ArticleResearch and practice in thrombosis and haemostasis2023
Determinants of successful immune tolerance induction in hemophilia A: systematic review and meta-analysis.
Article in Research and practice in thrombosis and haemostasis, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 17 papers, 1 of them a synthesis that pooled it.
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Who cites it
17 citing papers in PubMed, 1 synthesis or guideline pooled it, 18 citations in OpenAlex.
- Efficacy and Safety of Recombinant Factor VIII in Previously Untreated and Previously Treated Children with Hemophilia A: A Systematic Review.Advances in therapy · 2025Pooled it
- Determinants of Immune Tolerance Induction Outcomes in 237 Persons With Severe Hemophilia A.European journal of haematology · 2026Article
- Current perspectives on non-factor therapies for hemophilia.International journal of hematology · 2026Review
- Hemophilia in Mexico: Updated Consensus Recommendations on Diagnosis, Treatment and Gene Therapy.Diseases (Basel, Switzerland) · 2026Review
- Gene Therapy in Hemophilia: Clinical Advances, Immunological Challenges, and Emerging Therapeutic Perspectives.International journal of molecular sciences · 2026Review
- Outcomes of low-dose immune tolerance induction with single-dose rituximab in severe hemophilia A: a single-center retrospective experience.Frontiers in immunology · 2026Article
- Maternal microchimerism accelerates immune tolerance induction to factor VIII in children with hemophilia A and FVIII inhibitors.Frontiers in immunology · 2026Observational
- Large deletions in theResearch and practice in thrombosis and haemostasis · 2025Article
- Curative Therapies for Hemophilias and Hemoglobinopathies in Adults: Immune, Gene, and Stem Cell Approaches in a Global Context.Biomedicines · 2025Review
- Immune Tolerance Induction With a Recombinant Factor VIII Fc in Haemophilia A: Data From a Chart Review Study.European journal of haematology · 2025Article
- Dose-intensive therapy (DIT) for infantile Pompe disease: A pilot study.Molecular genetics and metabolism reports · 2025Article
- MHC class II presentation of FVIII-AnnexinA5 fusion proteins internalized by antigen presenting cells.Frontiers in immunology · 2025Article
- Rational design of FVIII sialylated peptides to target Siglec-3 and Siglec-9 and improve peptide formulations for reverse vaccines.Frontiers in bioengineering and biotechnology · 2025Article
- Advances in biopharmaceutical products for hemophilia.iScience · 2024Review
- Minor surgical procedures during immune tolerance induction in people with hemophilia A and inhibitors: results from the Brazilian Immune Tolerance (BrazIT) study cohort.Hematology, transfusion and cell therapy · 2024Article
- Prediction of the chance of successful immune tolerance induction in persons with severe hemophilia A and inhibitors: a clinical prediction model.Research and practice in thrombosis and haemostasis · 2024Article
- The impact of inhibitors on the quality of life in patients with hemophilia.SAGE open medicine · 2023Article
Corrections and comments
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Authors and funding
13 authors at 9 institutions in 5 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Background: Immune tolerance induction (ITI) aims to eradicate anti-factor VIII (FVIII) antibodies (inhibitors) in persons with hemophilia A. However, this burdensome treatment fails in 10% to 40%. To estimate the chance of ITI success in clinical decision making, it is important to identify the predictors of ITI success. Objectives: We performed a systematic review and meta-analysis to summarize the current evidence on determinants of ITI outcome in persons with hemophilia A. Methods: A literature search was conducted to identify randomized controlled trials, cohort, or case-control studies reporting on the predictors for ITI outcome in persons with hemophilia A. The main outcome was ITI success. Methodological quality was assessed using an adapted Joanna Briggs Institute checklist, rating as high if ≥11 of 13 criteria were met. Pooled odds ratios (ORs) for ITI success were calculated for each determinant. ITI success was defined as negative inhibitor titer (<0.6 BU/mL), FVIII recovery ≥66% of expected, and FVIII half-life ≥6 hours in 16 (59.3%) studies. Results: We included 27 studies, involving 1,734 participants. Methodological quality of 6 (22.2%) studies (418 participants) was rated as high. Twenty different determinants were assessed. Historical peak titer ≤100 BU/mL (compared with >100 BU/mL, OR, 1.7; 95% CI, 1.4-2.1), pre-ITI titer ≤10 BU/mL (compared with >10 BU/mL, OR, 1.8; 95% CI, 1.4-2.3), and peak titer during ITI ≤100 BU/mL (compared with >100 BU/mL, OR, 2.7; 95% CI, 1.9-3.8) were associated with a higher chance of ITI success. Conclusion: Our results suggest that determinants related to the inhibitor titer are associated with ITI success.
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