雷亚尔1
肌病
医学
肌肉活检
先天性肌病
近端肌无力
病理
外显子组测序
弱点
无症状的
突变
活检
遗传学
基因
内科学
解剖
生物
兰尼定受体
受体
作者
Leema Reddy Peddareddygari,Kinsi Oberoi,Leroy R. Sharer,Raji P. Grewal
标识
DOI:10.1097/cnd.0000000000000237
摘要
We describe a 57-year-old patient with mild diffuse weakness that was incidentally detected when he was evaluated for restless leg syndrome. An electromyography confirmed the presence of a myopathy without suggestion of inflammatory myopathy. A muscle biopsy demonstrated type 1 fiber predominance with minimal inflammatory features suggesting a genetic myopathy. Exome sequencing revealed c.10648C > T variant (p.R3550W), and a novel variant, c.10749_10753delGGAGG (E3584Rfs*3), in the ryanodine receptor 1 (RYR1) gene transmitted through his asymptomatic father indicating these mutations are in trans. Prompted by these results, a 47-year-old sister presented for evaluation. Her examination showed mild proximal muscle weakness, and an electromyography confirmed a noninflammatory myopathy. Her genotype was identical to her affected brother confirming that in these siblings, the RYR1 mutations, transmitted in an autosomal recessive pattern, are the cause of their myopathy. The adult age at diagnosis of these affected siblings likely reflects the mild and minimally progressive nature of the myopathy.
科研通智能强力驱动
Strongly Powered by AbleSci AI