Expanding the phenotypic and genotypic spectrum of GGPS1 related congenital muscular dystrophy

Am J Med Genet A. 2024 Apr;194(4):e63498. doi: 10.1002/ajmg.a.63498. Epub 2023 Dec 21.

Abstract

Congenital muscular dystrophies are a group of progressive disorders with wide range of symptoms associated with diverse cellular mechanisms. Recently, biallelic variants in GGPS1 were linked to a distinct autosomal recessive form of muscular dystrophy associated with hearing loss and ovarian insufficiency. In this report, we present a case of a young patient with a homozygous variant in GGPS1. The patient presented with only proximal muscle weakness, and elevated liver transaminases with spared hearing function. The hepatic involvement in this patient caused by a novel deleterious variant in the gene extends the phenotypic and genotypic spectrum of GGPS1 related muscular dystrophy.

Keywords: GGPS1; hepatopathy; muscular dystrophy; myopathy; normal hearing; ovarian insufficiency.

Publication types

  • Case Reports

MeSH terms

  • Deafness*
  • Dimethylallyltranstransferase* / genetics
  • Farnesyltranstransferase / genetics
  • Female
  • Geranyltranstransferase / genetics
  • Hearing Loss*
  • Homozygote
  • Humans
  • Muscular Dystrophies* / diagnosis
  • Muscular Dystrophies* / genetics
  • Primary Ovarian Insufficiency*

Substances

  • GGPS1 protein, human
  • Dimethylallyltranstransferase
  • Geranyltranstransferase
  • Farnesyltranstransferase