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Its genetic basis in many affected individuals remains unknown. Here, we found that <jats:italic>CCDC157<\/jats:italic> variants are associated with OAT. In two cohorts, a 21\u2010bp (g.30768132_30768152del21) and\/or 24\u2010bp (g.30772543_30772566del24) deletion of CCDC157 were identified in five sporadic OAT patients, and 2 cases within one pedigree. In a mouse model, loss of <jats:italic>Ccdc157<\/jats:italic> led to male sterility with OAT\u2010like phenotypes. Electron microscopy revealed misstructured acrosome and abnormal head\u2013tail coupling apparatus in the sperm of <jats:italic>Ccdc157<\/jats:italic>\u2010null mice. Comparative transcriptome analysis showed that the <jats:italic>Ccdc157<\/jats:italic> mutation alters the expressions of genes involved in cell migration\/motility and Golgi components. Abnormal Golgi apparatus and decreased expressions of genes involved in acrosome formation and lipid metabolism were detected in <jats:italic>Ccdc157<\/jats:italic>\u2010deprived mouse germ cells. Interestingly, we attempted to treat infertile patients and <jats:italic>Ccdc157<\/jats:italic> mutant mice with a Chinese medicine, Huangjin Zanyu, which improved the fertility in one patient and most mice that carried the heterozygous mutation in <jats:italic>CCDC157.<\/jats:italic> Healthy offspring were produced. Our study reveals CCDC157 is essential for sperm maturation and may serve as a marker for diagnosis of OAT.<\/jats:p>","DOI":"10.1111\/jcmm.18215","type":"journal-article","created":{"date-parts":[[2024,3,21]],"date-time":"2024-03-21T02:51:10Z","timestamp":1710989470000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["<scp>CCDC157<\/scp> is essential for sperm differentiation and shows oligoasthenoteratozoospermia\u2010related mutations in men"],"prefix":"10.1111","volume":"28","author":[{"given":"Huimei","family":"Zheng","sequence":"first","affiliation":[{"name":"Division of Human Reproduction and Developmental Genetics, the Women's Hospital Zhejiang University School of Medicine  Hangzhou China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Chenjia","family":"Gong","sequence":"additional","affiliation":[{"name":"Hefei National Laboratory for Physical Sciences at Microscale, the First Affiliated 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