Bornemann A, Besser R, Shin Y S, Goebel H H
Division of Neuropathology, University of Mainz, Germany.
Neuromuscul Disord. 1996 Mar;6(2):95-9. doi: 10.1016/0960-8966(95)00031-3.
Type IV glycogenosis is usually a rapidly progressive disease of early childhood, causing death before 4 years of age. It is characterized by hepatosplenomegaly, cirrhosis, and chronic hepatic failure. Muscle involvement is generally overshadowed by liver disease. A mild non-infantile variant of type IV glycogenosis has been described in a few patients. In some of them, the patients suffered foremost from chronic progressive myopathy. We here report on a female patient aged 51 years who had experienced difficulties in climbing stairs for 2 years due to leg weakness. EMG revealed a myopathic pattern. The muscle biopsy findings revealed polyglycosan bodies. Biochemical investigation showed absence of branching enzyme in muscle but not in leukocytes and fibroblasts.
IV型糖原贮积病通常是一种幼儿期快速进展的疾病,常在4岁前导致死亡。其特征为肝脾肿大、肝硬化和慢性肝衰竭。肌肉受累通常被肝脏疾病掩盖。少数患者中描述了一种轻度非婴儿型IV型糖原贮积病变异型。其中一些患者主要患有慢性进行性肌病。我们在此报告一名51岁女性患者,她因腿部无力爬楼梯困难已有2年。肌电图显示为肌病模式。肌肉活检结果显示有多聚糖体。生化检查表明肌肉中缺乏分支酶,但白细胞和成纤维细胞中不缺乏。