A novel COL6A2 mutation causing late-onset limb-girdle muscular dystrophy

J Neurol. 2019 Jul;266(7):1649-1654. doi: 10.1007/s00415-019-09307-y. Epub 2019 Apr 8.

Abstract

Limb-girdle muscular dystrophies (LGMD) are genetic disorders characterized by weakness of predominantly proximal limb and trunk muscles due to progressive loss of muscle tissue. Collagen VI-related muscular dystrophies usually display more generalized muscle involvement combined with contractures and/or hyperlaxity of distal finger joints. LGMD-like phenotype of collagenopathy has only rarely been described and as reported is usually of childhood onset. We identified a Finnish family with COL6A2-related LGMD with autosomal dominant inheritance and very late onset at 40-60 years of age. Since the mutation was previously unreported, the pathognomonic findings on muscle MRI were the decisive clue for the correct diagnosis.

Keywords: COL6A2; Collagenopathy; Limb-girdle muscular dystrophy; Myopathy.

Publication types

  • Case Reports

MeSH terms

  • Aged
  • Collagen Type VI / genetics*
  • Female
  • Humans
  • Male
  • Middle Aged
  • Muscular Dystrophies, Limb-Girdle / diagnostic imaging*
  • Muscular Dystrophies, Limb-Girdle / genetics*
  • Mutation / genetics*
  • Pedigree

Substances

  • COL6A2 protein, human
  • Collagen Type VI