  • to better understand the differences among cells within the same tumor.
  • because tumor heterogeneity is an important mechanism
    of emerging drug resistance
  • . “Broad-based tumor profiling and the use of sensitive testing platforms
    are essential
  • in identifying these potential mechanisms of disease resistance,
  • so that targeted approaches can be aimed at circumventing those mechanisms.”

Target Signaling

Also working to help physicians figure out which treatments among many
might work best for individual patients is Selventa. Focusing on gene expression
biomarkers, Selventa researchers

  • correlate gene expression patterns from patient data with
  • changes in target signaling mechanisms.

“We operate on the hypothesis that patients with high or low levels of
target (or downstream target)

  • pathway signaling correspond to potential responders or nonresponders
    to target therapy, respectively,”  said Renée Deehan Kenney, Ph.D.,
    VP of Research.

“If we know who responded and who did not respond to treatment,

  • we can use that information to hone the biomarker

Selventa is using its Systems Diagnostics (SysDx) platform

  • to identify biomarkers used in diagnosing immune disorders
    such as rheumatoid arthritis (RA).

Their product Clarify-RA is based on the SysDx approach using a blood
biomarker. It is designed to aid clinicians in

  • matching RA patients with those RA drugs that will be most beneficial to them.

Such matching is valuable because RA is a heterogeneous disease, but

  • different patients respond differently to the over 15 RA drugs that are available.

Moreover, RA is a debilitating disease that

  • cannot wait for a trial-and-error treatment approach.

.“To compound this clinical challenge, drugs approved for RA offer

  • about 50% improvement for only 40% of the patients,” said Dr. Deehan Kenney.

One biomarker Selventa found can identify RA patients

  • who are likely to respond to anti-TNF therapy.

Similarly, Selventa’s SysDx approach also found

  • a biomarker from tumor biopsy tissue that
  • identifies ER+ breast cancer patients whose cancer
  • tends to progress with tamoxifen treatment.

IHC-Based Testing

President and CEO of Precision Biologics, Philip Arlen, M.D., discussed
his company’s research on a new

  • monoclonal antibody (NPC-1C), which targets tumors in both
    pancreatic and colorectal cancer.

The antibody’s target is specific to tumors, and the antibody has

negligible reactions with normal tissue.

Precision Biologics took an unconventional tack to making NPC-1C,

  • using a cancer vaccine that had been developed from colorectal cancer tissue
    removed from patients with
  • varying stages of disease, and screened for
  • antibodies that were specific for tumors, but nonreactive with normal tissue.

In both cell cultures and in animal models, they found that

  • NPC-1C destroyed pancreatic cancer cells.

Phase I/IIa data demonstrating prolongation in overall survival in patients

  • that had exhausted all standards of therapy,” said Dr. Arlen.

Precision Biologics has developed an immunohistochemistry-based
diagnostic test for expression of NPC-1C’s target. “Patients’ tumors are tested, and

  • if the target is present, the patients can receive treatment with NPC-1C,” .

Dr. Arlen said: “We are also developing a diagnostic assay with NPC-1C for

  • early detection and prognosis of colorectal and pancreatic cancer.”

NMR Technology

LipoScience researchers using NMR technology to find cancer biomarkers that are

  • panels of metabolites coveringa range biochemical processes

They produced these 1H NMR spectra of unprocessed serum focusing on

(A) macromolecular signals and

(B) the small molecule metabolome.

LipoScience is also developing new ways to search for biomarkers. Specifically,
to find biomarkers of clinical value, they are using NMR technology. “We take
advantage of two of the key features of the NMR platform,” explained Thomas
O’Connell, Ph.D., senior director of research and development. “These are the

  • lack of required sample preparation for routine biofluids and
  • the inherently quantitative signals.”

This means that they can profile large sample sets very quickly.

LipoScience researchers are now using NMR to look for cancer biomarkers.
“Given the heterogeneity of most cancers,

  • it is not likely that a single biomarker will provide the clinical performance,”
  •  “so we are examining panels of metabolites that cover a range
    of biochemical processes, including
  • lipid and lipoprotein metabolism, energy perturbations, inflammatory
    processes, and others.”  said Dr. O’Connell,

They plan to use NMR and metabolomic profiling

  • to develop clinical assays that help to choose patient-specific therapies.

“We are hopeful that one day in the near future, panels of biomarkers
could provide clinicians with

  • much more objective, quantifiable, and personalized information
  • regarding the diagnosis and management of their patients,”

Single Molecule Arrays

The Simoa (for single molecule array) instrument from Quanterix uses

  •  a digital ELISA technique,
  • trapping fluorescent reaction product in individual wells,
    to speed blood testing for HIV.

Researchers at Quanterix have developed a method of testing for a
different type of biomarker—

  • faster, cheaper, and more sensitive than previous tests.
  • that indicates the early and acute (and most contagious) stage of HIV infection.

Previously, the gold standard HIV test with the highest sensitivity was nucleic acid
testing. Simoa works by preventing the sensitivity loss that can occur in conventional ELISAs

  • because of the dilution of reaction product into the reaction volume.

Simoa essentially miniaturizes the ELISA principle,

  • trapping fluorescent reaction product in individual wells to prevent dilution.

“The technology basically supercharges a standard ELISA to give 1,000-times
greater sensitivity, Due to this extreme sensitivity of Simoa to enzyme label,
label molecules can be reduced, which

  • lowers nonspecific interactions and improves signal background.

This drives the sensitivity of Simoa digital immunoassays down to the
level of nucleic acid testing.”

“A key need in many blood banking centers is high throughput,” Dr. Wilson said.
“Blood units are screened for a number of pathogens, so effective throughput
is measured in number of units processed in a given period of time.”

Simoa immunoassays can be multiplexed to test for up to 10 different target
proteins simultaneously, which may benefit blood banks. However, blood
banking is highly regulated, so introducing Simoa assays may take time.
“As with any new test used to ensure a blood unit is pathogen-free,” explained Dr. Wilson, “a substantial amount of data is needed

  1. to prove to regulatory bodies that the test exhibits the claimed
    performance, and that
  2. the manufacturing processes are fully validated and controlled.”

3.2  Personalized Medicine: Why We Are So Excited

http://www.genengnews.com/bioperspectives/personalized-medicine-
why-we-are-so-excited/4767

In this first chapter of a five-part series, learn about new scientific developments
and recent treatment successes.

Detlef Niese, M.D., Ph.D.

Same Symptoms: Same Disease

Patients diagnosed with the same disease may react very differently to the same treatment. This observation led   Sir William Osler (1849–1919) to the statement: “If it would not be about the variability among individuals
medicine could well be a science and not an art”. We are brought up with the idea
that

  • patients who have the same symptoms and whose course of disease follows similar patterns
  • are likely to suffer from the same disease.

Scientific disciplines such as anatomy, developmental anatomy, physiology and
biochemistry,

  • helped us to get a better understanding of how a normal organism functions.

Pathophysiology, micro and macro pathology and clinical chemistry

  • helped to describe, classify and understand diseases.

However, few of these scientific advances led to a

  • full understanding of the causes of diseases on a molecular, cellular or genetic basis.

Today’s medical text books still describe and define diseases on the basis of
combinations of objective and subjective symptoms, macroscopic, microscopic,
pathophysiological or biochemical findings, and the presence or absence of
specific pathogenic organisms or substances. Most diseases are also classified
according to the affected organ or organ system.

In consequence, patients diagnosed with the same disease

  • are considered to suffer from the same underlying pathology.

They are also expected to respond to the same treatments, although clinical
experience tells us that this is only true in a limited number of situations. What is
causing the symptoms on a molecular level, remains often unclear.

Effective Therapies Were Educated Guesses

We have to admit that our current knowledge of disease mechanisms unfortunately
has not always resulted in the development of highly effective medicines yet.
The effective therapies we have, were developed intuitively or empirically, or were
even discovered by chance. Examples include antibiotics, alkaloids, calcium channel blockers, immunosuppressants and cytotoxic agents.

There are many examples of medicines, which were developed on the basis of wrong assumptions, and some examples of medicines, which proved to be effective in an
unexpected condition. But there are also few examples of medicines, like oral
contraceptives or modern targeted medicines in oncology and rheumatology, of which the development is based on an exact understanding of the relevant mechanisms.

Different Symptoms: Same Underlying Disease

In reality patients diagnosed with the same disease based on the same diagnostic
criteria

  • show very different courses of the disease, and
  • respond very differently to the same treatment. In clinical practice,
  • doctors have no other choice than to optimize the treatment by trial and error.

We now know that these patients are probably suffering from different diseases.
In other words:

  • the diseases have a different underlying pathology.

While the scientific progress in basic scientific disciplines such as anatomy,
physiology and biochemistry

  • has been substantial over the last centuries,
  • we still have a very limited understanding of the molecular processes in diseases.
  • we know even less about the factors, which may determine whether a person has an increased risk
  • of developing a particular disease.

Is “type-2 diabetes” a single homogeneous disease, for instance?

New Understanding of Diseases

Over the last decades molecular research on sick and healthy cells

  1. gave new insights in cellular signalling pathways: mechanisms involving proteins and nuclear receptors,
  2. which are essential for the normal functioning of cells
  3. but under specific circumstances may lead to diseases.

These discoveries have helped us to understand and classify diseases in a new way.

We have learned a lot from rare monogenetic disorders, which allow us to study the
impact of specific genetic mutations on the clinical signs and symptoms of the disease.

This research allows us to gain insight in the

  • underlying genetic alterations and
  • the consequences of the presence and functioning of critical proteins.

The answer to the question ‘what causes diseases’ is revealing itself little by little

  • since we are able to decipher the genetic code of a person.

3.3 Personalized Medicine: Translation of Concepts

http://www.genengnews.com/bioperspectives/personalized-medicine-translation-of-concepts/4783

This second chapter of a five-part series shows how the search for predictive biomarkers could change drug
development research.

Lasse Tengbjerg Hansen  ,    Adam Heathfield, Ph.D.
Biomarker research activities should continue

  • in parallel with the clinical development program,
  • so that biomarkers can be improved by applying data
  • derived from early clinical trials to later stages of the program. [© taraki – Fotolia.com]

The previous chapter discussed why scientific developments and recent treatment
successes have made many of us so excited about personalised medicine. This
chapter describes how

  • the search for predictive biomarkers is likely to change drug development research.

It may lead to a reduction of costs and an increase in productivity of research &
development projects.

Predictive biomarkers will change the way clinical trials are performed.

We Need Predictive Biomarkers

Traditionally, biomarkers answer

  • critical questions that arise during various stages of drug development— questions such as:
  1. Does the drug reach the target?
  2. Does it have the desired biological effect?
  3. Does it have an influence on other expected or unexpected targets?
  4. Does the drug affect characteristics that predict desired or undesired effects?

However, the concept of personalised medicine centres around predictive:

  1. biomarkers that can help select a patient population
  2. with a higher chance for a favourable response
  3. to a specific kind of medicine.

In today’s drug development programmes, the identification and qualification of
biomarkers are integrated
.

If we are able to determine the

  • biologically relevant dose, range and selection of the optimal target population by means of biomarkers,
  • we are convinced that biomarkers will increase R&D productivity by reducing development timelines and
  • will prevent costly late stage attrition.

Unfortunately, predictive biomarkers are often not applied until rather

  • late in the clinical development programme, when clinical data show
  • that an optimal benefit-risk profile is only achieved in a subpopulation of patients.

In such situations,

  • the attention is suddenly turned to other available research data
  • that might explain the underlying biological nature of the research results.

Over the last years we gathered examples of biomarkers, which

  1. have proven to predict clinical response better
  2. because they are linked to the mode of actions of the compounds in question.

Examples include Her-2/neu (Trastuzumab/Lapatinib), KRAS (Cetuximab/Panitumomab), BRAF
(Vemurafenib), and CCR5 (Maraviroc).

We Lack Model Systems to Predict Drug Response

While it is crucial to identify and qualify biomarkers that predict clinical response

  • we lack good model systems that predict drug response.

Since efficacy is established in animal models, despite convincing data from animal models,

  • the translation from animals to humans is not always successful.
  • alternative models or an alternative technology are needed, and
  • this had been attempted with ex-vivo systems in areas such as rheumatoid arthritis and Crohn’s disease.

These assays have proven to be valuable for the evaluation of novel therapeutic
targets.While the application of such assays seems promising, it remains to be fully investigated.

A caveat of ex-ivo systems may be that

  • the generated biomarker signal derives from a local tissue environment, which
  • might be difficult to capture in peripheral blood samples.

The oncology field has the best access to tissue biopsies. That is one of the reasons why oncology is a front runner in personalized medicine with several tissue based
companion diagnostics. On the other hand, still very little is known

  • about the drug resistance as encountered in the oncology field.

This asks for biomarkers that elucidate more clearly what is going on in individual
tumors.

New Kind of Trials

Studying targeted medicines in traditionally defined patient populations is quite problematic. For example, if we want to test a medicine like gefitinib,

  • we do not need patients who are clinically diagnosed with non-small cell lung cancer,
  • but patients with a specific EGFR mutation.

If we were to test the medicine on a population which is

  • not preselected for the presence of the mechanism in question,
  • we might conclude that the medicine does not have an appropriate benefit-risk ratio.
  • but if tested on patients with the underlying mechanism we might come to the opposite conclusion.

Gefitinib is therefore indicated for patients with tumors showing specific EGFR
mutations, while

  • it is irrelevant whether the tumour is located in the lungs.

In order to develop targeted medicines, patients will have to be selected

  • based on presence or absence of specific biomarkers.

In some situations, that may be a

  • relatively small subset of the traditionally defined patient population.

For instance: traditionally we would test a drug on patients with breast cancer.
But now we would select patients with a

  • similar disease pathway: e.g. Her-2 positive.

Eventually, the patient population may be extended: for instance –

  • that we can treat patients with malignant tumours in other organs with the same medicine,
  • assuming the tumours are caused by the same molecular mechanisms (e.g. m-TOR mutations).

With this approach, patient populations participating in clinical trials would be

  • more homogenous, and response to treatment will be more consistent and predictable as well.

Integrating Biomarker Research with Target Evaluation

We think continuous biomarker research

  1. ideally starts at least two to four years prior to first-in-man clinical trials,
  2. we should ideally have identified a broad panel of biomarker candidates that
  3. subsequently can be further qualified in appropriate in-vitro, ex-vivo, in-vivo, or in-silico model systems.

Bioanalytical assays for the selected candidates can then be established and
validated

  • prior to implementation in clinical trials.

In order to be fully operational, this strategy requires competencies within three main areas:

  1. biomarker research,
  2. biomarker assay development and
  3. clinical biomarker implementation.

These areas should collaborate to guarantee the quality of the biomarker data derived from clinical trials. This will strengthen the basis on which a

  • personalised medicine program can be evaluated.
  • it is also advisable to continue the biomarker research activities
  • in parallel with the clinical development program.

In this way it is possible to improve the biomarkers by

  • applying data derived from early clinical trials to later stages of the program.

This approach clinically validates pre-selected biomarker candidates

  • but may also identify potential other and better biomarker candidates correlating with clinical outcomes.

Identifying Biomarkers is a Collaborative Effort

It is time consuming and costly to identify and qualify biomarkers for other uses,

  • such as predicting clinical outcomes.

A biomarker needs sufficient evidence of broader clinical utility and validation

  • in multiple independent studies,
  • across different cohorts,
  • ethnic groups and
  • clinical subgroups.

This is in line with recent guidance for biomarker qualification from the Food
& Drug Administration (FDA) and the European Medicines Agency (EMA).

In recent years, large consortia have succeeded in

  • the qualification and validation of various types of biomarkers.

Examples of collaborative biomarker onsortia are the Biomarker Consortium (US) and Innovative Medicines Initiative (EU).They are supported by representatives from
regulators, pharmaceutical industry, biotech industry and diagnostic industry,
academia and governmental organisations.

3.4 Personalized Medicine: From Biomarkers to Companion Diagnostics

This third chapter of a five-part series talks about coordinating diagnostic test kit development between
pharmaceutical companies and diagnostic manufacturers.

Peter Collins, Ph.D.

http://www.genengnews.com/gen-articles/personalized-medicine-from-biomarkers-to-companion-diagnostics/4820/

The previous chapter described how the search for predictive biomarkers

  • is likely to change drug development research.

This chapter talks about turning biomarkers into companion diagnostics.

Once biomarker tests have been turned into diagnostic test kits, we would

  • be able to routinely classify patients in clinics.

But the development of those diagnostic test kits has to be coordinated between two entities—pharmaceutical companies and diagnostic manufacturers—with rather different regulation.

As we have seen in the previous chapters, biomarkers can play a critical role in

  • classifying patients into subpopulations.

A biomarker can be used as the basis for

  • creating a routine diagnostic test for clinical use,

after it has been approved as an in-vitro diagnostic (IVD) test: i.e.

  • a certified product, reagent, application or other tool that analyses human material and helps to diagnose patients.

There is a tendency for regulators to require that

  • drugs and companion diagnostics are developed in tandem.

A companion diagnostic test is essentially a biomarker test

  • that enables better decision making on the use of a therapy.

In other words: it is a diagnostic test that is

  • specifically linked to a therapeutic drug.

The goal here is to increase the safety and the efficacy of the drug.

Pharmaceutical and biotechnology companies are trying to change their tack now and are working hard

  • on integrating the co-development concept but
  • true co-evelopment has been a rare phenomenon

There are two reasons for this.

  1. clinically useful biomarkers are usually established late in the drug validation process.
  2. the worlds of drug development and diagnostics, although both part of health care, are parallel universes

In the next paragraphs we will elaborate on these issues.

Clinically Useful Biomarkers Found Late in The Drug Validation Process

It is quite difficult to find clinically useful predictive biomarkers early on in a drug
development program, simply because

  • they can only be determined on the basis of the patients’ responses to the drug.

A number of biomarkers, such as KRAS and EGFR mutations,

  1. could only be established after a sufficient number of patients—
  2. well beyond the usual number for a Phase III trial—
  3. had elicited better understanding of differential drug response.

If the method of clinical trials changes consistent with our new understanding
of diseases and classifying patients (as we described in chapter one
and two), we might be able to find these biomarkers at an earlier stage. But

  • as long as still 30% of the drugs fail during Phase III,
  • diagnostic manufacturers will not be able to afford huge investments
  •  in the development of companion diagnostics.

Parallel Universes

The worlds of drugs and diagnostics are parallel universes: in general

  1. they have different development timelines, product lifecycles, return on investment, customers, and regulations.
  • Drugs are valued and reimbursed as products, typically of high value.
  • Diagnostics are valued and paid for as services,typically at a much lower value.
  • Relatively few if any models exist for valuing a drug diagnostic combination.

This issue will be discussed in the next chapter.

Drugs are protected by patents, but in the companion diagnostic arena,

  • biomarkers are considered to be within the public domain and there is less emphasis on intellectual property.

It is even debated whether biomarkers should be patented at all, because

  • biomarkers are not invented but already exist in cells.

Drugs and diagnostics are also regulated differently. It takes many years and

  • a large amount of data from expensive clinical trials to get a drug approved.

Under current rules, however in the current IVD Directive

  • there is little mention of clinical evidence.

However, the EU IVD Directive regulating in-vitro diagnostics is under revision.
The current IVD classification

  • does not take scientific and technological evolution into account
  •  which is expected to be changed towards a new risk-based approach.

A revised IVD Directive (which may even become a Regulation)

  • will improve safety and efficacy, but will also raise difficulties.

It is likely to state that an IVD assay must have the

  • performance characteristics  required for fulfilling a clinical purpose.

Moreover, requirements may be added on

  • how to demonstrate clinical validity, possibly proportionately with
  • the risk level of the test.

The intention to increase safety will inevitably

  • lead to higher costs and greater efforts for manufacturers.

It will take IVD manufacturers years and unusually high
investments to take an IVD past the regulators. Undoubtedly,

  • Phase III trials would benefit from well validated biomarker tests.

But it is almost impossible to comply

  • with regulations that require a clinically validated diagnostic test,
  • simply because clinical evidence will only be provided
  • by a clinical trial itself, and this is unlikely to be required for many IVDs.

Another point is that more regulation can severely impact innovation. The larger
investments that are needed might limit the ability

  • of (especially small) innovative companies to discover and develop biomarkers.

The Quality of Diagnostic Tests

In order to gain more insight into the consequences of stricter regulation, we must

  • dive into the universe of diagnostic practice in the EU a little deeper, because there is another challenging reality.

In the diagnostic arena there are, basically, two types of tests in use:

  1. manufactured diagnostic products, which are regulated in the EU, and
  2. lab developed tests (LDT’s), which are not regulated.

Thus, in the EU, there is a broad range of methodologies, often driven by

  • the individual laboratory’s capabilities, access to diagnostic testing platforms,
    reimbursement and preferences of the individual team.

How this situation works in practice was pointed out by a multicenter study. Various laboratories were asked to select patients 

  • with a positive EGFR or  KRAS biomarker.
  • Some of the tests were in close agreement but, others were not.
  • Due to the poor quality of so-called ‘home brew tests’ or inadequately validated tests in hospital laboratories,
  • patients with a positive EGFR biomarker might be excluded from an effective therapy on false grounds.

The result of this industry dynamic is a lack of standardization of testing and,

  1. inconsistent patient selection for therapy.
  2. Improper patient selections will lead to poor therapy outcomes, or worse, to adverse reactions to drugs.

This situation is not likely to change under a new EU IVD Regulation. IVD
manufacturers may have to make greater efforts in the future, including financially, to get an IVD past the regulators. However,

  • there is no legally enforced requirement
  • to use the companion diagnostic test approved in the clinical trial.

So, for as long as regulatory bodies do not sanction laboratories that

  1. do not conduct tests in line with pharmaceutical clinical trials,
  2. the quality of the testing will not improve.

Diagnostic partners cannot justify large investments in bringing products onto a market, in which there are

  • no rules for laboratories to perform substitute testing.

Regulation for testing  laboratories will be a more effective first step

  • towards increasing quality and safety than sharpening the rules for IVD manufacturers.

Discussion

The industry needs time to bring

  • diagnostic and therapeutic research together.

The regulation should leave room

  • to develop companion diagnostics in various business models,
  • instead of only requiring co-development.

Let’s not forget: many drugs

  • have proven to be effective without companion diagnostic tests.

We would not want to rule those out.

Regulation to increase safety is in itself a good thing, but

  • it is also important to provide opportunities for
  • small innovative enterprises to enter the market.

3.5 BioPerspectives

Apr 3, 2013

Personalized Medicine: Health Economic Aspects

This fourth chapter of a five-part series discusses personalized medicine’s
financial impact.

Alexander Roediger

http://www.genengnews.com/bioperspectives/personalized-medicine-health-economic-aspects/4824

There is a widespread skepticism about the financial impact of personalized medicine. [AlienForce – Fotolia.com]

From the previous discussion of turning biomarkers into companion diagnostics,

  • we now shift to a discussion of healthcare.

Personalized medicine has an impact on the healthcare budgets.

Are the tools that healthcare economists and pricing and reimbursement authorities
currently use

  • suitable for analysing personalized medicine?

In short, the concept of personalized medicine revolves around the idea of the
selection of patients who are likely to

  • respond the medicine, so that the treatment will offer better outcomes.

According to the recent Quintiles report “biopharma and managed care executives are

  • optimistic that personalised medicine will improve efficacy, safety and public health”.

However, there is a widespread skepticism about the financial impact of personalized medicine. According to the same report

  • 56% of managed care executives feel that personalized medicine will increase cost of prescription medicines.

Consequently, pricing and reimbursement of personalized medicines

  • need careful consideration and a balanced view
  • on cost-effectiveness and incentives innovation.

Another Economic Model is Needed

Much of our experience with economic evaluations of medicines is based on

  • medicines designed to treat the whole patient population.

Evaluations of the economic value of a particular drug

  • usually involve comparisons with treatment alternatives or palliative care.

Such comparative effectiveness are typically assessed

  • on the basis of data collected in
  • randomized controlled trials across a broad population of patients.

There have been attempts to

  • identify sub-sets of patients that benefit most noticeably from the medicine, i.e.
    patients over or under a certain age or patients judged to have a ‘severe’ form of a
    certain disease.

But in many cases these evaluations were

  1. not supported by robust evidence, and the sub-groups were not well characterized
  2. nor were they statistically well-sampled in the clinical data.

Testing May Be Economically Viable

In the personalized medicine concept,

  • a sub-group of responders is selected or screened out, which
  • raises the hope that economic evaluations will be more straightforward and positive.

This is particularly true for the cheapest use of costly molecular targeted agents.

Biomarkers can improve the ability

  • to identify responders and non-responders,
  • and insure that the information is used to make better treatment choices.

As a consequence, the respective drug gains a  more favorable risk-benefit ratio,

  • patients are expected to have better health outcomes and better quality of life and
  • healthcare resources are likely to be used more efficiently.

Much of the efficiency gains of personalized medicines depends on the testing—

  •  “testing before treating may be economically viable
  • if the savings gained by avoiding ineffective treatment and adverse events are greater than the cost of testing.”

Studies on the cost-effectiveness of personalized medicines are still on the way
but promising results are available. The introduction of

  • a companion diagnostic strategy in advanced non-small cell lung cancer
  • reduced overall treatment costs by more than € 800 compared to current treatment.

A cost-effectiveness analysis in the field of chronic myeloid leukemia showed that

  • the use of the FISH test reduced treatment costs by €12’500.6 In addition, it increased the life quality.

Reduction of R&D Costs

Personalized medicine may provide more value for money—

  1. not only because of improved drug effectiveness and reduced toxicity, but
  2. also decrease the average research and development costs for new medicines.

Clinical trials are the most expensive part of R&D (nearly 50% of the investment).
The costs of clinical trials seem

  • to have risen by one third between 2005 and 2007 due to
  • increasing regulatory and other requirements.

Biomarkers may enhance the efficacy of clinical trials of new drugs

  • by investing more heavily in early research to identify key biomarkers,
  • and in targeting relevant sub-groups of patients.

Smaller (and maybe even shorter) clinical trials are likely to reduce
development costs.

This sounds promising, but since personalized medicine is in its early stages,

  • efficiency gains may occur only in the long run. Furthermore,
  • companion diagnostics are likely to increase the additional costs and the complexity of
  • the risky process of drug discovery and development.

In oncology,  where personalized medicine is currently progressing most rapidly,

  • the late stage failure rate of new compounds has historically been higher than in any other area.

A well-known argument against personalized medicines is, that

  • they will lead to smaller groups of eligible patients and
  • therefore lead to higher unit prices in order to deliver competitive return on investment.

Additional value, therefore, “needs to be captured in terms of

  • premium pricing, faster adoption or longer effective patent life for a portfolio of targeted drugs,
  • to offset the reduction in potential revenues from market stratification”.

However, personalized medicines do not only diminish groups of eligible patients,

  • they enlarge them as well (see chapter 2).

The following problems still have to be solved:

  1. The costs of diagnostics are not easy to describe
  2. The evaluation of cost-effectiveness is difficult
  3. The regulatory pathway is fragmented

Costs of Diagnostics Are Not Easy to Describe

Implementing personalized medicines in healthcare is potentially a costly investment:

  • it requires testing a whole patient population to identify groups of responding patients
  • or to screen out patients likely to suffer adverse events or who need different dosing.

Some evaluations have attempted to tie the diagnostic

  • to the use of a specific new medicine (“co-dependent technologies”) and
  • have in some cases passed the costs of diagnosis on to the medicines developer.

But more and more multiple tests and multiple personalized medicines for particular
diseases become available. For example, in colorectal cancer and non-small cell
lung cancer,

  • a set of parallel tests are to be performed on a number of molecular biomarkers
  • to decide between a range of personalized medicines.

But more complex diagnostic and treatment pathways make it

  • less easy to ascribe costs of diagnosis to the use of a particular medicine.

Evaluation of Cost-Effectiveness is Difficult

To evaluate the cost-effectiveness of particular molecular diagnostic approaches
is also problematic. Diagnostics are normally supported by

  • analytical performance data and rarely by clinical or outcome data,
  • but in order to perform a health technology assessment you need the latter.

According to a systematic review, only eight studies evaluated the clinical validity, andnone of the studies was a prospective evaluation of a test’s clinical utility.

In addition, personalized medicines may also require

  • a different view on the clinical evidence available when a medicine is launched.

In some examples, personalized medicines have been approved on the basis of

  • a retrospective analysis of clinical data identifying the responsive sub-population.

In other cases, personalised medicines have been launched under

  • conditional approval mechanisms on the basis of phase 2 data alone (smaller studies, often
    without overall survival end points).

Regulatory Pathway is Fragmented

Discussion

Current health economic research shows that personalization of treatment, i.e.
identifying responders and non-responders, has

  • the potential to improve effectiveness and reduce costs.

In addition, biomarker testing may lead to more successful R&D projects.
However, the value of so-called tailor-made therapies depends to a large
extent on the quality of the tailor. In other words,

  • adaptations to both regulatory structures and market structures
  • are necessary to encourage the development of personalized medicine.

Part 4: Biomarkers as Diagnostics

There has been consolidation going on for over a decade in both the pharmaceutical and in the diagnostics industry, and at the same time the page is being rewritten for health care delivery.  I shall try to work through a clear picture of these not coincidental events.

Key notables:

  1. A growing segment of the US population is reaching Medicare age
  2. There is also a large underserved population in both metropolitan and nonurban areas and a fragmentation of the middle class after a growth slowdown in the economy since the 2008 deep recession.
  3. The deep recession affecting worldwide economies was only buffered by availability of oil or natural gas.
  4. In addition, there was a self-destructive strategy to cut spending on national scales that withdrew the support that would bolster support for infrastrucrue renewl.
  5. There has been a dramatic success in the clinical diagnostics industry, with a long history of being viewed as a loss leader, and this has been recently followed by the pharmaceutical industry faced with inability to introduce new products, leading to more competition in off-patent medications.
  6. The introduction of the Accountable Care Act has opened the opportunities for improved care, despite political opposition, and has probably sustained opportunity in the healthcare market.

Let’s take a look at this three headed serpent. – Pharma, Diagnostics, New Entity
?  The patient  ?
?  Insurance    ?
?  Physician    ?

4 Biomarkers in Medical Practice

4.1 Case in Point 1.

A Solid Prognostic Biomarker

HDL-C: Target of Therapy or Fuggedaboutit?

Steven E. Nissen, MD, MACC, Peter Libby, MD

DisclosuresNovember 06, 2014

Steven E. Nissen, MD, MACC: I am Steve Nissen, chairman of the Department of Cardiovascular Medicine at the Cleveland Clinic. I am here with Dr Peter Libby, chief of cardiology at the Brigham and Women’s Hospital and professor of medicine at Harvard Medical School. We are going to discuss high-density lipoprotein cholesterol (HDL-C), a topic that has been very controversial recently. Peter, HDL-C has been a pretty good biomarker. The question is whether it is a good target.

Peter Libby, MD: Since the early days in Berkley, when they were doing ultracentrifugation, and when it was reinforced and put on the map by the Framingham Study,[1] we have known that HDL-C is an extremely good biomarker of prospective cardiovascular risk with an inverse relationship with all kinds of cardiovascular events. That is as solid a finding as you can get in observational epidemiology. It is a very reliable prospective marker. It’s natural that the pharmaceutical industry and those of us who are interested in risk reduction would focus on HDL-C as a target. That is where the controversies come in.

Dr Nissen: It has been difficult. My view is that the trials that have attempted to modulate HDL-C or the drugs they used have been flawed. Although the results have not been promising, the jury is yet out. Torcetrapib, the cholesteryl ester transfer protein (CETP) inhibitor developed by Pfizer, had anoff-target toxicity.[2] Niacin is not very effective, and there are a lot of downsides to the drug. That has been an issue, but people are still working on this. We have done some studies. We did our ApoA-1 Milano infusion study[3]about a decade ago, which showed very promising results with respect to shrinking plaques in coronary arteries. I remain open to the possibility that the right drug in the right trial will work.

