Narisawa K, Igarashi Y, Tada K
Department of Pediatrics, Tohoku University School of Medicine, Sendai, Japan.
Enzyme. 1987;38(1-4):177-83. doi: 10.1159/000469203.
Glycogen storage diseases (GSD) type 1b is the first example of a genetic disorder involving the transport system of an intracellular membrane. It was revealed that the primary defect in GSD type 1b was a deficiency in the microsomal glucose-6-phosphate (G6P) translocase, based on the findings that the glucose-6-phosphatase activity was highly latent in the fresh liver homogenates. Further evidence of this defect in GSD type 1b has been provided by a membrane filter method which measures the uptake of 14C-G6P by microsomes. The clinical symptoms and enzymatic studies in our patients suggest that there is genetic heterogeneity in GSD 1b and the clinical severity depends on the level of residual activities of G6P translocase.
1b型糖原贮积病(GSD)是涉及细胞内膜转运系统的遗传性疾病的首个实例。基于新鲜肝脏匀浆中葡萄糖-6-磷酸酶活性高度潜伏的研究结果,发现1b型GSD的主要缺陷是微粒体葡萄糖-6-磷酸(G6P)转位酶缺乏。通过测量微粒体对14C-G6P摄取的膜滤过法,为1b型GSD的这一缺陷提供了进一步证据。我们患者的临床症状和酶学研究表明,1b型GSD存在遗传异质性,临床严重程度取决于G6P转位酶的残余活性水平。