Tomiyasu H, Takahashi W, Ohta T, Yoshii F, Shibuya M, Shinohara Y
Department of Neurology, Tokai University School of Medicine.
Rinsho Shinkeigaku. 2000 Apr;40(4):350-7.
We reported an autopsy case of neuronal ceroid-lipofuscinosis (NCL3) with dilatated cardiomyopathy. A 29-year-old male patient first noticed night-blindness at the age of four years. He was pointed out retinitis pigmentosa at the age of six years and developed ataxia, mental retardation, epilepsy and myoclonus, thereafter. T1 weighted MRI showed diffuse atrophy of the cerebellum, brainstem, and cerebrum, and dilatation of the ventricular system and T2-weighted MRI showed mild high signal intensity in the white matter around the trigones of the lateral ventricles. Autopsy findings showed an abundant accumulation of ceroid-lipofuscin-like lipopigments in most neurons in the central nervous system, and curvilinear bodies and lipofuscin like granules were confirmed by electron microscopy. The heart muscle showed an increase in the accumulation of ceroid-lipofuscin-like lipopigments, severe fibrosis and fatty infiltration in the myocardium. The peculiar point of this case is NCL3 with dilated cardiomyopathy.
我们报告了一例伴有扩张型心肌病的神经元蜡样脂褐质沉积症(NCL3)尸检病例。一名29岁男性患者4岁时首次出现夜盲。6岁时被诊断为色素性视网膜炎,此后出现共济失调、智力发育迟缓、癫痫和肌阵挛。T1加权磁共振成像(MRI)显示小脑、脑干和大脑弥漫性萎缩,脑室系统扩张;T2加权MRI显示侧脑室三角区周围白质呈轻度高信号。尸检结果显示,中枢神经系统大多数神经元中大量积聚类蜡样脂褐质脂色素,电子显微镜证实存在曲线体和类脂褐质颗粒。心肌显示类蜡样脂褐质脂色素积聚增加,心肌严重纤维化和脂肪浸润。该病例的特殊之处在于NCL3合并扩张型心肌病。