Myers Kenneth R, Liu Guanglu, Feng Yue, Zheng James Q
Department of Cell Biology, Emory University School of Medicine, Atlanta, Georgia, 30322.
Center for Neurodegenerative Diseases, Emory University School of Medicine, Atlanta, Georgia, 30322.
Dev Neurobiol. 2016 Sep;76(9):972-82. doi: 10.1002/dneu.22369. Epub 2015 Dec 22.
The selective RNA-binding protein Quaking I (QKI) has previously been implicated in RNA localization and stabilization, alternative splicing, cell proliferation, and differentiation. The spontaneously-occurring quakingviable (qkv) mutant mouse exhibits a sharply attenuated level of QKI in myelin-producing cells, including oligodendrocytes (OL) because of the loss of an OL-specific promoter. The disruption of QKI in OLs results in severe hypomyelination of the central nervous system, but the underlying cellular mechanisms remain to be fully elucidated. In this study, we used the qkv mutant mouse as a model to study myelination defects in the cerebellum. We found that oligodendroglial development and myelination are adversely affected in the cerebellum of qkv mice. Specifically, we identified an increase in the total number of oligodendroglial precursor cells in qkv cerebella, a substantial portion of which migrated into the grey matter. Furthermore, these mislocalized oligodendroglial precursor cells retained their migratory morphology late into development. Interestingly, a number of these presumptive oligodendrocyte precursors were found at the Purkinje cell layer in qkv cerebella, resembling Bergman glia. These findings indicate that QKI is involved in multiple aspects of oligodendroglial development. QKI disruption can impact the cell fate of oligodendrocyte precursor cells, their migration and differentiation, and ultimately myelination in the cerebellum. © 2015 Wiley Periodicals, Inc. Develop Neurobiol 76: 972-982, 2016.
选择性RNA结合蛋白震颤I(QKI)先前已被证明与RNA定位和稳定、可变剪接、细胞增殖及分化有关。自发产生的震颤存活(qkv)突变小鼠在包括少突胶质细胞(OL)在内的髓鞘生成细胞中,由于一个OL特异性启动子的缺失,QKI水平急剧下降。OL中QKI的破坏导致中枢神经系统严重髓鞘形成不足,但其潜在的细胞机制仍有待充分阐明。在本研究中,我们以qkv突变小鼠为模型,研究小脑的髓鞘形成缺陷。我们发现qkv小鼠小脑中少突胶质细胞的发育和髓鞘形成受到不利影响。具体而言,我们发现qkv小鼠小脑中少突胶质前体细胞总数增加,其中很大一部分迁移到灰质中。此外,这些定位错误的少突胶质前体细胞在发育后期仍保留其迁移形态。有趣的是,在qkv小鼠小脑的浦肯野细胞层发现了许多这些假定的少突胶质前体细胞,类似于伯格曼胶质细胞。这些发现表明QKI参与少突胶质细胞发育的多个方面。QKI的破坏会影响少突胶质前体细胞的细胞命运、迁移和分化,最终影响小脑的髓鞘形成。©2015威利期刊公司。《发育神经生物学》76:972 - 982,2016。