Dr Libby: What do you do with the genetic data that have come out in the past couple of years? Sekar Kathiresan masterminded and organized an enormous collaboration[4] in which they looked, with contemporary genetics, at whether HDL had the genetic markers of being a causal risk factor. They came up empty-handed.

Dr Nissen: I am cautious about interpreting those data, like I am cautious about interpreting animal studies of atherosclerosis. We have both lived through this problem in which something works extremely well in animals but doesn’t work in humans, or it doesn’t work in animals but it works in humans. The genetic studies don’t seal the fate of HDL. I have an open mind about this. Drugs are complex. They work by complex mechanisms. It is my belief that what we have to do is test these hypotheses in well-designed clinical trials, which are rigorously performed with drugs that are clean—unlike torcetrapib—and don’t have off-target toxicities.

An Unmet Need: High Lp(a) Levels

Dr Nissen: I’m going to push back on that and make a couple of points. The HPS2-THRIVE study was flawed. They studied the wrong people. It was not a good study, and AIM-HIGH[8] was underpowered. I am not putting people on niacin. What do you do with a patient whose Lp(a) is 200 mg/dL?

Dr Libby: I’m waiting for the results of the PCSK9 and anacetrapib studies. You can tell me about evacetrapib.[9]Reducing Lp(a) is an unmet medical need. We both care for kindreds with high Lp(a) levels and premature coronary artery disease. We have no idea what to do with them other than to treat them with statins and lower their LDL-C levels.

Dr Nissen: I have taken a more cautious approach with respect to taking people off of niacin. If I have patients who are doing well and tolerating it (depending on why it was started), I am discontinuing niacin in some people. I am starting very few people on the drug, but I worry about the quality of the trial.

Dr Libby: So you are of the “don’t start don’t stop” school?

Dr Nissen: Yes. It’s difficult when the trial is fatally flawed. There were 11,000 patients from China in this study. I have known for years that if you give niacin to people of Asiatic ethnic descent, they have terrible flushing and they won’t continue the drug. One question is, what was the adherence? The adverse events would have been tolerable had there been efficacy. The concern here is that this study was destined to fail because they studied a low LDL/high HDL population, a group of people for whom niacin just isn’t used.

Triglycerides and HDL: Do We Have It Backwards?

Dr Libby: What about the recent genetic[10] and epidemiologic data that support triglycerides, and apolipoprotein C3 in particular as a causal risk factor? Have we been misled through all of the generations in whom we have been adjusting triglycerides for HDL-C and saying that triglycerides are not a causal risk factor because once we adjust for HDL, the risk goes away? Do you think we got it backwards?

Dr Nissen: The tricky factor here is that because of this intimate inverse relationship between triglycerides and HDL, we may be talking about the same phenomenon. That is one of the reasons that I am not certain we are not going to be able to find a therapy. What if you had a therapy that lowered triglycerides and raised HDL-C? Could that work? Could that combination be favorable? I want answers from rigorous, well-designed clinical trials that ask the right questions in the right populations. I am disappointed, just as I have been disappointed by the fibrate trials.[11,12] There is a class of drugs that raises HDL-C a little and lowers triglycerides a lot.

Dr Nissen: But the gemfibrozil studies (VA-HIT[13] and Helsinki Heart[14]) showed benefit.

The Dyslipidemia Bar Has Been Raised

Dr Libby: Those studies were from the pre-statin era. We both were involved in trials in which patients were on high-dose statins at baseline. Do you think that this is too high a bar?

Dr Nissen: The bar has been raised, and for the pharmaceutical industry, the studies that we need to find out whether lowering triglycerides or raising HDL is beneficial are going to be large. We are doing a study with evacetrapib. It has 12,000 patients. It’s fully enrolled. Evacetrapib is a very clean-looking drug. It doesn’t have such a long biological half-life as anacetrapib, so I am very encouraged that it won’t have that baggage of being around for 2-4 years. We’ve got a couple of shots on goal here. Don’t forget that we have multiple ongoing studies of HDL-C infusion therapies that are still under development. Those have some promise too. The jury is still out.

Dr Libby: We agree on the need to do rigorous, large-scale endpoint trials. Do the biomarker studies, but don’t wait to start the endpoint trial because that’s the proof in the pudding.

Dr Nissen: Exactly. We have had a little controversy about HDL-C. We often agree, but not always, and we may have a different perspective. Thanks for joining me in this interesting discussion of what will continue to be a controversial topic for the next several years until we get the results of the current ongoing trials.

4.2 Case in Point 2.

NSTEMI? Honesty in Coding and Communication?

Melissa Walton-Shirley

November 07, 2014

The complaint at ER triage: Weakness, fatigue, near syncope of several days’ duration, vomiting, and decreased sensorium.

The findings: O2sat: 88% on room air. BP: 88 systolic. Telemetry: Sinus tachycardia 120 bpm. Blood sugar: 500 mg/dL. Chest X ray: atelectasis. Urinalysis: pyuria. ECG: T-wave-inversion anterior leads. Echocardiography: normal left ventricular ejection fraction (LVEF) and wall motion. Troponin I: 0.3 ng/mL. CT angiography: negative for pulmonary embolism (PE). White blood cell count: 20K with left shift. Blood cultures: positive for Gram-negative rods.

The treatment: Intravenous fluids and IV levofloxacin—changed to ciprofloxacin.

The communication at discharge: “You had a severe urinary-tract infection and grew bacteria in your bloodstream. Also, you’ve had a slight heart attack. See your cardiologist immediately upon discharge-no more than 5 days from now.”

The diagnoses coded at discharge: Urosepsis and non-ST segment elevation MI (NSTEMI) 410.1.

One year earlier: This moderately obese patient was referred to our practice for a preoperative risk assessment. The surgery planned was a technically simple procedure, but due to the need for precise instrumentation, general endotracheal anesthesia (GETA) was being considered. The patient was diabetic, overweight, and short of air. A stress exam was equivocal for CAD due to poor exercise tolerance and suboptimal imaging. Upon further discussion, symptoms were progressive; therefore, cardiac cath was recommended, revealing angiographically normal coronaries and a predictably elevated left ventricular end diastolic pressure (LVEDP) in the mid-20s range. The patient was given a diagnosis of diastolic dysfunction, a prescription for better hypertension control, and in-depth discussion on exercise and the Mediterranean and DASH diets for weight loss. Symptoms improved with a low dose of diuretic. The surgery was completed without difficulty. Upon follow-up visit, the patient felt well, had lost a few pounds, and blood pressure was well controlled.

Five days after ER workup: While out of town, the patient developed profound weakness and went to the ER as described above. Fast forward to our office visit in the designated time frame of “no longer than 5 days’ postdischarge,” where the patient and family asked me about the “slight heart attack” that literally came on the heels of a normal coronary angiogram.

But the patient really didn’t have a “heart attack,” did they? The cardiologist aptly stated that it was likely nonspecific troponin I leak in his progress notes. Yet the hospitalist framed the diagnosis of NSTEMI as item number 2 in the final diagnoses.

The motivations on behalf of personnel who code charts are largely innocent and likely a direct result of the lack of understanding of the coding system on behalf of us as healthcare providers. I have a feeling, though, that hospitals aren’t anxious to correct this misperception, due to an opportunity for increased reimbursement. I contacted a director of a coding department for a large hospital who prefers to remain anonymous. She explained that NSTEMI ICD9 code 410.1 falls in DRG 282 with a weight of .7562. The diagnosis of “demand ischemia,” code 411.89, a slightly less inappropriate code for a nonspecific troponin I leak, falls in DRG 311 with a weight of .5662. To determine reimbursement, one must multiply the weight by the average hospital Medicare base rate of $5370. Keep in mind that each hospital’s base rate and corresponding payment will vary. The difference in reimbursement for a large hospital bill between these two choices for coding is substantial, at over $1000 difference ($4060 vs $3040).

Although hospitals that are already reeling from shrinking revenues will make more money on the front end by coding the troponin leak incorrectly as an NSTEMI, when multiple unnecessary tests are generated to follow up on a nondiagnostic troponin leak, the amount of available Centers for Medicare & Medicaid Services (CMS) reimbursement pie shrinks in the long run. Furthermore, this inappropriate categorization generates extreme concern on behalf of patients and family members that is often never laid to rest. The emotional toll of a “heart-attack” diagnosis has an impact on work fitness, quality of life, cost of medication, and the cost of future testing. If the patient lived for another 100 years, they will likely still list a “heart attack” in their medical history.

As a cardiologist, I resent the loose utilization of one of “my” heart-attack codes when it wasn’t that at all. At discharge, we need to develop a better way of communicating what exactly did happen. Equally important, we need to communicate what exactly didn’t happen as well.

4.3 Case in Point 3.

Blood Markers Predict CKD Heart Failure 

Published: Oct 3, 2014 | Updated: Oct 3, 2014

Elevated levels of high-sensitivity troponin T (hsTnT) and N-terminal pro-B-type natriuretic peptide (NT-proBNP) strongly predicted heart failure in patients with chronic kidney disease followed for a median of close to 6 years, researchers reported.

Compared with patients with the lowest blood levels of hsTnT, those with the highest had a nearly five-fold higher risk for developing heart failure and the risk was 10-fold higher in patients with the highest NT-proBNP levels compared with those with the lowest levels of the protein, researcher Nisha Bansal, MD, of the University of Washington in Seattle, and colleagues wrote online in the Journal of the American Society of Nephrology.

A separate study, published online in theJournal of the American Medical Association earlier in the week, also examined the comorbid conditions of heart and kidney disease, finding no benefit to the practice of treating cardiac surgery patients who developed acute kidney injury with infusions of the antihypertensive drug fenoldopam.

The study, reported by researcher Giovanni Landoni, MD, of the IRCCS San Raffaele Scientific Institute, Milan, Italy, and colleagues, was stopped early “for futility,” according to the authors, and the incidence of hypotension during drug infusion was significantly higher in patients infused with fenoldopam than placebo (26% vs. 15%; P=0.001).

Blood Markers Predict CKD Heart Failure

The study in patients with mild to moderate chronic kidney disease (CKD) was conducted to determine if blood markers could help identify patients at high risk for developing heart failure.

Heart failure is the most common cardiovascular complication among people with renal disease, occurring in about a quarter of CKD patients.

The two markers, hsTnT and NT-proBNP, are associated with overworked cardiac myocytes and have been shown to predict heart failure in the general population.

However, Bansal and colleagues noted, the markers have not been widely used in diagnosing heart failure among patients with CKD due to concerns that reduced renal excretion may raise levels of these markers, and therefore do not reflect an actual increase in heart muscle strain.

To better understand the importance of elevated concentrations of hsTnT and NT-proBNP in CKD patients, the researchers examined their association with incident heart failure events in 3,483 participants in the ongoing observational Chronic Renal Insufficiency Cohort (CRIC) study.

All participants were recruited from June 2003 to August 2008, and all were free of heart failure at baseline. The researchers used Cox regression to examine the association of baseline levels of hsTnT and NT-proBNP with incident heart failure after adjustment for demographic influences, traditional cardiovascular risk factors, makers of kidney disease, pertinent medication use, and mineral metabolism markers.

At baseline, hsTnT levels ranged from ≤5.0 to 378.7 pg/mL and NT-proBNP levels ranged from ≤5 to 35,000 pg/mL. Compared with patients who had undetectable hsTnT, those in the highest quartile (>26.5 ng/mL) had a significantly higher rate of heart failure (hazard ratio 4.77; 95% CI 2.49-9.14).

Compared with those in the lowest NT-proBNP quintile (<47.6 ng/mL), patients in the highest quintile (>433.0 ng/mL) experienced an almost 10-fold increase in heart failure risk (HR 9.57; 95% CI 4.40-20.83).

The researchers noted that these associations remained robust after adjustment for potential confounders and for the other biomarker, suggesting that while hsTnT and NT-proBNP are complementary, they may be indicative of distinct biological pathways for heart failure.

Even Modest Increases in NP-proBNP Linked to Heart Failure

The findings are consistent with an earlier analysis that included 8,000 patients with albuminuria in the Prevention of REnal and Vascular ENd-stage Disease (PREVEND) study, which showed that hsTnT was associated with incident cardiovascular events, even after adjustment for eGFR and severity of albuminuria.

“Among participants in the CRIC study, those with the highest quartile of detectable hsTnT had a twofold higher odds of left ventricular hypertrophy compared with those in the lowest quartile,” Bansal and colleagues wrote, adding that the findings were similar after excluding participants with any cardiovascular disease at baseline.

Even modest elevations in NT-proBNP were associated with significantly increased rates of heart failure, including in subgroups stratified by eGFR, proteinuria, and diabetic status.

“NT-proBNP regulates blood pressure and body fluid volume by its natriuretic and diuretic actions, arterial dilation, and inhibition of the renin-aldosterone-angiotensin system and increased levels of this marker likely reflect myocardial stress induced by subclinical changes in volume or pressure, even in persons without clinical disease,” the researchers wrote.

The researchers concluded that further studies are needed to develop and validate risk prediction tools for clinical heart failure in patients with CKD, and to determine the potential role of these two biomarkers in a heart failure risk prediction and prevention strategy.

Fenoldopam ‘Widely Promoted’ in AKI Cardiac Surgery Setting

The JAMA study examined whether the selective dopamine receptor D agonist fenoldopam mesylate can reduce the need for dialysis in cardiac surgery patients who develop acute kidney injury (AKI).

Fenoldopam induces vasodilation of the renal, mesenteric, peripheral, and coronary arteries, and, unlike dopamine, it has no significant affinity for D2 receptors, meaning that it theoretically induces greater vasodilation in the renal medulla than in the cortex, the researchers wrote.

“Because of these hemodynamic effects, fenoldopam has been widely promoted for the prevention and therapy of AKI in the United States and many other countries with apparent favorable results in cardiac surgery and other settings,” Landoni and colleagues wrote.

The drug was approved in 1997 by the FDA for the indication of in-hospital, short-term management of severe hypertension. It has not been approved for renal indications, but is commonly used off-label in cardiac surgery patients who develop AKI.

Although a meta analysis of randomized trials, conducted by the researchers, indicated a reduction in the incidence and progression of AKI associated with the treatment, Landoni and colleagues wrote that the absence of a definitive trial “leaves clinicians uncertain as to whether fenoldopam should be prescribed after cardiac surgery to prevent deterioration in renal function.”

To address this uncertainty, the researchers conducted a prospective, randomized, parallel-group trial in 667 patients treated at 19 hospitals in Italy from March 2008 to April 2013.

All patients had been admitted to ICUs after cardiac surgery with early acute kidney injury (≥50% increase of serum creatinine level from baseline or low output of urine for ≥6 hours). A total of 338 received fenoldopam by continuous intravenous infusion for a total of 96 hours or until ICU discharge, while 329 patients received saline infusions.

The primary end point was the rate of renal replacement therapy, and secondary end points included mortality (intensive care unit and 30-day mortality) and the rate of hypotension during study drug infusion.

Study Showed No Benefit, Was Stopped Early

Part 5: Reality of the Diagnostics Industry in the US: Post Human Genome sequencing

5.1  Illimina and Roche

When Illumina Buys Roche: The Dawning Of The Era Of Diagnostics Dominance

Robert J. Easton, Alain J. Gilbert, Olivier Lesueur, Rachel Laing, and Mark Ratner
http://PharmaMedtechBI.com    | IN VIVO: The Business & Medicine Report Jul/Aug 2014; 32(7).

  • With current technology and resources, a well-funded IVD company can create and pursue a strategy of information gathering and informatics application to create medical knowledge, enabling it to assume the risk and manage certain segments of patients
  • We see the first step in the process as the emergence of new specialty therapy companies coming from an IVD legacy, most likely focused in cancer, infection, or critical care

When Illumina Inc. acquired the regulatory consulting firm Myraqa, a specialist in in vitro diagnostics (IVD), in July, the press release announcement characterized the deal as one that would bolster illumina’s in-house capabilities for clinical readiness and help prepare for its next growth phase in regulated markets. That’s not surprising given the US Food and Drug Administration’s (FDA) approval a year and a half ago of its MiSeq next-generation sequencer for clinical use. But the deal could also suggest illumina is beginning to move along the path toward taking on clinical risk – that is, eventually

  • advising physicians and patients, which would mean facing regulators directly

Such a move – by illumina, another life sciences tools firm, or an information specialist from the high-tech universe – is inevitable given

  • the emerging power of diagnostics and traditional health care players’ reluctance to themselves take on such risk.

Alternatively, we believe that a well-funded diagnostics company could establish this position. either way, such a champion would establish dominion over and earn higher valuation than less-aggressive players who

  • only supply compartmentalized drug and device solutions.

Diagnostics companies have long been dogged by a fundamental issue:

  1. they are viewed and valued more along the lines of a commodity business than as firms that deliver a unique product or service
  2. diagnostics companies are in position to do just that today because they are now advantaged by having access to more data points.
  3. if they were to cobble together the right capabilities, diagnostics companies would have the ability to turn information into true medical knowledge

Example: PathGEN PathChip

nucleic-acid-based platform detects 296 viruses, bacteria, fungi & parasites

http://ow.ly/d/2GvQhttp://ow.ly/DSORV

This puts the diagnostics player in an unfamiliar realm where it can ask the question of what value they offer compared with a therapeutic. The key is that diagnostics can now offer unique information and potentially unique tools to capture that information. In order to do so, it has to create information from the data it generates, and then to supply that knowledge to users who will value and act on that knowledge. Complex genomic tests, as much as physical examination, may be the first meaningful touch point for physicians’ classification of disease.

Even if lab tests are more expensive, it is a cheaper means for deciding what to do first for a patient than the trial and error of prescribing medication without adequate information. Information is gaining in value as the amount of treatment data available on genomically characterizable subpopulations increases. In such a circumstance
it is the ability to perform that advisory function that will add tremendous value above what any test provides, the leverage of being able to apply a proprietary diagnostics platform – and importantly, the data it generates. It is the ability to perform that advisory function that will add tremendous value above what any test provides.

Integrated Diagnostics Inc. and Biodesix Inc. with mass spectrometry has the tools for unraveling disease processes, and numerous players are quite visibly in or are getting into the business of providing medical knowledge and clinical decision support in pursuit of a huge payout for those who actually solve important disease mysteries. Of course one has to ask whether MS/MS is sufficient for the assigned task, and also whether the technology is ready for the kind of workload experienced in a clinical service compared to a research vehicle.  My impression (as a reviewer) is that it is not now the time to take this seriously.

Roche has not realized its intent with Ventana: failing to deliver on the promise of boosting Roche’s pipeline, which was a significant factor in the high price Roche paid. The combined company was to be “uniquely positioned to further expand Ventana’s business globally and together develop more cost-efficient, differentiated, and targeted medicines.  On the other hand,  Biodesix decided to use Veristrat to look back and analyze important trial data to try to ascertain which patients would benefit from ficlatuzumab (subset). The predictive effect for the otherwise unimpressive trial results was observed in both progression-free survival and overall survival endpoints, and encouraged the companies to conduct a proof-of-concept study of ficlatuzumab in combination with Tarceva in advanced Non Small Cell Lung Cancer Patients (NSCLC) selected using the Veristrat test.

A second phase of IVD evolution will be far more challenging to pharma, when the most accomplished companies begin to assemble and integrate much broader data
sets, thereby gaining knowledge sufficient to actually manage patients and dictate therapy, including drug selection. No individual physician has or will have access to all of this information on thousands of patients, combined with the informatics to tease out from trillions of data points the optimal personalized medical approach. When the IVD-origin knowledge integrator amasses enough data and understanding to guide therapy decisions in large categories, particularly drug choices, it will become more valuable than any of the drug suppliers.

This is an apparent reversal of fortune. The pharmaceutical industry has been considered the valued provider, while the IVD manufacturer has been the low valued cousin. Now, it is by an ability to make kore accurate the drug administration that the IVD company can control the drug bill, to the detriment of drug developers, by finding algorithms that generate equal-to-innovative-drug outcomes using generics for most of the patients, thereby limiting the margins of drug suppliers and the upsides for new drug discovery/development.

It is here that there appears to be a misunderstanding of the whole picture of the development of the healthcare industry.  The pharmaceutical industry had a high value added only insofar it could replace market leaders for treatment before or at the time of patent expiration, which largely depended either introducing a new class of drug, or by relieving the current drug in its class of undesired toxicities or “side effects”.  Otherwise, the drug armamentarium was time limited to the expiration date. In other words, the value was dependent on a window of no competition.  In addition, as the regulation of healthcare costs were tightening under managed care, the introduction of new products that were deemed to be only marginally better, could be substitued by “off-patent” drug products.

The other misunderstanding is related to the IVD sector.  Laboratory tests in the 1950’s were manual, and they could be done by “technicians” who might not have completed a specialized training in clinical laboratory sciences.  The first sign of progress was the introduction of continuous flow chemistry, with a sampling probe, tubing to bring the reacting reagents into a photocell, and the timing of the reaction controlled by a coiled glass tubing before introducing the colored product into a uv-visible photometer.  In perhaps a decade, the Technicon SMA 12 and 6 instruments were introduced that could do up to 18 tests from a single sample.

5.2  My Cancer Genome from Vanderbilt University: Matching Tumor Mutations to Therapies & Clinical Trials

Reporter: Aviva Lev-Ari, PhD, RN

My Cancer Genome from Vanderbilt University: Matching Tumor Mutations to Therapies & Clinical Trials


GenomOncology and Vanderbilt-Ingram Cancer Center (VICC) today announced a partnership for the exclusive commercial development of a decision support tool based on My Cancer Genome™, an online precision cancer medicine knowledge resource for physicians, patients, caregivers and researchers.

Through this collaboration, GenomOncology and VICC will enhance My Cancer Genome through the development of a new genomics content management tool. The MyCancerGenome.org website will remain free and open to the public. In addition, GenomOncology will develop a decision support tool based on My Cancer Genome™ data that will enable automated interpretation of mutations in the genome of a patient’s tumor, providing actionable results in hours versus days.

Vanderbilt-Ingram Cancer Center (VICC) launched My Cancer Genome™ in January 2011 as an integral part of their Personalized Cancer Medicine Initiative that helps physicians and researchers track the latest developments in precision cancer medicine and connect with clinical research trials. This web-based information tool is designed to quickly educate clinicians on the rapidly expanding list of genetic mutations that impact cancers and enable the research of treatment options based on specific mutations. For more information on My Cancer Genome™visit www.mycancergenome.org/about/what-is-my-cancer-genome.

Therapies based on the specific genetic alterations that underlie a patient’s cancer not only result in better outcomes but often have less adverse reactions

Up front fee

Nominal fee covers installation support, configuring the Workbench to your specification, designing and developing custom report(s) and training your team.

Per sample fee

GenomOncology is paid on signed-out clinical reports. This philosophy aligns GenomOncology with your Laboratory as we are incentivized to offer world-class support and solutions to differentiate your clinical NGS program. There is no annual license fee.

5.3 Clinical Trial Services: Foundation Medicine & EmergingMed to Partner

Reporter: Aviva Lev-Ari, PhD, RN
http://pharmaceuticalintelligence.com/2014/11/03/clinical-trial-services-foundation-medicine-emergingmed-to-partner/

Foundation Medicine and EmergingMed said today that they will partner to offer clinical trial navigation services for health care providers and their patients who have received one of Foundation Medicine’s tumor genomic profiling tests.

The firms will provide concierge services to help physicians

  • identify appropriate clinical trials for patients
  • based on the results of FoundationOne or FoundationOne Heme.

“By providing clinical trial navigation services, we aim to facilitate

  • timely and accurate clinical trial information and enrollment support services for physicians and patients,
  • enabling greater access to treatment options based on the unique genomic profile of a patient’s cancer

Currently, there are over 800 candidate therapies that target genomic alterations in clinical trials,

  • but “patients and physicians must identify and act on relevant options
  • when the patient’s clinical profile is aligned with the often short enrollment window for each trial.

These investigational therapies are an opportunity to engage patients with cancer whose cancer has progressed or returned following standard treatment in a most favorable second option after relapse.  The new service is unique in notifying when new clinical trials emerge that match a patient’s genomic and clinical profile.

Google signs on to Foundation Medicine cancer Dx by offering tests to employees

By Emily Wasserman

Diagnostics luminary Foundation Medicine ($FMI) is generating some upward momentum, fueled by growing revenues and the success of its clinical tests. Tech giant Google ($GOOG) has taken note and is signing onto the company’s cancer diagnostics by offering them to employees.

Foundation Medicine CEO Michael Pellini said during the company’s Q3 earnings call that Google will start covering its DNA tests for employees and their family members suffering from cancer as part of its health benefits portfolio, Reuters reports.

Both sides stand to benefit from the deal, as Google looks to keep a leg up on Silicon Valley competitors and Foundation Medicine expands its cancer diagnostics platform. Last month, Apple ($AAPL) and Facebook ($FB) announced that they would begin covering the cost of egg freezing for female employees. A diagnostics partnership and attractive health benefits could work wonders for Google’s employee retention rates and bottom line.

In the meantime, Cambridge, MA-based Foundation Medicine is charging full speed ahead with its cancer diagnostics platform after filing for an IPO in September 2013. The company chalked up 6,428 clinical tests during Q3 2014, an eye-popping 149% increase year over year, and brought in total revenue for the quarter of $16.4 million–a 100% leap from last year. Foundation Medicine credits the promising numbers in part to new diagnostic partnerships and extended coverage for its tests.

In January, the company teamed up with Novartis ($NVS) to help the drugmaker evaluate potential candidates for its cancer therapies. In April, Foundation Medicine announced that it would develop a companion diagnostic test for a Clovis Oncology ($CLVS) drug under development to treat patients with ovarian cancer, building on an ongoing collaboration between the two companies.

Foundation Medicine also has its sights set on China’s growing diagnostics market, inking a deal in October with WuXi PharmaTech ($WX) that allows the company to perform lab testing for its FoundationOne assay at WuXi’s Shanghai-based Genome Center.

Foundation Medicine teams with MD Anderson for new trial of cancer Dx

Second study to see if targeted therapy can change patient outcomes

August 15, 2014 | By   FierceDiagnostics

Foundation Medicine ($FMI) is teaming up with the MD Anderson Cancer Center in Texas for a new trial of the the Cambridge, MA-based company’s molecular diagnostic cancer test that targets therapies matched to individual patients.

The study is called IMPACT2 (Initiative for Molecular Profiling and Advanced Cancer Therapy) and is designed to build on results from the the first IMPACT study that found

  • 40% of the 1,144 patients enrolled had an identifiable genomic alteration.

The company said that

  • by matching specific gene alterations to therapies,
  • 27% of patients in the first study responded versus
  • 5% with an unmatched treatment, and
  • “progression-free survival” was longer in the matched group.

The FoundationOne molecular diagnostic test

  • combines genetic sequencing and data gathering
  • to help oncologists choose the best treatment for individual patients.

Costing $5,800 per test, FoundationOne’s technology can uncover a large number of genetic alterations for 200 cancer-related genes,

  • blending genomic sequencing, information and clinical practice.

“Based on the IMPACT1 data, a validated, comprehensive profiling approach has already been adopted by many academic and community-based oncology practices,” Vincent Miller, chief medical officer of Foundation Medicine, said in a release. “This study has the potential to yield sufficient evidence necessary to support broader adoption across most newly diagnosed metastatic tumors.”

The company got a boost last month when the New York State Department of Health approved Foundation Medicine’s two initial cancer tests: the FoundationOne test and FoundationOne Heme, which creates a genetic profile for blood cancers. Typically,

  • diagnostics companies struggle to win insurance approval for their tests
  • even after they gain a regulatory approval, leaving revenue growth relatively flat.

However, Foundation Medicine reported earlier this week its Q2 revenue reached $14.5 million compared to $5.9 million for the same period a year ago. Still,

  1. net losses continue to soar as the company ramps up
  2. its commercial and business development operation,
  • hitting $13.7 million versus a $10.1 million deficit in the second quarter of 2013.

Oncology

There has been a remarkable transformation in our understanding of

  • the molecular genetic basis of cancer and its treatment during the past decade or so.

In depth genetic and genomic analysis of cancers has revealed that

  • each cancer type can be sub-classified into many groups based on the genetic profiles and
  • this information can be used to develop new targeted therapies and treatment options for cancer patients.

This panel will explore the technologies that are facilitating our understanding of cancer, and

  • how this information is being used in novel approaches for clinical development and treatment.
Oncology – Reported by Dr. Aviva Lev-Ari, Founder, Leaders in Pharmaceutical Intelligence

Opening Speaker & Moderator:

Lynda Chin, M.D.
Department Chair, Department of Genomic Medicine
MD Anderson Cancer Center

Panelists:

Roy Herbst, M.D., Ph.D.
Ensign Professor of Medicine and Professor of Pharmacology;
Chief of Medical Oncology, Yale Cancer Center and Smilow Cancer Hospital

Development new drugs to match patient, disease and drug – finding the right patient for the right Clinical Trial

  • match patient to drugs
  • partnerships: out of 100 screened patients, 10 had the gene, 5 were able to attend the trial — without the biomarker — all 100 patients would participate for the WRONG drug for them (except the 5)
  • patients want to participate in trials next to home NOT to have to travel — now it is in the protocol
  • Annotated Databases – clinical Trial informed consent – adaptive design of Clinical Trial vs protocol
  • even Academic MD can’t read the reports on Genomics
  • patients are treated in the community — more training to MDs
  • Five companies collaborating – comparison og 6 drugs in the same class
  • if drug exist and you have the patient — you must apply PM

Summary and Perspective:

The current changes in Biotechnology have been reviewed with an open question about the relationship of In Vitro Diagnostics to Biopharmaceuticals switching, with the potential, particularly in cancer and infectious diseases, to added value in targeted therapy by matching patients to the best potential treatment for a favorable outcome.

This reviewer does not see the movement of the major diagnostics leaders entering into the domain of direct patient care, even though there are signals in that direction.  The Roche example is perhaps the most interesting because Roche already became the elephant in the room after the introduction of Valium,  subsequently bought out Boehringer Mannheim Diagnostics to gain entry into the IVD market, and established a huge presence in Molecular Diagnostics early.  If it did anything to gain a foothold in the treatment realm, it would more likely forge a relationship with Foundation Medicine.  Abbott Laboratories more than a decade ago was overextended, and it had become the leader in IVD as a result of the specialty tests, but it fell into difficulties with quality control of its products in the high volume testing market, and acceeded to Olympus, Roche, and in the mid volume market to Beckman and Siemens.  Of course, Dupont and Kodak, pioneering companies in IVD, both left the market.

The biggest challenge in the long run is identified by the ability to eliminate many treatments that would be failures for a large number of patients. That has already met the proof of concept.  However, when you look at the size of the subgroups, we are not anywhere near a large scale endeavor.  In addition, there is a lot that has to be worked out that is not related to genomic expression by the “classic” model, but has to take into account the emrging knowledge and greater understanding of regulation of cell metabolism, not only in cancer, but also in chronic inflammatory diseases.

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Milestones in the Evolution of Diagnostics in the US HealthCare System: 1920s to Pre-Genomics

Author and Curator: Larry H Bernstein, MD, FCAP

Diagnostics Industry and Drug Development in the Genomics Era: Mid 80s to Present

http://pharmaceuticalintelligence.com/2014/11/16/diagnostics-industry-and-drug-development-in-the-genomics-era-mid-80s-to-present/

Emergence of an IVD Clinical Automated Diagnostics Industry

Why tests are ordered

  1. Screening
  2. Diagnosis
  3. Monitoring

Historical Perspective

Part 1: The early 20th Century

Case in point 1: Arterial Blood Gases.  Van Slyke. National Academy of Sciences.

