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皮肌炎患者获得性凝血因子 VIII 抑制剂——病例报告

Acquired factor VIII inhibitor in a patient with dermatomyositis--a case study.

作者信息

Ayoola Ayodele, Mohsini Wajahath, Aung Sein

机构信息

Department of Medicine, Franklin Square Hospital Center, Baltimore, MD 21237, USA.

出版信息

Acta Haematol. 2008;119(1):8-11. doi: 10.1159/000112838. Epub 2008 Jan 4.

DOI:10.1159/000112838
PMID:18176073
Abstract

BACKGROUND

Acquired factor VIII inhibitor is an antibody formed against FVIII and seen in patients without FVIII replacement therapy. This entity has been associated with both autoimmune disease and infections.

CASE STUDY

We report the case of a 65-year-old man with dermatomyositis who presented with a bleeding episode following venipuncture and hematuria. His coagulation parameters were followed as well as the inhibitor level. His acquired FVIII inhibitor was managed successfully with rituximab.

CONCLUSION

Our patient's bleeding episodes resolved and the inhibitor level was undetectable after 12 months. This is the first case of acquired FVIII inhibitor in association with dermatomyositis.

摘要

背景

获得性凝血因子VIII抑制物是一种针对FVIII形成的抗体,见于未接受FVIII替代治疗的患者。该疾病与自身免疫性疾病和感染均有关联。

病例研究

我们报告了一名65岁皮肌炎男性患者的病例,该患者在静脉穿刺后出现出血事件及血尿。我们对其凝血参数以及抑制物水平进行了监测。他的获得性FVIII抑制物通过利妥昔单抗成功得到控制。

结论

我们患者的出血事件得到缓解,12个月后抑制物水平检测不到。这是首例与皮肌炎相关的获得性FVIII抑制物病例。

相似文献

1
Acquired factor VIII inhibitor in a patient with dermatomyositis--a case study.皮肌炎患者获得性凝血因子 VIII 抑制剂——病例报告
Acta Haematol. 2008;119(1):8-11. doi: 10.1159/000112838. Epub 2008 Jan 4.
2
Idiopathic factor VIII inhibitor autoantibody in a man presented after accident.男性患者因意外后出现特发性因子 VIII 抑制剂自身抗体。
Clin Appl Thromb Hemost. 2009 Oct;15(5):588-90. doi: 10.1177/1076029608319442. Epub 2008 Jun 11.
3
[Refractory acquired hemophilia A in which factor VIII inhibitor was not detected at onset and thereafter deteriorated after total hip replacement arthroplasty].[难治性获得性血友病A,其在发病时未检测到VIII因子抑制剂,之后在全髋关节置换术后病情恶化]
Rinsho Ketsueki. 2008 Nov;49(11):1548-51.
4
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.
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Rituximab alone or in association with corticosteroids in the treatment of acquired factor VIII inhibitors: report of two cases.
Transfus Med. 2006 Jun;16(3):209-12. doi: 10.1111/j.1365-3148.2006.00669.x.
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Rituximab for the treatment of patients with very high-titre acquired factor VIII inhibitors refractory to conventional chemotherapy.利妥昔单抗用于治疗对传统化疗难治的高滴度获得性凝血因子VIII抑制物患者。
Haemophilia. 2007 Jan;13(1):46-50. doi: 10.1111/j.1365-2516.2006.01342.x.
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Successful rituximab therapy of acquired factor VIII inhibitor in a patient with rheumatoid arthritis.利妥昔单抗成功治疗类风湿关节炎患者获得性凝血因子VIII抑制物
J Clin Rheumatol. 2007 Apr;13(2):89-91. doi: 10.1097/01.rhu.0000260656.05638.f7.
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[Fatal opportunistic infection following disappearance of antibodies by immunosuppressive therapy in a patient with acquired factor VIII inhibitor].[一名获得性因子VIII抑制剂患者接受免疫抑制治疗后抗体消失,继而发生致命性机会性感染]
Rinsho Ketsueki. 2004 Sep;45(9):1023-7.
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[Immunosuppressive treatment of a spontaneous inhibitor hemophilia A using cyclophosphamide, vincristine and prednisone following prior Factor VIII stimulation].[在先前使用凝血因子 VIII 刺激后,使用环磷酰胺、长春新碱和泼尼松对自发性抑制性血友病 A 进行免疫抑制治疗]
Schweiz Med Wochenschr. 1996 Nov 23;126(47):2026-31.
10
[Immunoblotting analysis of acquired factor VIII inhibitor in an elderly man].[一名老年男性获得性凝血因子VIII抑制物的免疫印迹分析]
Rinsho Ketsueki. 1993 Jul;34(7):876-8.

引用本文的文献

1
Acquired factor VIII deficiency: two case reports and a review of literature.获得性因子VIII缺乏症:两例病例报告及文献综述
Exp Hematol Oncol. 2017 Mar 24;6:8. doi: 10.1186/s40164-017-0068-3. eCollection 2017.