Nagase Tomoko, Shimozawa Nobuyuki, Takemoto Yasuhiko, Suzuki Yasuyuki, Komori Masayuki, Kondo Naomi
Department of Pediatrics, Gifu University School of Medicine, 40 Tsukasa-machi, Gifu 500-8705, Japan.
Biochim Biophys Acta. 2004 Mar 17;1671(1-3):26-33. doi: 10.1016/j.bbagen.2004.01.004.
In subjects with Zellweger syndrome, the most severe phenotype of peroxisomal biogenesis disorder, brain abnormalities include cortical dysplasia, neuronal heterotopia, and dysmyelination. To clarify the relationship between the lack of peroxisomes and neuronal abnormalities, we investigated peroxisomal localization in the mouse cerebellum, using double immunofluorescent staining for peroxisomal proteins. On immunostaining for peroxisomal matrix protein, while there are few peroxisomes in Purkinje cells, many locate in astroglia, especially soma of Bergmann glia. Clusters of peroxisomes were seen on the inferior side of the Purkinje cell layer in mice on postnatal days 3-5, and with time there was a shift to the superior side. The peroxisomal punctate pattern was seen to be radial and co-localized with Bergmann glial fibers. In cultured cells from the mouse cerebellum, peroxisomes were few in Purkinje cells, whereas many were evident in glial fibrillary acidic protein-positive cells. On the other hand, on immunostaining for peroxisomal membrane protein Pex14p, many particles were seen in Purkinje cells during all developmental stages, which means Purkinje cells possessed empty peroxisomal structures similar to findings of fibroblasts from the Zellweger patients. As peroxisomes in glial cells may control the development of neurons, the neuron-glial interaction and mechanisms of developing central nervous systems deserve ongoing attention.
在患有过氧化物酶体生物发生障碍最严重表型的脑肝肾综合征患者中,脑部异常包括皮质发育异常、神经元异位和髓鞘形成异常。为了阐明过氧化物酶体缺乏与神经元异常之间的关系,我们利用过氧化物酶体蛋白的双重免疫荧光染色,研究了小鼠小脑中过氧化物酶体的定位。在对过氧化物酶体基质蛋白进行免疫染色时,虽然浦肯野细胞中的过氧化物酶体很少,但许多过氧化物酶体位于星形胶质细胞中,尤其是伯格曼胶质细胞的胞体。在出生后第3至5天的小鼠中,在浦肯野细胞层的下侧可见过氧化物酶体簇,并且随着时间的推移,过氧化物酶体簇向上侧转移。过氧化物酶体的点状模式呈放射状,且与伯格曼胶质纤维共定位。在从小鼠小脑中培养的细胞中,浦肯野细胞中的过氧化物酶体很少,而在胶质纤维酸性蛋白阳性细胞中则有许多过氧化物酶体。另一方面,在对过氧化物酶体膜蛋白Pex14p进行免疫染色时,在所有发育阶段的浦肯野细胞中都可见到许多颗粒,这意味着浦肯野细胞拥有类似于脑肝肾综合征患者成纤维细胞的空过氧化物酶体结构。由于胶质细胞中的过氧化物酶体可能控制神经元的发育,神经元与胶质细胞的相互作用以及中枢神经系统发育的机制值得持续关注。