Yoshida T, Nakagawa S I, Tabata K, Yanagisawa N
Department of Neurology, Saku Central Hospital.
Rinsho Shinkeigaku. 1994 Sep;34(9):925-7.
A 30-year-old man had signs of a lumbosacral skin plaque, called "shagreen patch", and mental retardation. He had been treated as genuine epilepsy for more than twenty years. Neither significant abnormalities nor facial angiofibroma (adenoma sebaceum) were detected on physical and neurological examinations. Brain CT revealed calcified subependymal nodules. A T2-weighted brain MRI presented high signal intensity regions affecting the cerebral cortex, which suggested tuberous lesions. Echocardiography showed high echoic lesions on the left ventricular wall and papillary muscles, suggesting calcification. The case was diagnosed as suffering from "forme fruste" of tuberous sclerosis. The three criteria typical of tuberous sclerosis (facial angiofibroma, seizure and mental retardation) were not satisfied. The "forme fruste" variant is less known, because the pattern of involvement varies, and it may clinically simulate genuine epilepsy.
一名30岁男性有腰骶部皮肤斑块体征,称为“鲨革斑”,并有智力发育迟缓。他被当作真性癫痫治疗了二十多年。体格检查和神经检查均未发现明显异常,也未发现面部血管纤维瘤(皮脂腺瘤)。脑部CT显示室管膜下结节钙化。脑部T2加权磁共振成像显示影响大脑皮层的高信号强度区域,提示有结节性病变。超声心动图显示左心室壁和乳头肌有高回声病变,提示钙化。该病例被诊断为结节性硬化症的“顿挫型”。结节性硬化症的三个典型标准(面部血管纤维瘤、癫痫发作和智力发育迟缓)均未满足。“顿挫型”变体不太为人所知,因为受累模式各不相同,而且在临床上可能类似于真性癫痫。