An Adult Case of Severe Hypoglycemia Induced by Partially Impaired Hepatic Fructose-1, 6-diphosphatase Activity

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We have experienced an adult case who had been attacked by frequent hypoglycemia on fasting. The onset of this illness was the middle of July 1976 (46 years old). Resection of the body and tail of the pancreas with the splenectomy was done under the diagnosis of insulinoma February 2, 1977. The operation revealed no insulinoma and after the operation hypoglycemia appeared as frequent as before. Oral glucose tolerance test, arginine infusion test or glucagon loading test revealed no hyperinsulinemia. Binding antibody to insulin or abnormal sized insulin was not found in the patient plasma. Oral glycerol loading (50g) could not raise the lowered blood glucose level in fasting state. Biopsied liver showed ballooning of hepatic cells by large lipid containing vacuoles. Liver glycogen was normal. The activity of hepatic fructose-1, 6-diphosphatase of this patient was 0.706±0.135 pmoles FDP/g/min (pH 7.5) which value was lower than those of control livers (1.621±0.802).<BR>Oral administration of folic acid (30 mg daily) was started June 10, 1978. This treatment improved the illness and the patient continues this therapy with almost normal social activity.<BR>The hypoglycemia in this case is thought to be induced by the partially impaired activity of hepatic fructose-1, 6-diphosphatase.
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