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SCO-spondin淘汰赛小鼠表现出小脑室和轻微的脊柱变形

Huixin Xu, Guillaume P Dugué, Yasmine Cantaut-Belarif

bioRxiv : the preprint server for biology

|August 14, 2023

概括

SCO-脊柱蛋白 (SSPO) 形成赖斯纳纤维 (RF),对大脑发育至关重要. 在SSPO淘汰的小鼠中,心室尺寸缩小,这表明RF在调节大脑心室发育中的作用.

科学领域:

神经科学是一个神经科学.
发展生物学发展生物学
细胞外矩阵生物学细胞外矩阵生物学

背景情况:

赖斯纳纤维 (RF),由SCO-spondin (SSPO) 组成,是脑脊液 (CSF) 中的一个细胞外聚合物.
由于操纵大型SSPO基因的挑战,RF在哺乳动物发育中的作用尚未得到充分研究.

研究的目的:

研究SSPO和RF在哺乳动物发育中的功能.
为了生成和描述Sspo淘汰赛鼠标模型.

主要方法:

使用CRISPR/Cas9基因组编辑来创建Sspo淘汰赛小鼠.
现型分析包括对射频存在,大脑心室大小和脊柱曲率的评估.

主要成果:

在SSPO淘汰赛中,小鼠缺乏RF和相关的纤维状凝聚物.
在Sspo淘汰赛小鼠中,大脑心室的大小显著减少.
观察到轻微的胸脊曲缺陷,而不会影响运动行为.

结论:

在小鼠中,SSPO和RF对于正常大脑心室发育至关重要.
射频在调节脊髓几何学方面起到中等作用.

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