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流感疫苗接种后抗单识别颗粒介导的坏死性肌病病例

Case of Anti-Single Recognition Particle-Mediated Necrotizing Myopathy After Influenza Vaccination.

作者信息

Mamarabadi Mansoureh, Baisre Ada, Leitch Megan, Hsu Vivien, Kanduri Jaya S, Chen Shan

机构信息

Department of Neurology, Rutgers Robert Wood Johnson Medical School, New Brunswick, NJ.

Department of Pathology and Laboratory Medicine, Rutgers-New Jersey Medical School, Newark, NJ.

出版信息

J Clin Neuromuscul Dis. 2018 Jun;19(4):211-216. doi: 10.1097/CND.0000000000000208.

Abstract

Immune-mediated necrotizing myopathy is a very rare inflammatory disease affecting skeletal muscles. Immune-mediated necrotizing myopathy may be associated with myositis-specific autoantibodies including anti-single recognition particle and anti-3-hydroxy-3- methylglutaryl-coenzyme A reductase, infectious agents (HIV or hepatitis C), other connective tissue disorders (such as scleroderma), and malignancy. We reported a 28-year-old healthy woman presented with subacute onset ascending muscle weakness 2 weeks after an annual influenza vaccination. Cerebral Spinal Fluid study showed normal cell counts with elevated protein and nerve conduction study showed reduced diffuse compound muscle action potential amplitudes suggesting a diagnosis of Guillain-Barré syndrome. Despite treatment using intravenous immunoglobulin, her condition continued to get worse with new bulbar and respiratory muscle weakness. Eventually, the diagnosis of anti-single recognition particle-mediated necrotizing myopathy was made based on elevated creatine kinase, thigh magnetic resonance imaging, muscle biopsy, and positive antibody testing. Our patient responded to the combination of intravenous immunoglobulin, prednisone, and anti-CD20 monoclonal antibody, rituximab.

摘要

免疫介导的坏死性肌病是一种非常罕见的影响骨骼肌的炎症性疾病。免疫介导的坏死性肌病可能与肌炎特异性自身抗体有关,包括抗信号识别颗粒抗体和抗3-羟基-3-甲基戊二酰辅酶A还原酶抗体,还可能与感染因子(如HIV或丙型肝炎)、其他结缔组织疾病(如硬皮病)以及恶性肿瘤有关。我们报告了一名28岁的健康女性,在每年一次的流感疫苗接种后2周出现亚急性起病的进行性肌肉无力。脑脊液检查显示细胞计数正常但蛋白升高,神经传导检查显示复合肌肉动作电位弥漫性波幅降低,提示吉兰-巴雷综合征的诊断。尽管使用静脉注射免疫球蛋白进行了治疗,但她的病情仍继续恶化,出现了新的延髓和呼吸肌无力。最终,根据肌酸激酶升高、大腿磁共振成像、肌肉活检及抗体检测阳性,诊断为抗信号识别颗粒介导的坏死性肌病。我们的患者对静脉注射免疫球蛋白、泼尼松和抗CD20单克隆抗体利妥昔单抗的联合治疗有反应。

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