biostudies-literatureAlves MBRNICHD NIH HHSCanadian Institutes of Health Research241-257https://www.ebi.ac.uk/biostudies/studies/S-EPMC9930401biostudies-literatureUnknown108(2)Primary cilia play pivotal roles in embryonic patterning and organogenesis through transduction of the Hedgehog signaling pathway (Hh). Although mutations in Hh morphogens impair the development of the gonads and trigger male infertility, the contribution of Hh and primary cilia in the development of male reproductive ductules, including the epididymis, remains unknown. From a Pax2Cre; IFT88fl/fl knock-out mouse model, we found that primary cilia deletion is associated with imbalanced Hh signaling and morphometric changes in the Wolffian duct (WD), the embryonic precursor of the epididymis. Similar effects were observed following pharmacological blockade of primary cilia formation and Hh modulation on WD organotypic cultures. The expression of genes involved in extracellular matrix, mesencBiology of reproductionHedgehog signaling regulates Wolffian duct development through the primary cilium†.PMC9930401201803PJT-401278-E-CFBA-194130R03 HD101762Moon KHHinton BTBelleannee CBernet ALavoie-Ouellet CAlves MBRDroit AGirardet LBeauparlant CJRobert CAugiere CSoulet DCalvo ETeves MEBok JBastien AfalseHedgehog signaling regulates Wolffian duct development through the primary cilium†.Primary cilia play pivotal roles in embryonic patterning and organogenesis through transduction of the Hedgehog signaling pathway (Hh). Although mutations in Hh morphogens impair the development of the gonads and trigger male infertility, the contribution of Hh and primary cilia in the development of male reproductive ductules, including the epididymis, remains unknown. From a Pax2Cre; IFT88fl/fl knock-out mouse model, we found that primary cilia deletion is associated with imbalanced Hh signaling and morphometric changes in the Wolffian duct (WD), the embryonic precursor of the epididymis. Similar effects were observed following pharmacological blockade of primary cilia formation and Hh modulation on WD organotypic cultures. The expression of genes involved in extracellular matrix, mesenc2023-01-01T00:00:00Z2023 Feb2026-05-29T02:48:59.036Z2025-04-21T15:50:39.391ZS-EPMC99304013652534110.1093/biolre/ioac210