Adult Diagnosis of Type 1 Fiber Predominance Myopathy Caused by Novel Mutations in the RYR1 Gene.
J Clin Neuromuscul Dis
; 20(4): 214-216, 2019 Jun.
Article
in En
| MEDLINE
| ID: mdl-31135626
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.
Full text:
1
Collection:
01-internacional
Database:
MEDLINE
Main subject:
Ryanodine Receptor Calcium Release Channel
/
Muscular Diseases
Type of study:
Diagnostic_studies
Limits:
Humans
/
Male
/
Middle aged
Language:
En
Journal:
J Clin Neuromuscul Dis
Journal subject:
FISIOLOGIA
/
NEUROLOGIA
Year:
2019
Document type:
Article
Country of publication:
United States