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Dysfunction in certain dynein proteins has been implicated in asthenoteratozoospermia, while others exclusively induce asthenozoospermia in the absence of overt morphological abnormalities in the sperm. Dynein axonemal heavy chain 10 (DNAH10), an inner dynein arm heavy chain protein, was identified as being associated with asthenoteratozoospermia in our previous studies. However, the mechanism through which DNAH10 contributes to this condition remains unclear. In this study, we demonstrated that DNAH10 deficiency leads to abnormal morphology of the sperm head and flagella. Additionally, DNAH10 dysfunction leads to impaired manchette function and aberrant localisation of axonemal proteins. Mechanistically, ubiquitin carboxyl-terminal hydrolase L3 (UCHL3) binds to parkin co-regulated gene protein (PACRG) and stabilises it via deubiquitination. In this process, DNAH10 exerts a bridging effect, enhancing the interaction between the UCHL3-PACRG complex to facilitate their involvement in manchette function. Collectively, this study demonstrated the function of DNAH10 in intra-manchette transport, providing important guidance for genetic diagnosis and prognosis in patients with infertility.<\/jats:p>","DOI":"10.1242\/dev.204906","type":"journal-article","created":{"date-parts":[[2025,10,8]],"date-time":"2025-10-08T08:47:07Z","timestamp":1759913227000},"source":"Crossref","is-referenced-by-count":1,"title":["DNAH10 interacts with UCHL3-PACRG complex to coordinate sperm head and flagella development during spermiogenesis"],"prefix":"10.1242","volume":"152","author":[{"given":"Rui","family":"Zheng","sequence":"first","affiliation":[{"name":"1 Joint Lab of Reproductive Medicine of SCU-CUHK, Lab of Reproductive Genetics and Epigenetics, Department of Obstetrics\/Gynecology, Key Laboratory of Birth Defects and Related Disease of Women and Children of MOE, West China Second University Hospital, Sichuan University, Chengdu 610041, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jierui","family":"Yan","sequence":"additional","affiliation":[{"name":"1 Joint Lab of Reproductive Medicine of SCU-CUHK, Lab of Reproductive Genetics and Epigenetics, Department of Obstetrics\/Gynecology, Key Laboratory of Birth Defects and Related Disease of Women and Children of MOE, West China Second University Hospital, Sichuan University, Chengdu 610041, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuting","family":"Wen","sequence":"additional","affiliation":[{"name":"1 Joint Lab of Reproductive Medicine of SCU-CUHK, Lab of Reproductive Genetics and Epigenetics, Department of Obstetrics\/Gynecology, Key Laboratory of Birth Defects and Related Disease of Women and Children of MOE, West China Second University Hospital, Sichuan University, Chengdu 610041, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yazhen","family":"Wei","sequence":"additional","affiliation":[{"name":"West China School of Pharmacy, Sichuan University 2 , Chengdu 610041 ,","place":["China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Weiwei","family":"Zhi","sequence":"additional","affiliation":[{"name":"Key Laboratory of Reproductive Medicine, Sichuan Provincial Maternity and Child Health Care Hospital 3 , Chengdu 610000 ,","place":["China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chaofeng","family":"Tu","sequence":"additional","affiliation":[{"name":"Institute of Reproductive and Stem Cell Engineering, School of Basic Medical Science, Central South University 4 , Changsha 410000 ,","place":["China"]},{"name":"Reproductive and Genetic Hospital of CITIC-Xiangya 5 , Changsha 410000 ,","place":["China"]},{"name":"Clinical Research Center for Reproduction and Genetics in Hunan Province 6 , Changsha 410000 ,","place":["China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yueqiu","family":"Tan","sequence":"additional","affiliation":[{"name":"Institute of Reproductive and Stem Cell Engineering, School of Basic Medical Science, Central South University 4 , Changsha 410000 ,","place":["China"]},{"name":"Reproductive and Genetic Hospital of CITIC-Xiangya 5 , Changsha 410000 ,","place":["China"]},{"name":"Clinical Research Center for Reproduction and Genetics in Hunan Province 6 , Changsha 410000 ,","place":["China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3686-229X","authenticated-orcid":false,"given":"Wenming","family":"Xu","sequence":"additional","affiliation":[{"name":"1 Joint Lab of Reproductive Medicine of SCU-CUHK, Lab of Reproductive Genetics and Epigenetics, Department of Obstetrics\/Gynecology, Key Laboratory of Birth Defects and Related Disease of Women and Children of MOE, West China Second University Hospital, Sichuan University, Chengdu 610041, China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2025,10,31]]},"reference":[{"key":"2026090108000397200_DEV204906C1","doi-asserted-by":"publisher","first-page":"1885","DOI":"10.3390\/cells10081885","article-title":"Ciliary dyneins and dynein related ciliopathies","volume":"10","author":"Antony","year":"2021","journal-title":"Cells"},{"key":"2026090108000397200_DEV204906C2","doi-asserted-by":"publisher","first-page":"89","DOI":"10.1016\/j.mce.2010.06.006","article-title":"UBQLN1 interacts with SPEM1 and participates in spermiogenesis","volume":"327","author":"Bao","year":"2010","journal-title":"Mol. 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