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结节病:它是包涵体肌炎的可能触发因素吗?

Sarcoidosis: Is It a Possible Trigger of Inclusion Body Myositis?

作者信息

Zakaria Ali, Turk Issam, Leung Kenneth, Capatina-Rata Ana, Farra Waseem

机构信息

Department of Internal Medicine, Division of Pulmonology, Division of Rheumatology, Providence-Providence Park Hospital, Michigan State University College of Human Medicine, Southfield, MI, USA.

出版信息

Case Rep Rheumatol. 2017;2017:8469629. doi: 10.1155/2017/8469629. Epub 2017 Apr 24.

Abstract

Sarcoidosis is a multisystem disorder of unknown etiology, characterized pathologically by the presence of nonnecrotizing granulomatous inflammation in affected organs. Although skeletal muscle is involved in 50-80 percent of individuals with sarcoidosis, symptomatic myopathy has been shown to be a rare manifestation of the disease. Inclusion body myositis (IBM) is a rare acquired idiopathic inflammatory myopathy with the insidious onset of asymmetric and distal muscle weakness that characteristically involves the quadriceps, tibialis anterior, and forearm flexors. Moreover, dysphagia can be the presenting complaint in one-third of patients. Herein, we are presenting a case of 67-year-old African American female who presented with one-month history of new onset progressive dyspnea on exertion. She was diagnosed with stage IV sarcoidosis based on chest CT scan findings and transbronchial lung biopsy revealing nonnecrotizing granulomatous inflammation. Over the next three months after her diagnosis, she presented to the hospital with progressive dysphagia associated with asymmetrical distal muscle weakness. A quadriceps muscle biopsy revealed features consistent with inclusion body myositis. We are reporting this case as it may support the hypothesis of sarcoidosis being a trigger that possibly promotes the development of inclusion body myositis, leading to a very poor prognosis.

摘要

结节病是一种病因不明的多系统疾病,其病理特征为受累器官出现非坏死性肉芽肿性炎症。尽管50%至80%的结节病患者骨骼肌会受累,但有症状的肌病已被证明是该疾病的一种罕见表现。包涵体肌炎(IBM)是一种罕见的获得性特发性炎性肌病,隐匿起病,表现为不对称的远端肌无力,典型地累及股四头肌、胫前肌和前臂屈肌。此外,三分之一的患者可能以吞咽困难为首发症状。在此,我们报告一例67岁的非裔美国女性,她有1个月新发进行性劳力性呼吸困难的病史。根据胸部CT扫描结果和经支气管肺活检显示非坏死性肉芽肿性炎症,她被诊断为IV期结节病。在诊断后的接下来三个月里,她因进行性吞咽困难伴不对称远端肌无力入院。股四头肌活检显示符合包涵体肌炎的特征。我们报告该病例,因为它可能支持结节病是一种触发因素,可能促进包涵体肌炎发展的假说,从而导致非常差的预后。

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