Literature DB >> 31135626

Adult Diagnosis of Type 1 Fiber Predominance Myopathy Caused by Novel Mutations in the RYR1 Gene.

Leema Reddy Peddareddygari1, Kinsi Oberoi1, Leroy R Sharer2, Raji P Grewal3.   

Abstract

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.

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Year:  2019        PMID: 31135626     DOI: 10.1097/CND.0000000000000237

Source DB:  PubMed          Journal:  J Clin Neuromuscul Dis        ISSN: 1522-0443


  3 in total

1.  Correlation of Phenotype-Genotype and Protein Structure in RYR1-Related Myopathy.

Authors:  Xingzhi Chang; Risheng Wei; Cuijie Wei; Jieyu Liu; Lun Qin; Hui Yan; Yinan Ma; Zhaoxia Wang; Hui Xiong
Journal:  Front Neurol       Date:  2022-05-26       Impact factor: 4.086

Review 2.  Update on Congenital Myopathies in Adulthood.

Authors:  George Konstantinos Papadimas; Sophia Xirou; Evangelia Kararizou; Constantinos Papadopoulos
Journal:  Int J Mol Sci       Date:  2020-05-24       Impact factor: 5.923

3.  Clinical and genetic features of infancy-onset congenital myopathies from a Chinese paediatric centre.

Authors:  Yu Zhang; Hui Yan; Jieyu Liu; Huifang Yan; Yinan Ma; Cuijie Wei; Zhaoxia Wang; Hui Xiong; Xingzhi Chang
Journal:  BMC Pediatr       Date:  2022-01-26       Impact factor: 2.125

  3 in total

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