Very late-onset limb-girdle muscular dystrophy type 2D: A milder form with a normal muscle biopsy

J Clin Neurosci. 2020 Feb:72:471-473. doi: 10.1016/j.jocn.2019.12.003. Epub 2019 Dec 10.

Abstract

Sarcoglycanopathies are a genetically heterogeneous group of autosomal recessive limb-girdle muscular dystrophies (LGMD) caused by mutations in sarcoglycan genes. We report a Portuguese patient with a very late-onset LGMD phenotype, whose muscle biopsy and immunostaining, in particular for α-sarcoglycan, were unrevealing. Muscle MRI showed a predominant, bilateral and symmetric involvement of the tight muscles and also, to a lesser extent, of the posterior compartment of lower legs muscles. Next generation sequencing (NGS) revealed a known homozygous c.850C > T (p.Arg284Cys) mutation in SGCA gene. Milder forms of α-sarcoglycanopathies could be a challenging diagnosis; particularly if muscle histopathology and α-sarcoglycan immunohistochemistry are unhelpful. NGS plays a crucial role not only for aiding in the establishment of a definite diagnosis, but also for expanding clinical presentations.

Keywords: Limb-girdle muscular dystrophy type 2D; Next generation sequencing; α-Sarcoglycan immunohistochemistry; α-Sarcoglycanopathy.

Publication types

  • Case Reports

MeSH terms

  • Aged
  • Biopsy
  • Humans
  • Magnetic Resonance Imaging
  • Male
  • Muscle, Skeletal / pathology
  • Muscular Dystrophies, Limb-Girdle / genetics*
  • Muscular Dystrophies, Limb-Girdle / pathology*
  • Mutation
  • Phenotype
  • Sarcoglycanopathies / genetics*
  • Sarcoglycanopathies / pathology*
  • Sarcoglycans / genetics

Substances

  • Sarcoglycans

Supplementary concepts

  • Limb-girdle muscular dystrophy autosomal recessive