Title | Whole-exome sequencing detects PYGM variants in two adults with McArdle disease. |
Publication Type | Journal Article |
Year of Publication | 2022 |
Authors | Thomas-Wilson A, Dharmadhikari AV, Heymann JJ, Jobanputra V, DiMauro S, Hirano M, Naini AB, Ganapathi M |
Journal | Cold Spring Harb Mol Case Stud |
Volume | 8 |
Issue | 2 |
Date Published | 2022 Feb |
ISSN | 2373-2873 |
Keywords | Adolescent, Adult, Exome Sequencing, Genotype, Glycogen Phosphorylase, Muscle Form, Glycogen Storage Disease Type V, Homozygote, Humans |
Abstract | McArdle disease is a debilitating glycogen storage disease with typical onset in childhood. Here, we describe a former competitive athlete with early adult-onset McArdle disease and a septuagenarian with a history of exercise intolerance since adolescence who was evaluated for proximal muscle weakness. Exome sequencing identified biallelic variants in the PYGM gene for both cases. The former athlete has the common, well-known pathogenic variant p.(Arg50Ter) in trans with a novel missense variant, p.(Asp694Glu). The second individual has a previously described homozygous missense variant, p.(Arg771Gln). Here, we describe the clinical course, enzyme-testing results using muscle tissue, and molecular findings for the individuals and add to the knowledge of the genotypic spectrum of this disorder. |
DOI | 10.1101/mcs.a006173 |
Alternate Journal | Cold Spring Harb Mol Case Stud |
PubMed ID | 35022222 |
PubMed Central ID | PMC8958908 |
Related Faculty:
Jonas Heymann, M.D.