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利妥昔单抗未能诱导一名重度甲型血友病男孩产生免疫耐受并伴有同种免疫性凝血因子 VIII 抗体:病例报告及文献综述

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.

作者信息

Biss T T, Velangi M R, Hanley J P

机构信息

Newcastle Haemophilia Comprehensive Care Centre, Newcastle Hospitals NHS Trust, Royal Victoria Infirmary, Newcastle-upon-Tyne, UK.

出版信息

Haemophilia. 2006 May;12(3):280-4. doi: 10.1111/j.1365-2516.2006.01212.x.

Abstract

We report the use of rituximab (MabThera); 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); Grifols, Barcelona, Spain) followed by daily high-dose recombinant FVIII concentrate (Recombinate); 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.

摘要

我们报告了利妥昔单抗(美罗华;德国罗氏公司,格林扎赫-维伦)在一名6岁重度甲型血友病男孩中的应用,该男孩有高滴度的同种免疫性凝血因子VIII(FVIII)抗体,对标准免疫耐受治疗无反应。利妥昔单抗每周给药1次,共4次,同时给予大剂量静脉注射免疫球蛋白(Flebogamma;西班牙巴塞罗那基立福公司),随后每日给予大剂量重组FVIII浓缩物(Recombinate;美国加利福尼亚州百特公司)。尽管CD20阳性细胞计数降至检测不到的水平,但抑制剂仍然存在。我们讨论了免疫耐受诱导失败的可能原因,并回顾了有关利妥昔单抗用于该适应症的文献。

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