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一名接受阿仑单抗治疗多发性硬化症的患者发生获得性甲型血友病。

Development of acquired haemophilia A in a patient treated with alemtuzumab for multiple sclerosis.

作者信息

Madeley Jarrett, Hodges Georgina, Birchley Andrew

机构信息

Department of Haematology, Cairns Hospital, Cairns, Queensland, Australia.

Department of Haematology, Townsville Hospital, Townsville, Queensland, Australia.

出版信息

BMJ Case Rep. 2018 Oct 17;2018:bcr-2018-226588. doi: 10.1136/bcr-2018-226588.

DOI:10.1136/bcr-2018-226588
PMID:30337289
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6254386/
Abstract

This case illustrates a 36-year-old man who presented with a factor VIII (FVIII) inhibitor (acquired haemophilia A) with cutaneous bleeding and a significant thigh haematoma. No traditional risk factors for the development of a FVIII inhibitor were identified. However, previous treatment with alemtuzumab for multiple sclerosis was noted in the patient's history. Alemtuzumab is an anti-CD52 monoclonal antibody and is known to be associated with the development of a number of autoimmune conditions, with a delay in onset of these conditions as long as 5 years after the cessation of treatment. To our knowledge, there have only been three previously documented cases of a FVIII inhibitor in the setting of alemtuzumab therapy. This case adds further evidence to the current body of literature suggesting alemtuzumab as a causative agent for the development of an FVIII inhibitor.

摘要

该病例为一名36岁男性,表现为伴有皮肤出血和严重大腿血肿的VIII因子(FVIII)抑制物(获得性血友病A)。未发现导致FVIII抑制物形成的传统风险因素。然而,患者病史中记录有先前使用阿仑单抗治疗多发性硬化症。阿仑单抗是一种抗CD52单克隆抗体,已知与多种自身免疫性疾病的发生有关,这些疾病的发病延迟可长达治疗停止后5年。据我们所知,先前仅有3例记录在案的阿仑单抗治疗引发FVIII抑制物的病例。该病例为当前文献进一步提供了证据,表明阿仑单抗是导致FVIII抑制物形成的病因。

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本文引用的文献

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BMJ Case Rep. 2017 Dec 5;2017:bcr-2017-223016. doi: 10.1136/bcr-2017-223016.
2
Alemtuzumab in the long-term treatment of relapsing-remitting multiple sclerosis: an update on the clinical trial evidence and data from the real world.阿仑单抗用于复发缓解型多发性硬化症的长期治疗:临床试验证据及真实世界数据的最新情况
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Therapeutic outcomes using subcutaneous low dose alemtuzumab for acquired bone marrow failure conditions.皮下注射低剂量阿仑单抗治疗获得性骨髓衰竭疾病的疗效
Br J Haematol. 2018 Oct;183(1):133-136. doi: 10.1111/bjh.14907. Epub 2017 Sep 14.
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Urgent Lung Transplantation in Severe Acute Respiratory Failure Based on Rapidly Progressive Interstitial Lung Disease: A Case Report.基于快速进展性间质性肺疾病的严重急性呼吸衰竭患者的紧急肺移植:一例报告
Transplant Proc. 2017 Sep;49(7):1544-1548. doi: 10.1016/j.transproceed.2017.06.020.
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Chronic lymphocytic leukemia: 2017 update on diagnosis, risk stratification, and treatment.慢性淋巴细胞白血病:2017 年诊断、风险分层和治疗更新。
Am J Hematol. 2017 Sep;92(9):946-965. doi: 10.1002/ajh.24826.
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Development of a secondary autoimmune disorder after hematopoietic stem cell transplantation for autoimmune diseases: role of conditioning regimen used.自身免疫性疾病造血干细胞移植后继发性自身免疫性疾病的发生:所采用预处理方案的作用
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