Diagnosis of type 1a and type 1c glycogen storage diseases in adults

Lancet. 1987 May 9;1(8541):1059-62. doi: 10.1016/s0140-6736(87)90484-3.

Abstract

The hepatic glucose-6-phosphatase system was studied with a novel microanalytical technique in adult patients undergoing liver biopsy. 4 patients were diagnosed as having type 1 glycogen storage disease (GSD). 3 of these patients, who had hypoglycaemic symptoms, had variations of type 1a GSD, which is caused by a defect in the hepatic microsomal glucose-6-phosphatase enzyme. The fourth, with hepatomegaly and no hypoglycaemic symptoms, had a normal glucose-6-phosphatase enzyme but a defect in the hepatic microsomal phosphate/pyrophosphate translocase T2; this is the first report of an adult with type 1c GSD. Adult type 1 GSD should be considered in patients with unresolved hypoglycaemic symptoms and/or unresolved hepatomegaly.

Publication types

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

MeSH terms

  • Adult
  • Biopsy, Needle
  • Female
  • Glucose Tolerance Test
  • Glucose-6-Phosphatase / analysis
  • Glycogen Storage Disease / diagnosis*
  • Glycogen Storage Disease Type I / diagnosis*
  • Glycogen Storage Disease Type II / diagnosis*
  • Humans
  • Male
  • Microchemistry
  • Microsomes, Liver / analysis
  • Microsomes, Liver / enzymology
  • Microsomes, Liver / pathology
  • Middle Aged
  • Phosphoric Monoester Hydrolases / analysis

Substances

  • mannose-6-phosphatase
  • Phosphoric Monoester Hydrolases
  • Glucose-6-Phosphatase