Lack of the C-terminal domain of nebulin in a patient with nemaline myopathy

Muscle Nerve. 2002 May;25(5):747-752. doi: 10.1002/mus.10097.

Abstract

The most common autosomal recessive form of nemaline myopathy is due to mutations in the nebulin gene. Among eight patients studied, we identified one, a 14-year-old girl, with a specific pattern of diffuse rods in muscle fibers. Western blot analysis detected absence of the C-terminal domain of nebulin. Protein analysis may represent a good screening method to direct molecular studies in the case of very large and complex genes such as the large 1298 kb nebulin gene.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Blotting, Western
  • Child
  • Child, Preschool
  • Female
  • Fluorescent Antibody Technique
  • Humans
  • Male
  • Muscle Fibers, Skeletal / pathology
  • Muscle Proteins / genetics*
  • Muscle Proteins / metabolism
  • Mutation / physiology*
  • Myopathies, Nemaline / genetics*
  • Myopathies, Nemaline / pathology

Substances

  • Muscle Proteins
  • nebulin