Key result
Novel compound heterozygous mutations in the RYR1 gene (c.10648C > T and c.10749_10753delGGAGG) were identified as the cause of mild, adult-onset type 1 fiber predominance myopathy in two siblings.
Case Report (n=2)
Identifies novel RYR1 mutations causing a mild, adult-onset type 1 fiber predominance myopathy.
May aid RYR1 testing in adult-onset myopathies; leaves open validation in larger cohorts.
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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Peddareddygari et al. (2019) conducted a case report in Type 1 Fiber Predominance Myopathy (n=2). RYR1 gene mutations (c.10648C > T and c.10749_10753delGGAGG) was evaluated on Diagnosis of myopathy. Novel compound heterozygous mutations in the RYR1 gene (c.10648C > T and c.10749_10753delGGAGG) were identified as the cause of mild, adult-onset type 1 fiber predominance myopathy in two siblings.
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