The National Eye Institute (NEI) awarded a $462,430 Project Grant (CFDA 93.867 - Vision Research) to the University of Utah's Office of Sponsored Projects Division to conduct research on the role of centriole linkage in retinal degeneration and hearing loss. The project aims to determine the function of the CNAP1 protein isoforms in maintaining the ciliary structure and Usher syndrome protein complex in photoreceptors and hair cells. The research will utilize new CEP250 mutant mouse models developed during preliminary studies to explore the mechanistic connections between centriole linkage, retinal and auditory impairments, and the Usher syndrome pathology. This 5-year project is expected to provide new insights into the disease mechanisms underlying genetic mutations in CEP250, NEK2, and CEP78, which have been linked to retinitis pigmentosa and combined cone-rod dystrophy with hearing loss.