biostudies-literatureUnknown13(12)Pasquini MOdad3 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-right 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 itsCells1053https://www.ebi.ac.uk/biostudies/studies/S-EPMC11201558biostudies-literatureThe Odad3 Gene Is Necessary for Spermatozoa Development and Male Fertility in Mice.PMC11201558Chiani FPasquini MGambadoro APaoletti ROrsini TLa Sala GErmakova OPutti SDi Pietro CScavizzi FfalseThe Odad3 Gene Is Necessary for Spermatozoa Development and Male Fertility in Mice.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-right 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 its2024-01-01T00:00:00Z2024 Jun2026-05-23T03:08:46.714Z2025-04-05T17:53:46.604ZS-EPMC112015583892068110.3390/cells13121053