Trial reportBlood2013
The polygenic nature of inhibitors in hemophilia A: results from the Hemophilia Inhibitor Genetics Study (HIGS) Combined Cohort.
Trial report in Blood, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT00166387 (Hemophilia Inhibitor Genetics Study), which is not on this map. Cited by 30 papers.
What it found
Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.
The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
Hemophilia Inhibitor Genetics Study (HIGS)
Who cites it
30 citing papers in PubMed, 103 citations in OpenAlex.
- Large deletions in theResearch and practice in thrombosis and haemostasis · 2025Article
- Immune System-Related Genetic Risk Factors for Inhibitory Antibody Development in Patients With Hemophilia: Reviewing an Old Problem From a New Perspective-A Narrative Review.Health science reports · 2025Article
- Marginal Zone B Cells Represent a Conserved Initiating Player in the Immune Response to Factor VIII in Hemophilia A Mice.Thrombosis and haemostasis · 2024Article
- Mutation detection and inhibitor analysis of 43 children with severe hemophilia A in a single center: three novel mutations.Indian journal of hematology & blood transfusion : an official journal of Indian Society of Hematology and Blood Transfusion · 2024Article
- Article
- Immunogenicity of Current and New Therapies for Hemophilia A.Pharmaceuticals (Basel, Switzerland) · 2022Review
- Article
- Genome-Wide Association Study and Gene-Based Analysis of Participants With Hemophilia A and Inhibitors in the My Life, Our Future Research Repository.Frontiers in medicine · 2022Article
- Neutralizing Antibodies Against Factor VIII Can Occur Through a Non-Germinal Center Pathway.Frontiers in immunology · 2022Article
- Haemophilia.Nature reviews. Disease primers · 2021Review
- FVIII Immunogenicity-Bioinformatic Approaches to Evaluate Inhibitor Risk in Non-severe Hemophilia A.Frontiers in immunology · 2020Review
- Removal of Mannose-Ending Glycan at AsnFrontiers in immunology · 2020Article
- Polymorphisms in the cytotoxic T lymphocyte-associated protein-4 immune regulatory gene and their impact on inhibitor development in patients with hemophilia A.The Journal of international medical research · 2019Article
- Role of factor VIII-binding capacity of endogenous von Willebrand factor in the development of factor VIII inhibitors in patients with severe hemophilia A.Haematologica · 2019Article
- Risk stratification integrating genetic data for factor VIII inhibitor development in patients with severe hemophilia A.PloS one · 2019Article
- Factor VIII: Perspectives on Immunogenicity and Tolerogenic Strategies.Frontiers in immunology · 2019Review
- Analyses of the FranceCoag cohort support differences in immunogenicity among one plasma-derived and two recombinant factor VIII brands in boys with severe hemophilia A.Haematologica · 2018Article
- Marginal zone B cells are critical to factor VIII inhibitor formation in mice with hemophilia A.Blood · 2017Article
- The importance of genetic factors for the development of arthropathy: a longitudinal study of children and adolescents with haemophilia A.Thrombosis and haemostasis · 2017Article
- Engineering less immunogenic and antigenic FVIII proteins.Cellular immunology · 2016Review
Corrections and comments
- Commented on by
Authors and funding
13 authors at 6 institutions in 2 countries.
Funding
Abstract
Studies of determinants of development of inhibitory Abs to factor VIII in people with hemophilia A indicate a complex process involving multiple factors. The Hemophilia Inhibitor Genetics Study (HIGS) Combined Cohort was formed to extend our understanding of the genetic background of risk. The study group contains 833 subjects from 3 independent cohorts: brother pairs and singletons with and without a history of inhibitors, as well as 104 brother pairs discordant for inhibitor status. Using an Illumina iSelect platform, 13 331 single-nucleotide polymorphisms from 1081 genes, primarily immune response and immune modifier genes, were typed. Each cohort was analyzed separately with results combined using a meta-analytic technique. After adjustment for potential confounders, 53 single-nucleotide polymorphisms were found to be significant predictors of inhibitor status using the criteria of odds ratios in the same direction in all cohorts or allowing for a 20% interval around an odds ratio = 1 in 1 of the 3 and significant in at least 2. Of the 53 markers, 13 had meta P < .001. Eight of the 53 were significant predictors among the discordant pairs. Results support the complexity of the immune response and encourage further research with the goal of understanding the pathways involved.
Indexed as
Identifiers
What OpenQuestion holds
Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.