Cain A R, Ryman B E
Gut. 1971 Nov;12(11):929-32. doi: 10.1136/gut.12.11.929.
A case of hereditary fructose intolerance is reported in a girl aged 2 years at the time of her death. She had apparently progressed normally until the age of 14 months. At 19 months she was admitted to hospital with failure to thrive, hepatomegaly, and superficial infections. Investigations revealed hypoglycaemia, persistent acidosis, aminoaciduria, and a high liver glycogen level which suggested that she had glycogen storage disease. There was also some evidence of malabsorption. At necropsy the liver enzyme estimations showed that fructose 1-phosphate aldolase activity was absent and that fructose 1,6-diphosphate aldolase activity was reduced. Hereditary fructose intolerance and glycogen storage disease have been confused in the past on clinical grounds, but a high liver glycogen level has not previously been reported in hereditary fructose intolerance.
报告了一例遗传性果糖不耐受病例,患儿为2岁女童,死亡时发病。她在14个月大之前发育明显正常。19个月时,因发育不良、肝肿大和浅表感染入院。检查发现低血糖、持续性酸中毒、氨基酸尿和肝糖原水平升高,提示患有糖原贮积病。也有一些吸收不良的证据。尸检时肝脏酶测定显示,1-磷酸果糖醛缩酶活性缺失,1,6-二磷酸果糖醛缩酶活性降低。过去,遗传性果糖不耐受和糖原贮积病在临床上容易混淆,但遗传性果糖不耐受此前尚未有肝糖原水平升高的报道。