Open AccessWDR62 is involved in spindle assembly by interacting with CEP170 in spermatogenesis
Author(s) -
Yan Qin,
Yang Zhou,
Zhiming Shen,
BaoHua Xu,
Min Chen,
Yaqiong Li,
Axel Behrens,
Jingjing Zhou,
Xin Qi,
Wenxiang Meng,
Yaqing Wang,
Fei Gao
Publication year - 2019
Publication title -
development
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 3.754
H-Index - 325
eISSN - 1477-9129
pISSN - 0950-1991
DOI - 10.1242/dev.174128
Subject(s) - biology , spermatogenesis , microbiology and biotechnology , evolutionary biology , genetics , endocrinology
WDR62 is the second most common genetic alteration associated with microcephaly. Our previous study demonstrates that Wdr62 is required for germ cell meiosis initiation and the majority of male germ cells are lost as the meiotic defect of first wave spermatogenesis. Strikingly, in this study, we found that the initiation of meiosis of following spermatogenesis was not affected and the germ cells were gradually repopulated at later developmental stages. However, most germ cells were arrested at metaphase of meiosis I and no mature sperm were detected in epididymides. Further studies demonstrated that metaphase I arrest of Wdr62-deficient spermatocytes was caused by asymmetrical distribution of the centrosome and aberrant spindle assembly. Mechanistic studies demonstrated that WDR62 interacted with centriole-associated protein CEP170, and deletion of Wdr62 caused downregulation of the CEP170 protein which in turn led to the aberrant spindle assembly. In summary, this study indicates that the meiosis of first wave spermatogenesis and the following spermatogenesis started from spermatogonium is probably regulated by different mechanisms. We also demonstrated a new function of WDR62 in germ cell meiosis by interacting with CEP170.