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难治性巨细胞动脉炎合并主动脉炎:抗IL6单克隆抗体(托珠单抗)及升主动脉手术重建的挽救生命治疗

Refractory PMR with aortitis: life-saving treatment with anti-IL6 monoclonal antibody (tocilizumab) and surgical reconstruction of the ascending aorta.

作者信息

Ashraf Fahd Adeeb Mohamed, Anjum Shakeel, Hussaini Abid, Fraser Alexander

机构信息

Department of Rheumatology, University Hospital Limerick, Limerick, Ireland.

出版信息

BMJ Case Rep. 2013 Jun 18;2013:bcr2013009523. doi: 10.1136/bcr-2013-009523.

Abstract

Aortitis is uncommon but well described in patients with polymyalgia rheumatica (PMR). While glucocorticoid remains the mainstay therapy for large-vessel vasculitis, there have been cases where tocilizumab therapy led to clinical and serological improvement in patients with relapsing or refractory disease. We report a case of life-threatening PMR with aortitis in the absence of manifestations related to giant cell arteritis, which, having failed to respond to corticosteroid therapy, was successfully treated with tocilizumab and emergency reconstruction of the ascending aorta. This case adds to the literature supporting the potential value of interleukin-6 inhibition in rare rheumatological conditions such as inflammatory aortitis.

摘要

大动脉炎并不常见,但在风湿性多肌痛(PMR)患者中已有充分描述。虽然糖皮质激素仍然是大血管血管炎的主要治疗方法,但已有病例表明,托珠单抗治疗可使复发或难治性疾病患者在临床和血清学方面得到改善。我们报告一例危及生命的PMR合并大动脉炎病例,该病例无巨细胞动脉炎相关表现,对皮质类固醇治疗无效,经托珠单抗治疗及升主动脉紧急重建后成功治愈。该病例补充了文献资料,支持白细胞介素-6抑制在罕见风湿性疾病如炎性大动脉炎中的潜在价值。

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本文引用的文献

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Biologic treatment of large-vessel vasculitides.大血管血管炎的生物治疗。
Curr Opin Rheumatol. 2012 Jan;24(1):31-7. doi: 10.1097/BOR.0b013e32834dc392.
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Pathogenesis of Takayasu's arteritis: a 2011 update.Takayasu 动脉炎的发病机制:2011 年更新。
Autoimmun Rev. 2011 Nov;11(1):61-7. doi: 10.1016/j.autrev.2011.08.001. Epub 2011 Aug 9.
8
Comment on "Polymyalgia rheumatica as the manifestation of unclassified aortitis".
Mod Rheumatol. 2008;18(4):427-8. doi: 10.1007/s10165-008-0073-9. Epub 2008 Apr 25.
10
Polymyalgia rheumatica as the manifestation of unclassified aortitis.
Mod Rheumatol. 2008;18(1):105-8. doi: 10.1007/s10165-007-0019-7. Epub 2007 Dec 22.

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