ArticleResearch and practice in thrombosis and haemostasis2025
Effect of DNA methylation on inhibitor development in people with hemophilia A treated with FVIII concentrates.
Article in Research and practice in thrombosis and haemostasis, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers, 1 of them a synthesis that pooled it.
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Who cites it
2 citing papers in PubMed, 1 synthesis or guideline pooled it.
- TNF‑α Gene Polymorphisms as Determinants of Alloantibody Emergence in Hemophilia: A Systematic Review and Meta-Analysis.Haemophilia : the official journal of the World Federation of HemophiliaPooled it
- Shaping hemophilia care: lessons and legacy of the SIPPET trial after 10 years.Research and practice in thrombosis and haemostasis · 2026Review
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Authors and funding
6 authors.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Background: Hemophilia A (HA) is a hereditary X-linked hemorrhagic disorder. Following the first treatment with exogenous factor (F)VIII, one-third of patients with severe HA develop anti-FVIII antibodies (inhibitors), which render treatment ineffective. Recent findings underlined the critical role of DNA methylation in several autoimmune diseases by altering gene expression profiles. This study was designed to evaluate potential differences in DNA methylation profiles of previously untreated patients (PUPs) who develop inhibitors against FVIII and those who do not, with the aim of identifying immune-regulatory genes that may contribute to the risk of inhibitor formation. Objectives: In this study, we aimed to understand whether CpG sites are differentially methylated in peripheral blood mononuclear cells (PBMCs) of PUPs and have a role in inhibitor development to better understand the biological pathways that lead to inhibitor development. Methods: A case-control study was performed using 45 inhibitor-positive and 67 inhibitor-negative PUPs from the Survey of Inhibitors in Plasma-Product Exposed Toddlers study cohort. Enrichment bisulfite sequencing was performed on DNA samples from PBMCs of HA patients and differentially methylated CpG sites (DMCs) were identified with bioinformatic approach. Results: Overall, information on 621,121 CpG sites was obtained. Two thousand seven hundred seventy-two sites were significantly differentially methylated (unadjusted Conclusion: DMCs were identified in PBMC samples from HA patients with inhibitors. However, our data could not confirm the role of these CpG sites in affecting immune-regulatory pathways.
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