The test is used to determine the pH of the blood, the partial pressure of carbon dioxide and oxygen, and the bicarbonate level. Many blood gas analyzers will also report concentrations of lactate, hemoglobin, several electrolytes, oxyhemoglobin, carboxyhemoglobin and methemoglobin. ABG testing is mainly used in pulmonology and critical care medicine to determine gas exchange which reflect gas exchange across the alveolar-capillary membrane.

DONALD DEXTER VAN SLYKE died on May 4, 1971, after a long and productive career that spanned three generations of biochemists and physicians. He left behind not only a bibliography of 317 journal publications and 5 books, but also more than 100 persons who had worked with him and distinguished themselves in biochemistry and academic medicine. His doctoral thesis, with Gomberg at University of Michigan was published in the Journal of the American Chemical Society in 1907.  Van Slyke received an invitation from Dr. Simon Flexner, Director of the Rockefeller Institute, to come to New York for an interview. In 1911 he spent a year in Berlin with Emil Fischer, who was then the leading chemist of the scientific world. He was particularly impressed by Fischer’s performing all laboratory operations quantitatively —a procedure Van followed throughout his life. Prior to going to Berlin, he published the  classic nitrous acid method for the quantitative determination of primary aliphatic amino groups,  the first of the many gasometric procedures devised by Van Slyke, and made possible the determination of amino acids. It was the primary method used to study amino acid composition of proteins for years before chromatography. Thus, his first seven postdoctoral years were centered around the development of better methodology for protein composition and amino acid metabolism.

With his colleague G. M. Meyer, he first demonstrated that amino acids, liberated during digestion in the intestine, are absorbed into the bloodstream, that they are removed by the tissues, and that the liver alone possesses the ability to convert the amino acid nitrogen into urea.  From the study of the kinetics of urease action, Van Slyke and Cullen developed equations that depended upon two reactions: (1) the combination of enzyme and substrate in stoichiometric proportions and (2) the reaction of the combination into the end products. Published in 1914, this formulation, involving two velocity constants, was similar to that arrived at contemporaneously by Michaelis and Menten in Germany in 1913.

He transferred to the Rockefeller Institute’s Hospital in 2013, under Dr. Rufus Cole, where “Men who were studying disease clinically had the right to go as deeply into its fundamental nature as their training allowed, and in the Rockefeller Institute’s Hospital every man who was caring for patients should also be engaged in more fundamental study”.  The study of diabetes was already under way by Dr. F. M. Allen, but patients inevitably died of acidosis.  Van Slyke reasoned that if incomplete oxidation of fatty acids in the body led to the accumulation of acetoacetic and beta-hydroxybutyric acids in the blood, then a reaction would result between these acids and the bicarbonate ions that would lead to a lower than-normal bicarbonate concentration in blood plasma. The problem thus became one of devising an analytical method that would permit the quantitative determination of bicarbonate concentration in small amounts of blood plasma.  He ingeniously devised a volumetric glass apparatus that was easy to use and required less than ten minutes for the determination of the total carbon dioxide in one cubic centimeter of plasma.  It also was soon found to be an excellent apparatus by which to determine blood oxygen concentrations, thus leading to measurements of the percentage saturation of blood hemoglobin with oxygen. This found extensive application in the study of respiratory diseases, such as pneumonia and tuberculosis. It also led to the quantitative study of cyanosis and a monograph on the subject by C. Lundsgaard and Van Slyke.

In all, Van Slyke and his colleagues published twenty-one papers under the general title “Studies of Acidosis,” beginning in 1917 and ending in 1934. They included not only chemical manifestations of acidosis, but Van Slyke, in No. 17 of the series (1921), elaborated and expanded the subject to describe in chemical terms the normal and abnormal variations in the acid-base balance of the blood. This was a landmark in understanding acid-base balance pathology.  Within seven years after Van moved to the Hospital, he had published a total of fifty-three papers, thirty-three of them coauthored with clinical colleagues.

In 1920, Van Slyke and his colleagues undertook a comprehensive investigation of gas and electrolyte equilibria in blood. McLean and Henderson at Harvard had made preliminary studies of blood as a physico-chemical system, but realized that Van Slyke and his colleagues at the Rockefeller Hospital had superior techniques and the facilities necessary for such an undertaking. A collaboration thereupon began between the two laboratories, which resulted in rapid progress toward an exact physico-chemical description of the role of hemoglobin in the transport of oxygen and carbon dioxide, of the distribution of diffusible ions and water between erythrocytes and plasma,
and of factors such as degree of oxygenation of hemoglobin and hydrogen ion concentration that modified these distributions. In this Van Slyke revised his volumetric gas analysis apparatus into a manometric method.  The manometric apparatus proved to give results that were from five to ten times more accurate.

A series of papers on the CO2 titration curves of oxy- and deoxyhemoglobin, of oxygenated and reduced whole blood, and of blood subjected to different degrees of oxygenation and on the distribution of diffusible ions in blood resulted.  These developed equations that predicted the change in distribution of water and diffusible ions between blood plasma and blood cells when there was a change in pH of the oxygenated blood. A significant contribution of Van Slyke and his colleagues was the application of the Gibbs-Donnan Law to the blood—regarded as a two-phase system, in which one phase (the erythrocytes) contained a high concentration of nondiffusible negative ions, i.e., those associated with hemoglobin, and cations, which were not freely exchaThe importance of Vanngeable between cells and plasma. By changing the pH through varying the CO2 tension, the concentration of negative hemoglobin charges changed in a predictable amount. This, in turn, changed the distribution of diffusible anions such as Cl” and HCO3″ in order to restore the Gibbs-Donnan equilibrium. Redistribution of water occurred to restore osmotic equilibrium. The experimental results confirmed the predictions of the equations.

As a spin-off from the physico-chemical study of the blood, Van undertook, in 1922, to put the concept of buffer value of weak electrolytes on a mathematically exact basis.
This proved to be useful in determining buffer values of mixed, polyvalent, and amphoteric electrolytes, and put the understanding of buffering on a quantitative basis. A
monograph in Medicine entitled “Observation on the Courses of Different Types of Bright’s Disease, and on the Resultant Changes in Renal Anatomy,” was a landmark that related the changes occurring at different stages of renal deterioration to the quantitative changes taking place in kidney function. During this period, Van Slyke and R. M. Archibald identified glutamine as the source of urinary ammonia. During World War II, Van and his colleagues documented the effect of shock on renal function and, with R. A. Phillips, developed a simple method, based on specific gravity, suitable for use in the field.

Over 100 of Van’s 300 publications were devoted to methodology. The importance of Van Slyke’s contribution to clinical chemical methodology cannot be overestimated.
These included the blood organic constituents (carbohydrates, fats, proteins, amino acids, urea, nonprotein nitrogen, and phospholipids) and the inorganic constituents (total cations, calcium, chlorides, phosphate, and the gases carbon dioxide, carbon monoxide, and nitrogen). It was said that a Van Slyke manometric apparatus was almost all the special equipment needed to perform most of the clinical chemical analyses customarily performed prior to the introduction of photocolorimeters and spectrophotometers for such determinations.

The progress made in the medical sciences in genetics, immunology, endocrinology, and antibiotics during the second half of the twentieth century obscures at times the progress that was made in basic and necessary biochemical knowledge during the first half. Methods capable of giving accurate quantitative chemical information on biological material had to be painstakingly devised; basic questions on chemical behavior and metabolism had to be answered; and, finally, those factors that adversely modified the normal chemical reactions in the body so that abnormal conditions arise that we characterize as disease states had to be identified.

Viewed in retrospect, he combined in one scientific lifetime (1) basic contributions to the chemistry of body constituents and their chemical behavior in the body, (2) a chemical understanding of physiological functions of certain organ systems (notably the respiratory and renal), and (3) how such information could be exploited in the
understanding and treatment of disease. That outstanding additions to knowledge in all three categories were possible was in large measure due to his sound and broadly based chemical preparation, his ingenuity in devising means of accurate measurements of chemical constituents, and the opportunity given him at the Hospital of the Rockefeller Institute to study disease in company with physicians.

In addition, he found time to work collaboratively with Dr. John P. Peters of Yale on the classic, two-volume Quantitative Clinical Chemistry. In 1922, John P. Peters, who had just gone to Yale from Van Slyke’s laboratory as an Associate Professor of Medicine, was asked by a publisher to write a modest handbook for clinicians describing useful chemical methods and discussing their application to clinical problems. It was originally to be called “Quantitative Chemistry in Clinical Medicine.” He soon found that it was going to be a bigger job than he could handle alone and asked Van Slyke to join him in writing it. Van agreed, and the two men proceeded to draw up an outline and divide up the writing of the first drafts of the chapters between them. They also agreed to exchange each chapter until it met the satisfaction of both.At the time it was published in 1931, it contained practically all that could be stated with confidence about those aspects of disease that could be and had been studied by chemical means. It was widely accepted throughout the medical world as the “Bible” of quantitative clinical chemistry, and to this day some of the chapters have not become outdated.

Clinical Chemistry in Mid 20th Century

Case in Point 2:  Outstanding Contributions in Clinical Chemistry. 1991. Arthur Karmen.

Dr. Karmen was born in New York City in 1930. He graduated from the Bronx High School of Science in 1946 and earned an A.B. and M.D. in 1950 and 1954, respectively, from New York University. In 1952, while a medical student working on a summer project at Memorial-Sloan Kettering, he used paper chromatography of amino acids to demonstrate the presence of glutamic-oxaloacetic and glutaniic-pyruvic ransaminases (aspartate and alanine aminotransferases) in serum and blood. In 1954, he devised the spectrophotometric method for measuring aspartate aminotransferase in serum, which, with minor modifications, is still used for diagnostic testing today. When developing this assay, he studied the reaction of NADH with serum and demonstrated the presence of lactate and malate dehydrogenases, both of which were also later used in diagnosis. Using the spectrophotometric method, he found that aspartate aminotransferase increased in the period immediately after an acute myocardial infarction and did the pilot studies that showed its diagnostic utility in heart and liver diseases.  This became as important as the EKG. It was replaced in cardiology usage by the MB isoenzyme of creatine kinase, which was driven by Burton Sobel’s work on infarct size, and later by the troponins.

Case in Point 3. History of Laboratory Medicine at Yale University.

The roots of the Department of Laboratory Medicine at Yale can be traced back to John Peters, the head of what he called the “Chemical Division” of the Department of Internal Medicine, subsequently known as the Section of Metabolism, who co-authored with Donald Van Slyke the landmark 1931 textbook Quantitative Clinical Chemistry (2.3); and to Pauline Hald, research collaborator of Dr. Peters who subsequently served as Director of Clinical Chemistry at Yale-New Haven Hospital for many years. In 1947, Miss Hald reported the very first flame photometric measurements of sodium and potassium in serum (4). This study helped to lay the foundation for modern studies of metabolism and their application to clinical care.

The Laboratory Medicine program at Yale had its inception in 1958 as a section of Internal Medicine under the leadership of David Seligson. In 1965, Laboratory Medicine achieved autonomous section status and in 1971, became a full-fledged academic department. Dr. Seligson, who served as the first Chair, pioneered modern automation and computerized data processing in the clinical laboratory. In particular, he demonstrated the feasibility of discrete sample handling for automation that is now the basis of virtually all automated chemistry analyzers. In addition, Seligson and Zetner demonstrated the first clinical use of atomic absorption spectrophotometry. He was one of the founding members of the major Laboratory Medicine academic society, the Academy of Clinical Laboratory Physicians and Scientists.

Davenport fig 10.jpg

Case in Point 4.  Nathan Gochman.  Developer of Automated Chemistries.

Nathan Gochman, PhD, has over 40 years of experience in the clinical diagnostics industry. This includes academic teaching and research, and 30 years in the pharmaceutical and in vitro diagnostics industry. He has managed R & D, technical marketing and technical support departments. As a leader in the industry he was President of the American Association for Clinical Chemistry (AACC) and the National Committee for Clinical Laboratory Standards (NCCLS, now CLSI). He is currently a Consultant to investment firms and IVD companies.

Nathan Gochman

Nathan Gochman

The clinical laboratory has become so productive, particularly in chemistry and immunology, and the labor, instrument and reagent costs are well determined, that today a physician’s medical decisions are 80% determined by the clinical laboratory.  Medical information systems have lagged far behind.  Why is that?  Because the decision for a MIS has historical been based on billing capture.  Moreover, the historical use of chemical profiles were quite good at validating healthy status in an outpatient population, but the profiles became restricted under Diagnostic Related Groups.    Thus, it came to be that the diagnostics was considered a “commodity”.  In order to be competitive, a laboratory had to provide “high complexity” tests that were drawn in by a large volume of “moderate complexity”tests.

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Metabolomics Summary and Perspective

Metabolomics Summary and Perspective

Author and Curator: Larry H Bernstein, MD, FCAP 

 

This is the final article in a robust series on metabolism, metabolomics, and  the “-OMICS-“ biological synthesis that is creating a more holistic and interoperable view of natural sciences, including the biological disciplines, climate science, physics, chemistry, toxicology, pharmacology, and pathophysiology with as yet unforeseen consequences.

There have been impressive advances already in the research into developmental biology, plant sciences, microbiology, mycology, and human diseases, most notably, cancer, metabolic , and infectious, as well as neurodegenerative diseases.

Acknowledgements:

I write this article in honor of my first mentor, Harry Maisel, Professor and Emeritus Chairman of Anatomy, Wayne State University, Detroit, MI and to my stimulating mentors, students, fellows, and associates over many years:

Masahiro Chiga, MD, PhD, Averill A Liebow, MD, Nathan O Kaplan, PhD, Johannes Everse, PhD, Norio Shioura, PhD, Abraham Braude, MD, Percy J Russell, PhD, Debby Peters, Walter D Foster, PhD, Herschel Sidransky, MD, Sherman Bloom, MD, Matthew Grisham, PhD, Christos Tsokos, PhD,  IJ Good, PhD, Distinguished Professor, Raool Banagale, MD, Gustavo Reynoso, MD,Gustave Davis, MD, Marguerite M Pinto, MD, Walter Pleban, MD, Marion Feietelson-Winkler, RD, PhD,  John Adan,MD, Joseph Babb, MD, Stuart Zarich, MD,  Inder Mayall, MD, A Qamar, MD, Yves Ingenbleek, MD, PhD, Emeritus Professor, Bette Seamonds, PhD, Larry Kaplan, PhD, Pauline Y Lau, PhD, Gil David, PhD, Ronald Coifman, PhD, Emeritus Professor, Linda Brugler, RD, MBA, James Rucinski, MD, Gitta Pancer, Ester Engelman, Farhana Hoque, Mohammed Alam, Michael Zions, William Fleischman, MD, Salman Haq, MD, Jerard Kneifati-Hayek, Madeleine Schleffer, John F Heitner, MD, Arun Devakonda,MD, Liziamma George,MD, Suhail Raoof, MD, Charles Oribabor,MD, Anthony Tortolani, MD, Prof and Chairman, JRDS Rosalino, PhD, Aviva Lev Ari, PhD, RN, Rosser Rudolph, MD, PhD, Eugene Rypka, PhD, Jay Magidson, PhD, Izaak Mayzlin, PhD, Maurice Bernstein, PhD, Richard Bing, Eli Kaplan, PhD, Maurice Bernstein, PhD.

This article has EIGHT parts, as follows:

Part 1

Metabolomics Continues Auspicious Climb

Part 2

Biologists Find ‘Missing Link’ in the Production of Protein Factories in Cells

Part 3

Neuroscience

Part 4

Cancer Research

Part 5

Metabolic Syndrome

Part 6

Biomarkers

Part 7

Epigenetics and Drug Metabolism

Part 8

Pictorial

genome cartoon

genome cartoon

 iron metabolism

iron metabolism

personalized reference range within population range

personalized reference range within population range

Part 1.  MetabolomicsSurge

metagraph  _OMICS

metagraph _OMICS

Metabolomics Continues Auspicious Climb

Jeffery Herman, Ph.D.
GEN May 1, 2012 (Vol. 32, No. 9)

Aberrant biochemical and metabolite signaling plays an important role in

  • the development and progression of diseased tissue.

This concept has been studied by the science community for decades. However, with relatively

  1. recent advances in analytical technology and bioinformatics as well as
  2. the development of the Human Metabolome Database (HMDB),

metabolomics has become an invaluable field of research.

At the “International Conference and Exhibition on Metabolomics & Systems Biology” held recently in San Francisco, researchers and industry leaders discussed how

  • the underlying cellular biochemical/metabolite fingerprint in response to
  1. a specific disease state,
  2. toxin exposure, or
  3. pharmaceutical compound
  • is useful in clinical diagnosis and biomarker discovery and
  • in understanding disease development and progression.

Developed by BASF, MetaMap® Tox is

  • a database that helps identify in vivo systemic effects of a tested compound, including
  1. targeted organs,
  2. mechanism of action, and
  3. adverse events.

Based on 28-day systemic rat toxicity studies, MetaMap Tox is composed of

  • differential plasma metabolite profiles of rats
  • after exposure to a large variety of chemical toxins and pharmaceutical compounds.

“Using the reference data,

  • we have developed more than 110 patterns of metabolite changes, which are
  • specific and predictive for certain toxicological modes of action,”

said Hennicke Kamp, Ph.D., group leader, department of experimental toxicology and ecology at BASF.

With MetaMap Tox, a potential drug candidate

  • can be compared to a similar reference compound
  • using statistical correlation algorithms,
  • which allow for the creation of a toxicity and mechanism of action profile.

“MetaMap Tox, in the context of early pre-clinical safety enablement in pharmaceutical development,” continued Dr. Kamp,

  • has been independently validated “
  • by an industry consortium (Drug Safety Executive Council) of 12 leading biopharmaceutical companies.”

Dr. Kamp added that this technology may prove invaluable

  • allowing for quick and accurate decisions and
  • for high-throughput drug candidate screening, in evaluation
  1. on the safety and efficacy of compounds
  2. during early and preclinical toxicological studies,
  3. by comparing a lead compound to a variety of molecular derivatives, and
  • the rapid identification of the most optimal molecular structure
  • with the best efficacy and safety profiles might be streamlined.
Dynamic Construct of the –Omics

Dynamic Construct of the –Omics

Targeted Tandem Mass Spectrometry

Biocrates Life Sciences focuses on targeted metabolomics, an important approach for

  • the accurate quantification of known metabolites within a biological sample.

Originally used for the clinical screening of inherent metabolic disorders from dried blood-spots of newborn children, Biocrates has developed

  • a tandem mass spectrometry (MS/MS) platform, which allows for
  1. the identification,
  2. quantification, and
  3. mapping of more than 800 metabolites to specific cellular pathways.

It is based on flow injection analysis and high-performance liquid chromatography MS/MS.

Clarification of Pathway-Specific Inhibition by Fourier Transform Ion Cyclotron Resonance.Mass Spectrometry-Based Metabolic Phenotyping Studies F5.large

common drug targets

common drug targets

The MetaDisIDQ® Kit is a

  • “multiparamatic” diagnostic assay designed for the “comprehensive assessment of a person’s metabolic state” and
  • the early determination of pathophysiological events with regards to a specific disease.

MetaDisIDQ is designed to quantify

  • a diverse range of 181 metabolites involved in major metabolic pathways
  • from a small amount of human serum (10 µL) using isotopically labeled internal standards,

This kit has been demonstrated to detect changes in metabolites that are commonly associated with the development of

  • metabolic syndrome, type 2 diabetes, and diabetic nephropathy,

Dr. Dallman reports that data generated with the MetaDisIDQ kit correlates strongly with

  • routine chemical analyses of common metabolites including glucose and creatinine

Biocrates has also developed the MS/MS-based AbsoluteIDQ® kits, which are

  • an “easy-to-use” biomarker analysis tool for laboratory research.

The kit functions on MS machines from a variety of vendors, and allows for the quantification of 150-180 metabolites.

The SteroIDQ® kit is a high-throughput standardized MS/MS diagnostic assay,

  • validated in human serum, for the rapid and accurate clinical determination of 16 known steroids.

Initially focusing on the analysis of steroid ranges for use in hormone replacement therapy, the SteroIDQ Kit is expected to have a wide clinical application.

Hormone-Resistant Breast Cancer

Scientists at Georgetown University have shown that

  • breast cancer cells can functionally coordinate cell-survival and cell-proliferation mechanisms,
  • while maintaining a certain degree of cellular metabolism.

To grow, cells need energy, and energy is a product of cellular metabolism. For nearly a century, it was thought that

  1. the uncoupling of glycolysis from the mitochondria,
  2. leading to the inefficient but rapid metabolism of glucose and
  3. the formation of lactic acid (the Warburg effect), was

the major and only metabolism driving force for unchecked proliferation and tumorigenesis of cancer cells.

Other aspects of metabolism were often overlooked.

“.. we understand now that

  • cellular metabolism is a lot more than just metabolizing glucose,”

said Robert Clarke, Ph.D., professor of oncology and physiology and biophysics at Georgetown University. Dr. Clarke, in collaboration with the Waters Center for Innovation at Georgetown University (led by Albert J. Fornace, Jr., M.D.), obtained

  • the metabolomic profile of hormone-sensitive and -resistant breast cancer cells through the use of UPLC-MS.

They demonstrated that breast cancer cells, through a rather complex and not yet completely understood process,

  1. can functionally coordinate cell-survival and cell-proliferation mechanisms,
  2. while maintaining a certain degree of cellular metabolism.

This is at least partly accomplished through the upregulation of important pro-survival mechanisms; including

  • the unfolded protein response;
  • a regulator of endoplasmic reticulum stress and
  • initiator of autophagy.

Normally, during a stressful situation, a cell may

  • enter a state of quiescence and undergo autophagy,
  • a process by which a cell can recycle organelles
  • in order to maintain enough energy to survive during a stressful situation or,

if the stress is too great,

  • undergo apoptosis.

By integrating cell-survival mechanisms and cellular metabolism

  • advanced ER+ hormone-resistant breast cancer cells
  • can maintain a low level of autophagy
  • to adapt and resist hormone/chemotherapy treatment.

This adaptation allows cells

  • to reallocate important metabolites recovered from organelle degradation and
  • provide enough energy to also promote proliferation.

With further research, we can gain a better understanding of the underlying causes of hormone-resistant breast cancer, with

  • the overall goal of developing effective diagnostic, prognostic, and therapeutic tools.

NMR

Over the last two decades, NMR has established itself as a major tool for metabolomics analysis. It is especially adept at testing biological fluids. [Bruker BioSpin]

Historically, nuclear magnetic resonance spectroscopy (NMR) has been used for structural elucidation of pure molecular compounds. However, in the last two decades, NMR has established itself as a major tool for metabolomics analysis. Since

  • the integral of an NMR signal is directly proportional to
  • the molar concentration throughout the dynamic range of a sample,

“the simultaneous quantification of compounds is possible

  • without the need for specific reference standards or calibration curves,” according to Lea Heintz of Bruker BioSpin.

NMR is adept at testing biological fluids because of

  1.  high reproducibility,
  2. standardized protocols,
  3. low sample manipulation, and
  4. the production of a large subset of data,

Bruker BioSpin is presently involved in a project for the screening of inborn errors of metabolism in newborn children from Turkey, based on their urine NMR profiles. More than 20 clinics are participating to the project that is coordinated by INFAI, a specialist in the transfer of advanced analytical technology into medical diagnostics. The construction of statistical models are being developed

  • for the detection of deviations from normality, as well as
  • automatic quantification methods for indicative metabolites

Bruker BioSpin recently installed high-resolution magic angle spinning NMR (HRMAS-NMR) systems that can rapidly analyze tissue biopsies. The main objective for HRMAS-NMR is to establish a rapid and effective clinical method to assess tumor grade and other important aspects of cancer during surgery.

Combined NMR and Mass Spec

There is increasing interest in combining NMR and MS, two of the main analytical assays in metabolomic research, as a means

  • to improve data sensitivity and to
  • fully elucidate the complex metabolome within a given biological sample.
  •  to realize a potential for cancer biomarker discovery in the realms of diagnosis, prognosis, and treatment.

.

Using combined NMR and MS to measure the levels of nearly 250 separate metabolites in the patient’s blood, Dr. Weljie and other researchers at the University of Calgary were able to rapidly determine the malignancy of a  pancreatic lesion (in 10–15% of the cases, it is difficult to discern between benign and malignant), while avoiding unnecessary surgery in patients with benign lesions.

When performing NMR and MS on a single biological fluid, ultimately “we are,” noted Dr. Weljie,

  1. “splitting up information content, processing, and introducing a lot of background noise and error and
  2. then trying to reintegrate the data…
    It’s like taking a complex item, with multiple pieces, out of an IKEA box and trying to repackage it perfectly into another box.”

By improving the workflow between the initial splitting of the sample, they improved endpoint data integration, proving that

  • a streamlined approach to combined NMR/MS can be achieved,
  • leading to a very strong, robust and precise metabolomics toolset.

Metabolomics Research Picks Up Speed

Field Advances in Quest to Improve Disease Diagnosis and Predict Drug Response

John Morrow Jr., Ph.D.
GEN May 1, 2011 (Vol. 31, No. 9)

As an important discipline within systems biology, metabolomics is being explored by a number of laboratories for

  • its potential in pharmaceutical development.

Studying metabolites can offer insights into the relationships between genotype and phenotype, as well as between genotype and environment. In addition, there is plenty to work with—there are estimated to be some 2,900 detectable metabolites in the human body, of which

  1. 309 have been identified in cerebrospinal fluid,
  2. 1,122 in serum,
  3. 458 in urine, and
  4. roughly 300 in other compartments.

Guowang Xu, Ph.D., a researcher at the Dalian Institute of Chemical Physics.  is investigating the causes of death in China,

  • and how they have been changing over the years as the country has become a more industrialized nation.
  •  the increase in the incidence of metabolic disorders such as diabetes has grown to affect 9.7% of the Chinese population.

Dr. Xu,  collaborating with Rainer Lehman, Ph.D., of the University of Tübingen, Germany, compared urinary metabolites in samples from healthy individuals with samples taken from prediabetic, insulin-resistant subjects. Using mass spectrometry coupled with electrospray ionization in the positive mode, they observed striking dissimilarities in levels of various metabolites in the two groups.

“When we performed a comprehensive two-dimensional gas chromatography, time-of-flight mass spectrometry analysis of our samples, we observed several metabolites, including

  • 2-hydroxybutyric acid in plasma,
  •  as potential diabetes biomarkers,” Dr. Xu explains.

In other, unrelated studies, Dr. Xu and the German researchers used a metabolomics approach to investigate the changes in plasma metabolite profiles immediately after exercise and following a 3-hour and 24-hour period of recovery. They found that

  • medium-chain acylcarnitines were the most distinctive exercise biomarkers, and
  • they are released as intermediates of partial beta oxidation in human myotubes and mouse muscle tissue.

Dr. Xu says. “The traditional approach of assessment based on a singular biomarker is being superseded by the introduction of multiple marker profiles.”

Typical of the studies under way by Dr. Kaddurah-Daouk and her colleaguesat Duke University

  • is a recently published investigation highlighting the role of an SNP variant in
  • the glycine dehydrogenase gene on individual response to antidepressants.
  •  patients who do not respond to the selective serotonin uptake inhibitors citalopram and escitalopram
  • carried a particular single nucleotide polymorphism in the GD gene.

“These results allow us to pinpoint a possible

  • role for glycine in selective serotonin reuptake inhibitor response and
  • illustrate the use of pharmacometabolomics to inform pharmacogenomics.

These discoveries give us the tools for prognostics and diagnostics so that

  • we can predict what conditions will respond to treatment.

“This approach to defining health or disease in terms of metabolic states opens a whole new paradigm.

By screening hundreds of thousands of molecules, we can understand

  • the relationship between human genetic variability and the metabolome.”

Dr. Kaddurah-Daouk talks about statins as a current

  • model of metabolomics investigations.

It is now known that the statins  have widespread effects, altering a range of metabolites. To sort out these changes and develop recommendations for which individuals should be receiving statins will require substantial investments of energy and resources into defining the complex web of biochemical changes that these drugs initiate.
Furthermore, Dr. Kaddurah-Daouk asserts that,

  • “genetics only encodes part of the phenotypic response.

One needs to take into account the

  • net environment contribution in order to determine
  • how both factors guide the changes in our metabolic state that determine the phenotype.”

Interactive Metabolomics

Researchers at the University of Nottingham use diffusion-edited nuclear magnetic resonance spectroscopy to assess the effects of a biological matrix on metabolites. Diffusion-edited NMR experiments provide a way to

  • separate the different compounds in a mixture
  • based on the differing translational diffusion coefficients (which reflect the size and shape of the molecule).

The measurements are carried out by observing

  • the attenuation of the NMR signals during a pulsed field gradient experiment.

Clare Daykin, Ph.D., is a lecturer at the University of Nottingham, U.K. Her field of investigation encompasses “interactive metabolomics,”which she defines as

“the study of the interactions between low molecular weight biochemicals and macromolecules in biological samples ..

  • without preselection of the components of interest.

“Blood plasma is a heterogeneous mixture of molecules that

  1. undergo a variety of interactions including metal complexation,
  2. chemical exchange processes,
  3. micellar compartmentation,
  4. enzyme-mediated biotransformations, and
  5. small molecule–macromolecular binding.”

Many low molecular weight compounds can exist

  • freely in solution,
  • bound to proteins, or
  • within organized aggregates such as lipoprotein complexes.

Therefore, quantitative comparison of plasma composition from

  • diseased individuals compared to matched controls provides an incomplete insight to plasma metabolism.

“It is not simply the concentrations of metabolites that must be investigated,

  • but their interactions with the proteins and lipoproteins within this complex web.

Rather than targeting specific metabolites of interest, Dr. Daykin’s metabolite–protein binding studies aim to study

  • the interactions of all detectable metabolites within the macromolecular sample.

Such activities can be studied through the use of diffusion-edited nuclear magnetic resonance (NMR) spectroscopy, in which one can assess

  • the effects of the biological matrix on the metabolites.

“This can lead to a more relevant and exact interpretation

  • for systems where metabolite–macromolecule interactions occur.”

Diffusion-edited NMR experiments provide a way to separate the different compounds in a mixture based on

  • the differing translational diffusion coefficients (which reflect the size and shape of the molecule).

The measurements are carried out by observing

  • the attenuation of the NMR signals during a pulsed field gradient experiment.

Pushing the Limits

It is widely recognized that many drug candidates fail during development due to ancillary toxicity. Uwe Sauer, Ph.D., professor, and Nicola Zamboni, Ph.D., researcher, both at the Eidgenössische Technische Hochschule, Zürich (ETH Zürich), are applying

  • high-throughput intracellular metabolomics to understand
  • the basis of these unfortunate events and
  • head them off early in the course of drug discovery.

“Since metabolism is at the core of drug toxicity, we developed a platform for

  • measurement of 50–100 targeted metabolites by
  • a high-throughput system consisting of flow injection
  • coupled to tandem mass spectrometry.”

Using this approach, Dr. Sauer’s team focused on

  • the central metabolism of the yeast Saccharomyces cerevisiae, reasoning that
  • this core network would be most susceptible to potential drug toxicity.

Screening approximately 41 drugs that were administered at seven concentrations over three orders of magnitude, they observed changes in metabolome patterns at much lower drug concentrations without attendant physiological toxicity.

The group carried out statistical modeling of about

  • 60 metabolite profiles for each drug they evaluated.

This data allowed the construction of a “profile effect map” in which

  • the influence of each drug on metabolite levels can be followed, including off-target effects, which
  • provide an indirect measure of the possible side effects of the various drugs.

