Castro de Kolster C, Rolo M, Arias S, Guerreiro N, Carvajal A, Castro J, Kolster J
Departamento de Pediatría, Universidad de Carabobo, Hospital Central de Valencia, Venezuela.
G E N. 1992 Jul-Sep;46(3):191-8.
Nine children with clinical diagnosis of glycogenoses were studied, types were confirmed through determination of levels and structure of glycogen, stimulation with glucagon and enzymatic defect analyses. Eight patients suffered glycogenoses type III and one, type VI. The major age group un type III was 1 to 2 years old (62.5%), the type VI was diagnosed in a preschool boy. Mean clinical features were: hepatomegaly, doll-like facies and short height. Major biochemical alterations were: transaminases elevation in both types, hypertriglyceridemia, hyperglycemia, metabolic acidosis and hyperuricemia only in glycogenoses III. One III type patient presented cardiovascular alterations. All patients showed increased concentrations of erythrocyte glycogen, with normal structure in type VI and abnormal in 75% of type III. Tree fourths of type III patients had a positive response to glucagon stimulation. No one presented glucose 6 phosphatase deficiency.
对9名临床诊断为糖原贮积病的儿童进行了研究,通过测定糖原水平和结构、胰高血糖素刺激试验以及酶缺陷分析来确诊类型。8例患者为Ⅲ型糖原贮积病,1例为Ⅵ型。Ⅲ型的主要年龄组为1至2岁(62.5%),Ⅵ型在一名学龄前男孩中确诊。主要临床特征为:肝肿大、娃娃脸和身材矮小。主要生化改变为:两型均有转氨酶升高、高甘油三酯血症、高血糖,仅Ⅲ型糖原贮积病有代谢性酸中毒和高尿酸血症。1例Ⅲ型患者出现心血管改变。所有患者红细胞糖原浓度均升高,Ⅵ型结构正常,Ⅲ型75%异常。四分之三的Ⅲ型患者对胰高血糖素刺激试验呈阳性反应。无人表现出葡萄糖6磷酸酶缺乏。