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1例获得性凝血因子VIII抑制物合并多种治疗相关机会性感染病例及文献复习

A Case of an Acquired Factor VIII Inhibitor Complicated by Multiple Treatment-Related Opportunistic Infections and Review of the Literature.

作者信息

Hutchinson Anna L, Tan Yi Ling, Kidson-Gerber Giselle

机构信息

Prince of Wales Hospital, Barker Street, Randwick, NSW 2031, Australia.

Department of Medicine, University of New South Wales, Botany Street, Sydney, NSW 2052, Australia.

出版信息

Case Rep Hematol. 2013;2013:703027. doi: 10.1155/2013/703027. Epub 2013 Dec 18.

DOI:10.1155/2013/703027
PMID:24455336
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC3878284/
Abstract

This case report describes a patient with an idiopathic acquired Factor VIII inhibitor and severe bleeding. She was treated with rituximab after failing first-line treatment with steroids and cyclophosphamide. Two months following rituximab treatment, our patient developed a succession of severe opportunistic infections requiring intensive care unit admission. Over a period of 12 weeks she required treatment for Pseudomonas aeruginosa septicaemia, herpes simplex gingivostomatitis and pharyngotonsillitis, clostridium difficile-related diarrhoea, systemic cytomegalovirus infection, pneumocystis jiroveci, and invasive pulmonary aspergillosis lung infections. After significant rehabilitation, the patient was finally discharged following a 5-month admission. This case highlights the complexity of balancing a life-threatening condition with the side effects of treatment. It also raises the issue of routine prophylaxis for immunosuppression in nonmalignant conditions, which will become a common dilemma with the expanding indications for rituximab use.

摘要

本病例报告描述了一名患有特发性获得性凝血因子VIII抑制剂且严重出血的患者。在一线使用类固醇和环磷酰胺治疗失败后,她接受了利妥昔单抗治疗。利妥昔单抗治疗两个月后,我们的患者出现了一系列严重的机会性感染,需要入住重症监护病房。在12周的时间里,她需要治疗铜绿假单胞菌败血症、单纯疱疹性龈口炎和咽扁桃体炎、艰难梭菌相关性腹泻、系统性巨细胞病毒感染、耶氏肺孢子菌肺炎和侵袭性肺曲霉病肺部感染。经过大量康复治疗后,患者在入院5个月后最终出院。该病例突出了在危及生命的病情与治疗副作用之间进行平衡的复杂性。它还提出了在非恶性疾病中进行免疫抑制常规预防的问题,随着利妥昔单抗使用适应症的扩大,这将成为一个常见的困境。

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本文引用的文献

1
Rituximab and intravenous immunoglobulin (IVIG) for the management of acquired factor VIII inhibitor in multiple myeloma: case report and review of literature.利妥昔单抗和静脉注射免疫球蛋白(IVIG)治疗多发性骨髓瘤获得性因子 VIII 抑制剂:病例报告及文献复习。
Int J Hematol. 2012 Jan;95(1):102-6. doi: 10.1007/s12185-011-0968-7. Epub 2011 Dec 15.
2
The effect of rituximab on humoral and cell mediated immunity and infection in the treatment of autoimmune diseases.利妥昔单抗治疗自身免疫性疾病对体液和细胞介导免疫及感染的影响。
Br J Haematol. 2010 Apr;149(1):3-13. doi: 10.1111/j.1365-2141.2010.08076.x. Epub 2010 Feb 11.
3
Successful eradication of acquired factor-VIII-inhibitor using single low-dose rituximab.使用单次低剂量利妥昔单抗成功根除获得性凝血因子VIII抑制剂。
Haematologica. 2010 Mar;95(3):521-2. doi: 10.3324/haematol.2009.017749. Epub 2009 Nov 10.
4
Incidence and predictive factors for infectious disease after rituximab therapy in kidney-transplant patients.肾移植患者接受利妥昔单抗治疗后的感染性疾病发生率及预测因素。
Am J Transplant. 2010 Jan;10(1):89-98. doi: 10.1111/j.1600-6143.2009.02785.x. Epub 2009 Jul 28.
5
A review of the current use of rituximab in autoimmune diseases.利妥昔单抗在自身免疫性疾病中的当前应用综述。
Int Immunopharmacol. 2009 Jan;9(1):10-25. doi: 10.1016/j.intimp.2008.10.004. Epub 2008 Nov 8.
6
Current management of acquired factor VIII inhibitors.获得性凝血因子VIII抑制剂的当前管理
Curr Opin Hematol. 2008 Sep;15(5):451-5. doi: 10.1097/MOH.0b013e328309ecab.
7
Acquired factor VIII inhibitors.获得性凝血因子 VIII 抑制剂
Blood. 2008 Jul 15;112(2):250-5. doi: 10.1182/blood-2008-03-143586. Epub 2008 May 7.
8
Late-onset neutropenia associated with rituximab therapy: evidence for a maturation arrest at the (pro)myelocyte stage of granulopoiesis.与利妥昔单抗治疗相关的迟发性中性粒细胞减少症:粒细胞生成的(原)髓细胞阶段成熟停滞的证据。
Med Oncol. 2008;25(4):374-9. doi: 10.1007/s12032-008-9049-z. Epub 2008 Feb 16.
9
Safety and efficacy of additional courses of rituximab in patients with active rheumatoid arthritis: an open-label extension analysis.利妥昔单抗附加疗程用于活动性类风湿关节炎患者的安全性和有效性:一项开放标签扩展分析
Arthritis Rheum. 2007 Dec;56(12):3896-908. doi: 10.1002/art.23059.
10
Acquired hemophilia: a case report of 2 patients with acquired factor VIII inhibitor treated with rituximab plus a short course of steroid and review of the literature.获得性血友病:2例接受利妥昔单抗联合短期类固醇治疗的获得性凝血因子VIII抑制物患者的病例报告及文献复习
Clin Appl Thromb Hemost. 2007 Oct;13(4):443-8. doi: 10.1177/1076029607303777.