Dr. Sauer says.“We have found that this approach is

  • at least 100 times as fast as other omics screening platforms,”

“Some drugs, including many anticancer agents,

  • disrupt metabolism long before affecting growth.”
killing cancer cells

killing cancer cells

Furthermore, they used the principle of 13C-based flux analysis, in which

  • metabolites labeled with 13C are used to follow the utilization of metabolic pathways in the cell.

These 13C-determined intracellular responses of metabolic fluxes to drug treatment demonstrate

  • the functional performance of the network to be rather robust,
conformational changes leading to substrate efflux.

conformational changes leading to substrate efflux.

leading Dr. Sauer to the conclusion that

  • the phenotypic vigor he observes to drug challenges
  • is achieved by a flexible make up of the metabolome.

Dr. Sauer is confident that it will be possible to expand the scope of these investigations to hundreds of thousands of samples per study. This will allow answers to the questions of

  • how cells establish a stable functioning network in the face of inevitable concentration fluctuations.

Is Now the Hour?

There is great enthusiasm and agitation within the biotech community for

  • metabolomics approaches as a means of reversing the dismal record of drug discovery

that has accumulated in the last decade.

While the concept clearly makes sense and is being widely applied today, there are many reasons why drugs fail in development, and metabolomics will not be a panacea for resolving all of these questions. It is too early at this point to recognize a trend or a track record, and it will take some time to see how this approach can aid in drug discovery and shorten the timeline for the introduction of new pharmaceutical agents.

Degree of binding correlated with function

Degree of binding correlated with function

Diagram_of_a_two-photon_excitation_microscope_

Diagram_of_a_two-photon_excitation_microscope_

Part 2.  Biologists Find ‘Missing Link’ in the Production of Protein Factories in Cells

Biologists at UC San Diego have found

  • the “missing link” in the chemical system that
  • enables animal cells to produce ribosomes

—the thousands of protein “factories” contained within each cell that

  • manufacture all of the proteins needed to build tissue and sustain life.
‘Missing Link’

‘Missing Link’

Their discovery, detailed in the June 23 issue of the journal Genes & Development, will not only force

  • a revision of basic textbooks on molecular biology, but also
  • provide scientists with a better understanding of
  • how to limit uncontrolled cell growth, such as cancer,
  • that might be regulated by controlling the output of ribosomes.

Ribosomes are responsible for the production of the wide variety of proteins that include

  1. enzymes;
  2. structural molecules, such as hair,
  3. skin and bones;
  4. hormones like insulin; and
  5. components of our immune system such as antibodies.

Regarded as life’s most important molecular machine, ribosomes have been intensively studied by scientists (the 2009 Nobel Prize in Chemistry, for example, was awarded for studies of its structure and function). But until now researchers had not uncovered all of the details of how the proteins that are used to construct ribosomes are themselves produced.

In multicellular animals such as humans,

  • ribosomes are made up of about 80 different proteins
    (humans have 79 while some other animals have a slightly different number) as well as
  • four different kinds of RNA molecules.

In 1969, scientists discovered that

  • the synthesis of the ribosomal RNAs is carried out by specialized systems using two key enzymes:
  • RNA polymerase I and RNA polymerase III.

But until now, scientists were unsure if a complementary system was also responsible for

  • the production of the 80 proteins that make up the ribosome.

That’s essentially what the UC San Diego researchers headed by Jim Kadonaga, a professor of biology, set out to examine. What they found was the missing link—the specialized

  • system that allows ribosomal proteins themselves to be synthesized by the cell.

Kadonaga says that he and coworkers found that ribosomal proteins are synthesized via

  • a novel regulatory system with the enzyme RNA polymerase II and
  • a factor termed TRF2,”

“For the production of most proteins,

  1. RNA polymerase II functions with
  2. a factor termed TBP,
  3. but for the synthesis of ribosomal proteins, it uses TRF2.”
  •  this specialized TRF2-based system for ribosome biogenesis
  • provides a new avenue for the study of ribosomes and
  • its control of cell growth, and

“it should lead to a better understanding and potential treatment of diseases such as cancer.”

Coordination of the transcriptome and metabolome

Coordination of the transcriptome and metabolome

the potential advantages conferred by distal-site protein synthesis

the potential advantages conferred by distal-site protein synthesis

Other authors of the paper were UC San Diego biologists Yuan-Liang Wang, Sascha Duttke and George Kassavetis, and Kai Chen, Jeff Johnston, and Julia Zeitlinger of the Stowers Institute for Medical Research in Kansas City, Missouri. Their research was supported by two grants from the National Institutes of Health (1DP2OD004561-01 and R01 GM041249).

Turning Off a Powerful Cancer Protein

Scientists have discovered how to shut down a master regulatory transcription factor that is

  • key to the survival of a majority of aggressive lymphomas,
  • which arise from the B cells of the immune system.

The protein, Bcl6, has long been considered too complex to target with a drug since it is also crucial

  • to the healthy functioning of many immune cells in the body, not just B cells gone bad.

The researchers at Weill Cornell Medical College report that it is possible

  • to shut down Bcl6 in diffuse large B-cell lymphoma (DLBCL)
  • while not affecting its vital function in T cells and macrophages
  • that are needed to support a healthy immune system.

If Bcl6 is completely inhibited, patients might suffer from systemic inflammation and atherosclerosis. The team conducted this new study to help clarify possible risks, as well as to understand

  • how Bcl6 controls the various aspects of the immune system.

The findings in this study were inspired from

  • preclinical testing of two Bcl6-targeting agents that Dr. Melnick and his Weill Cornell colleagues have developed
  • to treat DLBCLs.

These experimental drugs are

  • RI-BPI, a peptide mimic, and
  • the small molecule agent 79-6.

“This means the drugs we have developed against Bcl6 are more likely to be

  • significantly less toxic and safer for patients with this cancer than we realized,”

says Ari Melnick, M.D., professor of hematology/oncology and a hematologist-oncologist at NewYork-Presbyterian Hospital/Weill Cornell Medical Center.

Dr. Melnick says the discovery that

  • a master regulatory transcription factor can be targeted
  • offers implications beyond just treating DLBCL.

Recent studies from Dr. Melnick and others have revealed that

  • Bcl6 plays a key role in the most aggressive forms of acute leukemia, as well as certain solid tumors.

Bcl6 can control the type of immune cell that develops in the bone marrow—playing many roles

  • in the development of B cells, T cells, macrophages, and other cells—including a primary and essential role in
  • enabling B-cells to generate specific antibodies against pathogens.

According to Dr. Melnick, “When cells lose control of Bcl6,

  • lymphomas develop in the immune system.

Lymphomas are ‘addicted’ to Bcl6, and therefore

  • Bcl6 inhibitors powerfully and quickly destroy lymphoma cells,” .

The big surprise in the current study is that rather than functioning as a single molecular machine,

  • Bcl6 functions like a Swiss Army knife,
  • using different tools to control different cell types.

This multifunction paradigm could represent a general model for the functioning of other master regulatory transcription factors.

“In this analogy, the Swiss Army knife, or transcription factor, keeps most of its tools folded,

  • opening only the one it needs in any given cell type,”

He makes the following analogy:

  • “For B cells, it might open and use the knife tool;
  • for T cells, the cork screw;
  • for macrophages, the scissors.”

“this means that you only need to prevent the master regulator from using certain tools to treat cancer. You don’t need to eliminate the whole knife,” . “In fact, we show that taking out the whole knife is harmful since

  • the transcription factor has many other vital functions that other cells in the body need.”

Prior to these study results, it was not known that a master regulator could separate its functions so precisely. Researchers hope this will be a major benefit to the treatment of DLBCL and perhaps other disorders that are influenced by Bcl6 and other master regulatory transcription factors.

The study is published in the journal Nature Immunology, in a paper titled “Lineage-specific functions of Bcl-6 in immunity and inflammation are mediated by distinct biochemical mechanisms”.

Part 3. Neuroscience

Vesicles influence function of nerve cells 
Oct, 06 2014        source: http://feeds.sciencedaily.com

Neurons (blue) which have absorbed exosomes (green) have increased levels of the enzyme catalase (red), which helps protect them against peroxides.

Neurons (blue) which have absorbed exosomes (green) have increased levels of the enzyme catalase (red), which helps protect them against peroxides.

Neurons (blue) which have absorbed exosomes (green) have increased levels of the enzyme catalase (red), which helps protect them against peroxides.

Tiny vesicles containing protective substances

  • which they transmit to nerve cells apparently
  • play an important role in the functioning of neurons.

As cell biologists at Johannes Gutenberg University Mainz (JGU) have discovered,

  • nerve cells can enlist the aid of mini-vesicles of neighboring glial cells
  • to defend themselves against stress and other potentially detrimental factors.

These vesicles, called exosomes, appear to stimulate the neurons on various levels:

  • they influence electrical stimulus conduction,
  • biochemical signal transfer, and
  • gene regulation.

Exosomes are thus multifunctional signal emitters

  • that can have a significant effect in the brain.
Exosome

Exosome

The researchers in Mainz already observed in a previous study that

  • oligodendrocytes release exosomes on exposure to neuronal stimuli.
  • these are absorbed by the neurons and improve neuronal stress tolerance.

Oligodendrocytes, a type of glial cell, form an

  • insulating myelin sheath around the axons of neurons.

The exosomes transport protective proteins such as

  • heat shock proteins,
  • glycolytic enzymes, and
  • enzymes that reduce oxidative stress from one cell type to another,
  • but also transmit genetic information in the form of ribonucleic acids.

“As we have now discovered in cell cultures, exosomes seem to have a whole range of functions,” explained Dr. Eva-Maria Krmer-Albers. By means of their transmission activity, the small bubbles that are the vesicles

  • not only promote electrical activity in the nerve cells, but also
  • influence them on the biochemical and gene regulatory level.

“The extent of activities of the exosomes is impressive,” added Krmer-Albers. The researchers hope that the understanding of these processes will contribute to the development of new strategies for the treatment of neuronal diseases. Their next aim is to uncover how vesicles actually function in the brains of living organisms.

http://labroots.com/user/news/article/id/217438/title/vesicles-influence-function-of-nerve-cells

The above story is based on materials provided by Universitt Mainz.

Universitt Mainz. “Vesicles influence function of nerve cells.” ScienceDaily. ScienceDaily, 6 October 2014. www.sciencedaily.com/releases/2014/10/141006174214.htm

Neuroscientists use snail research to help explain “chemo brain”

10/08/2014
It is estimated that as many as half of patients taking cancer drugs experience a decrease in mental sharpness. While there have been many theories, what causes “chemo brain” has eluded scientists.

In an effort to solve this mystery, neuroscientists at The University of Texas Health Science Center at Houston (UTHealth) conducted an experiment in an animal memory model and their results point to a possible explanation. Findings appeared in The Journal of Neuroscience.

In the study involving a sea snail that shares many of the same memory mechanisms as humans and a drug used to treat a variety of cancers, the scientists identified

  • memory mechanisms blocked by the drug.

Then, they were able to counteract or

  • unblock the mechanisms by administering another agent.

“Our research has implications in the care of people given to cognitive deficits following drug treatment for cancer,” said John H. “Jack” Byrne, Ph.D., senior author, holder of the June and Virgil Waggoner Chair and Chairman of the Department of Neurobiology and Anatomy at the UTHealth Medical School. “There is no satisfactory treatment at this time.”

Byrne’s laboratory is known for its use of a large snail called Aplysia californica to further the understanding of the biochemical signaling among nerve cells (neurons).  The snails have large neurons that relay information much like those in humans.

When Byrne’s team compared cell cultures taken from normal snails to

  • those administered a dose of a cancer drug called doxorubicin,

the investigators pinpointed a neuronal pathway

  • that was no longer passing along information properly.

With the aid of an experimental drug,

  • the scientists were able to reopen the pathway.

Unfortunately, this drug would not be appropriate for humans, Byrne said. “We want to identify other drugs that can rescue these memory mechanisms,” he added.

According the American Cancer Society, some of the distressing mental changes cancer patients experience may last a short time or go on for years.

Byrne’s UT Health research team includes co-lead authors Rong-Yu Liu, Ph.D., and Yili Zhang, Ph.D., as well as Brittany Coughlin and Leonard J. Cleary, Ph.D. All are affiliated with the W.M. Keck Center for the Neurobiology of Learning and Memory.

Byrne and Cleary also are on the faculty of The University of Texas Graduate School of Biomedical Sciences at Houston. Coughlin is a student at the school, which is jointly operated by UT Health and The University of Texas MD Anderson Cancer Center.

The study titled “Doxorubicin Attenuates Serotonin-Induced Long-Term Synaptic Facilitation by Phosphorylation of p38 Mitogen-Activated Protein Kinase” received support from National Institutes of Health grant (NS019895) and the Zilkha Family Discovery Fellowship.

Doxorubicin Attenuates Serotonin-Induced Long-Term Synaptic Facilitation by Phosphorylation of p38 Mitogen-Activated Protein Kinase

Source: Univ. of Texas Health Science Center at Houston

http://www.rdmag.com/news/2014/10/neuroscientists-use-snail-research-help-explain-E2_9_Cchemo-brain

Doxorubicin Attenuates Serotonin-Induced Long-Term Synaptic Facilitation by Phosphorylation of p38 Mitogen-Activated Protein Kinase

Rong-Yu Liu*,  Yili Zhang*,  Brittany L. Coughlin,  Leonard J. Cleary, and  John H. Byrne   +Show Affiliations
The Journal of Neuroscience, 1 Oct 2014, 34(40): 13289-13300;
http://dx.doi.org:/10.1523/JNEUROSCI.0538-14.2014

Doxorubicin (DOX) is an anthracycline used widely for cancer chemotherapy. Its primary mode of action appears to be

  • topoisomerase II inhibition, DNA cleavage, and free radical generation.

However, in non-neuronal cells, DOX also inhibits the expression of

  • dual-specificity phosphatases (also referred to as MAPK phosphatases) and thereby
  1. inhibits the dephosphorylation of extracellular signal-regulated kinase (ERK) and
  2. p38 mitogen-activated protein kinase (p38 MAPK),
  3. two MAPK isoforms important for long-term memory (LTM) formation.

Activation of these kinases by DOX in neurons, if present,

  • could have secondary effects on cognitive functions, such as learning and memory.

The present study used cultures of rat cortical neurons and sensory neurons (SNs) of Aplysia

  • to examine the effects of DOX on levels of phosphorylated ERK (pERK) and
  • phosphorylated p38 (p-p38) MAPK.

In addition, Aplysia neurons were used to examine the effects of DOX on

  • long-term enhanced excitability, long-term synaptic facilitation (LTF), and
  • long-term synaptic depression (LTD).

DOX treatment led to elevated levels of

  • pERK and p-p38 MAPK in SNs and cortical neurons.

In addition, it increased phosphorylation of

  • the downstream transcriptional repressor cAMP response element-binding protein 2 in SNs.

DOX treatment blocked serotonin-induced LTF and enhanced LTD induced by the neuropeptide Phe-Met-Arg-Phe-NH2. The block of LTF appeared to be attributable to

  • overriding inhibitory effects of p-p38 MAPK, because
  • LTF was rescued in the presence of an inhibitor of p38 MAPK
    (SB203580 [4-(4-fluorophenyl)-2-(4-methylsulfinylphenyl)-5-(4-pyridyl)-1H-imidazole]) .

These results suggest that acute application of DOX might impair the formation of LTM via the p38 MAPK pathway.
Terms: Aplysia chemotherapy ERK  p38 MAPK serotonin synaptic plasticity

Technology that controls brain cells with radio waves earns early BRAIN grant

10/08/2014

bright spots = cells with increased calcium after treatment with radio waves,  allows neurons to fire

bright spots = cells with increased calcium after treatment with radio waves, allows neurons to fire

BRAIN control: The new technology uses radio waves to activate or silence cells remotely. The bright spots above represent cells with increased calcium after treatment with radio waves, a change that would allow neurons to fire.

A proposal to develop a new way to

  • remotely control brain cells

from Sarah Stanley, a research associate in Rockefeller University’s Laboratory of Molecular Genetics, headed by Jeffrey M. Friedman, is

  • among the first to receive funding from U.S. President Barack Obama’s BRAIN initiative.

The project will make use of a technique called

  • radiogenetics that combines the use of radio waves or magnetic fields with
  • nanoparticles to turn neurons on or off.

The National Institutes of Health is one of four federal agencies involved in the BRAIN (Brain Research through Advancing Innovative Neurotechnologies) initiative. Following in the ambitious footsteps of the Human Genome Project, the BRAIN initiative seeks

  • to create a dynamic map of the brain in action,

a goal that requires the development of new technologies. The BRAIN initiative working group, which outlined the broad scope of the ambitious project, was co-chaired by Rockefeller’s Cori Bargmann, head of the Laboratory of Neural Circuits and Behavior.

Stanley’s grant, for $1.26 million over three years, is one of 58 projects to get BRAIN grants, the NIH announced. The NIH’s plan for its part of this national project, which has been pitched as “America’s next moonshot,” calls for $4.5 billion in federal funds over 12 years.

The technology Stanley is developing would

  • enable researchers to manipulate the activity of neurons, as well as other cell types,
  • in freely moving animals in order to better understand what these cells do.

Other techniques for controlling selected groups of neurons exist, but her new nanoparticle-based technique has a

  • unique combination of features that may enable new types of experimentation.
  • it would allow researchers to rapidly activate or silence neurons within a small area of the brain or
  • dispersed across a larger region, including those in difficult-to-access locations.

Stanley also plans to explore the potential this method has for use treating patients.

“Francis Collins, director of the NIH, has discussed

  • the need for studying the circuitry of the brain,
  • which is formed by interconnected neurons.

Our remote-control technology may provide a tool with which researchers can ask new questions about the roles of complex circuits in regulating behavior,” Stanley says.
Rockefeller University’s Laboratory of Molecular Genetics
Source: Rockefeller Univ.

Part 4.  Cancer

Two Proteins Found to Block Cancer Metastasis

Why do some cancers spread while others don’t? Scientists have now demonstrated that

  • metastatic incompetent cancers actually “poison the soil”
  • by generating a micro-environment that blocks cancer cells
  • from settling and growing in distant organs.

The “seed and the soil” hypothesis proposed by Stephen Paget in 1889 is now widely accepted to explain how

  • cancer cells (seeds) are able to generate fertile soil (the micro-environment)
  • in distant organs that promotes cancer’s spread.

However, this concept had not explained why some tumors do not spread or metastasize.

The researchers, from Weill Cornell Medical College, found that

  • two key proteins involved in this process work by
  • dramatically suppressing cancer’s spread.

The study offers hope that a drug based on these

  • potentially therapeutic proteins, prosaposin and Thrombospondin 1 (Tsp-1),

might help keep human cancer at bay and from metastasizing.

Scientists don’t understand why some tumors wouldn’t “want” to spread. It goes against their “job description,” says the study’s senior investigator, Vivek Mittal, Ph.D., an associate professor of cell and developmental biology in cardiothoracic surgery and director of the Neuberger Berman Foundation Lung Cancer Laboratory at Weill Cornell Medical College. He theorizes that metastasis occurs when

  • the barriers that the body throws up to protect itself against cancer fail.

But there are some tumors in which some of the barriers may still be intact. “So that suggests

  • those primary tumors will continue to grow, but that
  • an innate protective barrier still exists that prevents them from spreading and invading other organs,”

The researchers found that, like typical tumors,

  • metastasis-incompetent tumors also send out signaling molecules
  • that establish what is known as the “premetastatic niche” in distant organs.

These niches composed of bone marrow cells and various growth factors have been described previously by others including Dr. Mittal as the fertile “soil” that the disseminated cancer cell “seeds” grow in.

Weill Cornell’s Raúl Catena, Ph.D., a postdoctoral fellow in Dr. Mittal’s laboratory, found an important difference between the tumor types. Metastatic-incompetent tumors

  • systemically increased expression of Tsp-1, a molecule known to fight cancer growth.
  • increased Tsp-1 production was found specifically in the bone marrow myeloid cells
  • that comprise the metastatic niche.

These results were striking, because for the first time Dr. Mittal says

  • the bone marrow-derived myeloid cells were implicated as
  • the main producers of Tsp-1,.

In addition, Weill Cornell and Harvard researchers found that

  • prosaposin secreted predominantly by the metastatic-incompetent tumors
  • increased expression of Tsp-1 in the premetastatic lungs.

Thus, Dr. Mittal posits that prosaposin works in combination with Tsp-1

  • to convert pro-metastatic bone marrow myeloid cells in the niche
  • into cells that are not hospitable to cancer cells that spread from a primary tumor.
  • “The very same myeloid cells in the niche that we know can promote metastasis
  • can also be induced under the command of the metastatic incompetent primary tumor to inhibit metastasis,”

The research team found that

  • the Tsp-1–inducing activity of prosaposin
  • was contained in only a 5-amino acid peptide region of the protein, and
  • this peptide alone induced Tsp-1 in the bone marrow cells and
  • effectively suppressed metastatic spread in the lungs
  • in mouse models of breast and prostate cancer.

This 5-amino acid peptide with Tsp-1–inducing activity

  • has the potential to be used as a therapeutic agent against metastatic cancer,

The scientists have begun to test prosaposin in other tumor types or metastatic sites.

Dr. Mittal says that “The clinical implications of the study are:

  • “Not only is it theoretically possible to design a prosaposin-based drug or drugs
  • that induce Tsp-1 to block cancer spread, but
  • you could potentially create noninvasive prognostic tests
  • to predict whether a cancer will metastasize.”

The study was reported in the April 30 issue of Cancer Discovery, in a paper titled “Bone Marrow-Derived Gr1+ Cells Can Generate a Metastasis-Resistant Microenvironment Via Induced Secretion of Thrombospondin-1”.

Disabling Enzyme Cripples Tumors, Cancer Cells

First Step of Metastasis

First Step of Metastasis

Published: Sep 05, 2013  http://www.technologynetworks.com/Metabolomics/news.aspx?id=157138

Knocking out a single enzyme dramatically cripples the ability of aggressive cancer cells to spread and grow tumors.

The paper, published in the journal Proceedings of the National Academy of Sciences, sheds new light on the importance of lipids, a group of molecules that includes fatty acids and cholesterol, in the development of cancer.

Researchers have long known that cancer cells metabolize lipids differently than normal cells. Levels of ether lipids – a class of lipids that are harder to break down – are particularly elevated in highly malignant tumors.

“Cancer cells make and use a lot of fat and lipids, and that makes sense because cancer cells divide and proliferate at an accelerated rate, and to do that,

  • they need lipids, which make up the membranes of the cell,”

said study principal investigator Daniel Nomura, assistant professor in UC Berkeley’s Department of Nutritional Sciences and Toxicology. “Lipids have a variety of uses for cellular structure, but what we’re showing with our study is that

  • lipids can send signals that fuel cancer growth.”

In the study, Nomura and his team tested the effects of reducing ether lipids on human skin cancer cells and primary breast tumors. They targeted an enzyme,

  • alkylglycerone phosphate synthase, or AGPS,
  • known to be critical to the formation of ether lipids.

The researchers confirmed that

  1. AGPS expression increased when normal cells turned cancerous.
  2. inactivating AGPS substantially reduced the aggressiveness of the cancer cells.

“The cancer cells were less able to move and invade,” said Nomura.

The researchers also compared the impact of

  • disabling the AGPS enzyme in mice that had been injected with cancer cells.

Nomura. observes -“Among the mice that had the AGPS enzyme inactivated,

  • the tumors were nonexistent,”

“The mice that did not have this enzyme

  • disabled rapidly developed tumors.”

The researchers determined that

  • inhibiting AGPS expression depleted the cancer cells of ether lipids.
  • AGPS altered levels of other types of lipids important to the ability of the cancer cells to survive and spread, including
    • prostaglandins and acyl phospholipids.

“What makes AGPS stand out as a treatment target is that the enzyme seems to simultaneously

  • regulate multiple aspects of lipid metabolism
  • important for tumor growth and malignancy.”

Future steps include the

  • development of AGPS inhibitors for use in cancer therapy,

“This study sheds considerable light on the important role that AGPS plays in ether lipid metabolism in cancer cells, and it suggests that

  • inhibitors of this enzyme could impair tumor formation,”

said Benjamin Cravatt, Professor and Chair of Chemical Physiology at The Scripps Research Institute, who is not part of the UC.

Agilent Technologies Thought Leader Award Supports Translational Research Program
Published: Mon, March 04, 2013

The award will support Dr DePinho’s research into

  • metabolic reprogramming in the earliest stages of cancer.

Agilent Technologies Inc. announces that Dr. Ronald A. DePinho, a world-renowned oncologist and researcher, has received an Agilent Thought Leader Award.

DePinho is president of the University of Texas MD Anderson Cancer Center. DePinho and his team hope to discover and characterize

  • alterations in metabolic flux during tumor initiation and maintenance, and to identify biomarkers for early detection of pancreatic cancer together with
  • novel therapeutic targets.

Researchers on his team will work with scientists from the university’s newly formed Institute of Applied Cancer Sciences.

The Agilent Thought Leader Award provides funds to support personnel as well as a state-of-the-art Agilent 6550 iFunnel Q-TOF LC/MS system.

“I am extremely pleased to receive this award for metabolomics research, as the survival rates for pancreatic cancer have not significantly improved over the past 20 years,” DePinho said. “This technology will allow us to

  • rapidly identify new targets that drive the formation, progression and maintenance of pancreatic cancer.

Discoveries from this research will also lead to

  • the development of effective early detection biomarkers and novel therapeutic interventions.”

“We are proud to support Dr. DePinho’s exciting translational research program, which will make use of

  • metabolomics and integrated biology workflows and solutions in biomarker discovery,”

said Patrick Kaltenbach, Agilent vice president, general manager of the Liquid Phase Division, and the executive sponsor of this award.

The Agilent Thought Leader Program promotes fundamental scientific advances by support of influential thought leaders in the life sciences and chemical analysis fields.

The covalent modifier Nedd8 is critical for the activation of Smurf1 ubiquitin ligase in tumorigenesis

Ping Xie, Minghua Zhang, Shan He, Kefeng Lu, Yuhan Chen, Guichun Xing, et al.
Nature Communications
  2014; 5(3733).  http://dx.doi.org:/10.1038/ncomms4733

Neddylation, the covalent attachment of ubiquitin-like protein Nedd8, of the Cullin-RING E3 ligase family

  • regulates their ubiquitylation activity.

However, regulation of HECT ligases by neddylation has not been reported to date. Here we show that

  • the C2-WW-HECT ligase Smurf1 is activated by neddylation.

Smurf1 physically interacts with

  1. Nedd8 and Ubc12,
  2. forms a Nedd8-thioester intermediate, and then
  3. catalyses its own neddylation on multiple lysine residues.

Intriguingly, this autoneddylation needs

  • an active site at C426 in the HECT N-lobe.

Neddylation of Smurf1 potently enhances

  • ubiquitin E2 recruitment and
  • augments the ubiquitin ligase activity of Smurf1.

The regulatory role of neddylation

  • is conserved in human Smurf1 and yeast Rsp5.

Furthermore, in human colorectal cancers,

  • the elevated expression of Smurf1, Nedd8, NAE1 and Ubc12
  • correlates with cancer progression and poor prognosis.

These findings provide evidence that

  • neddylation is important in HECT ubiquitin ligase activation and
  • shed new light on the tumour-promoting role of Smurf1.
 Swinging domains in HECT E3

Swinging domains in HECT E3

Subject terms: Biological sciences Cancer Cell biology

Figure 1: Smurf1 expression is elevated in colorectal cancer tissues.

Smurf1 expression is elevated in colorectal cancer tissues.

Smurf1 expression is elevated in colorectal cancer tissues.

(a) Smurf1 expression scores are shown as box plots, with the horizontal lines representing the median; the bottom and top of the boxes representing the 25th and 75th percentiles, respectively; and the vertical bars representing the ra

Figure 2: Positive correlation of Smurf1 expression with Nedd8 and its interacting enzymes in colorectal cancer.

Positive correlation of Smurf1 expression with Nedd8 and its interacting enzymes in colorectal cancer

Positive correlation of Smurf1 expression with Nedd8 and its interacting enzymes in colorectal cancer

(a) Representative images from immunohistochemical staining of Smurf1, Ubc12, NAE1 and Nedd8 in the same colorectal cancer tumour. Scale bars, 100 μm. (b–d) The expression scores of Nedd8 (b, n=283 ), NAE1 (c, n=281) and Ubc12 (d, n=19…

Figure 3: Smurf1 interacts with Ubc12.

Smurf1 interacts with Ubc12

Smurf1 interacts with Ubc12

(a) GST pull-down assay of Smurf1 with Ubc12. Both input and pull-down samples were subjected to immunoblotting with anti-His and anti-GST antibodies. Smurf1 interacted with Ubc12 and UbcH5c, but not with Ubc9. (b) Mapping the regions…

Figure 4: Nedd8 is attached to Smurf1through C426-catalysed autoneddylation.

Nedd8 is attached to Smurf1through C426-catalysed autoneddylation

Nedd8 is attached to Smurf1through C426-catalysed autoneddylation

(a) Covalent neddylation of Smurf1 in vitro.Purified His-Smurf1-WT or C699A proteins were incubated with Nedd8 and Nedd8-E1/E2. Reactions were performed as described in the Methods section. Samples were analysed by western blotting wi…

Figure 5: Neddylation of Smurf1 activates its ubiquitin ligase activity.

Neddylation of Smurf1 activates its ubiquitin ligase activity.

Neddylation of Smurf1 activates its ubiquitin ligase activity.

(a) In vivo Smurf1 ubiquitylation assay. Nedd8 was co-expressed with Smurf1 WT or C699A in HCT116 cells (left panels). Twenty-four hours post transfection, cells were treated with MG132 (20 μM, 8 h). HCT116 cells were transfected with…

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The deubiquitylase USP33 discriminates between RALB functions in autophagy and innate immune response

M Simicek, S Lievens, M Laga, D Guzenko, VN. Aushev, et al.
Nature Cell Biology 2013; 15, 1220–1230    http://dx.doi.org:/10.1038/ncb2847

The RAS-like GTPase RALB mediates cellular responses to nutrient availability or viral infection by respectively

  • engaging two components of the exocyst complex, EXO84 and SEC5.
  1. RALB employs SEC5 to trigger innate immunity signalling, whereas
  2. RALB–EXO84 interaction induces autophagocytosis.

How this differential interaction is achieved molecularly by the RAL GTPase remains unknown.

We found that whereas GTP binding

  • turns on RALB activity,

ubiquitylation of RALB at Lys 47

  • tunes its activity towards a particular effector.

Specifically, ubiquitylation at Lys 47

  • sterically inhibits RALB binding to EXO84, while
  • facilitating its interaction with SEC5.

Double-stranded RNA promotes

  • RALB ubiquitylation and
  • SEC5–TBK1 complex formation.

In contrast, nutrient starvation

  • induces RALB deubiquitylation
  • by accumulation and relocalization of the deubiquitylase USP33
  • to RALB-positive vesicles.

Deubiquitylated RALB

  • promotes the assembly of the RALB–EXO84–beclin-1 complexes
  • driving autophagosome formation. Thus,
  • ubiquitylation within the effector-binding domain
  • provides the switch for the dual functions of RALB in
    • autophagy and innate immune responses.

Part 5. Metabolic Syndrome

Single Enzyme is Necessary for Development of Diabetes

Published: Aug 20, 2014 http://www.technologynetworks.com/Metabolomics/news.aspx?ID=169416

12-LO enzyme promotes the obesity-induced oxidative stress in the pancreatic cells.

An enzyme called 12-LO promotes the obesity-induced oxidative stress in the pancreatic cells that leads

  • to pre-diabetes, and diabetes.

12-LO’s enzymatic action is the last step in

  • the production of certain small molecules that harm the cell,

according to a team from Indiana University School of Medicine, Indianapolis.

