Exp Eye Res 2021 May 5:108603. Epub 2021 May 5.
Nuffield Laboratory of Ophthalmology, Department of Clinical Neurosciences, Oxford University, West Wing, John Radcliffe Hospital, Oxford, OX3 9DU, UK; Oxford Eye Hospital, John Radcliffe Hospital, Oxford University Hospitals NHS Foundation Trust, Headley Way, Oxford, OX3 9DU, UK. Electronic address:
Purpose: To validate the Cdhr1 mouse as a model for human CDHR1-associated retinal degeneration, which may present as cone-rod dystrophy or geographic atrophy.
Methods: Deep phenotyping of Cdhr1(n = 56) and C57BL6J wildtype control mice (n = 45) was undertaken using in vivo multimodal retinal imaging and dark- and light-adapted electroretinography (ERG) over 15 months to evaluate rod- and cone-photoreceptor responses and retinal morphology.
Results: Cdhr1 retinas exhibited outer retinal thinning on optical coherence tomography (OCT) at 1-month versus C57BL6J (mean 14. Read More