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As one of the most multifaceted male infertility resulting from sperm problems, its genetic etiology remains unknown in most cases. In this review, we systematically sort out the latest literature on clinical reports and animal models leading to OAT, summarise the expression profiles of causative genes for OAT, and highlight the important role of the protein transport system during spermiogenesis, spermatid cell\u2010specific genes, Golgi and acrosome\u2010related genes, manchette\u2010related genes, HTCA\u2010related genes, and axoneme\u2010related genes in OAT development. These causative genes would be instrumental in genetic etiological screening, genetic counseling, and pre\u2010implantation genetic testing of patients with clinical OAT.<\/jats:p>","DOI":"10.1111\/cge.14652","type":"journal-article","created":{"date-parts":[[2025,1,9]],"date-time":"2025-01-09T02:46:35Z","timestamp":1736390795000},"page":"243-260","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Genetic Underpinnings of Oligoasthenoteratozoospermia"],"prefix":"10.1111","volume":"107","author":[{"given":"Yanting","family":"Feng","sequence":"first","affiliation":[{"name":"School of Medicine Yunnan University  Kunming Yunnan China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7527-5920","authenticated-orcid":false,"given":"Wensheng","family":"Liu","sequence":"additional","affiliation":[{"name":"Department of Reproductive Medicine, NHC Key Laboratory of Healthy Birth and 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