Méndez Aparicio F M
An Esp Pediatr. 1980 Oct;13(10):901-8.
A case of liver glycogen storage disease with amylo 1,6-glucosidase deficiency is reported. Enlarged liver was found at birth, and it is now accompanied by splenomegaly, low fasting blood glucose with ketonuria, elevation of transaminase values and glycogen accumulation with connective periportal tissue in liver histological study. In this glucogenosis results of functional tests on carbohidrate metabolism and glycogen enzymatic assay showed a direct relationship between functional and biochemical behaviour of liver cells. Amylo 1,6-glucosidase deficiency is accompanied by absence of glucogenolysis when glucagon is administrated after a long fast, and an increase of blood glucose when glucagon is administrated after food ingestion. Glycolisis tests show blood lactate elevation when some hexose or alanine are administrated; glyconeogenesis tests show blood glucose elevation when hexose, alanine or glycerol are administrated.
报告了一例伴有淀粉-1,6-葡萄糖苷酶缺乏的肝糖原贮积病病例。患儿出生时即发现肝脏肿大,目前伴有脾肿大、空腹血糖低伴酮尿、转氨酶值升高以及肝脏组织学研究显示糖原在肝门周围结缔组织中蓄积。在这种糖原贮积病中,碳水化合物代谢功能试验和糖原酶分析结果表明肝细胞的功能和生化行为之间存在直接关系。淀粉-1,6-葡萄糖苷酶缺乏伴有长时间禁食后注射胰高血糖素时糖原分解缺失,以及进食后注射胰高血糖素时血糖升高。糖酵解试验显示,给予某些己糖或丙氨酸时血乳酸升高;糖异生试验显示,给予己糖、丙氨酸或甘油时血糖升高。