Katz M L, Sanders D N, Mooney B P, Johnson Gary S
Mason Eye Institute, University of Missouri School of Medicine, One Hospital Dr., Columbia, MO 65212, USA.
J Inherit Metab Dis. 2007 Nov;30(6):952-63. doi: 10.1007/s10545-007-0683-y. Epub 2007 Nov 15.
The neuronal ceroid lipofuscinoses (NCLs) are inherited neurodegenerative diseases characterized by massive accumulation of autofluorescent storage bodies in neurons and other cells. A late-onset form of NCL occurs in Tibetan terrier dogs. Gel electrophoretic analyses of isolated storage body proteins from brains of affected dogs indicated that a protein of approximately 50 kDa was consistently prominent and a 16 kDa component was present in some brain storage body preparations. Mass spectral analysis identified the 50 kDa protein as glial fibrillary acidic protein (GFAP), isoform 2. GFAP identification was supported by immunoblot and immunohistochemical analyses. Histone H4 was the major protein in the 16 kDa component. Specific accumulation of GFAP and histone H4 in storage bodies has not been previously reported for any of the NCLs. Tibetan terrier NCL may be the canine correlate of one of the human adult-onset NCLs for which the genetic bases and storage body compositions have not yet been determined.
神经元蜡样脂褐质沉积症(NCLs)是遗传性神经退行性疾病,其特征是在神经元和其他细胞中大量积累自发荧光储存体。一种迟发性NCL发生在西藏梗犬身上。对患病犬大脑中分离出的储存体蛋白进行凝胶电泳分析表明,一种约50 kDa的蛋白始终占主导地位,并且在一些脑储存体制剂中存在一种16 kDa的成分。质谱分析将50 kDa的蛋白鉴定为胶质纤维酸性蛋白(GFAP),异构体2。免疫印迹和免疫组织化学分析支持了GFAP的鉴定。组蛋白H4是16 kDa成分中的主要蛋白。此前尚未在任何一种NCL中报道过GFAP和组蛋白H4在储存体中的特异性积累。西藏梗犬NCL可能是人类成年发病的NCL之一的犬类对应物,其遗传基础和储存体组成尚未确定。