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,56NPHP5,51RPGRIP1,52 or CCDC66,34 using U-ExM. Here, CCDC66 is linked to retinal ciliopathy.