Biochemical diagnosis of type 1b glycogen storage disease

Aust Paediatr J. 1984 Aug;20(3):217-20. doi: 10.1111/j.1440-1754.1984.tb00082.x.

Abstract

A child with the classical signs and symptoms of Type 1 glycogen storage disease is presented, who on investigation was shown to have a recently described variant of this disease known as Type 1b glycogen storage disease. A reliable and simple procedure for the diagnosis and differentiation of Types 1 and 1b glycogen storage disease is described, as the conventional diagnostic approach of assaying glucose-6-phosphate phosphohydrolase in frozen tissue will not diagnose Type 1b glycogen storage disease. A portion of biopsy tissue should be maintained at a temperature near 0 degrees C (but not frozen) and the remainder frozen. Glucose-6-phosphate phosphohydrolase assays are carried out on the tissue homogenates of both portions. In Type 1 glycogen storage disease, glucose-6-phosphate phosphohydrolase activity will be low or absent in both frozen and unfrozen tissues. In Type 1b glycogen storage disease the frozen tissue homogenate will exhibit normal glucose-6-phosphate phosphohydrolase activity due to the disruption of the microsomes by ice crystals, while in the unfrozen tissue low levels of glucose-6-phosphate phosphohydrolase activity will be detected.

Publication types

  • Case Reports

MeSH terms

  • Clinical Enzyme Tests
  • Diagnosis, Differential
  • Female
  • Glycogen Storage Disease Type I / diagnosis*
  • Glycogen Storage Disease Type I / metabolism
  • Histocytochemistry
  • Humans
  • Infant
  • Infant, Newborn
  • Liver / enzymology
  • Liver Glycogen / metabolism
  • Phosphoric Monoester Hydrolases / deficiency
  • Phosphoric Monoester Hydrolases / metabolism*

Substances

  • Liver Glycogen
  • Phosphoric Monoester Hydrolases
  • sugar-phosphatase