Failure of rituximab to induce immune tolerance in a boy with severe haemophilia A and an alloimmune factor VIII antibody: a case report and review of the literature

被引:13
作者
Biss, TT [1 ]
Velangi, MR [1 ]
Hanley, JP [1 ]
机构
[1] Newcastle Hosp NHS Trust, Royal Victoria Infirm, Newcastle Haemophilia Comprehens Care Ctr, Newcastle Upon Tyne NE1 4LP, Tyne & Wear, England
关键词
acquired haemophilia; alloimmune FVIII antibody; haemophilia A; immune tolerance induction; rituximab;
D O I
10.1111/j.1365-2516.2006.01212.x
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
We report the use of rituximab (MabThera((R)); Roche Grenzach-Wyhlen, Germany) in a 6-year-old boy with severe haemophilia A and a high titre alloimmune factor VIII (FVIII) antibody, which had failed to respond to standard immune tolerance therapy. Rituximab was administered in 4 weekly doses with concurrent high-dose i.v. immunoglobulin (Flebogamma((R)); Grifols, Barcelona, Spain) followed by daily high-dose recombinant FVIII concentrate (Recombinate((R)); Baxter, CA, USA). Despite a fall in CD20 positive cell count to undetectable levels the inhibitor persisted. We discuss the possible reasons for failure of immune tolerance induction and review the literature concerning the use of rituximab for this indication.
引用
收藏
页码:280 / 284
页数:5
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