{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,23]],"date-time":"2026-09-23T23:46:24Z","timestamp":1790207184672,"version":"4.1.0"},"reference-count":56,"publisher":"MDPI AG","issue":"12","license":[{"start":{"date-parts":[[2024,6,18]],"date-time":"2024-06-18T00:00:00Z","timestamp":1718668800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Consiglio Nazionale delle Ricerche Progetto d\u2019 Interesse Strategico Invecchiamento 2012\u20132016","award":["DSB.AD009.001"],"award-info":[{"award-number":["DSB.AD009.001"]}]},{"name":"Consiglio Nazionale delle Ricerche Progetto d\u2019 Interesse Strategico Invecchiamento 2012\u20132016","award":["261492"],"award-info":[{"award-number":["261492"]}]},{"name":"Consiglio Nazionale delle Ricerche Progetto d\u2019 Interesse Strategico Invecchiamento 2012\u20132016","award":["223263"],"award-info":[{"award-number":["223263"]}]},{"name":"Consiglio Nazionale delle Ricerche Progetto d\u2019 Interesse Strategico Invecchiamento 2012\u20132016","award":["ME.P05.001.003"],"award-info":[{"award-number":["ME.P05.001.003"]}]},{"name":"European Union Framework Programmes 6\u20137: EUCOMM and EUCOMMTools","award":["DSB.AD009.001"],"award-info":[{"award-number":["DSB.AD009.001"]}]},{"name":"European Union Framework Programmes 6\u20137: EUCOMM and EUCOMMTools","award":["261492"],"award-info":[{"award-number":["261492"]}]},{"name":"European Union Framework Programmes 6\u20137: EUCOMM and EUCOMMTools","award":["223263"],"award-info":[{"award-number":["223263"]}]},{"name":"European Union Framework Programmes 6\u20137: EUCOMM and EUCOMMTools","award":["ME.P05.001.003"],"award-info":[{"award-number":["ME.P05.001.003"]}]},{"name":"PHENOSCALE","award":["DSB.AD009.001"],"award-info":[{"award-number":["DSB.AD009.001"]}]},{"name":"PHENOSCALE","award":["261492"],"award-info":[{"award-number":["261492"]}]},{"name":"PHENOSCALE","award":["223263"],"award-info":[{"award-number":["223263"]}]},{"name":"PHENOSCALE","award":["ME.P05.001.003"],"award-info":[{"award-number":["ME.P05.001.003"]}]},{"name":"EMMA\u2014Sviluppo Internazionale Campus Monterotondo, Consiglio Nazionale delle Ricerche","award":["DSB.AD009.001"],"award-info":[{"award-number":["DSB.AD009.001"]}]},{"name":"EMMA\u2014Sviluppo Internazionale Campus Monterotondo, Consiglio Nazionale delle Ricerche","award":["261492"],"award-info":[{"award-number":["261492"]}]},{"name":"EMMA\u2014Sviluppo Internazionale Campus Monterotondo, Consiglio Nazionale delle Ricerche","award":["223263"],"award-info":[{"award-number":["223263"]}]},{"name":"EMMA\u2014Sviluppo Internazionale Campus Monterotondo, Consiglio Nazionale delle Ricerche","award":["ME.P05.001.003"],"award-info":[{"award-number":["ME.P05.001.003"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Cells"],"abstract":"<jats:p>Odad3 gene loss-of-function mutation leads to Primary Ciliary Dyskinesia (PCD), a disease caused by motile cilia dysfunction. Previously, we demonstrated that knockout of the Odad3 gene in mice replicates several features of PCD, such as hydrocephalus, defects in left\u2013right body symmetry, and male infertility, with a complete absence of sperm in the reproductive tract. The majority of Odad3 knockout animals die before sexual maturation due to severe hydrocephalus and failure to thrive, which precludes fertility studies. Here, we performed the expression analysis of the Odad3 gene during gonad development and in adult testes. We showed that Odad3 starts its expression during the first wave of spermatogenesis, specifically at the meiotic stage, and that its expression is restricted to the germ cells in the adult testes, suggesting that Odad3 plays a role in spermatozoa formation. Subsequently, we conditionally deleted the Odad3 gene in adult males and demonstrated that even partial ablation of the Odad3 gene leads to asthenoteratozoospermia with multiple morphological abnormalities of sperm flagella (MMAF) in mice. The analysis of the seminiferous tubules in Odad3-deficient mice revealed defects in spermatogenesis with accumulation of seminiferous tubules at the spermiogenesis and spermiation phases. Furthermore, analysis of fertility in heterozygous Odad3+\/\u2212 knockout mice revealed a reduction in sperm count and motility as well as abnormal sperm morphology. Additionally, Odad3+\/\u2212 males exhibited a shorter fertile lifespan. Overall, these results suggest the important role of Odad3 and Odad3 gene dosage in male fertility. These findings may have an impact on the genetic and fertility counseling practice of PCD patients carrying Odad3 loss-of-function mutations.