Hou Yanan, Wu Zhimao, Zhang Yingying, Chen Huicheng, Hu Jinghua, Guo Yi, Peng Ying, Wei Qing
Laboratory for Reproductive Health, Institute of Biomedicine and Biotechnology, Shenzhen Institutes of Advanced Technology, Chinese Academy of Sciences (CAS), Shenzhen, China.
Chinese Academy of Sciences Key Laboratory of Insect Developmental and Evolutionary Biology, Chinese Academy of Sciences Center for Excellence in Molecular Plant Sciences, Institute of Plant Physiology and Ecology, Chinese Academy of Sciences, Shanghai, China.
Front Cell Dev Biol. 2020 May 21;8:301. doi: 10.3389/fcell.2020.00301. eCollection 2020.
Cilia and flagella are conserved subcellular organelles, which arise from centrioles and play critical roles in development and reproduction of eukaryotes. Dysfunction of cilia leads to life-threatening ciliopathies. HYLS1 is an evolutionarily conserved centriole protein, which is critical for ciliogenesis, and its mutation causes ciliopathy-hydrolethalus syndrome. However, the molecular function of HYLS1 remains elusive. Here, we investigated the function of HYLS1 in cilia formation using the model. We demonstrated that HYLS1 is a conserved centriole and basal body protein. Deletion of HYLS1 led to sensory cilia dysfunction and spermatogenesis abnormality. Importantly, we found that HYLS1 is essential for giant centriole/basal body elongation in spermatocytes and is required for spermatocyte centriole to efficiently recruit pericentriolar material and for spermatids to assemble the proximal centriole-like structure (the precursor of the second centriole for zygote division). Hence, by taking advantage of the giant centriole/basal body of spermatocyte, we uncover previously uncharacterized roles of HYLS1 in centriole elongation and assembly.
纤毛和鞭毛是保守的亚细胞器，起源于中心粒，在真核生物的发育和繁殖中发挥关键作用。纤毛功能障碍会导致危及生命的纤毛病。HYLS1是一种进化上保守的中心粒蛋白，对纤毛发生至关重要，其突变会导致纤毛病——水脑致死综合征。然而，HYLS1的分子功能仍不清楚。在这里，我们使用该模型研究了HYLS1在纤毛形成中的功能。我们证明HYLS1是一种保守的中心粒和基体蛋白。HYLS1的缺失导致感觉纤毛功能障碍和精子发生异常。重要的是，我们发现HYLS1对于精母细胞中巨大中心粒/基体的延长至关重要，并且是精母细胞中心粒有效募集中心粒周围物质以及精子组装近端中心粒样结构（合子分裂第二个中心粒的前体）所必需的。因此，通过利用精母细胞的巨大中心粒/基体，我们揭示了HYLS1在中心粒延长和组装中以前未被描述的作用。
