Percy A K, Zoghbi H, Riccardi V M
Brain Dev. 1985;7(3):300-4.
Five female patients have been presented with the typical clinical appearance of the Rett syndrome. A systematic evaluation of a variety of anatomic, physiologic and biochemical parameters has revealed two potential leads regarding the pathogenesis of this disorder. First, a reduction in the levels of neurotransmitter metabolites in cerebrospinal fluid was noted in each patient, a dopamine metabolite, homovanillic acid, being reduced to the greatest extent. Second, a sural nerve biopsy from one patient examined by electron microscopy revealed intact myelin with an axonopathy. Confirmation of these studies is necessary. This initial description of patients from the United States is quite consistent with previous reports by Professor Rett and Professor Hagberg and colleagues.
五名女性患者出现了雷特综合征的典型临床表现。对各种解剖学、生理学和生化参数进行的系统评估揭示了有关该疾病发病机制的两条潜在线索。首先,在每位患者中均发现脑脊液中神经递质代谢物水平降低,其中多巴胺代谢物高香草酸降低幅度最大。其次,对一名患者的腓肠神经活检进行电子显微镜检查发现髓鞘完整,但存在轴索性病变。这些研究结果需要得到证实。来自美国的这些患者的初步描述与雷特教授、哈格伯格教授及其同事之前的报告非常一致。