Kolodny E H, Cable W J
Ann Neurol. 1982 Mar;11(3):221-32. doi: 10.1002/ana.410110302.
Inborn errors of metabolism often cause neurological dysfunction. These disorders are most common in childhood, but adult-onset forms with a different clinical presentation are encountered, examples being Pompe disease, Tay-Sachs disease, metachromatic leukodystrophy, Gaucher disease, and Maroteaux-Lamy disease. In the evaluation of a patient with a possible inborn error of metabolism, simple screening tests may aid in the diagnosis and provide direction for more comprehensive laboratory analysis. In most cases, diagnosis can be established without a brain biopsy through biochemical and ultrastructural analysis of peripheral tissues, blood, and urine. New clinical, genetic, and biochemical variants of inherited metabolic disorders are being recognized through wider application of screening tests, improved specificity of laboratory analysis, cell complementation experiments, and the identification of enzyme activator factors. Accurate diagnosis is important for medical management, determining prognosis, and genetic counseling.
先天性代谢缺陷常导致神经功能障碍。这些疾病在儿童期最为常见,但也会遇到临床表现不同的成人发病形式,如庞贝病、泰-萨克斯病、异染性脑白质营养不良、戈谢病和马罗-拉米病。在评估可能患有先天性代谢缺陷的患者时,简单的筛查试验可能有助于诊断,并为更全面的实验室分析提供方向。在大多数情况下,通过对周围组织、血液和尿液进行生化和超微结构分析,无需进行脑活检即可确诊。通过更广泛地应用筛查试验、提高实验室分析的特异性、细胞互补实验以及鉴定酶激活因子,人们正在认识到遗传性代谢疾病新的临床、遗传和生化变异。准确诊断对于医疗管理、判断预后和遗传咨询都很重要。