Department of Neurology, National Center Hospital, National Center of Neurology and Psychiatry, 4-1-1 Ogawahigashimachi, Kodaira, Tokyo 187-8551, Japan.
Department of Neurology, National Center Hospital, National Center of Neurology and Psychiatry, 4-1-1 Ogawahigashimachi, Kodaira, Tokyo 187-8551, Japan.
Neuromuscul Disord. 2022 Jun;32(6):516-520. doi: 10.1016/j.nmd.2022.02.010. Epub 2022 Feb 24.
Myoglobinopathy is a rare autosomal dominant myopathy that manifests in adulthood with proximal and axial weakness and variable respiratory and cardiac failure. Muscle pathology features associated with myoglobinopathy include characteristic sarcoplasmic bodies in skeletal and cardiac muscles. Here we present the first case of myoglobinopathy in an Asian individual. Although myoglobinopathy patients were reported not to have facial muscle weakness, our patient had orbicularis oculi muscle weakness, tongue weakness and atrophy, poor movement of the soft palate, and dysarthria. This is also the first reported case of tube feeding in a patient with myoglobinopathy. The patient started NPPV 18 years after onset, indicating that an older age of onset may have resulted in slow disease progression. Muscle selectivity, characteristic muscle pathology, and progressive cardiopulmonary dysfunction and dysphagia are hallmarks of this disease.
肌红蛋白尿症是一种罕见的常染色体显性遗传性肌病,成年起病,表现为近端和轴性肌无力,并伴有不同程度的呼吸和心力衰竭。与肌红蛋白尿症相关的肌肉病理学特征包括骨骼肌和心肌中特征性的肌浆小体。本文报道了首例亚洲人肌红蛋白尿症病例。尽管肌红蛋白尿症患者报道无面肌无力,但我们的患者存在眼轮匝肌无力、舌肌无力和萎缩、软腭运动不良和构音障碍。这也是首例肌红蛋白尿症患者行管饲的病例报道。该患者发病 18 年后开始使用 NPPV,提示发病年龄较晚可能导致疾病进展缓慢。肌肉选择性、特征性肌肉病理学以及进行性心肺功能障碍和吞咽困难是该疾病的标志。