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Divergent phenotypes of vision and accessory visual function in mice with visual cycle dysfunction (Rpe65 rd12) or
Stewart Thompson1, Robert F Mullins, Alisdair R Philp
1Department of Cell and Systems Biology, The University of Toronto, Toronto, Ontario, Canada. stewart-thompson@uiowa.edu
Investigative Ophthalmology & Visual Science
|June 3, 2008
Summary
A novel vision assay reveals that residual retinal function in mouse models of retinal disease can guide locomotion and suggests potential for early gene therapy. Masking responses are recommended for assessing vision restoration therapies.
Area of Science:
- Ophthalmology
- Neuroscience
- Genetics
Background:
- Assessing vision in mice is crucial for understanding retinal disorders.
- Current methods are limited, hindering the study of visual phenotypes.
Purpose of the Study:
- To develop and utilize a novel vision assay to evaluate visual phenotypes in mouse models of retinal disease.
- To determine how the extent and type of retinal disease impact visual function.
Main Methods:
- Assessed retinal histology, light-induced suppression of locomotion (masking), and visual guidance of locomotion.
- Utilized mouse models with progressive photoreceptor degeneration (rd/rd) and visual cycle dysfunction (Rpe65 rd12).
Main Results:
- Wild-type mice exhibited light-dependent locomotor control. rd/rd mice showed vision for locomotion until P46, with enhanced masking in bright light.
- Rpe65 rd12 mice lacked dim light responses but retained vision for locomotion under high illumination across all ages.
Conclusions:
- Functional visual pathways persist in rd/rd and Rpe65 rd12 mice, indicating potential for vision restoration therapies.
- Residual vision in Rpe65 rd12 mice under high illumination is useful, and early vision in rd/rd mice suggests therapeutic windows.
- Masking responses should be incorporated into assessments of gene and cell replacement therapies for retinal disorders.
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