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1例单中心Castleman病患者并发脊柱关节炎,经手术切除及托珠单抗治疗有效控制

A Case of Spondyloarthritis in Patient Affected by Unicentric Castleman's Disease Effectively Managed with Surgery Resection and Tocilizumab Treatment.

作者信息

Filippini M, Cartella S, Bonzanini O, Morello E, Tincani A

机构信息

Unità Operativa di Reumatologia, Spedali Civili di Brescia, Brescia, Italy.

Unità Operativa di Medicina Interna, Azienda Ospedaliera di Desenzano del Garda, Desenzano del Garda, Italy.

出版信息

Case Rep Rheumatol. 2018 Jan 23;2018:5067239. doi: 10.1155/2018/5067239. eCollection 2018.

Abstract

A 38-year-old woman was referred to our hospital for rheumatologic manifestations (migrant arthritis and tenosynovitis), without psoriasis or family history of psoriasis, gastroenteric manifestations, or recent genitourinary infections. The instrumental and laboratory tests have suggested a diagnosis of undifferentiated seronegative HLA-B27-positive spondyloarthritis with predominantly peripheral involvement. The symptoms were very severe and resistant to anti-inflammatory drugs and steroids. She had a history of hyaline-vascular unicentric Castleman's disease (HBV, HIV, and HHV-8 negative) treated with surgery resection. After a first pharmacological attempt with sulfasalazine (suspended for urticarial rash), we managed the patient with monotherapy tocilizumab 8 mg/kg, with full response of rheumatologic manifestations. The efficacy of tocilizumab was confirmed even after a follow-up of three years. Our experience seems to describe a new late-onset autoimmune disease (only 21 cases described in literature) potentially related to Castleman's disease. The patient experienced marked improvement from IL-6-based therapy (tocilizumab).

摘要

一名38岁女性因风湿性表现(游走性关节炎和腱鞘炎)被转诊至我院,无银屑病或银屑病家族史、胃肠道表现或近期泌尿生殖系统感染。仪器检查和实验室检查提示诊断为未分化血清阴性HLA - B27阳性脊柱关节炎,主要累及外周关节。症状非常严重,对抗炎药和类固醇耐药。她有透明血管型单中心Castleman病(乙肝病毒、艾滋病毒和HHV - 8均为阴性)病史,接受过手术切除治疗。在首次尝试使用柳氮磺胺吡啶(因荨麻疹性皮疹停药)后,我们采用托珠单抗8mg/kg单药治疗该患者,风湿性表现完全缓解。即使经过三年随访,托珠单抗的疗效仍得到证实。我们的经验似乎描述了一种新的迟发性自身免疫性疾病(文献中仅描述了21例),可能与Castleman病有关。该患者接受基于IL - 6的治疗(托珠单抗)后有显著改善。

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本文引用的文献

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A case of Castleman's disease with myasthenia gravis.1例伴重症肌无力的Castleman病。
Ann Thorac Cardiovasc Surg. 2014;20 Suppl:585-8. doi: 10.5761/atcs.cr.12.02165. Epub 2013 Apr 5.
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Castleman disease and associated autoimmune disease.卡斯尔曼病及相关自身免疫性疾病。
Curr Opin Rheumatol. 2012 Jan;24(1):76-83. doi: 10.1097/BOR.0b013e32834db525.

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