ArticleInvestigative ophthalmology & visual science2019
Rescue of M-cone Function in Aged Opn1mw-/- Mice, a Model for Late-Stage Blue Cone Monochromacy.
Article in Investigative ophthalmology & visual science, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 16 papers.
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Who cites it
16 citing papers in PubMed.
- IMPC impact on preclinical mouse models.Mammalian genome : official journal of the International Mammalian Genome Society · 2025Article
- The Ercc1Aging cell · 2025Article
- Evaluation of Retinal Structure and Visual Function in Blue Cone Monochromacy to Develop Clinical Endpoints for L-opsin Gene Therapy.International journal of molecular sciences · 2024Article
- Review
- Structural and functional rescue of cones carrying the most common cone opsin C203R missense mutation.JCI insight · 2024Article
- Dyschromatopsia: a comprehensive analysis of mechanisms and cutting-edge treatments for color vision deficiency.Frontiers in neuroscience · 2024Review
- Color Vision in Blue Cone Monochromacy: Outcome Measures for a Clinical Trial.Translational vision science & technology · 2023Article
- Retinal Cone Mosaic in sws1-Mutant Medaka (Oryzias latipes), A Teleost.Investigative ophthalmology & visual science · 2022Article
- Gene Therapy inHuman gene therapy · 2022Article
- Blue Cone Monochromatism: A Case Report with Opsoclonus and Light Exposure.Journal of pediatric genetics · 2022Article
- Article
- A mouse model of cone photoreceptor function lossFrontiers in molecular neuroscience · 2022Article
- Gene therapy in color vision deficiency: a review.International ophthalmology · 2021Review
- Limited time window for retinal gene therapy in a preclinical model of ciliopathy.Human molecular genetics · 2020Article
- Deletion of M-Opsin Prevents M Cone Degeneration in a Mouse Model of Leber Congenital Amaurosis.The American journal of pathology · 2020Article
- Foveal Therapy in Blue Cone Monochromacy: Predictions of Visual Potential From Artificial Intelligence.Frontiers in neuroscience · 2020Article
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Authors and funding
7 authors.
Funding
Abstract
Purpose: Previously we showed that AAV5-mediated expression of either human M- or L-opsin promoted regrowth of cone outer segments and rescued M-cone function in the treated M-opsin knockout (Opn1mw-/-) dorsal retina. In this study, we determined cone viability and window of treatability in aged Opn1mw-/- mice. Methods: Cone viability was assessed with antibody against cone arrestin and peanut agglutinin (PNA) staining. The rate of cone degeneration in Opn1mw-/- mice was quantified by PNA staining. AAV5 vector expressing human L-opsin was injected subretinally into one eye of Opn1mw-/- mice at 1, 7, and 15 months old, while the contralateral eyes served as controls. M-cone-mediated retinal function was analyzed 2 and 13 months postinjection by full-field ERG. L-opsin transgene expression and cone outer segment structure were examined by immunohistochemistry. Results: We showed that dorsal M-opsin dominant cones exhibit outer segment degeneration at an early age in Opn1mw-/- mice, whereas ventral S-opsin dominant cones were normal. The remaining M-opsin dominant cones remained viable for at least 15 months, albeit having shortened or no outer segments. We also showed that AAV5-mediated expression of human L-opsin was still able to rescue function and outer segment structure in the remaining M-opsin dominant cones when treatment was initiated at 15 months of age. Conclusions: Our results showing that the remaining M-opsin dominant cones in aged Opn1mw-/- mice can still be rescued by gene therapy is helpful for establishing the window of treatability in future blue cone monochromacy clinical trials.
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