Mahmoud Shekari Khaniani, Khorrami Aziz, Rafeey Mandana, Ghergherehchi Robabeh, Sima Mansoori Derakhshan
Liver and Gastrointestinal Disease Research Center, Tabriz University of Medical Sciences, Tabriz 5166/15731, Iran.
J Genet. 2017 Mar;96(1):19-23. doi: 10.1007/s12041-016-0734-y.
Glycogen storage diseases (GSDs) are caused by abnormalities in enzymes that are involved in the regulation of gluconeogenesis and glycogenolysis. GSD I, an autosomal recessive metabolic disorder, is the most common GSD and has four subtypes. Here, we examined GSD Ia caused by the defective glucose-6-phosphatase catalytic (G6PC) gene. We investigated the frequency of GSD Ia and clarified its molecular aspect in patients with the main clinical and biochemical characteristics of GSD, including 37 unrelated patients with a mean age of three years at the time of diagnosis. All patients belonged to the Azeri Turkish population. Hypoglycaemia and hypertriglyceridaemia were the most frequent laboratory findings. Mutations were detected by performing direct sequencing. Mutation analysis of the G6PC gene revealed that GSD Ia accounted for 11% in GSD patients with involvement of liver. Three patients were homozygous for R83C mutation. In addition, a novel stop mutation, Y85X, was identified in a patient with the typical features of GSD Ia.
糖原贮积病(GSDs)是由参与糖异生和糖原分解调节的酶异常引起的。GSD I是一种常染色体隐性代谢紊乱疾病,是最常见的糖原贮积病,有四种亚型。在此,我们研究了由缺陷型葡萄糖-6-磷酸酶催化亚基(G6PC)基因引起的GSD Ia。我们调查了GSD Ia的发病率,并在具有GSD主要临床和生化特征的患者中阐明了其分子层面情况,这些患者包括37名无亲缘关系的患者,诊断时平均年龄为三岁。所有患者均为阿塞拜疆土耳其族人群。低血糖和高甘油三酯血症是最常见的实验室检查结果。通过直接测序检测突变。G6PC基因的突变分析显示,GSD Ia在累及肝脏的GSD患者中占11%。三名患者为R83C突变纯合子。此外,在一名具有GSD Ia典型特征的患者中发现了一种新的终止突变Y85X。