Glycogenosis type IB: possible membrane transport defect

South Med J. 1981 Jun;74(6):761-4. doi: 10.1097/00007611-198106000-00033.

Abstract

We have described a 20-month-old child with type IB glycogen storage disease, based on clinical and biochemical manifestations. Functional testing data were similar to those found in glucose-6-phosphatase deficiency, but in vitro studies showed normal hepatic glucose-6-phosphatase activity. Disruption of membranes with deoxycholic acid was followed by an increase in enzyme activity compared to a control liver tissue, suggesting "latency" of enzyme. We suggest that this patient had glycogen storage type IB and that this disorder may represent a specific glucose-6-phosphate transport defect.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Biological Transport, Active / drug effects
  • Blood Glucose / metabolism
  • Deoxycholic Acid / pharmacology
  • Female
  • Glucose-6-Phosphatase / metabolism
  • Glycogen Storage Disease Type I / etiology*
  • Glycogen Storage Disease Type I / metabolism
  • Humans
  • Lactates / blood
  • Liver / drug effects
  • Liver / enzymology

Substances

  • Blood Glucose
  • Lactates
  • Deoxycholic Acid
  • Glucose-6-Phosphatase