Akintayo Richard Oluyinka, Agbola Olanrewaju Festus, Adeyemo Abiodun Waliyullah, Adelowo Olufemi
Department of Medicine, University of Ilorin Teaching Hospital, Ilorin, Nigeria.
Department of Medicine, Rheumatology Unit, Lagos State University Teaching Hospital, Lagos, Nigeria.
BMJ Case Rep. 2017 Jul 18;2017:bcr-2017-219221. doi: 10.1136/bcr-2017-219221.
Idiopathic inflammatory myopathies are a heterogeneous group of systemic diseases characterised by variable phenotypes of chronic progressive muscle weakness. Myositis-specific antibodies (MSAs) include antibodies to cytoplasmic signal recognition particle (SRP) and various tRNA synthetases as well as the nuclear helicase protein Mi-2. These antibodies are typically found only in a fraction of true myositis cases and they tend to be mutually exclusive. Few cases of coexistence of two MSAs in the same patient have been reported and these cases all involve an antisynthetase antibody coexisting with either anti-SRP or anti-Mi-2 antibody. Peculiar clinical manifestations may be associated with different combinations of MSAs but the rarity of these cases makes their characterisation difficult. We report the first ever case of anti-SRP and anti-Mi-2 copositive polymyositis in a 19-year-old boy who presented with a week history of profound muscle weakness that attained its peak within 24 hours of onset.
特发性炎性肌病是一组异质性的全身性疾病,其特征为慢性进行性肌无力的可变表型。肌炎特异性抗体(MSAs)包括抗细胞质信号识别颗粒(SRP)抗体、各种tRNA合成酶抗体以及核解旋酶蛋白Mi-2抗体。这些抗体通常仅在一部分真正的肌炎病例中发现,而且它们往往相互排斥。同一患者同时存在两种MSAs的病例报告很少,这些病例均涉及抗合成酶抗体与抗SRP或抗Mi-2抗体同时存在。特殊的临床表现可能与MSAs的不同组合有关,但这些病例罕见,难以对其进行特征描述。我们报告了首例19岁男孩抗SRP和抗Mi-2双阳性多发性肌炎病例,该男孩有一周的严重肌无力病史,肌无力在发病后24小时内达到高峰。