Dystrophic Myopathy of the Diaphragm with Recurrent Severe Respiratory Failure is Congenital Myasthenic Syndrome 11.

J J Kramer, H T M Boon, Q H Leijten,Henk Ter Laak, L Eshuis, B Kusters,J L M van Doorn, E J Kamsteeg, B Eymard,J Doorduin,N C Voermans

Journal of neuromuscular diseases(2023)

引用 0|浏览7
暂无评分
摘要
We here present the case of a patient with a congenital myasthenic syndrome (CMS) due to pathogenic variants in the RAPSN gene. During childhood he experienced recurrent episodes of respiratory failure during respiratory infections. This and other cases were reported as isolated dystrophy of the diaphragmatic musculature. In adulthood, whole exome sequencing revealed two heterozygous pathogenic variants in the RAPSN gene. This led to the revision of the diagnosis to rapsyn CMS11 (OMIM:616326, MONDO:0014588). EMG, muscle ultrasound and the revision of muscle biopsies taken in childhood support this diagnosis. After the revision of the diagnosis, treatment with pyridostigmine was started. This resulted in a reduction of fatigability and an improvement in functional abilities and quality of life.
更多
查看译文
关键词
CMS11,Congenital myasthenic syndrome,RAPSN,Rapsyn,Whole exome sequencing,diaphragmatic dystrophy
AI 理解论文
溯源树
样例
生成溯源树,研究论文发展脉络
Chat Paper
正在生成论文摘要