Myopathic evolution of an exertional muscle pain syndrome with phosphorylase b kinase deficiency

Acta Neuropathol. 1990;81(1):84-8. doi: 10.1007/BF00662642.

Abstract

The symptoms of a myopathy permanently affecting limb girdle muscles are reported in a 31-year-old woman who has been presenting an exertional muscle pain syndrome with myoglobinuria for 20 years. Investigations revealed a slightly decreased utilization of glycogen in muscle, while its storage affected only rare type 2 fibers. Active phosphorylase was undetectable and phosphorylase b kinase activity was clearly decreased in muscle cells, but normal in erythocytes, lymphocytes and cultured fibroblasts.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Atrophy
  • Female
  • Glycogen / analysis
  • Glycogen Storage Disease / enzymology
  • Glycogen Storage Disease / pathology*
  • Glycogen Storage Disease / physiopathology
  • Glycolysis
  • Humans
  • Microscopy, Electron
  • Muscles / metabolism
  • Muscles / pathology*
  • Muscles / ultrastructure
  • Muscular Diseases / etiology
  • Muscular Diseases / pathology*
  • Muscular Diseases / physiopathology
  • Pain / etiology
  • Pain / metabolism
  • Pain / pathology*
  • Phosphorylase Kinase / deficiency*
  • Phosphorylase Kinase / metabolism
  • Phosphorylase b / metabolism
  • Physical Exertion
  • Syndrome

Substances

  • Glycogen
  • Phosphorylase b
  • Phosphorylase Kinase