AUTHOR=Hou Yanan , Wu Zhimao , Zhang Yingying , Chen Huicheng , Hu Jinghua , Guo Yi , Peng Ying , Wei Qing 

TITLE=Functional Analysis of Hydrolethalus Syndrome Protein HYLS1 in Ciliogenesis and Spermatogenesis in Drosophila

JOURNAL=Frontiers in Cell and Developmental Biology

VOLUME=Volume 8 - 2020

YEAR=2020

URL=https://www.frontiersin.org/journals/cell-and-developmental-biology/articles/10.3389/fcell.2020.00301

DOI=10.3389/fcell.2020.00301

ISSN=2296-634X

ABSTRACT=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 defects in sensory cilia and in spermatogenesis. 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 (PCM) and for spermatids to assemble the proximal centriole-like structure (PCL, 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.