Whole-exome sequencing detects PYGM variants in two adults with McArdle disease.

TitleWhole-exome sequencing detects PYGM variants in two adults with McArdle disease.
Publication TypeJournal Article
Year of Publication2022
AuthorsThomas-Wilson A, Dharmadhikari AV, Heymann JJ, Jobanputra V, DiMauro S, Hirano M, Naini AB, Ganapathi M
JournalCold Spring Harb Mol Case Stud
Volume8
Issue2
Date Published2022 Feb
ISSN2373-2873
KeywordsAdolescent, 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.

DOI10.1101/mcs.a006173
Alternate JournalCold Spring Harb Mol Case Stud
PubMed ID35022222
PubMed Central IDPMC8958908
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