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Clin Pediatr Hematol Oncol. 2015 Apr;22(1):67-71. English. Case Report. https://doi.org/10.15264/cpho.2015.22.1.67
Kim CY , Lee KN , Park YS .
Department of Pediatrics, Kyung Hee University Hospital at Gangdong, Seoul, Korea. pysmd@khnmc.or.kr
Abstract

Inhibitor development is one of the major adverse events associated with increased morbidity and mortality in patients with congenital hemophilia. Recent treatment for them is immune tolerance induction (ITI), which involves the administration of high doses of factor concentrates over a prolonged period, sometimes combined with immunosuppressive agents. We report a case of inhibitor elimination with Rituximab, and high-dose factor VIII concentrates in a 5-year-old boy with hemophilia A. The patient improved clinically, with fewer bleeding episodes. However, he continued to have low immunoglobulin levels, which led to recurrent infections. After an infusion of intravenous immunoglobulin, inhibitor titers increased rapidly and his ITI was deemed a failure. In conclusion, even though it failed in the present study, Rituximab may be an alternative adjuvant therapy to eliminate the inhibitor in patients with hemophilia. The appropriate schedule and long-term side effects need further investigation.

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