The findings will enable the development of drugs that can interfere with this enzyme, preventing or even reversing diabetes. The research is published ahead of print in the journal Molecular and Cellular Biology.

In earlier studies, these researchers and their collaborators at Eastern Virginia Medical School showed that

  • 12-LO (which stands for 12-lipoxygenase) is present in these cells
  • only in people who become overweight.

The harmful small molecules resulting from 12-LO’s enzymatic action are known as HETEs, short for hydroxyeicosatetraenoic acid.

  1. HETEs harm the mitochondria, which then
  2. fail to produce sufficient energy to enable
  3. the pancreatic cells to manufacture the necessary quantities of insulin.

For the study, the investigators genetically engineered mice that

  • lacked the gene for 12-LO exclusively in their pancreas cells.

Mice were either fed a low-fat or high-fat diet.

Both the control mice and the knockout mice on the high fat diet

  • developed obesity and insulin resistance.

The investigators also examined the pancreatic beta cells of both knockout and control mice, using both microscopic studies and molecular analysis. Those from the knockout mice were intact and healthy, while

  • those from the control mice showed oxidative damage,
  • demonstrating that 12-LO and the resulting HETEs
  • caused the beta cell failure.

Mirmira notes that fatty diet used in the study was the Western Diet, which comprises mostly saturated-“bad”-fats. Based partly on a recent study of related metabolic pathways, he says that

  • the unsaturated and mono-unsaturated fats-which comprise most fats in the healthy,
  • relatively high fat Mediterranean diet-are unlikely to have the same effects.

“Our research is the first to show that 12-LO in the beta cell

  • is the culprit in the development of pre-diabetes, following high fat diets,” says Mirmira.

“Our work also lends important credence to the notion that

  • the beta cell is the primary defective cell in virtually all forms of diabetes and pre-diabetes.”

A New Player in Lipid Metabolism Discovered

Published: Aug18, 2014  http://www.technologynetworks.com/Metabolomics/news.aspx?ID=169356

Specially engineered mice gained no weight, and normal counterparts became obese

  • on the same high-fat, obesity-inducing Western diet.

Specially engineered mice that lacked a particular gene did not gain weight

  • when fed a typical high-fat, obesity-inducing Western diet.

Yet, these mice ate the same amount as their normal counterparts that became obese.

The mice were engineered with fat cells that lacked a gene called SEL1L,

  • known to be involved in the clearance of mis-folded proteins
  • in the cell’s protein making machinery called the endoplasmic reticulum (ER).

When mis-folded proteins are not cleared but accumulate,

  • they destroy the cell and contribute to such diseases as
  1. mad cow disease,
  2. Type 1 diabetes and
  3. cystic fibrosis.

“The million-dollar question is why don’t these mice gain weight? Is this related to its inability to clear mis-folded proteins in the ER?” said Ling Qi, associate professor of molecular and biochemical nutrition and senior author of the study published online July 24 in Cell Metabolism. Haibo Sha, a research associate in Qi’s lab, is the paper’s lead author.

Interestingly, the experimental mice developed a host of other problems, including

  • postprandial hypertriglyceridemia,
  • and fatty livers.

“Although we are yet to find out whether these conditions contribute to the lean phenotype, we found that

  • there was a lipid partitioning defect in the mice lacking SEL1L in fat cells,
  • where fat cells cannot store fat [lipids], and consequently
  • fat goes to the liver.

During the investigation of possible underlying mechanisms, we discovered

  • a novel function for SEL1L as a regulator of lipid metabolism,” said Qi.

Sha said “We were very excited to find that

  • SEL1L is required for the intracellular trafficking of
  • lipoprotein lipase (LPL), acting as a chaperone,” .

and added that “Using several tissue-specific knockout mouse models,

  • we showed that this is a general phenomenon,”

Without LPL, lipids remain in the circulation;

  • fat and muscle cells cannot absorb fat molecules for storage and energy combustion,

People with LPL mutations develop

  • postprandial hypertriglyceridemia similar to
  • conditions found in fat cell-specific SEL1L-deficient mice, said Qi.

Future work will investigate the

  • role of SEL1L in human patients carrying LPL mutations and
  • determine why fat cell-specific SEL1L-deficient mice remain lean under Western diets, said Sha.

Co-authors include researchers from Cedars-Sinai Medical Center in Los Angeles; Wageningen University in the Netherlands; Georgia State University; University of California, Los Angeles; and the Medical College of Soochow University in China.

The study was funded by the U.S. National Institutes of Health, the Netherlands Organization for Health Research and Development National Institutes of Health, the Cedars-Sinai Medical Center, Chinese National Science Foundation, the American Diabetes Association, Cornell’s Center for Vertebrate Genomics and the Howard Hughes Medical Institute.

Part 6. Biomarkers

Biomarkers Take Center Stage

Josh P. Roberts
GEN May 1, 2013 (Vol. 33, No. 9)  http://www.genengnews.com/

While work with biomarkers continues to grow, scientists are also grappling with research-related bottlenecks, such as

  1. affinity reagent development,
  2. platform reproducibility, and
  3. sensitivity.

Biomarkers by definition indicate some state or process that generally occurs

  • at a spatial or temporal distance from the marker itself, and

it would not be an exaggeration to say that biomedicine has become infatuated with them:

  1. where to find them,
  2. when they may appear,
  3. what form they may take, and
  4. how they can be used to diagnose a condition or
  5. predict whether a therapy may be successful.

Biomarkers are on the agenda of many if not most industry gatherings, and in cases such as Oxford Global’s recent “Biomarker Congress” and the GTC “Biomarker Summit”, they hold the naming rights. There, some basic principles were built upon, amended, and sometimes challenged.

In oncology, for example, biomarker discovery is often predicated on the premise that

  • proteins shed from a tumor will traverse to and persist in, and be detectable in, the circulation.

By quantifying these proteins—singularly or as part of a larger “signature”—the hope is

  1. to garner information about the molecular characteristics of the cancer
  2. that will help with cancer detection and
  3. personalization of the treatment strategy.

Yet this approach has not yet turned into the panacea that was hoped for. Bottlenecks exist in

  • affinity reagent development,
  • platform reproducibility, and
  • sensitivity.

There is also a dearth of understanding of some of the

  • fundamental principles of biomarker biology that we need to know the answers to,

said Parag Mallick, Ph.D., whose lab at Stanford University is “working on trying to understand where biomarkers come from.”

There are dogmas saying that

  • circulating biomarkers come solely from secreted proteins.

But Dr. Mallick’s studies indicate that fully

  • 50% of circulating proteins may come from intracellular sources or
  • proteins that are annotated as such.

“We don’t understand the processes governing

  • which tumor-derived proteins end up in the blood.”

Other questions include “how does the size of a tumor affect how much of a given protein will be in the blood?”—perhaps

  • the tumor is necrotic at the center, or
  • it’s hypervascular or hypovascular.

He points out “The problem is that these are highly nonlinear processes at work, and

  • there is a large number of factors that might affect the answer to that question,” .

Their research focuses on using

  1. mass spectrometry and
  2. computational analysis
  • to characterize the biophysical properties of the circulating proteome, and
  • relate these to measurements made of the tumor itself.

Furthermore, he said – “We’ve observed that the proteins that are likely to

  • first show up and persist in the circulation, ..
  • are more stable than proteins that don’t,”
  • “we can quantify how significant the effect is.”

The goal is ultimately to be able to

  1. build rigorous, formal mathematical models that will allow something measured in the blood
  2. to be tied back to the molecular biology taking place in the tumor.

And conversely, to use those models

  • to predict from a tumor what will be found in the circulation.

“Ultimately, the models will allow you to connect the dots between

  • what you measure in the blood and the biology of the tumor.”

Bound for Affinity Arrays

Affinity reagents are the main tools for large-scale protein biomarker discovery. And while this has tended to mean antibodies (or their derivatives), other affinity reagents are demanding a place in the toolbox.

Affimers, a type of affinity reagent being developed by Avacta, consist of

  1. a biologically inert, biophysically stable protein scaffold
  2. containing three variable regions into which
  3. distinct peptides are inserted.

The resulting three-dimensional surface formed by these peptides

  • interacts and binds to proteins and other molecules in solution,
  • much like the antigen-binding site of antibodies.

Unlike antibodies, Affimers are relatively small (13 KDa),

  • non-post-translationally modified proteins
  • that can readily be expressed in bacterial culture.

They may be made to bind surfaces through unique residues

  • engineered onto the opposite face of the Affimer,
  • allowing the binding site to be exposed to the target in solution.

“We don’t seem to see in what we’ve done so far

  • any real loss of activity or functionality of Affimers when bound to surfaces—

they’re very robust,” said CEO Alastair Smith, Ph.D.

Avacta is taking advantage of this stability and its large libraries of Affimers to develop

  • very large affinity microarrays for
  • drug and biomarker discovery.

To date they have printed arrays with around 20–25,000 features, and Dr. Smith is “sure that we can get toward about 50,000 on a slide,” he said. “There’s no real impediment to us doing that other than us expressing the proteins and getting on with it.”

Customers will be provided with these large, complex “naïve” discovery arrays, readable with standard equipment. The plan is for the company to then “support our customers by providing smaller arrays with

  • the Affimers that are binding targets of interest to them,” Dr. Smith foretold.

And since the intellectual property rights are unencumbered,

  • Affimers in those arrays can be licensed to the end users
  • to develop diagnostics that can be validated as time goes on.

Around 20,000-Affimer discovery arrays were recently tested by collaborator Professor Ann Morgan of the University of Leeds with pools of unfractionated serum from patients with symptoms of inflammatory disease. The arrays

  • “rediscovered” elevated C-reactive protein (CRP, the clinical gold standard marker)
  • as well as uncovered an additional 22 candidate biomarkers.
  • other candidates combined with CRP, appear able to distinguish between different diseases such as
  1. rheumatoid arthritis,
  2. psoriatic arthritis,
  3. SLE, or
  4. giant cell arteritis.

Epigenetic Biomarkers

Methylation of adenine

Sometimes biomarkers are used not to find disease but

  • to distinguish healthy human cell types, with
  •  examples being found in flow cytometry and immunohistochemistry.

These widespread applications, however, are difficult to standardize, being

  • subject to arbitrary or subjective gating protocols and other imprecise criteria.

Epiontis instead uses an epigenetic approach. “What we need is a unique marker that is

  • demethylated only in one cell type and
  • methylated in all the other cell types,”

Each cell of the right cell type will have

  • two demethylated copies of a certain gene locus,
  • allowing them to be enumerated by quantitative PCR.

The biggest challenge is finding that unique epigenetic marker. To do so they look through the literature for proteins and genes described as playing a role in the cell type’s biology, and then

  • look at the methylation patterns to see if one can be used as a marker,

They also “use customized Affymetrix chips to look at the

  • differential epigenetic status of different cell types on a genomewide scale.”

explained CBO and founder Ulrich Hoffmueller, Ph.D.

The company currently has a panel of 12 assays for 12 immune cell types. Among these is an assay for

  • regulatory T (Treg) cells that queries the Foxp3 gene—which is uniquely demethylated in Treg
  • even though it is transiently expressed in activated T cells of other subtypes.

Also assayed are Th17 cells, difficult to detect by flow cytometry because

  • “the cells have to be stimulated in vitro,” he pointed out.

Developing New Assays for Cancer Biomarkers

Researchers at Myriad RBM and the Cancer Prevention Research Institute of Texas are collaborating to develop

  • new assays for cancer biomarkers on the Myriad RBM Multi-Analyte Profile (MAP) platform.

The release of OncologyMAP 2.0 expanded Myriad RBM’s biomarker menu to over 250 analytes, which can be measured from a small single sample, according to the company. Using this menu, L. Stephen et al., published a poster, “Analysis of Protein Biomarkers in Prostate and Colorectal Tumor Lysates,” which showed the results of

  • a survey of proteins relevant to colorectal (CRC) and prostate (PC) tumors
  • to identify potential proteins of interest for cancer research.

The study looked at CRC and PC tumor lysates and found that 102 of the 115 proteins showed levels above the lower limit of quantification.

  • Four markers were significantly higher in PC and 10 were greater in CRC.

For most of the analytes, duplicate sections of the tumor were similar, although some analytes did show differences. In four of the CRC analytes, tumor number four showed differences for CEA and tumor number 2 for uPA.

Thirty analytes were shown to be

  • different in CRC tumor compared to its adjacent tissue.
  • Ten of the analytes were higher in adjacent tissue compared to CRC.
  • Eighteen of the markers examined demonstrated  —-

significant correlations of CRC tumor concentration to serum levels.

“This suggests.. that the Oncology MAP 2.0 platform “provides a good method for studying changes in tumor levels because many proteins can be assessed with a very small sample.”

Clinical Test Development with MALDI-ToF

While there have been many attempts to translate results from early discovery work on the serum proteome into clinical practice, few of these efforts have progressed past the discovery phase.

Matrix-assisted laser desorption/ionization-time of flight (MALDI-ToF) mass spectrometry on unfractionated serum/plasma samples offers many practical advantages over alternative techniques, and does not require

  • a shift from discovery to development and commercialization platforms.

Biodesix claims it has been able to develop the technology into

  • a reproducible, high-throughput tool to
  • routinely measure protein abundance from serum/plasma samples.

“.. we improved data-analysis algorithms to

  • reproducibly obtain quantitative measurements of relative protein abundance from MALDI-ToF mass spectra.

Heinrich Röder, CTO points out that the MALDI-ToF measurements

  • are combined with clinical outcome data using
  • modern learning theory techniques
  • to define specific disease states
  • based on a patient’s serum protein content,”

The clinical utility of the identification of these disease states can be investigated through a retrospective analysis of differing sample sets. For example, Biodesix clinically validated its first commercialized serum proteomic test, VeriStrat®, in 85 different retrospective sample sets.

Röder adds that “It is becoming increasingly clear that

  • the patients whose serum is characterized as VeriStrat Poor show
  • consistently poor outcomes irrespective of
  1. tumor type,
  2. histology, or
  3. molecular tumor characteristics,”

MALDI-ToF mass spectrometry, in its standard implementation,

  • allows for the observation of around 100 mostly high-abundant serum proteins.

Further, “while this does not limit the usefulness of tests developed from differential expression of these proteins,

  • the discovery potential would be greatly enhanced
  • if we could probe deeper into the proteome
  • while not giving up the advantages of the MALDI-ToF approach,”

Biodesix reports that its new MALDI approach, Deep MALDI™, can perform

  • simultaneous quantitative measurement of more than 1,000 serum protein features (or peaks) from 10 µL of serum in a high-throughput manner.
  • it increases the observable signal noise ratio from a few hundred to over 50,000,
  • resulting in the observation of many lower-abundance serum proteins.

Breast cancer, a disease now considered to be a collection of many complexes of symptoms and signatures—the dominant ones are labeled Luminal A, Luminal B, Her2, and Basal— which suggests different prognose, and

  • these labels are considered too simplistic for understanding and managing a woman’s cancer.

Studies published in the past year have looked at

  1. somatic mutations,
  2. gene copy number aberrations,
  3. gene expression abnormalities,
  4. protein and miRNA expression, and
  5. DNA methylation,

coming up with a list of significantly mutated genes—hot spots—in different categories of breast cancers. Targeting these will inevitably be the focus of much coming research.

“We’ve been taking these large trials and profiling these on a variety of array or sequence platforms. We think we’ll get

  1. prognostic drivers
  2. predictive markers for taxanes and
  3. monoclonal antibodies and
  4. tamoxifen and aromatase inhibitors,”
    explained Brian Leyland-Jones, Ph.D., director of Edith Sanford Breast Cancer Research. “We will end up with 20–40 different diseases, maybe more.”

Edith Sanford Breast Cancer Research is undertaking a pilot study in collaboration with The Scripps Research Institute, using a variety of tests on 25 patients to see how the information they provide complements each other, the overall flow, and the time required to get and compile results.

Laser-captured tumor samples will be subjected to low passage whole-genome, exome, and RNA sequencing (with targeted resequencing done in parallel), and reverse-phase protein and phosphorylation arrays, with circulating nucleic acids and circulating tumor cells being queried as well. “After that we hope to do a 100- or 150-patient trial when we have some idea of the best techniques,” he said.

Dr. Leyland-Jones predicted that ultimately most tumors will be found

  • to have multiple drivers,
  • with most patients receiving a combination of two, three, or perhaps four different targeted therapies.

Reduce to Practice

According to Randox, the evidence Investigator is a sophisticated semi-automated biochip sys­tem designed for research, clinical, forensic, and veterinary applications.

Once biomarkers that may have an impact on therapy are discovered, it is not always routine to get them into clinical practice. Leaving regulatory and financial, intellectual property and cultural issues aside, developing a diagnostic based on a biomarker often requires expertise or patience that its discoverer may not possess.

Andrew Gribben is a clinical assay and development scientist at Randox Laboratories, based in Northern Ireland, U.K. The company utilizes academic and industrial collaborators together with in-house discovery platforms to identify biomarkers that are

  • augmented or diminished in a particular pathology
  • relative to appropriate control populations.

Biomarkers can be developed to be run individually or

  • combined into panels of immunoassays on its multiplex biochip array technology.

Specificity can also be gained—or lost—by the affinity of reagents in an assay. The diagnostic potential of Heart-type fatty acid binding protein (H-FABP) abundantly expressed in human myocardial cells was recognized by Jan Glatz of Maastricht University, The Netherlands, back in 1988. Levels rise quickly within 30 minutes after a myocardial infarction, peaking at 6–8 hours and return to normal within 24–30 hours. Yet at the time it was not known that H-FABP was a member of a multiprotein family, with which the polyclonal antibodies being used in development of an assay were cross-reacting, Gribben related.

Randox developed monoclonal antibodies specific to H-FABP, funded trials investigating its use alone, and multiplexed with cardiac biomarker assays, and, more than 30 years after the biomarker was identified, in 2011, released a validated assay for H-FABP as a biomarker for early detection of acute myocardial infarction.

Ultrasensitive Immunoassays for Biomarker Development

Research has shown that detection and monitoring of biomarker concentrations can provide

  • insights into disease risk and progression.

Cytokines have become attractive biomarkers and candidates

  • for targeted therapies for a number of autoimmune diseases, including rheumatoid arthritis (RA), Crohn’s disease, and psoriasis, among others.

However, due to the low-abundance of circulating cytokines, such as IL-17A, obtaining robust measurements in clinical samples has been difficult.

Singulex reports that its digital single-molecule counting technology provides

  • increased precision and detection sensitivity over traditional ELISA techniques,
  • helping to shed light on biomarker verification and validation programs.

The company’s Erenna® immunoassay system, which includes optimized immunoassays, offers LLoQ to femtogram levels per mL resolution—even in healthy populations, at an improvement of 1-3 fold over standard ELISAs or any conventional technology and with a dynamic range of up to 4-logs, according to a Singulex official, who adds that

  • this sensitivity improvement helps minimize undetectable samples that
  • could otherwise delay or derail clinical studies.

The official also explains that the Singulex solution includes an array of products and services that are being applied to a number of programs and have enabled the development of clinically relevant biomarkers, allowing translation from discovery to the clinic.

In a poster entitled “Advanced Single Molecule Detection: Accelerating Biomarker Development Utilizing Cytokines through Ultrasensitive Immunoassays,” a case study was presented of work performed by Jeff Greenberg of NYU to show how the use of the Erenna system can provide insights toward

  • improving the clinical utility of biomarkers and
  • accelerating the development of novel therapies for treating inflammatory diseases.

A panel of inflammatory biomarkers was examined in DMARD (disease modifying antirheumatic drugs)-naïve RA (rheumatoid arthritis) vs. knee OA (osteoarthritis) patient cohorts. Markers that exhibited significant differences in plasma concentrations between the two cohorts included

  • CRP, IL-6R alpha, IL-6, IL-1 RA, VEGF, TNF-RII, and IL-17A, IL-17F, and IL-17A/F.

Among the three tested isoforms of IL-17,

  • the magnitude of elevation for IL-17F in RA patients was the highest.

“Singulex provides high-resolution monitoring of baseline IL-17A concentrations that are present at low levels,” concluded the researchers. “The technology also enabled quantification of other IL-17 isoforms in RA patients, which have not been well characterized before.”

The Singulex Erenna System has also been applied to cardiovascular disease research, for which its

  • cardiac troponin I (cTnI) digital assay can be used to measure circulating
  • levels of cTnI undetectable by other commercial assays.

Recently presented data from Brigham and Women’s Hospital and the TIMI-22 study showed that

  • using the Singulex test to serially monitor cTnI helps
  • stratify risk in post-acute coronary syndrome patients and
  • can identify patients with elevated cTnI
  • who have the most to gain from intensive vs. moderate-dose statin therapy,

according to the scientists involved in the research.

The study poster, “Prognostic Performance of Serial High Sensitivity Cardiac Troponin Determination in Stable Ischemic Heart Disease: Analysis From PROVE IT-TIMI 22,” was presented at the 2013 American College of Cardiology (ACC) Annual Scientific Session & Expo by R. O’Malley et al.

Biomarkers Changing Clinical Medicine

Better Diagnosis, Prognosis, and Drug Targeting Are among Potential Benefits

  1. John Morrow Jr., Ph.D.

Researchers at EMD Chemicals are developing biomarker immunoassays

  • to monitor drug-induced toxicity including kidney damage.

The pace of biomarker development is accelerating as investigators report new studies on cancer, diabetes, Alzheimer disease, and other conditions in which the evaluation and isolation of workable markers is prominently featured.

Wei Zheng, Ph.D., leader of the R&D immunoassay group at EMD Chemicals, is overseeing a program to develop biomarker immunoassays to

  • monitor drug-induced toxicity, including kidney damage.

“One of the principle reasons for drugs failing during development is because of organ toxicity,” says Dr. Zheng.
“proteins liberated into the serum and urine can serve as biomarkers of adverse response to drugs, as well as disease states.”

Through collaborative programs with Rules-Based Medicine (RBM), the EMD group has released panels for the profiling of human renal impairment and renal toxicity. These urinary biomarker based products fit the FDA and EMEA guidelines for assessment of drug-induced kidney damage in rats.

The group recently performed a screen for potential protein biomarkers in relation to

  • kidney toxicity/damage on a set of urine and plasma samples
  • from patients with documented renal damage.

Additionally, Dr. Zheng is directing efforts to move forward with the multiplexed analysis of

  • organ and cellular toxicity.

Diseases thought to involve compromised oxidative phosphorylation include

  • diabetes, Parkinson and Alzheimer diseases, cancer, and the aging process itself.

Good biomarkers allow Dr. Zheng to follow the mantra, “fail early, fail fast.” With robust, multiplexible biomarkers, EMD can detect bad drugs early and kill them before they move into costly large animal studies and clinical trials. “Recognizing the severe liability that toxicity presents, we can modify the structure of the candidate molecule and then rapidly reassess its performance.”

Scientists at Oncogene Science a division of Siemens Healthcare Diagnostics, are also focused on biomarkers. “We are working on a number of antibody-based tests for various cancers, including a test for the Ca-9 CAIX protein, also referred to as carbonic anhydrase,” Walter Carney, Ph.D., head of the division, states.

CAIX is a transmembrane protein that is

  • overexpressed in a number of cancers, and, like Herceptin and the Her-2 gene,
  • can serve as an effective and specific marker for both diagnostic and therapeutic purposes.
  • It is liberated into the circulation in proportion to the tumor burden.

Dr. Carney and his colleagues are evaluating patients after tumor removal for the presence of the Ca-9 CAIX protein. If

  • the levels of the protein in serum increase over time,
  • this suggests that not all the tumor cells were removed and the tumor has metastasized.

Dr. Carney and his team have developed both an immuno-histochemistry and an ELISA test that could be used as companion diagnostics in clinical trials of CAIX-targeted drugs.

The ELISA for the Ca-9 CAIX protein will be used in conjunction with Wilex’ Rencarex®, which is currently in a

  • Phase III trial as an adjuvant therapy for non-metastatic clear cell renal cancer.

Additionally, Oncogene Science has in its portfolio an FDA-approved test for the Her-2 marker. Originally approved for Her-2/Neu-positive breast cancer, its indications have been expanded over time, and was approved

  • for the treatment of gastric cancer last year.

It is normally present on breast cancer epithelia but

  • overexpressed in some breast cancer tumors.

“Our products are designed to be used in conjunction with targeted therapies,” says Dr. Carney. “We are working with companies that are developing technology around proteins that are

  • overexpressed in cancerous tissues and can be both diagnostic and therapeutic targets.”

The long-term goal of these studies is to develop individualized therapies, tailored for the patient. Since the therapies are expensive, accurate diagnostics are critical to avoid wasting resources on patients who clearly will not respond (or could be harmed) by the particular drug.

“At this time the rate of response to antibody-based therapies may be very poor, as

  • they are often employed late in the course of the disease, and patients are in such a debilitated state
  • that they lack the capacity to react positively to the treatment,” Dr. Carney explains.

Nanoscale Real-Time Proteomics

Stanford University School of Medicine researchers, working with Cell BioSciences, have developed a

  • nanofluidic proteomic immunoassay that measures protein charge,
  • similar to immunoblots, mass spectrometry, or flow cytometry.
  • unlike these platforms, this approach can measure the amount of individual isoforms,
  • specifically, phosphorylated molecules.

“We have developed a nanoscale device for protein measurement, which I believe could be useful for clinical analysis,” says Dean W. Felsher, M.D., Ph.D., associate professor at Stanford University School of Medicine.

Critical oncogenic transformations involving

  • the activation of the signal-related kinases ERK-1 and ERK-2 can now be followed with ease.

“The fact that we measure nanoquantities with accuracy means that

  • we can interrogate proteomic profiles in clinical patients,

by drawing tiny needle aspirates from tumors over the course of time,” he explains.

“This allows us to observe the evolution of tumor cells and

  • their response to therapy
  • from a baseline of the normal tissue as a standard of comparison.”

According to Dr. Felsher, 20 cells is a large enough sample to obtain a detailed description. The technology is easy to automate, which allows

  • the inclusion of hundreds of assays.

Contrasting this technology platform with proteomic analysis using microarrays, Dr. Felsher notes that the latter is not yet workable for revealing reliable markers.

Dr. Felsher and his group published a description of this technology in Nature Medicine. “We demonstrated that we could take a set of human lymphomas and distinguish them from both normal tissue and other tumor types. We can

  • quantify changes in total protein, protein activation, and relative abundance of specific phospho-isoforms
  • from leukemia and lymphoma patients receiving targeted therapy.

Even with very small numbers of cells, we are able to show that the results are consistent, and

  • our sample is a random profile of the tumor.”

Splice Variant Peptides

“Aberrations in alternative splicing may generate

  • much of the variation we see in cancer cells,”

says Gilbert Omenn, Ph.D., director of the center for computational medicine and bioinformatics at the University of Michigan School of Medicine. Dr. Omenn and his colleague, Rajasree Menon, are

  • using this variability as a key to new biomarker identification.

It is becoming evident that splice variants play a significant role in the properties of cancer cells, including

  • initiation, progression, cell motility, invasiveness, and metastasis.

Alternative splicing occurs through multiple mechanisms

  • when the exons or coding regions of the DNA transcribe mRNA,
  • generating initiation sites and connecting exons in protein products.

Their translation into protein can result in numerous protein isoforms, and

  • these isoforms may reflect a diseased or cancerous state.

Regulatory elements within the DNA are responsible for selecting different alternatives; thus

  • the splice variants are tempting targets for exploitation as biomarkers.
Analyses of the splice-site mutation

Analyses of the splice-site mutation

Despite the many questions raised by these observations, splice variation in tumor material has not been widely studied. Cancer cells are known for their tremendous variability, which allows them to

  • grow rapidly, metastasize, and develop resistance to anticancer drugs.

Dr. Omenn and his collaborators used

  • mass spec data to interrogate a custom-built database of all potential mRNA sequences
  • to find alternative splice variants.

When they compared normal and malignant mammary gland tissue from a mouse model of Her2/Neu human breast cancers, they identified a vast number (608) of splice variant proteins, of which

  • peptides from 216 were found only in the tumor sample.

“These novel and known alternative splice isoforms

  • are detectable both in tumor specimens and in plasma and
  • represent potential biomarker candidates,” Dr. Omenn adds.

Dr. Omenn’s observations and those of his colleague Lewis Cantley, Ph.D., have also

  • shed light on the origins of the classic Warburg effect,
  • the shift to anaerobic glycolysis in tumor cells.

The novel splice variant M2, of muscle pyruvate kinase,

  • is observed in embryonic and tumor tissue.

It is associated with this shift, the result of

  • the expression of a peptide splice variant sequence.

It is remarkable how many different areas of the life sciences are tied into the phenomenon of splice variation. The changes in the genetic material can be much greater than point mutations, which have been traditionally considered to be the prime source of genetic variability.

“We now have powerful methods available to uncover a whole new category of variation,” Dr. Omenn says. “High-throughput RNA sequencing and proteomics will be complementary in discovery studies of splice variants.”

Splice variation may play an important role in rapid evolutionary changes, of the sort discussed by Susumu Ohno and Stephen J. Gould decades ago. They, and other evolutionary biologists, argued that

  • gene duplication, combined with rapid variability, could fuel major evolutionary jumps.

At the time, the molecular mechanisms of variation were poorly understood, but today

  • the tools are available to rigorously evaluate the role of
  • splice variation and other contributors to evolutionary change.

“Biomarkers derived from studies of splice variants, could, in the future, be exploited

  • both for diagnosis and prognosis and
  • for drug targeting of biological networks,
  • in situations such as the Her-2/Neu breast cancers,” Dr. Omenn says.

Aminopeptidase Activities

“By correlating the proteolytic patterns with disease groups and controls, we have shown that

  • exopeptidase activities contribute to the generation of not only cancer-specific
  • but also cancer type specific serum peptides.

according to Paul Tempst, Ph.D., professor and director of the Protein Center at the Memorial Sloan-Kettering Cancer Center.

So there is a direct link between peptide marker profiles of disease and differential protease activity.” For this reason Dr. Tempst argues that “the patterns we describe may have value as surrogate markers for detection and classification of cancer.”

To investigate this avenue, Dr. Tempst and his colleagues have followed

  • the relationship between exopeptidase activities and metastatic disease.

“We monitored controlled, de novo peptide breakdown in large numbers of biological samples using mass spectrometry, with relative quantitation of the metabolites,” Dr. Tempst explains. This entailed the use of magnetic, reverse-phase beads for analyte capture and a MALDI-TOF MS read-out.

“In biomarker discovery programs, functional proteomics is usually not pursued,” says Dr. Tempst. “For putative biomarkers, one may observe no difference in quantitative levels of proteins, while at the same time, there may be substantial differences in enzymatic activity.”

In a preliminary prostate cancer study, the team found a significant difference

  • in activity levels of exopeptidases in serum from patients with metastatic prostate cancer
  • as compared to primary tumor-bearing individuals and normal healthy controls.

However, there were no differences in amounts of the target protein, and this potential biomarker would have been missed if quantitative levels of protein had been the only criterion of selection.

It is frequently stated that “practical fusion energy is 30 years in the future and always will be.” The same might be said of functional, practical biomarkers that can pass muster with the FDA. But splice variation represents a new handle on this vexing problem. It appears that we are seeing the emergence of a new approach that may finally yield definitive diagnostic tests, detectable in serum and urine samples.

Part 7. Epigenetics and Drug Metabolism

DNA Methylation Rules: Studying Epigenetics with New Tools

The tools to unravel the epigenetic control mechanisms that influence how cells control access of transcriptional proteins to DNA are just beginning to emerge.

Patricia Fitzpatrick Dimond, Ph.D.

http://www.genengnews.com/media/images/AnalysisAndInsight/Feb7_2013_24454248_GreenPurpleDNA_EpigeneticsToolsII3576166141.jpg

New tools may help move the field of epigenetic analysis forward and potentially unveil novel biomarkers for cellular development, differentiation, and disease.

