Nakamura I, Kurachi M, Fukutani Y, Kawasaki Y, Yamaguchi N, Torii H
Department of Neuropsychiatry, Kanazawa University School of Medicine, Japan.
Jpn J Psychiatry Neurol. 1988 Jun;42(2):277-89. doi: 10.1111/j.1440-1819.1988.tb01978.x.
We report here the clinical features of 3 affected siblings and neuropathological findings of the CNS from an autopsied case among them. Their common clinical features consisted of cerebellar ataxia and tremors through movements and postures. Two of the 3 siblings showed autonomic abnormalities, hard-of-hearing, pyramidal sign and areflexia. Then they always had xanthochromia and an elevated protein without pleocytosis in their CSFs. Neuropathologically, intracytoplasmic eosinophilic inclusion bodies were found in the neurons of some restricted nuclei or nerve cell groups of the brain stem, cerebellum and spinal cord. Similar bodies appeared also in glial, ependymal and choroid plexus epithelial cells. Such eosinophilic bodies are thought to consist of protein-rich substances from histochemical properties, and result from an unknown inherited metabolic error.
我们在此报告3名患病兄弟姐妹的临床特征以及其中1例尸检病例中枢神经系统的神经病理学发现。他们的共同临床特征包括运动和姿势方面的小脑共济失调和震颤。3名兄弟姐妹中有2名出现自主神经异常、听力减退、锥体束征和无反射。此外,他们的脑脊液始终存在黄变现象,蛋白升高但无细胞增多。神经病理学检查发现,在脑干、小脑和脊髓的一些特定核团或神经细胞群的神经元中存在胞浆内嗜酸性包涵体。类似的包涵体也出现在神经胶质细胞、室管膜细胞和脉络丛上皮细胞中。从组织化学特性来看,这种嗜酸性包涵体被认为由富含蛋白质的物质组成,是由一种未知的遗传性代谢错误导致的。