BackgroundLow-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.AimTo 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.MethodsA 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%.ResultsA 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%.
Frontiers in Immunology published a clinical update in Infectious Disease on 30 Apr 2026.
The item focuses on Outcomes of low-dose immune tolerance induction with single-dose rituximab in severe hemophilia A: a single-center retrospective experience.
Review the original article for the full source wording and details.