DNA sequencing has had the power of technology behind it as novel platforms to produce more sequencing faster and at lower cost have been introduced. But the tools to unravel the epigenetic control mechanisms that influence how cells control access of transcriptional proteins to DNA are just beginning to emerge.

Among these mechanisms, DNA methylation, or the enzymatically mediated addition of a methyl group to cytosine or adenine dinucleotides,

  • serves as an inherited epigenetic modification that
  • stably modifies gene expression in dividing cells.

The unique methylomes are largely maintained in differentiated cell types, making them critical to understanding the differentiation potential of the cell.

In the DNA methylation process, cytosine residues in the genome are enzymatically modified to 5-methylcytosine,

  • which participates in transcriptional repression of genes during development and disease progression.

5-methylcytosine can be further enzymatically modified to 5-hydroxymethylcytosine by the TET family of methylcytosine dioxygenases. DNA methylation affects gene transcription by physically

  • interfering with the binding of proteins involved in gene transcription.

Methylated DNA may be bound by methyl-CpG-binding domain proteins (MBDs) that can

  • then recruit additional proteins. Some of these include histone deacetylases and other chromatin remodeling proteins that modify histones, thereby
  • forming compact, inactive chromatin, or heterochromatin.

While DNA methylation doesn’t change the genetic code,

  • it influences chromosomal stability and gene expression.

Epigenetics and Cancer Biomarkers

multistage chemical carcinogenesis

multistage chemical carcinogenesis

And because of the increasing recognition that DNA methylation changes are involved in human cancers, scientists have suggested that these epigenetic markers may provide biological markers for cancer cells, and eventually point toward new diagnostic and therapeutic targets. Cancer cell genomes display genome-wide abnormalities in DNA methylation patterns,

  • some of which are oncogenic and contribute to genome instability.

In particular, de novo methylation of tumor suppressor gene promoters

  • occurs frequently in cancers, thereby silencing them and promoting transformation.

Cytosine hydroxymethylation (5-hydroxymethylcytosine, or 5hmC), the aforementioned DNA modification resulting from the enzymatic conversion of 5mC into 5-hydroxymethylcytosine by the TET family of oxygenases, has been identified

  • as another key epigenetic modification marking genes important for
  • pluripotency in embryonic stem cells (ES), as well as in cancer cells.

The base 5-hydroxymethylcytosine was recently identified as an oxidation product of 5-methylcytosine in mammalian DNA. In 2011, using sensitive and quantitative methods to assess levels of 5-hydroxymethyl-2′-deoxycytidine (5hmdC) and 5-methyl-2′-deoxycytidine (5mdC) in genomic DNA, scientists at the Department of Cancer Biology, Beckman Research Institute of the City of Hope, Duarte, California investigated

  • whether levels of 5hmC can distinguish normal tissue from tumor tissue.

They showed that in squamous cell lung cancers, levels of 5hmdC showed

  • up to five-fold reduction compared with normal lung tissue.

In brain tumors,5hmdC showed an even more drastic reduction

  • with levels up to more than 30-fold lower than in normal brain,
  • but 5hmdC levels were independent of mutations in isocitrate dehydrogenase-1, the enzyme that converts 5hmC to 5hmdC.

Immunohistochemical analysis indicated that 5hmC is “remarkably depleted” in many types of human cancer.

  • there was an inverse relationship between 5hmC levels and cell proliferation with lack of 5hmC in proliferating cells.

Their data suggest that 5hmdC is strongly depleted in human malignant tumors,

  • a finding that adds another layer of complexity to the aberrant epigenome found in cancer tissue.

In addition, a lack of 5hmC may become a useful biomarker for cancer diagnosis.

Enzymatic Mapping

But according to New England Biolabs’ Sriharsa Pradhan, Ph.D., methods for distinguishing 5mC from 5hmC and analyzing and quantitating the cell’s entire “methylome” and “hydroxymethylome” remain less than optimal.

The protocol for bisulphite conversion to detect methylation remains the “gold standard” for DNA methylation analysis. This method is generally followed by PCR analysis for single nucleotide resolution to determine methylation across the DNA molecule. According to Dr. Pradhan, “.. bisulphite conversion does not distinguish 5mC and 5hmC,”

Recently we found an enzyme, a unique DNA modification-dependent restriction endonuclease, AbaSI, which can

  • decode the hydryoxmethylome of the mammalian genome.

You easily can find out where the hydroxymethyl regions are.”

AbaSI, recognizes 5-glucosylatedmethylcytosine (5gmC) with high specificity when compared to 5mC and 5hmC, and

  • cleaves at narrow range of distances away from the recognized modified cytosine.

By mapping the cleaved ends, the exact 5hmC location can, the investigators reported, be determined.

Dr. Pradhan and his colleagues at NEB; the Department of Biochemistry, Emory University School of Medicine, Atlanta; and the New England Biolabs Shanghai R&D Center described use of this technique in a paper published in Cell Reports this month, in which they described high-resolution enzymatic mapping of genomic hydroxymethylcytosine in mouse ES cells.

In the current report, the authors used the enzyme technology for the genome-wide high-resolution hydroxymethylome, describing simple library construction even with a low amount of input DNA (50 ng) and the ability to readily detect 5hmC sites with low occupancy.

As a result of their studies, they propose that

factors affecting the local 5mC accessibility to TET enzymes play important roles in the 5hmC deposition

  • including include chromatin compaction, nucleosome positioning, or TF binding.
  •  the regularly oscillating 5hmC profile around the CTCF-binding sites, suggests 5hmC ‘‘writers’’ may be sensitive to the nucleosomal environment.
  • some transiently stable 5hmCs may indicate a poised epigenetic state or demethylation intermediate, whereas others may suggest a locally accessible chromosomal environment for the TET enzymatic apparatus.

“We were able to do complete mapping in mouse embryonic cells and are pleased about what this enzyme can do and how it works,” Dr. Pradhan said.

And the availability of novel tools that make analysis of the methylome and hypomethylome more accessible will move the field of epigenetic analysis forward and potentially novel biomarkers for cellular development, differentiation, and disease.

Patricia Fitzpatrick Dimond, Ph.D. (pdimond@genengnews.com), is technical editor at Genetic Engineering & Biotechnology News.

Epigenetic Regulation of ADME-Related Genes: Focus on Drug Metabolism and Transport

Published: Sep 23, 2013

Epigenetic regulation of gene expression refers to heritable factors that are functionally relevant genomic modifications but that do not involve changes in DNA sequence.

Examples of such modifications include

  • DNA methylation, histone modifications, noncoding RNAs, and chromatin architecture.

Epigenetic modifications are crucial for

packaging and interpreting the genome, and they have fundamental functions in regulating gene expression and activity under the influence of physiologic and environmental factors.

In this issue of Drug Metabolism and Disposition, a series of articles is presented to demonstrate the role of epigenetic factors in regulating

  • the expression of genes involved in drug absorption, distribution, metabolism, and excretion in organ development, tissue-specific gene expression, sexual dimorphism, and in the adaptive response to xenobiotic exposure, both therapeutic and toxic.

The articles also demonstrate that, in addition to genetic polymorphisms, epigenetics may also contribute to wide inter-individual variations in drug metabolism and transport. Identification of functionally relevant epigenetic biomarkers in human specimens has the potential to improve prediction of drug responses based on patient’s epigenetic profiles.

http://www.technologynetworks.com/Metabolomics/news.aspx?ID=157804

This study is published online in Drug Metabolism and Disposition

Part 8.  Pictorial Maps

 Prediction of intracellular metabolic states from extracellular metabolomic data

MK Aurich, G Paglia, Ottar Rolfsson, S Hrafnsdottir, M Magnusdottir, MM Stefaniak, BØ Palsson, RMT Fleming &

Ines Thiele

Metabolomics Aug 14, 2014;

http://dx.doi.org:/10.1007/s11306-014-0721-3

http://link.springer.com/article/10.1007/s11306-014-0721-3/fulltext.html#Sec1

http://link.springer.com/static-content/images/404/art%253A10.1007%252Fs11306-014-0721-3/MediaObjects/11306_2014_721_Fig1_HTML.gif

Metabolic models can provide a mechanistic framework

  • to analyze information-rich omics data sets, and are
  • increasingly being used to investigate metabolic alternations in human diseases.

An expression of the altered metabolic pathway utilization is the selection of metabolites consumed and released by cells. However, methods for the

  • inference of intracellular metabolic states from extracellular measurements in the context of metabolic models remain underdeveloped compared to methods for other omics data.

Herein, we describe a workflow for such an integrative analysis

  • emphasizing on extracellular metabolomics data.

We demonstrate,

  • using the lymphoblastic leukemia cell lines Molt-4 and CCRF-CEM,

how our methods can reveal differences in cell metabolism. Our models explain metabolite uptake and secretion by predicting

  • a more glycolytic phenotype for the CCRF-CEM model and
  • a more oxidative phenotype for the Molt-4 model,
  • which was supported by our experimental data.

Gene expression analysis revealed altered expression of gene products at

  • key regulatory steps in those central metabolic pathways, and

literature query emphasized the role of these genes in cancer metabolism.

Moreover, in silico gene knock-outs identified unique

  •  control points for each cell line model, e.g., phosphoglycerate dehydrogenase for the Molt-4 model.

Thus, our workflow is well suited to the characterization of cellular metabolic traits based on

  • -extracellular metabolomic data, and it allows the integration of multiple omics data sets
  • into a cohesive picture based on a defined model context.

Keywords Constraint-based modeling _ Metabolomics _ Multi-omics _ Metabolic network _ Transcriptomics

1 Introduction

Modern high-throughput techniques have increased the pace of biological data generation. Also referred to as the ‘‘omics avalanche’’, this wealth of data provides great opportunities for metabolic discovery. Omics data sets

  • contain a snapshot of almost the entire repertoire of mRNA, protein, or metabolites at a given time point or

under a particular set of experimental conditions. Because of the high complexity of the data sets,

  • computational modeling is essential for their integrative analysis.

Currently, such data analysis is a bottleneck in the research process and methods are needed to facilitate the use of these data sets, e.g., through meta-analysis of data available in public databases [e.g., the human protein atlas (Uhlen et al. 2010) or the gene expression omnibus (Barrett et al.  2011)], and to increase the accessibility of valuable information for the biomedical research community.

Constraint-based modeling and analysis (COBRA) is

  • a computational approach that has been successfully used to
  • investigate and engineer microbial metabolism through the prediction of steady-states (Durot et al.2009).

The basis of COBRA is network reconstruction: networks are assembled in a bottom-up fashion based on

  • genomic data and extensive
  • organism-specific information from the literature.

Metabolic reconstructions capture information on the

  • known biochemical transformations taking place in a target organism
  • to generate a biochemical, genetic and genomic knowledge base (Reed et al. 2006).

Once assembled, a

  • metabolic reconstruction can be converted into a mathematical model (Thiele and Palsson 2010), and
  • model properties can be interrogated using a great variety of methods (Schellenberger et al. 2011).

The ability of COBRA models

  • to represent genotype–phenotype and environment–phenotype relationships arises
  • through the imposition of constraints, which
  • limit the system to a subset of possible network states (Lewis et al. 2012).

Currently, COBRA models exist for more than 100 organisms, including humans (Duarte et al. 2007; Thiele et al. 2013).

Since the first human metabolic reconstruction was described [Recon 1 (Duarte et al. 2007)],

  • biomedical applications of COBRA have increased (Bordbar and Palsson 2012).

One way to contextualize networks is to

  • define their system boundaries according to the metabolic states of the system, e.g., disease or dietary regimes.

The consequences of the applied constraints can

  • then be assessed for the entire network (Sahoo and Thiele 2013).

Additionally, omics data sets have frequently been used

  • to generate cell-type or condition-specific metabolic models.

Models exist for specific cell types, such as

  1. enterocytes (Sahoo and Thiele2013),
  2. macrophages (Bordbar et al. 2010),
  3. adipocytes (Mardinoglu et al. 2013),
  4. even multi-cell assemblies that represent the interactions of brain cells (Lewis et al. 2010).

All of these cell type specific models, except the enterocyte reconstruction

  • were generated based on omics data sets.

Cell-type-specific models have been used to study

  • diverse human disease conditions.

For example, an adipocyte model was generated using

  • transcriptomic, proteomic, and metabolomics data.

This model was subsequently used to investigate metabolic alternations in adipocytes

  • that would allow for the stratification of obese patients (Mardinoglu et al. 2013).

The biomedical applications of COBRA have been

  1. cancer metabolism (Jerby and Ruppin, 2012).
  2. predicting drug targets (Folger et al. 2011; Jerby et al. 2012).

A cancer model was generated using

  • multiple gene expression data sets and subsequently used
  • to predict synthetic lethal gene pairs as potential drug targets
  • selective for the cancer model, but non-toxic to the global model (Recon 1),

a consequence of the reduced redundancy in the cancer specific model (Folger et al. 2011).

In a follow up study, lethal synergy between FH and enzymes of the heme metabolic pathway

  • were experimentally validated and resolved the mechanism by which FH deficient cells,
    e.g., in renal-cell cancer cells survive a non-functional TCA cycle (Frezza et al. 2011).

Contextualized models, which contain only the subset of reactions active in a particular tissue (or cell-) type,

  • can be generated in different ways (Becker and Palsson, 2008; Jerby et al. 2010).

However, the existing algorithms mainly consider

  • gene expression and proteomic data
  • to define the reaction sets that comprise the contextualized metabolic models.

These subset of reactions are usually defined

  • based on the expression or absence of expression of the genes or proteins (present and absent calls),
  • or inferred from expression values or differential gene expression.

Comprehensive reviews of the methods are available (Blazier and Papin, 2012; Hyduke et al. 2013). Only the compilation of a large set of omics data sets

  • can result in a tissue (or cell-type) specific metabolic model, whereas

the representation of one particular experimental condition is achieved

  • through the integration of omics data set generated from one experiment only (condition-specific cell line model).

Recently, metabolomic data sets have become more comprehensive and

  • using these data sets allow direct determination of the metabolic network components (the metabolites).

Additionally, metabolomics has proven to be stable, relatively inexpensive, and highly reproducible (Antonucci et al. 2012). These factors make metabolomic data sets particularly valuable for

  • interrogation of metabolic phenotypes.

Thus, the integration of these data sets is now an active field of research (Li et al. 2013; Mo et al. 2009; Paglia et al. 2012b; Schmidt et al. 2013).

Generally, metabolomic data can be incorporated into metabolic networks as

  • qualitative, quantitative, and thermodynamic constraints (Fleming et al. 2009; Mo et al. 2009).

Mo et al. used metabolites detected in the

  • spent medium of yeast cells to determine intracellular flux states through a sampling analysis (Mo et al. 2009),
  • which allowed unbiased interrogation of the possible network states (Schellenberger and Palsson 2009) and
  • prediction of internal pathway use.
Modes of transcriptional regulation during the YMC

Modes of transcriptional regulation during the YMC

Such analyses have also been used to reveal the effects of

  1. enzymopathies on red blood cells (Price et al. 2004),
  2. to study effects of diet on diabetes (Thiele et al. 2005) and
  3. to define macrophage metabolic states (Bordbar et al. 2010).

This type of analysis is available as a function in the COBRA toolbox (Schellenberger et al. 2011).

In this study, we established a workflow

  • for the generation and analysis of condition-specific metabolic cell line models
  • that can facilitate the interpretation of metabolomic data.

Our modeling yields meaningful predictions regarding

  • metabolic differences between two lymphoblastic leukemia cell lines (Fig. 1A).

Fig. 1

metabol leukem cell lines11306_2014_721_Fig1_HTML

metabol leukem cell lines11306_2014_721_Fig1_HTML

A Combined experimental and computational pipeline to study human metabolism.

  1. Experimental work and omics data analysis steps precede computational modeling.
  2. Model predictions are validated based on targeted experimental data.
  3. Metabolomic and transcriptomic data are used for model refinement and submodel extraction.
  4. Functional analysis methods are used to characterize the metabolism of the cell-line models and compare it to additional experimental data.
  5. The validated models are subsequently used for the prediction of drug targets.

B Uptake and secretion pattern of model metabolites. All metabolite uptakes and secretions that were mapped during model generation are shown.

  • Metabolite uptakes are depicted on the left, and
  • secreted metabolites are shown on the right.
  1. A number of metabolite exchanges mapped to the model were unique to one cell line.
  2. Differences between cell lines were used to set quantitative constraints for the sampling analysis.

C Statistics about the cell line-specific network generation.

D Quantitative constraints.

For the sampling analysis, an additional set of constraints was imposed on the cell line specific models,

  • emphasizing the differences in metabolite uptake and secretion between cell lines.

Higher uptake of a metabolite was allowed

  • in the model of the cell line that consumed more of the metabolite in vitro, whereas
  • the supply was restricted for the model with lower in vitro uptake.

This was done by establishing the same ratio between the models bounds as detected in vitro.

X denotes the factor (slope ratio) that distinguishes the bounds, and

  • which was individual for each metabolite.

(a) The uptake of a metabolite could be x times higher in CCRF-CEM cells,

(b) the metabolite uptake could be x times higher in Molt-4,

(c) metabolite secretion could be x times higher in CCRF-CEM, or

(d) metabolite secretion could be x times higher in Molt-4 cells.LOD limit of detection.

The consequence of the adjustment was, in case of uptake, that one model was constrained to a lower metabolite uptake (A, B), and the difference depended on the ratio detected in vitro. In case of secretion, one model

  • had to secrete more of the metabolite, and again
  • the difference depended on the experimental difference detected between the cell lines

2 Results

We set up a pipeline that could be used to infer intracellular metabolic states

  • from semi-quantitative data regarding metabolites exchanged between cells and their environment.

Our pipeline combined the following four steps:

  1. data acquisition,
  2. data analysis,
  3. metabolic modeling and
  4. experimental validation of the model predictions (Fig. 1A).

We demonstrated the pipeline and the predictive potential to predict metabolic alternations in diseases such as cancer based on

^two lymphoblastic leukemia cell lines.

The resulting Molt-4 and CCRF-CEM condition-specific cell line models could explain

^  metabolite uptake and secretion
^  by predicting the distinct utilization of central metabolic pathways by the two cell lines.
^  the CCRF-CEM model resembled more a glycolytic, commonly referred to as ‘Warburg’ phenotype,
^  our model predicted a more respiratory phenotype for the Molt-4 model.

We found these predictions to be in agreement with measured gene expression differences

  • at key regulatory steps in the central metabolic pathways, and they were also
  • consistent with additional experimental data regarding the energy and redox states of the cells.

After a brief discussion of the data generation and analysis steps, the results derived from model generation and analysis will be described in detail.

2.1 Pipeline for generation of condition-specific metabolic cell line models

integration of exometabolomic (EM) data

integration of exometabolomic (EM) data

2.1.1 Generation of experimental data

We monitored the growth and viability of lymphoblastic leukemia cell lines in serum-free medium (File S2, Fig. S1). Multiple omics data sets were derived from these cells.Extracellular metabolomics (exo-metabolomic) data,

integration of exometabolomic (EM) data

integration of exometabolomic (EM) data

^  comprising measurements of the metabolites in the spent medium of the cell cultures (Paglia et al. 2012a),
^ were collected along with transcriptomic data, and these data sets were used to construct the models.

2.1.4 Condition-specific models for CCRF-CEM and Molt-4 cells

To determine whether we had obtained two distinct models, we evaluated the reactions, metabolites, and genes of the two models. Both the Molt-4 and CCRF-CEM models contained approximately half of the reactions and metabolites present in the global model (Fig. 1C). They were very similar to each other in terms of their reactions, metabolites, and genes (File S1, Table S5A–C).

(1) The Molt-4 model contained seven reactions that were not present in the CCRF-CEM model (Co-A biosynthesis pathway and exchange reactions).
(2) The CCRF-CEM contained 31 unique reactions (arginine and proline metabolism, vitamin B6 metabolism, fatty acid activation, transport, and exchange reactions).
(3) There were 2 and 15 unique metabolites in the Molt-4 and CCRF-CEM models, respectively (File S1, Table S5B).
(4) Approximately three quarters of the global model genes remained in the condition-specific cell line models (Fig. 1C).
(5) The Molt-4 model contained 15 unique genes, and the CCRF-CEM model had 4 unique genes (File S1, Table S5C).
(6) Both models lacked NADH dehydrogenase (complex I of the electron transport chain—ETC), which was determined by the absence of expression of a mandatory subunit (NDUFB3, Entrez gene ID 4709).

Rather, the ETC was fueled by FADH2 originating from succinate dehydrogenase and from fatty acid oxidation, which through flavoprotein electron transfer

FADH2

FADH2

  • could contribute to the same ubiquinone pool as complex I and complex II (succinate dehydrogenase).

Despite their different in vitro growth rates (which differed by 11 %, see File S2, Fig. S1) and
^^^ differences in exo-metabolomic data (Fig. 1B) and transcriptomic data,
^^^ the internal networks were largely conserved in the two condition-specific cell line models.

2.1.5 Condition-specific cell line models predict distinct metabolic strategies

Despite the overall similarity of the metabolic models, differences in their cellular uptake and secretion patterns suggested distinct metabolic states in the two cell lines (Fig. 1B and see “Materials and methods” section for more detail). To interrogate the metabolic differences, we sampled the solution space of each model using an Artificial Centering Hit-and-Run (ACHR) sampler (Thiele et al. 2005). For this analysis, additional constraints were applied, emphasizing the quantitative differences in commonly uptaken and secreted metabolites. The maximum possible uptake and maximum possible secretion flux rates were reduced
^^^ according to the measured relative differences between the cell lines (Fig. 1D, see “Materials and methods” section).

We plotted the number of sample points containing a particular flux rate for each reaction. The resulting binned histograms can be understood as representing the probability that a particular reaction can have a certain flux value.

A comparison of the sample points obtained for the Molt-4 and CCRF-CEM models revealed

  • a considerable shift in the distributions, suggesting a higher utilization of glycolysis by the CCRF-CEM model
    (File S2, Fig. S2).

This result was further supported by differences in medians calculated from sampling points (File S1, Table S6).
The shift persisted throughout all reactions of the pathway and was induced by the higher glucose uptake (34 %) from the extracellular medium in CCRF-CEM cells.

The sampling median for glucose uptake was 34 % higher in the CCRF-CEM model than in Molt-4 model (File S2, Fig. S2).

The usage of the TCA cycle was also distinct in the two condition-specific cell-line models (Fig. 2). Interestingly,
the models used succinate dehydrogenase differently (Figs. 2, 3).

TCA_reactions

TCA_reactions

The Molt-4 model utilized an associated reaction to generate FADH2, whereas

  • in the CCRF-CEM model, the histogram was shifted in the opposite direction,
  • toward the generation of succinate.

Additionally, there was a higher efflux of citrate toward amino acid and lipid metabolism in the CCRF-CEM model (Fig. 2). There was higher flux through anaplerotic and cataplerotic reactions in the CCRF-CEM model than in the Molt-4 model (Fig. 2); these reactions include

(1) the efflux of citrate through ATP-citrate lyase,
(2) uptake of glutamine,
(3) generation of glutamate from glutamine,
(4) transamination of pyruvate and glutamate to alanine and to 2-oxoglutarate,
(5) secretion of nitrogen, and
(6) secretion of alanine.

energetics-of-cellular-respiration

energetics-of-cellular-respiration

The Molt-4 model showed higher utilization of oxidative phosphorylation (Fig. 3), again supported by
elevated median flux through ATP synthase (36 %) and other enzymes, which contributed to higher oxidative metabolism. The sampling analysis therefore revealed different usage of central metabolic pathways by the condition-specific models.

Fig. 2

Differences in the use of  the TCA cycle by the CCRF-CEM model (red) and the Molt-4 model (blue).

Differences in the use of the TCA cycle by the CCRF-CEM model (red) and the Molt-4 model (blue).

Differences in the use of the TCA cycle by the CCRF-CEM model (red) and the Molt-4 model (blue).

The table provides the median values of the sampling results. Negative values in histograms and in the table describe reversible reactions with flux in the reverse direction. There are multiple reversible reactions for the transformation of isocitrate and α-ketoglutarate, malate and fumarate, and succinyl-CoA and succinate. These reactions are unbounded, and therefore histograms are not shown. The details of participating cofactors have been removed.

Figure 3.

Molt-4 has higher median flux through ETC reactions II–IV 11306_2014_721_Fig3_HTML

Molt-4 has higher median flux through ETC reactions II–IV 11306_2014_721_Fig3_HTML

Atp ATP, cit citrate, adp ADP, pi phosphate, oaa oxaloacetate, accoa acetyl-CoA, coa coenzyme-A, icit isocitrate, αkg α-ketoglutarate, succ-coa succinyl-CoA, succ succinate, fumfumarate, mal malate, oxa oxaloacetate,
pyr pyruvate, lac lactate, ala alanine, gln glutamine, ETC electron transport chain

Ingenuity network analysis showing up (red) and downregulation (green) of miRNAs involved in PC and their target genes

Ingenuity network analysis showing up (red) and downregulation (green) of miRNAs involved in PC and their target genes

metabolic pathways 1476-4598-10-70-1

metabolic pathways 1476-4598-10-70-1

Metabolic Systems Research Team fig2

Metabolic Systems Research Team fig2

Metabolic control analysis of respiration in human cancer tissue. fphys-04-00151-g001

Metabolic control analysis of respiration in human cancer tissue. fphys-04-00151-g001

Metabolome Informatics Research fig1

Metabolome Informatics Research fig1

Modelling of Central Metabolism network3

Modelling of Central Metabolism network3

N. gaditana metabolic pathway map ncomms1688-f4

N. gaditana metabolic pathway map ncomms1688-f4

protein changes in biological mechanisms

protein changes in biological mechanisms

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Larry H. Bernstein, MD, FCAP, Curator

Leaders in Pharmaceutical Intelligence

Early discharge using single cardiac troponin and copeptin testing in patients
with suspected 
acute coronary syndrome (ACS): a randomized, controlled
clinical process study
M Mockel, J Searle, Christian Hamm, A Slagman, S Blankenberg, et al.
EurHeartJ Apr 2014.  http://dx.doi.org:/10.1093/eurheartj/ehu178

This randomized controlled trial (RCT) evaluated whether a process with single
combined testing of copeptin and troponin at admission in patients with low-to-
intermediate risk and suspected acute coronary syndrome (ACS)  does not lead to a higher proportion of major adverse cardiac events (MACE) than
the current standard process (non-inferiority design). After clinical work-up and  single combined testing of troponin and copeptin to rule-out AMI,  early  discharge
of low- to intermediate risk patients with suspected ACS seems to be safe and has
the potential to shorten length of stay in the ED.

Diagnostic accuracy of combined cardiac troponin and copeptin
assessment for 
early rule-out of myocardial infarction: a systematic
review and meta-analysis
T Raskovalova, R Twerenbold, PO Collinson, T Keller, H Bouvaist, et al.
http://acc.sagepub.com/content/3/1/18
EurHeartJ: Acute Cardiovascular Care 2014; 3(1): 18-27.
http://dx.doi.org:/10.1177/2048872613514015

This systematic review aimed to investigate the diagnostic accuracy of combined
cardiac troponin (cTn) and  copeptin assessment in comparison to cTn alone for
early rule-out of acute myocardial infarction (AMI).  In 15  studies totalling 8740
patients (prevalence of   AMI 16%), adding copeptin improved the sensitivity
of cTn assays  (from 0.87 to 0.96, p=0.003) at the expense of lower specificity
(from 0.84 to 0.56, p<0.001).

In 12 studies providing for 6988 patients without ST-segment elevation,
the summary sensitivity and specificity  estimates were 0.95 (95% CI 0.89 to
0.98) and 0.57 (95% CI 0.49 to   0.65) for the combined assessment of cTn
and copeptin. When a high-sensitivity cTnT assay was used in combination
with copeptin,  the summary sensitivity  and specificity estimates were 0.98
(95% CI 0.96 to 1.00) and 0.50 (95% CI 0.42 to 0.58). The result indicates
that  copeptin significantly improves baseline cTn sensitivity.

Diagnostic accuracy of copeptin sensitivity and specificity in patients with
suspected non-ST-elevation myocardial infarction with troponin I below
the 
99th centile at presentation
J Duchenne, S Mestres, N Dublanchet, N Combaret, G Marceau, et al.
BMJ Open 2014;4:e004449.
http://dx.doi.org:/10.1136/bmjopen-2013-004449

To our knowledge, our prospective multicentric study is the only one that includes
only patients with suspected non-ST-segment elevation myocardial infarction and
high-sensitive cardiac troponin I below  the 99th centile  at presentation to the
emergency department, to limit spectrum bias. Our study included only patients
with negative ultrasensitive troponin at admission. However, this is the only group
of patients for which a multimarker rule-out strategy could add diagnostic value.
Serial clinical, electrographical and biochemical investigations were performed at
admission and after 2, 4, 6 and 12 h. Hs-cTnT was measured using an assay with
Dimension VISTA, Siemens. Copeptin was measured by the BRAHMS copeptin-us
assay on the KRYPTOR Compact Plus system. The follow-up period was 90 days.

The final diagnosis was adjudicated blinded to copeptin result. During 12 months,
102 patients were analysed. Final diagnosis was NSTEMI for 7.8% (n=8), unstable
angina for 3.9% (n=4), cardiac but non coronary artery disease for 8.8% (n=9),
non-cardiac chest pain for 52% (n=53) and unknown for 27.5% (n=28).

There was no statistical difference for copeptin values between patients with
NSTEMI and others (respectively 5.5 pmol/L IQR (3.1–7.9) and 6.5 pmol/L IQR
(3.9–12.1), p=0.49). Only one patient with NSTEMI had a copeptin value
above the cut-off
 of 95th centile at admission.

In this study, copeptin does not add a diagnostic value at admission to ED for patients
with suspected acute coronary syndrome without ST-
segment elevation and with hs-cTnT below the 99th centile.

Can a Second Measurement of Copeptin Improve Acute Myocardial
Infarction Rule Out?

N Marston, K Shah, C Mueller, Sean-Xavier Neath, R Christenson, J McCord, et al.
J Am Coll Cardiol. 2014;63(12_S):A202.  Presentation Number: 1226-242
http://dx.doi.org:/10.1016/S0735-1097(14)60202-3

Of the 494 patients analyzed, 378 (76.5%) had a persistently elevated copeptin at 2
hours and 116 patients (23.5%) had a copeptin that fell below the cutoff of 14 pmol/l.
The AMI rate was 6.1% when the second copeptin was elevated compared to 0%
when the second copeptin was not (p=.006), yielding a sensitivity of 100%. This
strategy identified 23.5% of patients with an intermediate risk who could be ruled
out for AMI while still in the ED. In contrast, a second troponin measurement had a
sensitivity of 74%. A negative second copeptin drawn 2 hours after presentation
demonstrated 100% sensitivity for AMI, allowing for effective rule out in this
intermediate risk cohort. This strategy identified nearly 25% of intermediate risk
patients who could be considered for discharge.

 The role of copeptin as a diagnostic and prognostic biomarker for risk
stratification in the emergency department.
Nickel CH, Bingisser R and Morgenthaler NG
BMC Medicine 2012, 10:7
http://www.biomedcentral.com/1741-7015/10/7

Copeptin, the C-terminal part of the arginine vasopressin precursor peptide,
is a sensitive and stable surrogate marker for arginine vasopressin release.
Measurement of copeptin levels has been shown to be useful in a variety of
clinical scenarios, particularly as a prognostic marker in patients with acute
diseases such as lower respiratory tract infection, heart disease and stroke.

