ArticleNature chemical biology2013
Identification of DES1 as a vitamin A isomerase in Müller glial cells of the retina.
Article in Nature chemical biology, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 65 papers.
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Who cites it
65 citing papers in PubMed, 107 citations in OpenAlex.
- Long-term effect of gene therapy on Leber's congenital amaurosis.The New England journal of medicine · 2015Trial
- Intracellular Retinoid-Binding Proteins (CRBP, CRALBP, CRABP) as Emerging Drug Targets in Retinal Disease.International journal of molecular sciences · 2026Review
- The cone visual cycle and its disorders: insights from zebrafish.Frontiers in molecular neuroscience · 2025Review
- Ablation of Fatty Acid Transport Protein-4 Enhances Cone Survival, M-cone Vision, and Synthesis of Cone-Tropic 9-The Journal of neuroscience : the official journal of the Society for Neuroscience · 2024Article
- Cellular and subcellular optogenetic approaches towards neuroprotection and vision restoration.Progress in retinal and eye research · 2023Review
- Rapid RGR-dependent visual pigment recycling is mediated by the RPE and specialized Müller glia.Cell reports · 2023Article
- Adaptation memory in photoreceptors: different mechanisms in rods and cones.Frontiers in molecular neuroscience · 2023Article
- Acyl-CoA:wax alcohol acyltransferase 2 modulates the cone visual cycle in mouse retina.FASEB journal : official publication of the Federation of American Societies for Experimental Biology · 2022Article
- The Role of Vitamin A in Retinal Diseases.International journal of molecular sciences · 2022Review
- Non-visual Opsins and Novel Photo-Detectors in the Vertebrate Inner Retina Mediate Light Responses Within the Blue Spectrum Region.Cellular and molecular neurobiology · 2022Review
- Function of mammalian M-cones depends on the level of CRALBP in Müller cells.The Journal of general physiology · 2021Article
- Molecular components affecting ocular carotenoid and retinoid homeostasis.Progress in retinal and eye research · 2021Review
- The molecular aspects of absorption and metabolism of carotenoids and retinoids in vertebrates.Biochimica et biophysica acta. Molecular and cell biology of lipids · 2020Review
- Vitamin A aldehyde-taurine adduct and the visual cycle.Proceedings of the National Academy of Sciences of the United States of America · 2020Article
- Shedding new light on the generation of the visual chromophore.Proceedings of the National Academy of Sciences of the United States of America · 2020Review
- RNA-based therapies in animal models of Leber congenital amaurosis causing blindness.Precision clinical medicine · 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
- Photic generation of 11-The Journal of biological chemistry · 2019Article
- Light-Driven Regeneration of Cone Visual Pigments through a Mechanism Involving RGR Opsin in Müller Glial Cells.Neuron · 2019Article
- Conditional deletion of Des1 in the mouse retina does not impair the visual cycle in cones.FASEB journal : official publication of the Federation of American Societies for Experimental Biology · 2019Article
5 more citing papers are in PubMed but not listed here.
Corrections and comments
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Authors and funding
13 authors at 1 institution in 1 country.
Funding
Abstract
Absorption of a light particle by an opsin-pigment causes photoisomerization of its retinaldehyde chromophore. Restoration of light sensitivity to the resulting apo-opsin requires chemical re-isomerization of the photobleached chromophore. This is carried out by a multistep enzyme pathway called the visual cycle. Accumulating evidence suggests the existence of an alternative visual cycle for regenerating opsins in daylight. Here we identified dihydroceramide desaturase-1 (DES1) as a retinol isomerase and an excellent candidate for isomerase-2 in this alternative pathway. DES1 is expressed in retinal Müller cells, where it coimmunoprecipitates with cellular retinaldehyde binding protein (CRALBP). Adenoviral gene therapy with DES1 partially rescued the biochemical and physiological phenotypes in Rpe65(-/-) mice lacking isomerohydrolase (isomerase-1). Knockdown of DES1 expression by RNA interference concordantly reduced isomerase-2 activity in cultured Müller cells. Purified DES1 had very high isomerase-2 activity in the presence of appropriate cofactors, suggesting that DES1 by itself is sufficient for isomerase activity.
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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.