ArticleThe FEBS journal2011
An alternative isomerohydrolase in the retinal Müller cells of a cone-dominant species.
Article in The FEBS journal, 2011. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers.
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Who cites it
14 citing papers in PubMed, 27 citations in OpenAlex.
- Long-term effect of gene therapy on Leber's congenital amaurosis.The New England journal of medicine · 2015Trial
- The cone visual cycle and its disorders: insights from zebrafish.Frontiers in molecular neuroscience · 2025Review
- Retina Organoid Transplants Develop Photoreceptors and Improve Visual Function in RCS Rats With RPE Dysfunction.Investigative ophthalmology & visual science · 2020Article
- Non-photopic and photopic visual cycles differentially regulate immediate, early, and late phases of cone photoreceptor-mediated vision.The Journal of biological chemistry · 2020Article
- Retinoid isomerase inhibitors impair but do not block mammalian cone photoreceptor function.The Journal of general physiology · 2018Article
- Enhancing Understanding of the Visual Cycle by Applying CRISPR/Cas9 Gene Editing in Zebrafish.Frontiers in cell and developmental biology · 2018Review
- Development of an optimized AAV2/5 gene therapy vector for Leber congenital amaurosis owing to defects in RPE65.Gene therapy · 2016Article
- Structure of zebrafish IRBP reveals fatty acid binding.Experimental eye research · 2015Article
- Interphotoreceptor retinoid-binding protein (IRBP) promotes retinol uptake and release by rat Müller cells (rMC-1) in vitro: implications for the cone visual cycle.Investigative ophthalmology & visual science · 2014Article
- Immuno-histochemical analysis of rod and cone reaction to RPE65 deficiency in the inferior and superior canine retina.PloS one · 2014Article
- Complementation test of Rpe65 knockout and tvrm148.Investigative ophthalmology & visual science · 2013Article
- Slowed recovery of human photopic ERG a-wave amplitude following intense bleaches: a slowing of cone pigment regeneration?Documenta ophthalmologica. Advances in ophthalmology · 2012Article
- Cultured Müller cells from mammals can synthesize and accumulate retinyl esters.Experimental eye research · 2012Article
- Identification of the key residues determining the product specificity of isomerohydrolase.Biochemistry · 2012Article
Corrections and comments
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Authors and funding
5 authors at 1 institution in 1 country.
Funding
Abstract
Cone photoreceptors have faster light responses than rods and a higher demand for 11-cis retinal (11cRAL), the chromophore of visual pigments. RPE65 is the isomerohydrolase in the retinal pigment epithelium (RPE) that converts all-trans retinyl ester to 11-cis retinol, a key step in the visual cycle for regenerating 11cRAL. Accumulating evidence suggests that cone-dominant species express an alternative isomerase, likely in retinal Müller cells, to meet the high demand for the chromophore by cones. In the present study, we describe the identification and characterization of a novel isomerohydrolase, RPE65c, from the cone-dominant zebrafish retina. RPE65c shares 78% amino acid sequence identity with RPE-specific zebrafish RPE65a (orthologue of human RPE65) and retains all of the known key residues for the enzymatic activity of RPE65. Similar to the other RPE-specific RPE65, RPE65c was present in both the membrane and cytosolic fractions, used all-trans retinyl ester as its substrate and required iron for its enzymatic activity. However, immunohistochemistry detected RPE65c in the inner retina, including Müller cells, but not in the RPE. Furthermore, double-immunostaining of dissociated retinal cells using antibodies for RPE65c and glutamine synthetase (a Müller cell marker), showed that RPE65c co-localized with the Müller cell marker. These results suggest that RPE65c is the alternative isomerohydrolase in the intra-retinal visual cycle, providing 11cRAL to cone photoreceptors in cone-dominant species. Identification of an alternative visual cycle will contribute to the understanding of the functional differences of rod and cone photoreceptors.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.