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Larry H Bernstein, MD, FCAP, Curator

Leaders in Pharmaceutical Innovation

High sensitivity c-Reactive Protein

High sensitivity C-reactive protein (hsCRP)
Author: Larry Bernstein, M.D.,  (see Reviewers/Authors page)
Revised: 12 December 2010, last major update December 2010
Copyright: (c) 2003-2010, PathologyOutlines.com, Inc.

http://dx.doi.org:/PathologyOutlines.com/cardiac

General
=========================================================================

  • hsCRP is an enhanced sensitivity C-reactive protein (CRP) immunoassay with a lowered measurement cutoff

Methodology
=========================================================================

  • Laser nephelometry

Indications
=========================================================================

  • In the JUPITER trial of apparently healthy persons without hyperlipidemia but with elevated
    high-sensitivity C-reactive protein levels, rosuvastatin significantly reduced the incidence of major
    cardiovascular events ( N Engl J Med 2008;359:2195)
  • This effect is thought to be due to the effect of statins on inflammation, which is detected by hsCRP
  • hsCRP assessment for cardiovascular disease in asymptomatic individuals seems to be most useful for
    those classified as intermediate risk on the basis of traditional risk factors (e.g. an NCEP-ATP III global
    risk score between 5% and 20%), and who do not already warrant chronic treatment with aspirin and a statin

Limitations
=========================================================================

  • Most useful for patients with intermediate risk for cardiovascular disease (Circ Cardiovasc Qual Outcomes
    2008;1:92, Ann Intern Med 2009;151:483)
  • For low risk patients, if their risk increases 3x (e.g. from 1% to 3%), their absolute cardiovascular risk
    is still low, so the hsCRP test has no practical value
  • High risk patients are candidates for chronic aspirin and lipid-lowering therapy regardless of their hsCRP test results
  • However, a recent study concludes that risk based statin treatment without hs-CRP testing is more cost-effective
    than hs-CRP screening, assuming that statins have good long-term safety and provide benefits among low-risk
    people with normal hs-CRP (Circulation 2010;122:1478)

Reference ranges
=========================================================================

  • Low risk: under 1 mg/L
  • Intermediate risk: 1-3 mg/L
  • High risk: > 3 mg/L

Additional references
=========================================================================

  • Wikipedia, Circulation 2006;113:2335, N Engl J Med 2001;344:1959

How to use C-reactive protein in acute coronary care
LM. Biasucci,W Koenig, J Mair, C Mueller, M Plebani, B Lindahl, N Rifai, P Venge, C Hamm, et al.
Eur Heart J  Nov 2013;  http://dx.doi.org:/10.1093/eurheartj/eht435

In patients with acute myocardial infarction (AMI), C-reactive protein increases within 4–6 h of symptoms,
peaks 2–4 days later, and returns to baseline after 7–10 days. Because of evidence that atherosclerosis
is an inflammatory disease, high-sensitivity C-reactive protein can be used as a biomarker of risk
in primary prevention
and in patients with known cardiovascular disease.
The upper reference limit is method-dependent but usually 8 mg/L for standard assays. The distribution of high-
sensitivity C-reactive protein concentrations is skewed in both genders with a 50th percentile of 1.5 mg/L (excluding
women on hormone replacement therapy).  C-reactive protein concentrations are increased by smoking, obesity, and
hormone replacement therapy and reduced by exercise, moderate alcohol drinking, and statin use. Correction for these
factors is essential in reference range studies.
C-reactive protein assays are not standardized. We recommend the use of third-generation high-sensitivity C-reactive
protein assays that combine features of standard and high-sensitivity C-reactive protein assays. Required assay precision
should be < 10% in the range of 3 and 10 mg/L.

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Larry H Bernstein, MD, Curator

Leaders in Pharmaceutical Intelligence

 

Natriuretic Peptides (BNP and Amino-terminal proBNP)

Author: Larry Bernstein, M.D.,
(see Reviewers/Authors page)
Revised: 12 December 2010, last major update December 2010
Copyright: (c) 2003-2010, PathologyOutlines.com, Inc.
http://dx.doi.org:/PathologyOutlines.com/cardiac

General
=========================================================================

  • Brain natriuretic peptide (BNP), now known as B-type natriuretic peptide (also BNP),
    is a 32 amino acid polypeptide secreted by the cardiac ventricles in response to
    excessive stretching of cardiomyocytes (Wikipedia)
  • BNP was originally identified in extracts of porcine brain, although in humans
    it is produced mainly in the cardiac ventricles
  • BNP is co-secreted with a 76 amino acid N-terminal fragment (NT-proBNP),
    which is biologically inactive Indications

=========================================================================

  • Evaluation of dyspneic patient with suspected congestive heart failure,
    regardless of renal function (J Am Coll Cardiol 2006;47:91)
  • B-type natriuretic peptide levels are higher in patients with congestive heart
    failure than in dyspnea from other causes (J Am Coll Cardiol 2002;39:202,
    N Engl J Med 2004;350:647)
  • NT-proBNP measurement is a valuable addition to standard clinical
    assessment for the identification and exclusion of acute CHF in the
    emergency department setting (Am J Cardiol 2005;95:9480)

Clinical features
=========================================================================

  • Reduces misdiagnosis of congestive heart failure, which occurs
    50% to 75% of the time
  • NT-proBNP is superior to BNP for predicting mortality and morbidity for heart
    failure (Clin Chem 2006;52:1528), and coexisting renal disease and heart failure
    (Clin Chem 2007;53:1511)

Reference ranges
=========================================================================

  • BNP levels below 100 pg/mL indicate no heart failure

Limitations
=========================================================================

  • Determination of endogenous BNP with the AxSYM assay using frozen
    plasma samples may not be valid after 1 day, but NT-proBNP as
    measured by the Elecsys assay may be stored at -20 degrees C for
    at least four months without a relevant loss of the immunoreactive
    analyte (Clin Chem Lab Med 2004;42:942)

Additional references
=========================================================================

  • Clin Chem 2007;53:1928, Am J Kidney Dis 2005;46:610,
    Hypertension 2005;46:118, Hypertension 2006;47:874,
    Eur J Heart Fail 2004;6:269

Natriuretic peptides for risk stratification of patients with acute
coronary
 syndromes  
M Galvani,  D Ferrini, F Ottani. Eur J Heart Fail 2004;  6: 327–333.
http://eurjhf.oxfordjournals.org

Both BNP and NT-proBNP possess several characteristics of the ideal biomarker,
showing independent and incremental prognostic value above traditional clinical,
electrocardiographic, and biochemical (particularly troponin) risk indicators. Specifically,
in ACS patients, BNP and NT-proBNP have powerful prognostic value both in patients
without a history of previous heart failure or without clinical or instrumental signs of
left ventricular dysfunction on admission or during hospital stay.

Our results show that the prognostic value of natriuretic peptides is similar:
(1) both at short- and long-term;
(2) when natriuretic peptides are measured at first patient contact or during hospital stay;
(3) for BNP or NT-proBNP; and
(4) in patients with ST elevation myocardial infarction or no ST elevation ACS.

 

Steady-State Levels of Troponin and Brain Natriuretic Peptide for Prediction
of Long-Term
 Outcome after Acute Heart Failure with or without Stage 3 to 4
Chronic Kidney Disease

Y Endo, S Kohsaka, T Nagai, K Koide, M Takahashi, et al.
Br J Med Med Res 2012; 2(4): 490-500.
http://dx.doi.org:/10.9734/BJMMR/2012/1384

The population was predominantly male (69.3%), and the mean age was 66.6±15.3 years.
Patients with higher BNP levels or detectable TnT had a worse prognosis (BNP45.0% vs.
18.8%, p<0.001; TnT 43.8% vs. 25.1%, p=0.002, respectively). The primary event rate
was additively worse among patients with both increased BNP levels and detectable TnT
compared to those with increased levels of BNP or detectable TnT alone (log-rank p<0.001).
A similar trend was observed in the subgroup of patients with CKD stage III–V (n=172).

The Effect of Correction of Mild Anemia in Severe, Resistant Congestive
Heart Failure
 Using Subcutaneous Erythropoietin and Intravenous Iron:
A Randomized Controlled Study

DS. Silverberg, D Wexler, D Sheps, M Blum, G Keren, et al.  JACC 2001; 37(7).
PII S0735-1097(01)01248-7  http://www.ncbi.nlm.nih.gov/pubmed/11401110

When anemia in CHF is treated with EPO and IV iron, a marked improvement in
cardiac and patient function is seen, associated with less hospitalization and renal
impairment and less need for diuretics. (J Am Coll Cardiol 2001;37:1775– 80)

 

 

 

Hemoglobin on NT proBNP

Hemoglobin on NT proBNP

 

 

 

 

What is the best approximation of reference normal for NT-proBNP?
Clinical levels for enhanced assessment
 of NT-proBNP (CLEAN) 

Larry H. Bernstein1*, Michael Y. Zions1,4, Mohammed E. Alam1,5, Salman A. Haq1,
John F. Heitner1, Stuart Zarich2, Bette Seamonds3 and Stanley Berger3
1New York Methodist Hospital, Brooklyn, NY; 2Bridgeport Hospital, Bridgeport, CT;
3Mercy Catholic Medical Center, Darby, Phila, PA;  4Touro College, &  5Medgar
Evers College, Brooklyn, NY
Journal of Medical Laboratory and Diagnosis 04/2011; 2:16-21.
http://www.academicjournals.org/jmld

The natriuretic peptides, B-type natriuretic peptide (BNP) and NT-proBNP that
have emerged as tools for diagnosing congestive heart failure (CHF) are affected
by age and renal insufficiency (RI).  NTproBNP is used in rejecting CHF and as a
marker of risk for patients with acute coronary syndromes. This observational study
was undertaken to evaluate the reference value for interpreting NT-proBNP
concentrations. The hypothesis is that increasing concentrations of NT-proBNP
are associated with the effects of multiple co-morbidities, not merely CHF,
resulting in altered volume status or myocardial filling pressures.

NT-proBNP was measured in a population with normal trans-thoracic echocardiograms
(TTE) and free of anemia or renal impairment. Exclusion conditions were the following
co-morbidities:

  • anemia as defined by WHO,
  • atrial fibrillation (AF),
  • elevated troponin T exceeding 0.070 mg/dl,
  • systolic or diastolic blood pressure exceeding 140 and 90 respectively,
  • ejection fraction less than 45%,
  • left ventricular hypertrophy (LVH),
  • left ventricular wall relaxation impairment, and
  • renal insufficiency (RI) defined by creatinine clearance < 60ml/min using
    the MDRD formula .

Study participants were seen in acute care for symptoms of shortness of breath
suspicious for CHF requiring evaluation with cardiac NTproBNP assay. The median
NT-proBNP for patients under 50 years is 60.5 pg/ml with an upper limit of 462 pg/ml,
and for patients over 50 years the median was 272.8 pg/ml with an upper limit of
998.2 pg/ml.
We suggest that NT-proBNP levels can be more accurately interpreted only after
removal of the major co-morbidities that affect an increase in this  peptide in serum.
The PRIDE study guidelines should be applied until presence or absence of
comorbidities is diagnosed. With no comorbidities, the reference range for normal
over 50 years of age remains steady at ~1000 pg/ml. The effect shown in previous
papers likely is due to increasing concurrent comorbidity with age.

NT-proBNP profile of combined population taken from 3 sites and donors.

Age    Under 50 years 50-69 years 70 and over
NT-proBNP
N 
Mean   
95% CI of Mean
Median   
95% CI of median
2.5-97.5 percentile   
25-75 percentile
209
35.9
29.8-43.3
27.6
24.8-33.6
5.0-1364
14.9-55.8
126
182.4
132.1-251.9
142.3
92.3-219.0
10.8-11604
42.1-565
82
611.7
425.2-880.1
564.2
419.7-1007.7
28.8-14242
210.2-2062

 

We observe the following changes with respect to NTproBNP and age:

(i) Sharp increase in NT-proBNP at over age 50

(ii) Increase in NT-proBNP at 7% per decade over 50

(iii) Decrease in eGFR at 4% per decade over 50

(iv) Slope of NT-proBNP increase with age is related to proportion of patients with
eGFR less than 90

(v) NT-proBNP increase can be delayed or accelerated based on disease
comorbidities

NT-proBNP sensitivity and specificity with RI prevalence

NT-proBNP sensitivity and specificity with RI prevalence

Figure 1. Plot of NT-proBNP sensitivity and specificity with RI prevalence.
GFRe scale: 0, > 120; 1, 90- 119; 2, 60-89; 3, 40-59; 4, 15-39; 5, under 15 ml/min.

NKF staging by GFRe interval and NT-proBNP (CHF removed).

NKF staging by GFRe interval and NT-proBNP (CHF removed).

 

Figure 2  plots the mean and 95% CI of NTproBNP (CHF removed) by the National Kidney Foundation
staging for eGFR interval (eGFR scale: 0, > 120; 1, 90 to 119;2, 60 to 89; 3, 40 to 59; 4, 15 to 39; 5,
under 15 ml/min). We created a new variable to minimize the effects of age and eGFR variability by
correcting these large effects in the whole sample population.

Adjustment of the NT-proBNP for eGFR and for age over 50 differences. We have
carried out a normalization to adjust for both eGFR and for age over 50:

(i) Take Log of NT-proBNP and multiply by 1000

(ii) Divide the result by eGFR (using MDRD9 or Cockroft Gault10)

(iii) Compare results for age under 50, 50-70, and over 70 years

(iv) Adjust to age under 50 years by multiplying by 0.66 and 0.56.

The equation does not require weight because the results are reported normalized
to 1.73 m2 body surface area, which is an accepted average adult surface area.

 

fn.log-NT-proBNP vs age

fn.log-NT-proBNP vs age

Figure 3.  Plot of 1000*log (NT-proBNP)/GFR vs age at  eGFR over 90  and 60 ml/min

scatterplot and regression line with centroid and confidence interval for fn.logNTproBNP vs age

scatterplot and regression line with centroid and confidence interval for fn.logNTproBNP vs age

Figure 4. Superimposed scatterplot and regression line with centroid and
confidence interval for 1000*log(NT-proBNP)/eGFR vs age (anemia removed)
at eGFR over 40 and 90 ml/min. (Black: eGFR > 90, Blue:  eGFR > 40)  

 

Ref Range NTpro NKLogNTpro

Ref Range NTpro NKLogNTpro

 

Reference range for NT-proBNP before and after adjusting

 

Amino-Terminal Pro-Brain Natriuretic Peptide, Renal Function, and
Outcomes in Acute Heart Failure
RRJ. van Kimmenade,  JL. Januzzi, JR,  AL. Baggish, et al. JACC 2006; 48(8).: 1621-7.

We sought to study the individual and integrative role of amino-terminal pro-brain natriuretic
peptide (NT-proBNP) and parameters of renal function for prognosis in heart failure. The
combination of NT-proBNP with measures of renal function better predicts short-term outcome
in acute heart failure than either parameter alone. Among heart failure patients, the objective
parameter of NT-proBNP seems more useful to delineate the “cardiorenal syndrome” than the
previous criteria of a clinical diagnosis of heart failure.

 

NT-proBNP testing for diagnosis and short-term prognosis in acute destabilized
heart failure: an international pooled analysis of 1256 patients The International
Collaborative of NT-proBNP Study
Januzzi, R van Kimmenade, J Lainchbury, A Bayes-Genis, J Ordonez-Llanos, et al.
Eur Heart J 2006; 27, 330–337. http://dx.doi.org:/10.1093/eurheartj/ehi631

Differences in NT-proBNP levels among 1256 patients with and without acute HF and the relationship
between NT-proBNPlevels and HF symptomswere examined.Optimal cut-points for diagnosis and
prognosis were identified and verified using bootstrapping and multi-variable logistic regression techniques.

Seven hundred and twenty subjects (57.3%) had acute HF, whose median NT-proBNP was considerably
higher than those without (4639 vs. 108 pg/mL, P < 0.001), and levels of NT-proBNP correlated with HF
symptom severity (P < 0.008). An optimal strategy to identify acute HF was to use age-related cut-points
of 450, 900, and 1800 pg/mL for ages < 50, 50–75, and  > 75, which yielded 90% sensitivity and 84% specificity
for acute HF. An age-independent cut-point of 300 pg/mL had 98% negative predictive value to exclude acute
HF. Among those with acute HF, a presenting NT-proBNP concentration > 5180 pg/mL was strongly predictive
of death by 76 days [odds ratio = 5.2, 95% confidence interval (CI) =2.2 – 8.1, P < 0.001].

Effect of B-type natriuretic peptide-guided treatment of chronic heart failure on total mortality
and hospitalization: an individual patient meta-analysis
RW. Troughton, CM. Frampton, HP Brunner-La Rocca, M Pfisterer, LW.M. Eurlings, et al.
Eur Heart J Mar 2014; 35, 1559–1567.
http://dx.doi.org:/10.1093/eurheartj/ehu090

We sought to perform an individual patient data meta-analysis to evaluate the effect of NP-guided treatment
of heart failure on all-cause mortality.  The survival benefit from NP-guided therapy was seen in younger (< 75
years) patients [0.62 (0.45–0.85); P = 0.004] but not older (≥75 years) patients [0.98 (0.75–1.27); P = 0.96].
Hospitalization due to heart failure [0.80 (0.67–0.94); P = 0.009] or cardiovascular disease [0.82 (0.67–0.99);
P = 0.048] was significantly lower in NP-guided patients with no heterogeneity between studies and no interaction
with age or LVEF.

 

Diagnostic and prognostic evaluation of left ventricular systolic heart failure by plasma N-terminal
pro-brain natriuretic peptide concentrations in a large sample of the general population

BA Groenning, I Raymond, PR Hildebrandt, JC Nilsson, M Baumann, F Pedersen.
Heart 2004; 90:297–303.  http://dx.doi.org:/10.1136/hrt.2003.026021

Value of NT-proBNP in evaluating patients with symptoms of heart failure and impaired left ventricular (LV) systolic
function; prognostic value of NT-proBNP for mortality and hospital admissions. In 38 (5.6%) participants LV ejection
fraction (LVEF) was ( 40%. NT-proBNP identified patients with symptoms of heart failure and LVEF ( 40% with a
sensitivity of 0.92, a specificity of 0.86, AUC of 0.94.  NT-proBNP was the strongest independent predictor of mortality
(hazard ratio (HR) = 5.70, p , 0.0001), hospital admissions for heart failure (HR = 13.83, p , 0.0001), and other cardiac
admissions (HR = 3.69, p , 0.0001). Mortality (26 v 6, p = 0.0003), heart failure admissions (18 v 2, p = 0.0002), and
admissions for other cardiac causes (44 v 13, p , 0.0001) were significantly higher in patients with NTproBNP above the
study median (32.5 pmol/l).

 

Testing for BNP and NT-proBNP in the Diagnosis and Prognosis of Heart Failure
Evidence Report/Technology Assessment – Number 142. Agency for Healthcare Research and Quality.
Prepared by: McMaster University Evidence-based Practice Center, Hamilton, ON, Canada
C Balion, PL. Santaguida, S Hill, A Worster, M McQueen, et al.
http://archive.ahrq.gov/downloads/pub/evidence/pdf/bnp/bnp.pdf

Question 1: What are the determinants of both BNP and NT-proBNP?
Question 2a: What are the clinical performance characteristics of both BNP and NTproBNP
measurement in patients with symptoms suggestive of HF or with known HF?
Question 2b: Does measurement of BNP or NT-proBNP add independent diagnostic information
to the traditional diagnostic measures of HF in patients with suggestive HF?
Question 3a: Do BNP or NT-proBNP levels predict cardiac events in populations at risk of CAD,
with diagnosed CAD and HF?
Question 3b: What are the screening performance characteristics of BNP or NT-proBNP in
general asymptomatic populations?
Question 4: Can BNP or NT-proBNP measurement be used to monitor response to therapy?        

Diagnosis: In all settings both BNP and NT-proBNP show good diagnostic properties as a rule out test for HF.
Prognosis: BNP and NT-proBNP are consistent independent predictors of mortality and other cardiac composite
endpoints for populations with risk of CAD, diagnosed CAD, and diagnosed HF. There is insufficient evidence to
determine the value of B-type natriuretic peptides for screening of HF.
Monitoring Treatment: There is insufficient evidence to demonstrate that BNP and NT-proBNP levels
show change in response to therapies to manage stable chronic HF patients.

Guide-IT Trial

Biomarker-Guided HF Therapy: Is It Cost-Effective
www.medscape.org/viewarticle/764686_transcript

Jan 29, 2013 – Uploaded by DCLRI
Michael Felker, MD, MHS
Associate Professor in the Division of Cardiology
Duke University Medical Center
www.youtube.com/watch?v=AW0480EE2kw

GUIDE-IT will last five years and involve approximately 45 trial sites in the United States. The first group of
patients will be enrolled by the end of 2012.

The trial tests NT-proBNP guided therapy with a COMPANION diagnostic biomarker used to optimize already
available and effective therapies for heart failure. It may identify  patients who will benefit from intensified therapy,
and  who would not have been known using only signs and symptoms of heart failure as it is currently the practice.
The NT-proBNP biomarker would enable doctors to create personalized treatment plans for patients to substantially
reduce mortality and morbidity

 Risk stratification in acute heart failure: Rationale and design of the
STRATIFY and DECIDE studies 

SP. Collins, CJ. Lindsell, CA. Jenkins, FE. Harrell, et al.
Am Heart J 2012;164:825-34.
http://dx.doi.org/10.1016/j.ahj.2012.07.033

Two studies (STRATIFY and DECIDE) have been funded by the National Heart Lung and Blood Institute with
the goal of developing prediction rules to facilitate early decision making in AHF. Using prospectively gathered
evaluation and treatment data from the acute setting (STRATIFY) and early inpatient stay (DECIDE), rules will
be generated to predict risk for death and serious complications.
A rigorous analysis plan has been developed to construct the prediction rules that will maximally extract both the
statistical and clinical properties of every data element. Upon completion of this study we will subsequently externally
test the prediction rules in a heterogeneous patient cohort.

N-terminal pro-B-type natriuretic peptide and the prediction of primary cardiovascular
events: results from 15-year follow-up of WOSCOPS

P Welsh, O Doolin, P Willeit, C Packard, P Macfarlane, S Cobbe, et al.
Eur Heart J Aug  2012.
http://dx.doi.org:/10.1093/eurheartj/ehs239

To test whether N-terminal pro-B-type natriuretic peptide (NT-proBNP) was independently associated with, and
improved the prediction of, cardiovascular disease (CVD) in a primary prevention cohort. N-terminal pro-B-type
natriuretic peptide predicts CVD events in men without clinical evidence of CHD, angina, or history of stroke,
and appears related more strongly to the risk for fatal events.
NT-proBNP was associated with an increased risk of all CVD [HR: 1.17 (95% CI: 1.11–1.23) per standard deviation
increase in log NT-proBNP] after adjustment for classical and clinical cardiovascular risk factors plus C-reactive protein.
N-terminal pro-B-type natriuretic peptide was more strongly related to the rsk of fatal [HR: 1.34 (95% CI: 1.19–1.52)]
than non-fatal CVD [HR: 1.17 (95% CI: 1.10–1.24)] (P = 0.022). The addition of NT-proBNP to traditional risk factors
improved the C-index (+0.013; P = 0.001). The continuous net reclassification index improved with the addition of NT-
proBNP by 19.8% (95% CI: 13.6–25.9%) compared with 9.8% (95% CI: 4.2–15.6%) with the addition of C-reactive protein.

 

Utility of B-Natriuretic Peptide in Detecting Diastolic Dysfunction: Comparison With
Doppler Velocity Recordings
E Lubien, A DeMaria, P Krishnaswamy, P Clopton, J Koon…A Maisel.
http://circ.ahajournals.org/content/105/5/595
Circulation. 2002;105:595-601
http://dx.doi.org:/10.1161/hc0502.103010

Although Doppler echocardiography has been used to identify abnormal left ventricular (LV) diastolic filling dynamics,
inherent limitations suggest the need for additional measures of diastolic dysfunction. Because data suggest that B-natriuretic
peptide (BNP) partially reflects ventricular pressure, we hypothesized that BNP levels could predict diastolic abnormalities
in patients with normal systolic function. A rapid assay for BNP can reliably detect the presence of diastolic abnormalities
on echocardiography. In  patients with normal systolic function, elevated BNP levels and diastolic filling abnormalities might
help to reinforce the diagnosis diastolic dysfunction

Association of common variants in NPPA and NPPB with circulating natriuretic
peptides and blood pressure.
C Newton-Cheh, MG Larson, RS Vasan, D Levy, KD Bloch, et al.
Nat Genet. 2009 Mar; 41(3): 348–353.
http://dx.doi.org:/10.1038/ng.328

We examined the association of common variants at the NPPA-NPPB locus with circulating concentrations of the
natriuretic peptides, which have blood pressure–lowering properties. In 29,717 individuals, the alleles of rs5068
and rs198358 that showed association with increased circulating natriuretic peptide concentrations were also found
to be associated with lower systolic (P = 2 ×10−6 and 6 × 10−5, respectively) and diastolic blood pressure (P = 1 × 10−6
and 5 × 10−5), as well as reduced odds of hypertension (OR = 0.85, 95% CI = 0.79–0.92, P = 4 × 10−5; OR = 0.90, 95%
CI = 0.85–0.95, P = 2 × 10−4, respectively).

2013 ACC/AHA Guideline on the Assessment of Cardiovascular Risk
DC. Goff, Jr, DM. Lloyd-Jones, G Bennett, S Coady, RB. D’Agostino, Sr, et al.
Circulation. 2013;  http://circ.ahajournals.org/content/early/2013/11/11/01.cir.0000437741.48606.98.citation
http://dx.doi.org:/10.1161/01.cir.0000437741.48606.98

The ACC and AHA have collaborated with the National Heart, Lung, and Blood Institute (NHLBI) and stakeholder
and professional organizations to develop clinical practice guidelines for assessment of CV risk, lifestyle modifications
to reduce CV risk, and management of blood cholesterol, overweight and obesity in adults.
Although the Task Force led the final development of these prevention guidelines, they differ from other ACC/AHA
guidelines. First, as opposed to an extensive compendium of clinical information, these documents are significantly
more limited in scope and focus on selected CQs in each topic, based on the highest quality evidence available.
Recommendations were derived from randomized trials, meta-analyses, and observational studies evaluated for quality,
and were not formulated when sufficient evidence was not available. Second, the text accompanying each recommendation
is succinct, summarizing the evidence for each question. Third, the format of the recommendations differs from other
ACC/AHA guidelines. Each recommendation has been mapped from the NHLBI grading format to the ACC/AHA Class
of Recommendation/Level of Evidence (COR/LOE) construct (Table 1) and is expressed in both formats.

 

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Why did this occur? The matter of Individual Actions Undermining Trust, The Patent Dilemma and The Value of a Clinical Trials

Why did this occur? The matter of Individual Actions Undermining Trust, The Patent Dilemma and The Value of a Clinical Trials

Reporter and Curator: Larry H. Bernstein, MD, FCAP

 

he large amount of funding tied to continued research and support of postdoctoral fellows leads one to ask how following the money can lead to discredited work in th elite scientific community.

Moreover, the pressure to publish in prestigious journals with high impact factors is a road to academic promotion.  In the last twenty years, it is unusual to find submissions for review with less than 6-8 authors, with the statement that all contributed to the work.  These factors can’t be discounted outright, but it is easy for work to fall through the cracks when a key investigator has over 200 publications and holds tenure in a great research environment.  But that is where we find ourselves today.

There is another issue that comes up, which is also related to the issue of carrying out research, and then protecting the work for commercialization.  It is more complicated in the sense that it is necessary to determine whether there is prior art, and then there is the possibility that after the cost of filing patent and a 6 year delay in obtaining protection, there is as great a cost in bringing the patent to finasl production.

I.  Individual actions undermining trust.

II. The patent dilemma.

III. The value of a clinical trial.

IV. The value contributions of RAP physicians
(radiologists, anesthesiologists, and pathologists – the last for discussion)
Those who maintain and inform the integrity of medical and surgical decisions

 

I. Top heart lab comes under fire

Kelly Servick

Science 18 July 2014: Vol. 345 no. 6194 p. 254 DOI: 10.1126/science.345.6194.25

 

In the study of cardiac regeneration, Piero Anversa is among the heavy hitters. His research into the heart’s repair mechanisms helped kick-start the field of cardiac cell therapy (see main story). After more than 4 decades of research and 350 papers, he heads a lab at Harvard Medical School’s Brigham and Women’s Hospital (BWH) in Boston that has more than $6 million in active grant funding from the National Institutes of Health (NIH). He is also an outspoken voice in a field full of disagreement.

So when an ongoing BWH investigation of the lab came to light earlier this year, Anversa’s colleagues were transfixed. “Reactions in the field run the gamut from disbelief to vindication,” says Mark Sussman, a cardiovascular researcher at San Diego State University in California who has collaborated with Anversa. By Sussman’s account, Anversa’s reputation for “pushing the envelope” and “challenging existing dogma” has generated some criticism. Others, however, say that the disputes run deeper—to doubts about a cell therapy his lab has developed and about the group’s scientific integrity. Anversa told Science he was unable to comment during the investigation.

“People are talking about this all the time—at every scientific meeting I go to,” says Charles Murry, a cardiovascular pathologist at the University of Washington, Seattle. “It’s of grave concern to people in the field, but it’s been frustrating,” because no information is available about BWH’s investigation. BWH would not comment for this article, other than to say that it addresses concerns about its researchers confidentially.

In April, however, the journal Circulation agreed to Harvard’s request to retract a 2012 paper on which Anversa is a corresponding author, citing “compromised” data. The Lancet also issued an “Expression of Concern” about a 2011 paper reporting results from a clinical trial, known as SCIPIO, on which Anversa collaborated. According to a notice from the journal, two supplemental figures are at issue.

For some, Anversa’s status has earned him the benefit of the doubt. “Obviously, this is very disconcerting,” says Timothy Kamp, a cardiologist at the University of Wisconsin, Madison, but “I would be surprised if it was an implication of a whole career of research.”

Throughout that career, Anversa has argued that the heart is a prolific, lifelong factory for new muscle cells. Most now accept the view that the adult heart can regenerate muscle, but many have sparred with Anversa over his high estimates for the rate of this turnover, which he maintained in the retracted Circulation paper.

Anversa’s group also pioneered a method of separating cells with potential regenerative abilities from other cardiac tissue based on the presence of a protein called c-kit. After publishing evidence that these cardiac c-kit+cells spur new muscle growth in rodent hearts, the group collaborated in the SCIPIO trial to inject them into patients with heart failure. In The Lancet, the scientists reported that the therapy was safe and showed modest ability to strengthen the heart—evidence that many found intriguing and provocative. Roberto Bolli, the cardiologist whose group at the University of Louisville in Kentucky ran the SCIPIO trial, plans to test c-kit+ cells in further clinical trials as part of the NIH-funded Cardiovascular Cell Therapy Research Network.

But others have been unable to reproduce the dramatic effects Anversa saw in animals, and some have questioned whether these cells really have stem cell–like properties. In May, a group led by Jeffery Molkentin, a molecular biologist at Cincinnati Children’s Hospital Medical Center in Ohio, published a paper in Nature tracing the genetic lineage of c-kit+ cells that reside in the heart. He concluded that although they did make new muscle cells, the number is “astonishingly low” and likely not enough to contribute to the repair of damaged hearts. Still, Molkentin says that he “believe[s] in their therapeutic potential” and that he and Anversa have discussed collaborating.

