Retinol dehydrogenase (RDH12) protects photoreceptors from light-induced degeneration in mice
| Autor: | H.J. Winkens, Wolfgang Baehr, C.A.G.G. Driessen, Akiko Maeda, Wenyu Sun, Krzysztof Palczewski, Beata Jastrzebska, Tadao Maeda, Yoshikazu Imanishi, Denise A. Hatala, Klaus Peter Hofmann, J.J.M. Janssen |
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| Rok vydání: | 2006 |
| Předmět: |
Male
Retinal degeneration Light genetic structures Biology Retinol dehydrogenase medicine.disease_cause Biochemistry Article Mice Retinoids chemistry.chemical_compound Perception and Action [DCN 1] Electroretinography medicine Neurosensory disorders [UMCN 3.3] Animals Humans Molecular Biology Gene Loss function DNA Primers Mice Knockout Genetics Mutation Base Sequence medicine.diagnostic_test Retinal Degeneration Retinol Cell Biology medicine.disease eye diseases Cell biology Mice Inbred C57BL Alcohol Oxidoreductases chemistry Female sense organs Cellular energy metabolism [UMCN 5.3] Photoreceptor Cells Vertebrate Visual phototransduction |
| Zdroj: | Journal of Biological Chemistry, 281, 37697-704 Journal of Biological Chemistry, 281, 49, pp. 37697-704 |
| ISSN: | 0021-9258 |
| Popis: | Contains fulltext : 50335.pdf (Publisher’s version ) (Open Access) RDH12 has been suggested to be one of the retinol dehydrogenases (RDH) involved in the vitamin A recycling system (visual cycle) in the eye. Loss of function mutations in the RDH12 gene were recently reported to be associated with autosomal recessive childhood-onset severe retinal dystrophy. Here we show that RDH12 localizes to the photoreceptor inner segments and that deletion of this gene in mice slows the kinetics of all-trans-retinal reduction, delaying dark adaptation. However, accelerated 11-cis-retinal production and increased susceptibility to light-induced photoreceptor apoptosis were also observed in Rdh12(-/-) mice, suggesting that RDH12 plays a unique, nonredundant role in the photoreceptor inner segments to regulate the flow of retinoids in the eye. Thus, severe visual impairments of individuals with null mutations in RDH12 may likely be caused by light damage(1). |
| Databáze: | OpenAIRE |
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