Perioperative management of benign hepatic tumors in patients with glycogen storage disease type Ia

J Hepatobiliary Pancreat Surg. 2008;15(2):200-3. doi: 10.1007/s00534-007-1244-3. Epub 2008 Apr 6.

Abstract

Glycogen storage disease type Ia (GSD-Ia; von Gierke disease) is an inherited disorder caused by glucose-6-phosphatase deficiency, and there have been some reports of hepatic tumors in patients with this disease. We report two patients with benign hepatic tumors with GSD-Ia. One is a 19-year-old man who underwent segmentectomy 4 for a focal nodular hyperplasia, and the other is a 31-year-old woman who underwent segmentectomies 3, 5, and 6 for hepatic adenomas. Two significant perioperative complications, resulting from the carbohydrate metabolic disorders, hypoglycemia and metabolic acidosis, occurred in both patients. We managed the metabolic complications successfully by administering a sufficient volume of glucose intravenously. Close perioperative monitoring of blood glucose and lactate concentrations is essential in the perioperative management of patients with GSD-Ia. The intravenous administration of glucose, starting with a smaller dose and then increasing the dose, is adequate management for lactic acidosis with or without hypoglycemia during the perioperative period.

Publication types

  • Case Reports

MeSH terms

  • Acidosis / etiology
  • Acidosis / therapy
  • Adenoma, Liver Cell / complications
  • Adult
  • Female
  • Focal Nodular Hyperplasia / complications
  • Glucose Metabolism Disorders / etiology
  • Glucose Metabolism Disorders / therapy
  • Glycogen Storage Disease Type I / complications*
  • Hepatectomy / adverse effects*
  • Humans
  • Hypoglycemia / etiology
  • Hypoglycemia / therapy
  • Liver Diseases / complications
  • Liver Diseases / surgery*
  • Liver Neoplasms / complications
  • Liver Neoplasms / surgery
  • Male
  • Perioperative Care