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Case Report
The Use of Rituximab with Immune Tolerance Induction Therapy for Hemophilia A with Inhibitors
Clin Pediatr Hematol Oncol 2015;22:67-71.
Published online April 30, 2015
© 2015 Korean Society of Pediatric Hematology-Oncology and Korean Society for Pediatric Neuro-Oncology

Chae Young Kim, M.D., Keum Nho Lee, M.D. and Young Shil Park, M.D., Ph.D.

Department of Pediatrics, Kyung Hee University Hospital at Gangdong, Seoul, Korea
Correspondence to: Young Shil Park
Department of Pediatrics, Kyung Hee
University Hospital at Gangdong, 892,
Dongnam-ro, Gangdong-gu, Seoul
134-727, Korea
Tel: +82-2-440-6133
Fax: +82-2-440-7175
E-mail: pysmd@khnmc.or.kr
Received March 5, 2015; Revised April 15, 2015; Accepted April 20, 2015.
This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
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.
Keywords: Hemophilia, Inhibitor, Immune tolerance induction, Rituximab
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  • Korea Hemophilia Foundation