Primary cilia: biosensors of the male reproductive tract

Abstract

Background: The primary cilium is a microtubule-based organelle that extends transiently from the apical cell surface to act as a sensory antenna. Initially viewed as a cellular appendage of obscure significance, the primary cilium is now acknowledged as a key coordinator of signaling pathways during development and in tissue homeostasis. Objectives: The aim of this review was to present the structure and function of this overlooked organelle,with an emphasis on its epididymal context and contribution to male infertility issues. Materials and Methods: A systematic review has been performed in order to include main references relevant to the aforementioned topic. Results: Increasing evidence demonstrates that primary cilia dysfunctions are associated with impaired male reproductive system development and male infertility issues. Discussion: While a large amount of data exists regarding the role of primary cilia in most organs and tissues, few studies investigated the contribution of these organelles to male reproductive tract development and homeostasis. Conclusion: Functional studies of primary cilia constitute an emergent and exciting new area in reproductive biology research.

Hedgehog signaling pathway regulates gene expression profile of epididymal principal cells through the primary cilium

“…41 While the origin and the modulation of the secretory routes used by Hh ligands remains to be determined, the presence of both SHH and IHH in the epididymal fluid suggests that these two Hh ligands could potentially interact with specific receptors located on PC in the epididymis. In that regard, previous studies indicate that PC organelles extend from distinct epithelial cells of the epididymis at different stages of development 12,14,27 and could potentially transduce Hh signaling in response to secreted Hh ligands in this organ.…”

“…Motile cilia and PC are found in the different organs of the male reproductive tract (for review 12), and their presence can be related to reproductive pathologies. For instance, PC are present in basal cells of the prostate, but are absent in cases of preinvasive and invasive prostate cancers, 13 indicating that these organelles could represent potent sentinels and/or markers of human pathologies.…”

Targeting E3 Ubiquitin Ligases and Deubiquitinases in Ciliopathy and Cancer

“…The impairment of the sperm tail affects sperm motility. The impairment of motile and primary cilia also affects the function of the reproductive tract in both males and females [ 54 , 56 ]. Structural and functional studies of cilia associated with infertility constitute an important area in reproductive research [ 55 , 56 ].…”

Characterization of the pVHL Interactome in Human Testis Using High-Throughput Library Screening

“…In contrast with the VHL murine model, no infertility or defect of spermatogenesis are reported in humans except for mechanical obstruction of seminal ducts due to epididymal cysts. On the other hand, pVHL is required for the correct cilia formation [ 23 ], while it is well understood that multiple male infertility syndromes arise from congenital defects in ciliogenesis [ 24 ]. Similarly, defects in ciliogenesis are also reported in cancer and linked to deregulation of the cellular response to signals from multiple pathways [ 25 , 26 ].…”