<\/jats:p>","DOI":"10.3390\/cells13121053","type":"journal-article","created":{"date-parts":[[2024,6,18]],"date-time":"2024-06-18T07:33:20Z","timestamp":1718696000000},"page":"1053","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["The Odad3 Gene Is Necessary for Spermatozoa Development and Male Fertility in Mice"],"prefix":"10.3390","volume":"13","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6500-8909","authenticated-orcid":false,"given":"Miriam","family":"Pasquini","sequence":"first","affiliation":[{"name":"Institute of Biochemistry and Cell Biology (IBBC), National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"},{"name":"European Mouse Mutant Archive (EMMA), INFRAFRONTIER, Monterotondo Mouse Clinic, National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8887-3900","authenticated-orcid":false,"given":"Francesco","family":"Chiani","sequence":"additional","affiliation":[{"name":"Institute of Biochemistry and Cell Biology (IBBC), National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"},{"name":"European Mouse Mutant Archive (EMMA), INFRAFRONTIER, Monterotondo Mouse Clinic, National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3563-2759","authenticated-orcid":false,"given":"Alessia","family":"Gambadoro","sequence":"additional","affiliation":[{"name":"Institute of Biochemistry and Cell Biology (IBBC), National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"},{"name":"European Mouse Mutant Archive (EMMA), INFRAFRONTIER, Monterotondo Mouse Clinic, National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2680-1821","authenticated-orcid":false,"given":"Chiara","family":"Di Pietro","sequence":"additional","affiliation":[{"name":"Institute of Biochemistry and Cell Biology (IBBC), National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"},{"name":"European Mouse Mutant Archive (EMMA), INFRAFRONTIER, Monterotondo Mouse Clinic, National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Renata","family":"Paoletti","sequence":"additional","affiliation":[{"name":"Institute of Biochemistry and Cell Biology (IBBC), National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0023-7916","authenticated-orcid":false,"given":"Tiziana","family":"Orsini","sequence":"additional","affiliation":[{"name":"Institute of Biochemistry and Cell Biology (IBBC), National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"},{"name":"European Mouse Mutant Archive (EMMA), INFRAFRONTIER, Monterotondo Mouse Clinic, National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3335-043X","authenticated-orcid":false,"given":"Sabrina","family":"Putti","sequence":"additional","affiliation":[{"name":"Institute of Biochemistry and Cell Biology (IBBC), National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"},{"name":"European Mouse Mutant Archive (EMMA), INFRAFRONTIER, Monterotondo Mouse Clinic, National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4483-6753","authenticated-orcid":false,"given":"Ferdinando","family":"Scavizzi","sequence":"additional","affiliation":[{"name":"Institute of Biochemistry and Cell Biology (IBBC), National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"},{"name":"European Mouse Mutant Archive (EMMA), INFRAFRONTIER, Monterotondo Mouse Clinic, National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gina","family":"La Sala","sequence":"additional","affiliation":[{"name":"Institute of Biochemistry and Cell Biology (IBBC), National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"},{"name":"European Mouse Mutant Archive (EMMA), INFRAFRONTIER, Monterotondo Mouse Clinic, National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Olga","family":"Ermakova","sequence":"additional","affiliation":[{"name":"Institute of Biochemistry and Cell Biology (IBBC), National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"},{"name":"European Mouse Mutant Archive (EMMA), INFRAFRONTIER, Monterotondo Mouse Clinic, National Research Council of Italy (CNR), Adriano Buzzati-Traverso Campus, Via Ramarini, 32, 00015 Monterotondo, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2024,6,18]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","unstructured":"Lyu, Q., Li, Q., Zhou, J., and Zhao, H. 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