Kanatani Masahiro, Adachi Tadashi, Sakata Ryoichi, Watanabe Yasuhiro, Hanajima Ritsuko
Division of Neurology, Department of Brain and Neurosciences, Faculty of Medicine, Tottori University.
Rinsho Shinkeigaku. 2020 Jul 31;60(7):489-494. doi: 10.5692/clinicalneurol.60.cn-001427. Epub 2020 Jun 13.
A 66-year-old woman who had myasthenia gravis (MG) admitted for type II respiratory failure and right heart failure. Although she had neither ptosis, eye movement disorder, nor diplopia, she had orbital muscles weakness, reduction of gag reflex, dysarthria, dysphagia, and mild proximal muscle weakness. Blood tests showed anti-striated muscle antibodies (anti-titin antibody and anti-Kv1.4 antibody). A muscle biopsy of the left biceps showed a marked variation in fiber size, mild mononuclear cell infiltration was seen surrounding blood vessels in perimysium and nemaline bodies in some fibers. Immunohistochemical stains showed many muscle fibers express HLA-ABC. The patient was diagnosed as sporadic late-onset nemaline myopathy (SLONM) with MG, and treated by tacrolimus. After treatment, her respiratory function gradually improved and she discharged. In the case of atypical MG, measurement of anti-striated muscle antibody or muscle biopsy should be considered.
一名66岁患有重症肌无力(MG)的女性因II型呼吸衰竭和右心衰竭入院。尽管她既没有上睑下垂、眼球运动障碍,也没有复视,但她存在眶肌无力、咽反射减弱、构音障碍、吞咽困难以及轻度近端肌无力。血液检查显示有抗横纹肌抗体(抗肌联蛋白抗体和抗Kv1.4抗体)。左侧二头肌的肌肉活检显示纤维大小存在明显差异,在肌束膜的血管周围可见轻度单核细胞浸润,部分纤维中有杆状体。免疫组织化学染色显示许多肌纤维表达HLA-ABC。该患者被诊断为伴有MG的散发性晚发型杆状体肌病(SLONM),并接受了他克莫司治疗。治疗后,她的呼吸功能逐渐改善并出院。对于非典型MG病例,应考虑检测抗横纹肌抗体或进行肌肉活检。