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获得性血友病A并发阿仑单抗治疗多发性硬化症。

Acquired haemophilia A complicating alemtuzumab therapy for multiple sclerosis.

作者信息

McCaughan Georgia, Massey Jennifer, Sutton Ian, Curnow Jennifer

机构信息

Haematology Department, Westmead Hospital, Sydney, New South Wales, Australia.

Sydney Medical School, Sydney, Australia.

出版信息

BMJ Case Rep. 2017 Dec 5;2017:bcr-2017-223016. doi: 10.1136/bcr-2017-223016.

DOI:10.1136/bcr-2017-223016
PMID:29212866
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC5728240/
Abstract

Alemtuzumab is a highly efficacious therapy used in the treatment of multiple sclerosis (MS), but uncoupling of T and B cell repopulation during immune reconstitution associates with an increasing range of secondary B cell-mediated autoimmune complications. A 34-year-old woman developed Graves' disease 11 months following an initial course of alemtuzumab treatment for MS. Nine months following the second treatment with alemtuzumab, the patient presented with spontaneous intramuscular and subcutaneous haemorrhage due to development of an inhibitory autoantibody to coagulation factor VIII. Acquired haemophilia A (AHA) is an extremely rare complication in patients treated with alemtuzumab. Treatment with rituximab may induce a rapid remission of AHA; however, the patient's high John Cunningham virus (JCV) antibody index and alemtuzumab-induced T cell lymphopenia may lead to an increased risk of progressive multifocal leucoencephalopathy, a potential complication which was unacceptable to the patient.

摘要

阿仑单抗是一种用于治疗多发性硬化症(MS)的高效疗法,但免疫重建过程中T细胞和B细胞再增殖的解偶联与越来越多的继发性B细胞介导的自身免疫并发症相关。一名34岁女性在接受阿仑单抗初始疗程治疗MS 11个月后患上格雷夫斯病。在第二次使用阿仑单抗治疗9个月后,该患者因产生针对凝血因子VIII的抑制性自身抗体而出现自发性肌肉内和皮下出血。获得性血友病A(AHA)是接受阿仑单抗治疗患者中极为罕见的并发症。使用利妥昔单抗治疗可能会使AHA迅速缓解;然而,患者较高的约翰·坎宁安病毒(JCV)抗体指数以及阿仑单抗诱导的T细胞淋巴细胞减少可能会导致进行性多灶性白质脑病风险增加,而这是患者无法接受的一种潜在并发症。

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本文引用的文献

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Neutropenia in Patients With Multiple Sclerosis Treated With Alemtuzumab.用阿仑单抗治疗的多发性硬化症患者的中性粒细胞减少症
JAMA Neurol. 2017 Sep 1;74(9):1143-1144. doi: 10.1001/jamaneurol.2017.1456.
2
Interpreting Lymphocyte Reconstitution Data From the Pivotal Phase 3 Trials of Alemtuzumab.解读阿仑单抗关键3期试验中的淋巴细胞重建数据。
JAMA Neurol. 2017 Aug 1;74(8):961-969. doi: 10.1001/jamaneurol.2017.0676.
3
Treatment effectiveness of alemtuzumab compared with natalizumab, fingolimod, and interferon beta in relapsing-remitting multiple sclerosis: a cohort study.在复发缓解型多发性硬化症中,与那他珠单抗、芬戈莫德和干扰素β相比,阿仑单抗的治疗效果:一项队列研究。
Lancet Neurol. 2017 Apr;16(4):271-281. doi: 10.1016/S1474-4422(17)30007-8. Epub 2017 Feb 11.
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Factor VIII inhibitor eradication with bortezomib in acquired haemophilia A.硼替佐米用于获得性血友病A中消除VIII因子抑制剂
Br J Haematol. 2017 Sep;178(6):986-987. doi: 10.1111/bjh.14185. Epub 2016 Jul 19.
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JC virus reactivation in patients with autoimmune rheumatic diseases treated with rituximab.接受利妥昔单抗治疗的自身免疫性风湿病患者中的JC病毒再激活。
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