Now, an anonymous blogger claims that problems in the Anversa lab go beyond controversial findings. In a letter published on the blog Retraction Watch on 30 May, a former research fellow in the Anversa lab described a lab culture focused on protecting the c-kit+ cell hypothesis: “[A]ll data that did not point to the ‘truth’ of the hypothesis were considered wrong,” the person wrote. But another former lab member offers a different perspective. “I had a great experience,” says Federica Limana, a cardiovascular disease researcher at IRCCS San Raffaele Pisana in Rome who spent 2 years of her Ph.D. work with the group in 1999 and 2000, as it was beginning to investigate c-kit+ cells. “In that period, there was no such pressure” to produce any particular result, she says.

Accusations about the lab’s integrity, combined with continued silence from BWH, are deeply troubling for scientists who have staked their research on theories that Anversa helped pioneer. Some have criticized BWH for requesting retractions in the midst of an investigation. “Scientific reputations and careers hang in the balance,” Sussman says, “so everyone should wait until all facts are clearly and fully disclosed.”

 

II.  Trolling Along: Recent Commotion About Patent Trolls

July 17, 2014

PriceWaterhouseCoopers recently released a study about 2014 Patent Litigation. PwC’s ultimate conclusion was that case volume increased vastly and damages continue a general decline, but what’s making headlines everywhere is that “patent trolls” now account for 67% of all new patent lawsuits (see, e.g., Washington Post and Fast Company).

Surprisingly, looking at PwC’s study, the word “troll” is not to be found. So, with regard to patent trolls, what does this study really mean for companies, patent owners and casual onlookers?

First of all, who are these trolls?

“Patent Troll” is a label applied to patent owners who do not make or manufacture a product, or offer a service. Patent trolls live (and die) by suing others for allegedly practicing an invention that is claimed by their patents.

The politically correct term is Non-practicing Entity (NPE). PwC solely uses the term NPE, which it defines as an entity that does not have the capability to design, manufacture, or distribute products with features protected by the patent.

So, what’s so bad about them?

The common impression of an NPEs is a business venture looking to collect and monetize assets (i.e., patents). In the most basic strategy, an NPE typically buys patents with broad claims that cover a wide variety of technologies and markets, and then sues a large group of alleged patent infringers in the hope to collect a licensing royalty or a settlement. NPEs typically don’t want to spend money on a trial unless they have to, and one tactic uses settlements with smaller businesses to build a “war chest” for potential suits with larger companies.

NPEs initiating a lawsuit can be viewed positively, such as a just defense of the lowly inventor who sold his patent to someone (with deeper pockets) who could fund the litigation to protect the inventor’s hard work against a mega-conglomerate who ripped off his idea.

Or NPE litigation can be seen negatively, such as an attorney’s demand letter on behalf of an anonymous shell corporation to shake down dozens of five-figure settlements from all the local small businesses that have ever used a fax machine.

NPEs can waste a company’s valuable time and resources with lawsuits, yet also bring value to their patent portfolios by energizing a patent sales and licensing market. There are unscrupulous NPEs, but it’s hardly the black and white situation that some media outlets are depicting.

What did PwC say about trolls?

Well, the PwC study looked at the success rates and awards of patent litigation decisions. One conclusion is that damages awards for NPEs averaged more than triple those for practicing entities over the last four years. We’ll come back to this statistic.

Another key observation is that NPEs have been successful 25% of the time overall, versus 35% for practicing entities. This makes sense because of the burden of proof the NPEs carry as a plaintiff at trial and the relative lack of success for NPEs at summary judgment. However, PwC’s report states that both types of entities win about two-thirds of their trials.

But what about this “67% of all patent trials are initiated by trolls” discussion?

The 67% number comes from the RPX Corporation’s litigation report (produced January 2014) that quantified the percentage of NPE cases filed in 2013 as 67%, compared to 64% in 2012, 47% in 2011, 30% in 2010 and 28% in 2009.

PwC refers to the RPX statistics to accentuate that this new study indicates that only 20% ofdecisions in 2013 involved NPE-filed cases, so the general conclusion would be that NPE cases tend to settle or be dismissed prior to a court’s decision. Admittedly, this is indicative of the prevalent “spray and pray” strategy where NPEs prefer to collect many settlement checks from several “targets” and avoid the courtroom.

In this study, who else is an NPE?

If someone were looking to dramatize the role of “trolls,” the name can be thrown around liberally (and hurtfully) to anyone who owns and asserts a patent without offering a product or a service. For instance, colleges and universities fall under the NPE umbrella as their research and development often ends with a series of published papers rather than a marketable product on an assembly line.

In fact, PwC distinguishes universities and non-profits from companies and individuals within their NPE analysis, with only about 5% of the NPE cases from 1995 to 2013 being attributed to universities and non-profits. Almost 50% of the NPE cases are attributed to an “individual,” who could be the listed inventor for the patent or a third-party assignee.

The word “troll” is obviously a derogatory term used to connote greed and hiding (under a bridge), but the term has adopted a newer, meme-like status as trolls are currently depicted as lacking any contribution to society and merely living off of others’ misfortunes and fears. [Three Billy Goats Gruff]. This is not always the truth with NPEs (e.g., universities).

No one wants to be called a troll—especially in front of a jury—so we’ve even recently seen courts bar defendants from referring to NPEs as such colorful terms as a “corporate shell,” “bounty hunter,” “privateer,” or someone “playing the lawsuit lottery.” [Judge Koh Bans Use Of Term ” Patent Troll” In Apple Jury Trial]

Regardless of the portrayal of an NPE, most people in the patent world distinguish the “trolls” by the strength of the patent, merits of the alleged infringement and their behavior upon notification. Often these are expressed as “frivolity” of the case and “gamesmanship” of the attorneys. Courts are able to punish plaintiffs who bring frivolous claims against a party and state bar associations are tasked with monitoring the ethics of attorneys. The USPTO is tasked with working to strengthen the quality of patents.

What’s the take-away from this study regarding NPEs?

The study focuses on patent litigation that produced a decision, therefore the most important and relevant conclusion is that, over the last four years, average damages awards for NPEs are more than triple the damages for practicing entities. Everything else in these articles, such as the initiation of litigation by NPEs, settlement percentages, and the general behavior of patent trolls is pure inference beyond the scope of the study.

This may sound sympathetic to trolls, but keep in mind that the study highlights that NPEs have more than triple the damages on average compared to practicing entities and it is meant to shock the reader a bit. One explanation for this is that NPEs are in the best position to choose the patents they want to assert and choose the targets they wish to sue—especially when the NPE is willing to ride that patent all the way to the end of a long, expensive trial. Sometimes settling is not an option. Chart 2b indicates that the disparity in the damages awarded to NPEs relative to practicing entities has always been big (since 2000), but perhaps going from two-fold from 2000 – 2009 to three times as much in the past 4 years indicates that NPEs are improving at finding patents and/or picking battles to take all the way to a court decision. More than anything, this seems to reflect the growth in the concept of patents as a business asset.

The PwC report is chock full of interesting patterns and trends of litigation results, so it’s a shame that the 67% number makes the headlines—far more interesting are the charts comparing success rates by 4-year periods (Chart 6b) or success rates for NPEs and practicing entities in front of a jury verusin front of a bench (Chart 6c), as well as other tables that reveal statistics for specific districts of the federal courts. Even the stats that look at the success rates of each type of NPE are telling because the reader sees that universities and non-profits have a higher success rate than non-practicing companies or individuals.

What do we do about the trolls?

The White House has recently called for Congress to do something about the trolls as horror stories of scams and shake-downs are shared. A bill was gaining momentum in the Senate, when Senator Leahy took it off the agenda in early July. That bill had miraculously passed 325-91 in the House and President Obama was willing to sign it if the Senate were to pass it. The bill was opposed by trial attorneys, universities, and bio-pharmaceutical businesses who felt as though the law would severely inhibit everyone’s access to the courts in order to hinder just the trolls. Regardless, most people think that the sitting Congressmen merely wanted a “win” prior to the mid-term elections and that patent reform is unlikely to reappear until next term.

In the meantime, the Supreme Court has recently reiterated rules concerning attorney fee-shifting on frivolous patent cases, as well as clarifying the validity of software patents. Time will tell if these changes have any effects on the damages awards that PwC’s study examined or even if they cause a chilling of the number of patent lawsuit filings.

Furthermore, new ways to challenge the validity of asserted patents have been initiated via the America Invents Act. For example, the Inter Partes Review (IPR) has yielded frightening preliminary statistics as to slowing, if not killing, patents that have been asserted in a suit. While these administrative trials are not cheap, many view these new tools at the Patent Trial and Appeals Board as anti-troll measures. It will be interesting to watch how the USPTO implements these procedures in the near future, especially while former Google counsel, Acting Director Michelle K. Lee, oversees the office.

In the private sector, Silicon Valley has recently seen a handful of tech companies come together as the License on Transfer Network, a group hoping to disarm the “Patent Assertion Entities.” Joining the LOT Network comes via an agreement that creates a license for use of a patent by anyone in the LOT network once that patent is sold. The thought is that the NPEs who consider purchasing patents from companies in the LOT Network will have fewer companies to sue since the license to the other active LOT participants will have triggered upon the transfer and, thus, the NPE will not be as inclined to “troll.” For instance, if a member-company such as Google were to sell a patent to a non-member company and an NPE bought that patent, the NPE would not be able to sue any members of the LOT Network with that patent.

Other notes

NPEs are only as evil as the people who run them—that being said, there are plenty of horror stories of small businesses receiving phantom demand letters that threaten a patent infringement suit without identifying themselves or the patent. This is an out-and-out scam and a plague on society that results in wasted time and resource, and inevitably higher prices on the consumer end.

It is a sin and a shame that patent rights can be misused in scams and shake-downs of businesses around us, but there is a reason that U.S. courts are so often used to defend patent rights. The PwC study, at minimum, reflects the high stakes of the patent market and perhaps the fragility. Nevertheless, merely monitoring the courts may not keep the trolls at bay.

I’d love to hear your thoughts.

*This is provided for informational purposes only, and does not constitute legal or financial advice. The information expressed is subject to change at any time and should be checked for completeness, accuracy and current applicability. For advice, consult a suitably licensed attorney or patent agent.

 

III. Large-scale analysis finds majority of clinical trials don’t provide meaningful evidence

Ineffective Treatments, Medical Ethics • Tags: Center for Drug Evaluation and Research, Clinical trial, CTTI, Duke University Hospital, FDA, Food and Drug Administration, National Institutes of Health, United States National Library of Medicine

04 May 2012

DURHAM, N.C.— The largest comprehensive analysis of ClinicalTrials.gov finds that clinical trials are falling short of producing high-quality evidence needed to guide medical decision-making. The analysis, published today in JAMA, found the majority of clinical trials is small, and there are significant differences among methodical approaches, including randomizing, blinding and the use of data monitoring committees.

“Our analysis raises questions about the best methods for generating evidence, as well as the capacity of the clinical trials enterprise to supply sufficient amounts of high quality evidence to ensure confidence in guideline recommendations,” said Robert Califf, M.D., first author of the paper, vice chancellor for clinical research at Duke University Medical Center, and director of the Duke Translational Medicine Institute.

The analysis was conducted by the Clinical Trials Transformation Initiative (CTTI), a public private partnership founded by the Food and Drug Administration (FDA) and Duke. It extends the usability of the data in ClinicalTrials.gov for research by placing the data through September 27, 2010 into a database structured to facilitate aggregate analysis. This publically accessible database facilitates the assessment of the clinical trials enterprise in a more comprehensive manner than ever before and enables the identification of trends by study type.

 

The National Library of Medicine (NLM), a part of the National Institutes of Health, developed and manages ClinicalTrials.gov. This site maintains a registry of past, current, and planned clinical research studies.

“Since 2007, the Food and Drug Administration Amendment Act has required registration of clinical trials, and the expanded scope and rigor of trial registration policies internationally is producing more complete data from around the world,” stated Deborah Zarin, MD, director, ClinicalTrials.gov, and assistant director for clinical research projects, NLM. “We have amassed over 120,000 registered clinical trials. This rich repository of data has a lot to say about the national and international research portfolio.”

This CTTI project was a collaborative effort by informaticians, statisticians and project managers from NLM, FDA and Duke. CTTI comprises more than 60 member organizations with the goal of identifying practices that will improve the quality and efficiency of clinical trials.

“Since the ClinicalTrials.gov registry contains studies sponsored by multiple entities, including government, industry, foundations and universities, CTTI leaders recognized that it might be a valuable source for benchmarking the state of the clinical trials enterprise,” stated Judith Kramer, MD, executive director of CTTI.

The project goal was to produce an easily accessible database incorporating advances in informatics to permit a detailed characterization of the body of clinical research and facilitate analysis of groups of studies by therapeutic areas, by type of sponsor, by number of participants and by many other parameters.

“Analysis of the entire portfolio will enable the many entities in the clinical trials enterprise to examine their practices in comparison with others,” says Califf. “For example, 96% of clinical trials have ≤1000 participants, and 62% have ≤ 100. While there are many excellent small clinical trials, these studies will not be able to inform patients, doctors and consumers about the choices they must make to prevent and treat disease.”

The analysis showed heterogeneity in median trial size, with cardiovascular trials tending to be twice as large as those in oncology and trials in mental health falling in the middle. It also showed major differences in the use of randomization, blinding, and data monitoring committees, critical issues often used to judge the quality of evidence for medical decisions in clinical practice guidelines and systematic overviews.

“These results reinforce the importance of exploration, analysis and inspection of our clinical trials enterprise,” said Rachel Behrman Sherman, MD, associate director for the Office of Medical Policy at the FDA’s Center for Drug Evaluation and Research. “Generation of this evidence will contribute to our understanding of the number of studies in different phases of research, the therapeutic areas, and ways we can improve data collection about clinical trials, eventually improving the quality of clinical trials.”

Related articles

 

IV.  Lawmakers urge CMS to extend MU hardship exemption for pathologists

 

Eighty-nine members of Congress have asked the Centers for Medicare & Medicaid Services to give pathologists a break and extend the hardship exemption they currently enjoy for all of Stage 3 of the Meaningful Use program.In the letter–dated July 10 and addressed to CMS Administrator Marilyn Tavenner–the lawmakers point out that CMS had recognized in its 2012 final rule implementing Stage 2 of the program that it was difficult for pathologists to meet the Meaningful Use requirements and granted a one year exception for 2015, the first year that penalties will be imposed. They now are asking that the exception be expanded to include the full five-year maximum allowed under the American Recovery and Reinvestment Act.

“Pathologists have limited direct contact with patients and do not operate in EHRs,” the letter states. “Instead, pathologists use sophisticated computerized laboratory information systems (LISs) to support the work of analyzing patient specimens and generating test results. These LISs exchange laboratory and pathology data with EHRs.”

Interestingly, the lawmakers’ exemption request is only on behalf of pathologists, even though CMS had granted the one-year hardship exception to pathologists, radiologists and anesthesiologists.

Rep. Tom Price (R-Ga.), one of the members spearheading the letter, had also introduced a bill (H.R. 1309) in March 2013 that would exclude pathologists from the incentives and penalties of the Meaningful Use program. The bill, which has 31 cosponsors, is currently sitting in committee. That bill also does not include relief for radiologists or anesthesiologists.

CMS has provided some flexibility about the hardship exceptions in the past, most recently by allowing providers to apply for one due to EHR vendor delays in upgrading to Stage 2 of the program.

However, CMS also noted in the 2012 rule granting the one-year exception that it was granting the exception in large part because of the then-current lack of health information exchange and that “physicians in these three specialties should not expect that this exception will continue indefinitely, nor should they expect that we will grant the exception for the full 5-year period permitted by statute.”

To learn more:
– read the letter (.pdf)

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USPTO Guidance On Patentable Subject Matter

USPTO Guidance On Patentable Subject Matter

Curator and Reporter: Larry H Bernstein, MD, FCAP

LH Bernstein

LH Bernstein

 

 

 

 

 

 

Revised 4 July, 2014

http://pharmaceuticalintelligence.com/2014/07/03/uspto-guidance-on-patentable-subject-matter

 

I came across a few recent articles on the subject of US Patent Office guidance on patentability as well as on Supreme Court ruling on claims. I filed several patents on clinical laboratory methods early in my career upon the recommendation of my brother-in-law, now deceased.  Years later, after both brother-in-law and patent attorney are no longer alive, I look back and ask what I have learned over $100,000 later, with many trips to the USPTO, opportunities not taken, and a one year provisional patent behind me.

My conclusion is

(1) that patents are for the protection of the innovator, who might realize legal protection, but the cost and the time investment can well exceed the cost of startup and building a small startup enterprize, that would be the next step.

(2) The other thing to consider is the capability of the lawyer or firm that represents you.  A patent that is well done can be expected to take 5-7 years to go through with due diligence.   I would not expect it to be done well by a university with many other competing demands. I might be wrong in this respect, as the climate has changed, and research universities have sprouted engines for change.  Experienced and productive faculty are encouraged or allowed to form their own such entities.

(3) The emergence of Big Data, computational biology, and very large data warehouses for data use and integration has changed the landscape. The resources required for an individual to pursue research along these lines is quite beyond an individuals sole capacity to successfully pursue without outside funding.  In addition, the changed designated requirement of first to publish has muddied the water.

Of course, one can propose without anything published in the public domain. That makes it possible for corporate entities to file thousands of patents, whether there is actual validation or not at the time of filing.  It would be a quite trying experience for anyone to pursue in the USPTO without some litigation over ownership of patent rights. At this stage of of technology development, I have come to realize that the organization of research, peer review, and archiving of data is still at a stage where some of the best systems avalailable for storing and accessing data still comes considerably short of what is needed for the most complex tasks, even though improvements have come at an exponential pace.

I shall not comment on the contested views held by physicists, chemists, biologists, and economists over the completeness of guiding theories strongly held.  Only history will tell.  Beliefs can hold a strong sway, and have many times held us back.

I am not an expert on legal matters, but it is incomprehensible to me that issues concerning technology innovation can be adjudicated in the Supreme Court, as has occurred in recent years. I have postgraduate degrees in  Medicine, Developmental Anatomy, and post-medical training in pathology and laboratory medicine, as well as experience in analytical and research biochemistry.  It is beyond the competencies expected for these type of cases to come before the Supreme Court, or even to the Federal District Courts, as we see with increasing frequency,  as this has occurred with respect to the development and application of the human genome.

I’m not sure that the developments can be resolved for the public good without a more full development of an open-access system of publishing. Now I present some recent publication about, or published by the USPTO.

DR ANTHONY MELVIN CRASTO

Dr. Melvin Castro - Organic Chemistry and New Drug Development

Dr. Melvin Castro – Organic Chemistry and New Drug Development

 

 

 

 

 

 

 

 

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USPTO Guidance On Patentable Subject Matter: Impediment to Biotech Innovation

Joanna T. Brougher, David A. Fazzolare J Commercial Biotechnology 2014 20(3):Brougher

jcbiotech-patents

jcbiotech-patents

 

 

 

 

 

 

 

 

 

 

 

Abstract In June 2013, the U.S. Supreme Court issued a unanimous decision upending more than three decades worth of established patent practice when it ruled that isolated gene sequences are no longer patentable subject matter under 35 U.S.C. Section 101.While many practitioners in the field believed that the USPTO would interpret the decision narrowly, the USPTO actually expanded the scope of the decision when it issued its guidelines for determining whether an invention satisfies Section 101.

The guidelines were met with intense backlash with many arguing that they unnecessarily expanded the scope of the Supreme Court cases in a way that could unduly restrict the scope of patentable subject matter, weaken the U.S. patent system, and create a disincentive to innovation. By undermining patentable subject matter in this way, the guidelines may end up harming not only the companies that patent medical innovations, but also the patients who need medical care.  This article examines the guidelines and their impact on various technologies.

Keywords:   patent, patentable subject matter, Myriad, Mayo, USPTO guidelines

Full Text: PDF

References

35 U.S.C. Section 101 states “Whoever invents or discovers any new and useful process, machine, manufacture, or composition of matter, or any new and useful improvement thereof, may obtain a patent therefor, subject to the conditions and requirements of this title.

” Prometheus Laboratories, Inc. v. Mayo Collaborative Services, 566 U.S. ___ (2012)

Association for Molecular Pathology et al., v. Myriad Genetics, Inc., 569 U.S. ___ (2013).

Parke-Davis & Co. v. H.K. Mulford Co., 189 F. 95, 103 (C.C.S.D.N.Y. 1911)

USPTO. Guidance For Determining Subject Matter Eligibility Of Claims Reciting Or Involving Laws of Nature, Natural Phenomena, & Natural Products.

http://www.uspto.gov/patents/law/exam/myriad-mayo_guidance.pdf

Funk Brothers Seed Co. v. Kalo Inoculant Co., 333 U.S. 127, 131 (1948)

USPTO. Guidance For Determining Subject Matter Eligibility Of Claims Reciting Or Involving Laws of Nature, Natural Phenomena, & Natural Products.

http://www.uspto.gov/patents/law/exam/myriad-mayo_guidance.pdf

Courtney C. Brinckerhoff, “The New USPTO Patent Eligibility Rejections Under Section 101.” PharmaPatentsBlog, published May 6, 2014, accessed http://www.pharmapatentsblog.com/2014/05/06/the-new-patent-eligibility-rejections-section-101/

Courtney C. Brinckerhoff, “The New USPTO Patent Eligibility Rejections Under Section 101.” PharmaPatentsBlog, published May 6, 2014, accessed http://www.pharmapatentsblog.com/2014/05/06/the-new-patent-eligibility-rejections-section-101/

DOI: http://dx.doi.org/10.5912/jcb664

 

Science 4 July 2014; 345 (6192): pp. 14-15  DOI: http://dx.doi.org/10.1126/science.345.6192.14
  • IN DEPTH

INTELLECTUAL PROPERTY

Biotech feels a chill from changing U.S. patent rules

A 2013 Supreme Court decision that barred human gene patents is scrambling patenting policies.

PHOTO: MLADEN ANTONOV/AFP/GETTY IMAGES

A year after the U.S. Supreme Court issued a landmark ruling that human genes cannot be patented, the biotech industry is struggling to adapt to a landscape in which inventions derived from nature are increasingly hard to patent. It is also pushing back against follow-on policies proposed by the U.S. Patent and Trademark Office (USPTO) to guide examiners deciding whether an invention is too close to a natural product to deserve patent protection. Those policies reach far beyond what the high court intended, biotech representatives say.

“Everything we took for granted a few years ago is now changing, and it’s generating a bit of a scramble,” says patent attorney Damian Kotsis of Harness Dickey in Troy, Michigan, one of more than 15,000 people who gathered here last week for the Biotechnology Industry Organization’s (BIO’s) International Convention.

At the meeting, attorneys and executives fretted over the fate of patent applications for inventions involving naturally occurring products—including chemical compounds, antibodies, seeds, and vaccines—and traded stories of recent, unexpected rejections by USPTO. Industry leaders warned that the uncertainty could chill efforts to commercialize scientific discoveries made at universities and companies. Some plan to appeal the rejections in federal court.

USPTO officials, meanwhile, implored attendees to send them suggestions on how to clarify and improve its new policies on patenting natural products, and even announced that they were extending the deadline for public comment by a month. “Each and every one of you in this room has a moral duty … to provide written comments to the PTO,” patent lawyer and former USPTO Deputy Director Teresa Stanek Rea told one audience.

At the heart of the shake-up are two Supreme Court decisions: the ruling last year in Association for Molecular Pathology v. Myriad Genetics Inc. that human genes cannot be patented because they occur naturally (Science, 21 June 2013, p. 1387); and the 2012 Mayo v. Prometheus decision, which invalidated a patent on a method of measuring blood metabolites to determine drug doses because it relied on a “law of nature” (Science, 12 July 2013, p. 137).

Myriad and Mayo are already having a noticeable impact on patent decisions, according to a study released here. It examined about 1000 patent applications that included claims linked to natural products or laws of nature that USPTO reviewed between April 2011 and March 2014. Overall, examiners rejected about 40%; Myriad was the basis for rejecting about 23% of the applications, and Mayo about 35%, with some overlap, the authors concluded. That rejection rate would have been in the single digits just 5 years ago, asserted Hans Sauer, BIO’s intellectual property counsel, at a press conference. (There are no historical numbers for comparison.) The study was conducted by the news service Bloomberg BNA and the law firm Robins, Kaplan, Miller & Ciseri in Minneapolis, Minnesota.

USPTO is extending the decisions far beyond diagnostics and DNA?

The numbers suggest USPTO is extending the decisions far beyond diagnostics and DNA, attorneys say. Harness Dickey’s Kotsis, for example, says a client recently tried to patent a plant extract with therapeutic properties; it was different from anything in nature, Kotsis argued, because the inventor had altered the relative concentrations of key compounds to enhance its effect. Nope, decided USPTO, too close to nature.

In March, USPTO released draft guidance designed to help its examiners decide such questions, setting out 12 factors for them to weigh. For example, if an examiner deems a product “markedly different in structure” from anything in nature, that counts in its favor. But if it has a “high level of generality,” it gets dinged.

The draft has drawn extensive criticism. “I don’t think I’ve ever seen anything as complicated as this,” says Kevin Bastian, a patent attorney at Kilpatrick Townsend & Stockton in San Francisco, California. “I just can’t believe that this will be the standard.”

USPTO officials appear eager to fine-tune the draft guidance, but patent experts fear the Supreme Court decisions have made it hard to draw clear lines. “The Myriad decision is hopelessly contradictory and completely incoherent,” says Dan Burk, a law professor at the University of California, Irvine. “We know you can’t patent genetic sequences,” he adds, but “we don’t really know why.”

Get creative in using Draft Guidelines!

For now, Kostis says, applicants will have to get creative to reduce the chance of rejection. Rather than claim protection for a plant extract itself, for instance, an inventor could instead patent the steps for using it to treat patients. Other biotech attorneys may try to narrow their patent claims. But there’s a downside to that strategy, they note: Narrower patents can be harder to protect from infringement, making them less attractive to investors. Others plan to wait out the storm, predicting USPTO will ultimately rethink its guidance and ease the way for new patents.

 

Public comment period extended

USPTO has extended the deadline for public comment to 31 July, with no schedule for issuing final language. Regardless of the outcome, however, Stanek Rea warned a crowd of riled-up attorneys that, in the world of biopatents, “the easy days are gone.”

 

United States Patent and Trademark Office

Today we published and made electronically available a new edition of the Manual of Patent Examining Procedure (MPEP). Manual of Patent Examining Procedure uspto.gov http://www.uspto.gov/web/offices/pac/mpep/index.html Summary of Changes

PDF Title Page
PDF Foreword
PDF Introduction
PDF Table of Contents
PDF Chapter 600 –
PDF   Parts, Form, and Content of Application Chapter 700 –
PDF    Examination of Applications Chapter 800 –
PDF   Restriction in Applications Filed Under 35 U.S.C. 111; Double Patenting Chapter 900 –
PDF   Prior Art, Classification, and Search Chapter 1000 –
PDF  Matters Decided by Various U.S. Patent and Trademark Office Officials Chapter 1100 –
PDF   Statutory Invention Registration (SIR); Pre-Grant Publication (PGPub) and Preissuance Submissions Chapter 1200 –
PDF    Appeal Chapter 1300 –
PDF   Allowance and Issue Appendix L –
PDF   Patent Laws Appendix R –
PDF   Patent Rules Appendix P –
PDF   Paris Convention Subject Matter Index 
PDF Zipped version of the MPEP current revision in the PDF format.

Manual of Patent Examining Procedure (MPEP)Ninth Edition, March 2014

The USPTO continues to offer an online discussion tool for commenting on selected chapters of the Manual. To participate in the discussion and to contribute your ideas go to:
http://uspto-mpep.ideascale.com.

Manual of Patent Examining Procedure (MPEP) Ninth Edition, March 2014
The USPTO continues to offer an online discussion tool for commenting on selected chapters of the Manual. To participate in the discussion and to contribute your ideas go to: http://uspto-mpep.ideascale.com.

Note: For current fees, refer to the Current USPTO Fee Schedule.
Consolidated Laws – The patent laws in effect as of May 15, 2014. Consolidated Rules – The patent rules in effect as of May 15, 2014.  MPEP Archives (1948 – 2012)
Current MPEP: Searchable MPEP

The documents updated in the Ninth Edition of the MPEP, dated March 2014, include changes that became effective in November 2013 or earlier.
All of the documents have been updated for the Ninth Edition except Chapters 800, 900, 1000, 1300, 1700, 1800, 1900, 2000, 2300, 2400, 2500, and Appendix P.
More information about the changes and updates is available from the “Blue Page – Introduction” of the Searchable MPEP or from the “Summary of Changes” link to the HTML and PDF versions provided below. Discuss the Manual of Patent Examining Procedure (MPEP) Welcome to the MPEP discussion tool!

We have received many thoughtful ideas on Chapters 100-600 and 1800 of the MPEP as well as on how to improve the discussion site. Each and every idea submitted by you, the participants in this conversation, has been carefully reviewed by the Office, and many of these ideas have been implemented in the August 2012 revision of the MPEP and many will be implemented in future revisions of the MPEP. The August 2012 revision is the first version provided to the public in a web based searchable format. The new search tool is available at http://mpep.uspto.gov. We would like to thank everyone for participating in the discussion of the MPEP.

We have some great news! Chapters 1300, 1500, 1600 and 2400 of the MPEP are now available for discussion. Please submit any ideas and comments you may have on these chapters. Also, don’t forget to vote on ideas and comments submitted by other users. As before, our editorial staff will periodically be posting proposed new material for you to respond to, and in some cases will post responses to some of the submitted ideas and comments.Recently, we have received several comments concerning the Leahy-Smith America Invents Act (AIA). Please note that comments regarding the implementation of the AIA should be submitted to the USPTO via email t aia_implementation@uspto.gov or via postal mail, as indicated at the America Invents Act Web site. Additional information regarding the AIA is available at www.uspto.gov/americainventsact  We have also received several comments suggesting policy changes which have been routed to the appropriate offices for consideration. We really appreciate your thinking and recommendations!

FDA Guidance for Industry:Electronic Source Data in Clinical Investigations

Electronic Source Data

Electronic Source Data

 

 

 

 

 

 

 

The FDA published its new Guidance for Industry (GfI) – “Electronic Source Data in Clinical Investigations” in September 2013.
The Guidance defines the expectations of the FDA concerning electronic source data generated in the context of clinical trials. Find out more about this Guidance.
http://www.gmp-compliance.org/enews_4288_FDA%20Guidance%20for%20Industry%3A%20Electronic%20Source%20Data%20in%20Clinical%20Investigations
_8534,8457,8366,8308,Z-COVM_n.html

After more than 5 years and two draft versions, the final version of the Guidance for
Industry (GfI) – “Electronic Source Data in Clinical Investigations” was published in
September 2013. This new FDA Guidance defines the FDA’s expectations for sponsors,
CROs, investigators and other persons involved in the capture, review and retention of
electronic source data generated in the context of FDA-regulated clinical trials.In an
effort to encourage the modernization and increased efficiency of processes in clinical
trials, the FDA clearly supports the capture of electronic source data and emphasizes
the agency’s intention to support activities aimed at ensuring the reliability, quality,
integrity and traceability of this source data, from its electronic source to the electronic
submission of the data in the context of an authorization procedure. The Guidance
addresses aspects as data capture, data review and record retention. When the
computerized systems used in clinical trials are described, the FDA recommends
that the description not only focus on the intended use of the system, but also on
data protection measures and the flow of data across system components and
interfaces. In practice, the pharmaceutical industry needs to meet significant
requirements regarding organisation, planning, specification and verification of
computerized systems in the field of clinical trials. The FDA also mentions in the
Guidance that it does not intend to apply 21 CFR Part 11 to electronic health records
(EHR). Author: Oliver Herrmann Q-Infiity Source: http://www.fda.gov/downloads/Drugs/GuidanceComplianceRegulatoryInformation/
Guidances/UCM328691.pdf
Webinar: https://collaboration.fda.gov/p89r92dh8wc

 

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