These insights gained from this study pave the way for a better understanding of the alterations in PSC molecular architecture and the efficacy of AAV-based gene augmentation therapies in established canine models of retinal ciliopathy, such as those with mutations in RPGR [61], NPHP5 [56], RPGRIP1 [57], and CCDC66 [31], using U-ExM. This evidence concerns the gene CCDC66 and retinal ciliopathy.