A novel mutation in the GNE gene and a linkage disequilibrium in Japanese pedigrees

Ann Neurol. 2002 Oct;52(4):516-9. doi: 10.1002/ana.10341.

Abstract

Distal myopathy with rimmed vacuoles (DMRV) is an autosomal recessive muscular disorder characterized by weakness of the anterior compartment of the lower limbs with onset in early adulthood and sparing of the quadricep muscles. The UDP-N-acetylglucosamine-2-epimerase/N-acetylmannosamine kinase (GNE) gene was recently identified as the causative gene for hereditary inclusion body myopathy (HIBM). To investigate whether DMRV and HIBM are allelic diseases, we conducted mutational analysis of the GNE gene of six Japanese DMRV pedigrees and found that all the pedigrees share a homozygous mutation (V572L) associated with a strong linkage disequilibrium, suggesting a strong founder effect in Japanese DMRV pedigrees.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Carbohydrate Epimerases / genetics*
  • Founder Effect
  • Haplotypes
  • Humans
  • Japan
  • Linkage Disequilibrium*
  • Muscular Dystrophies / genetics*
  • Muscular Dystrophies / pathology
  • Mutation
  • Pedigree
  • Phosphotransferases (Alcohol Group Acceptor) / genetics*
  • Vacuoles / pathology

Substances

  • Phosphotransferases (Alcohol Group Acceptor)
  • N-acylmannosamine kinase
  • Carbohydrate Epimerases