The localization of Usher proteins (PCDH15 and Whirlin), as well as actin filament–related proteins in canine photoreceptors, aligns with the patterns reported in NHP photoreceptors, suggesting that a canine IRD model may provide valuable insights into pathogenesis of Usher syndrome, for which effective pathological models are challenging to develop in rodents. Here, PCDH15 is linked to Usher syndrome.