Farmer P M
Ann Clin Lab Sci. 1982 Nov-Dec;12(6):431-8.
Of the 12 known genetic disorders of glycogen metabolism, five consistently involve the neuromuscular system. Pompe's disease is a generalized, fatal, lysosomal storage disease caused by absence of acid maltase. Structurally abnormal glycogen accumulates in Forbes-Cori and Andersen's diseases, resulting from deficient debranching and branching enzymes, respectively. Exercise intolerance, muscle cramps, and myoglobinuria characterize McArdle's syndrome or myophosphorylase deficiency. In Tauri's disease, absence of phosphofructokinase leads to glycogen accumulation indirectly owing to a metabolic block in glycolysis. Diagnosis of the symptomatic patient, antenatal diagnosis, and detection of heterozygous genetic carriers are accomplished using a variety of laboratory methods. Tissue enzyme assays, chemical analysis of glycogen, and studies of carbohydrate metabolism are available. Recent advances in biophysics, such as nuclear magnetic resonance, have opened up a new approach for the study of metabolic diseases.
在已知的12种糖原代谢遗传性疾病中,有5种始终累及神经肌肉系统。庞贝氏病是一种全身性、致命的溶酶体贮积病,由酸性麦芽糖酶缺乏引起。结构异常的糖原分别在福布斯 - 科里病和安徒生病中积累,这两种病分别是由脱支酶和分支酶缺乏所致。运动不耐受、肌肉痉挛和肌红蛋白尿是麦克尔综合征或肌磷酸化酶缺乏症的特征。在塔auri病中,磷酸果糖激酶缺乏由于糖酵解代谢阻滞而间接导致糖原积累。有症状患者的诊断、产前诊断以及杂合子遗传携带者的检测可通过多种实验室方法完成。可进行组织酶测定、糖原化学分析以及碳水化合物代谢研究。生物物理学的最新进展,如核磁共振,为代谢疾病的研究开辟了新途径。