ArticleFrontiers in immunology2026
Outcomes of low-dose immune tolerance induction with single-dose rituximab in severe hemophilia A: a single-center retrospective experience.
Article in Frontiers in immunology, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
Background: Low-dose immune tolerance induction (ITI) combined with standard-dose rituximab-based immunosuppression (LD-ITI + RTX-IS) is an effective strategy for factor VIII (FVIII) inhibitor eradication in severe hemophilia A with poor-risk prognostic factors for ITI, but optimal rituximab dosing remains undefined. Prolonged rituximab exposure increases adverse effects and financial burden. Aim: To describe the outcomes and tolerability of LD-ITI combined with single-dose rituximab (LD-ITI + SD-RTX-IS) for FVIII inhibitor eradication in severe hemophilia A. Methods: A single-center retrospective cohort study was conducted in patients with severe hemophilia A and inhibitors who received LD-ITI + SD-RTX-IS. Treatment comprised FVIII (50 IU/kg every other day), rituximab (375 mg/m², single dose), and prednisone (1 mg/kg daily, tapering). Success was defined as inhibitor titer <0.6 BU/mL and FVIII recovery ≥66%. Results: A total of 16 patients (median age 17 years) were enrolled, comprising 8 adults and 8 children. Eleven of 16 patients (68.8%) achieved successful inhibitor eradication with a median time of 9.3 months (IQR: 2.8-15.2; range: 1.8-18.6); median FVIII recovery was 82%. The monthly bleeding rate decreased by 84.2% from pre-ITI (median: 4 episodes) to during ITI (median: 0.66 episodes), with a corresponding reduction in monthly joint bleeds from 3 to 0.46 episodes. At 24-month follow-up, no recurrence was documented. Adverse events were predominantly mild (rituximab-related infusion reactions, n=2; fever/pneumonia, n=4). Conclusions: In this cohort, LD-ITI + SD-RTX-IS was associated with an inhibitor eradication rate consistent with prior LD-ITI + RTX-IS reports, using less rituximab. These preliminary findings warrant further prospective evaluation.
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