Functional Analysis of Hydrolethalus Syndrome Protein HYLS1 in Ciliogenesis and Spermatogenesis in Drosophila
Functional Analysis of Hydrolethalus Syndrome Protein HYLS1 in Ciliogenesis and Spermatogenesis in Drosophila
复制标题
Hydrolethalus综合征蛋白HYLS1在果蝇纤毛发生和精子发生中的功能分析
DOI：
10.3389/fcell.2020.00301
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发表时间：
2020-05-21
影响因子：
5.5
通讯作者：
Wei, Qing
中科院分区：
文献类型：
--
作者：
Hou, Yanan;Wu, Zhimao;Wei, Qing
纤毛和鞭毛是由中心粒产生的保守的亚细胞器，在真核生物的发育和繁殖中起着至关重要的作用。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，在纤毛发生中起关键作用，它的突变会导致睫状肌病变-脑积水综合征。然而，HYLS1的分子功能仍然难以捉摸。在这里，我们利用果蝇模型研究了HYLS1在纤毛形成中的功能。我们证明果蝇HYLS1是一种保守的中心粒和基底体蛋白。HYLS1基因缺失导致感觉纤毛功能障碍和精子发生异常。重要的是，我们发现果蝇HYLS1对于精母细胞中巨大的中心粒/基体的伸长是必不可少的，并且是精母细胞中心粒有效地招募中心粒周围物质和精子细胞组装近端中心粒样结构(第二中心粒用于合子分裂的前体)所必需的。因此，通过利用果蝇精母细胞的巨大中心粒/基本体，我们揭示了HYLS1在中心粒伸长和组装中先前未知的作用。
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 Drosophila model. We demonstrated that Drosophila HYLS1 is a conserved centriole and basal body protein. Deletion of HYLS1 led to sensory cilia dysfunction and spermatogenesis abnormality. Importantly, we found that Drosophila 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 Drosophila spermatocyte, we uncover previously uncharacterized roles of HYLS1 in centriole elongation and assembly.