Rdh12 Activity and Effects on Retinoid Processing in the Murine Retina
DC Field | Value | Language |
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dc.contributor.author | Chrispell, Jared D. | - |
dc.contributor.author | Feathers, Kecia L. | - |
dc.contributor.author | Kane, Maureen A. | - |
dc.contributor.author | Kim, Chul Y. | - |
dc.contributor.author | Brooks, Matthew | - |
dc.contributor.author | Khanna, Ritu | - |
dc.contributor.author | Kurth, Ingo | - |
dc.contributor.author | Huebner, Christian A. | - |
dc.contributor.author | Gal, Andreas | - |
dc.contributor.author | Mears, Alan J. | - |
dc.contributor.author | Swaroop, Anand | - |
dc.contributor.author | Napoli, Joseph L. | - |
dc.contributor.author | Sparrow, Janet R. | - |
dc.contributor.author | Thompson, Debra A. | - |
dc.date.accessioned | 2021-06-23T15:05:27Z | - |
dc.date.available | 2021-06-23T15:05:27Z | - |
dc.date.created | 2021-01-21 | - |
dc.date.issued | 2009-08 | - |
dc.identifier.issn | 0021-9258 | - |
dc.identifier.uri | https://scholarworks.bwise.kr/erica/handle/2021.sw.erica/40968 | - |
dc.description.abstract | RDH12 mutations are responsible for early-onset autosomal recessive retinal dystrophy, which results in profound retinal pathology and severe visual handicap in patients. To investigate the function of RDH12 within the network of retinoid dehydrogenases/reductases (RDHs) present in retina, we studied the retinal phenotype of Rdh12-deficient mice. In vivo rates of all-trans-retinal reduction and 11-cis-retinal formation during recovery from bleaching were similar in Rdh12-deficient and wild-type mice matched for an Rpe65 polymorphism that impacts visual cycle efficiency. However, retinal homogenates from Rdh12-deficient mice exhibited markedly decreased capacity to reduce exogenous retinaldehydes in vitro. Furthermore, in vivo levels of the bisretinoid compound diretinoid-pyridinium-ethanolamine (A2E) were increased in Rdh12-deficient mice of various genetic backgrounds. Conversely, in vivo levels of retinoic acid and total retinol were significantly decreased. Rdh12 transcript levels in wild-type mice homozygous for the Rpe65-Leu(450) polymorphism were greater than in Rpe65-Met(450) mice and increased during postnatal development in wild-type mice and Nrl-deficient mice having an all-cone retina. Rdh12-deficient mice did not exhibit increased retinal degeneration relative to wild-type mice at advanced ages, when bred on the light-sensitive BALB/c background, or when heterozygous for a null allele of superoxide dismutase 2 (Sod2(+/-)). Our findings suggest that a critical function of RDH12 is the reduction of all-trans-retinal that exceeds the reductive capacity of the photoreceptor outer segments. | - |
dc.language | 영어 | - |
dc.language.iso | en | - |
dc.publisher | AMER SOC BIOCHEMISTRY MOLECULAR BIOLOGY INC | - |
dc.title | Rdh12 Activity and Effects on Retinoid Processing in the Murine Retina | - |
dc.type | Article | - |
dc.contributor.affiliatedAuthor | Kim, Chul Y. | - |
dc.identifier.doi | 10.1074/jbc.M109.020966 | - |
dc.identifier.scopusid | 2-s2.0-69249129452 | - |
dc.identifier.wosid | 000268564400041 | - |
dc.identifier.bibliographicCitation | JOURNAL OF BIOLOGICAL CHEMISTRY, v.284, no.32, pp.21468 - 21477 | - |
dc.relation.isPartOf | JOURNAL OF BIOLOGICAL CHEMISTRY | - |
dc.citation.title | JOURNAL OF BIOLOGICAL CHEMISTRY | - |
dc.citation.volume | 284 | - |
dc.citation.number | 32 | - |
dc.citation.startPage | 21468 | - |
dc.citation.endPage | 21477 | - |
dc.type.rims | ART | - |
dc.type.docType | Article | - |
dc.description.journalClass | 1 | - |
dc.description.isOpenAccess | Y | - |
dc.description.journalRegisteredClass | scie | - |
dc.description.journalRegisteredClass | scopus | - |
dc.relation.journalResearchArea | Biochemistry & Molecular Biology | - |
dc.relation.journalWebOfScienceCategory | Biochemistry & Molecular Biology | - |
dc.subject.keywordPlus | LEBER CONGENITAL AMAUROSIS | - |
dc.subject.keywordPlus | ALL-TRANS-RETINOL | - |
dc.subject.keywordPlus | HUMAN RETINOL-DEHYDROGENASE-12 RDH12 | - |
dc.subject.keywordPlus | SHORT-CHAIN DEHYDROGENASE/REDUCTASE | - |
dc.subject.keywordPlus | PERFORMANCE LIQUID-CHROMATOGRAPHY | - |
dc.subject.keywordPlus | MANGANESE SUPEROXIDE-DISMUTASE | - |
dc.subject.keywordPlus | PHOTORECEPTOR OUTER SEGMENTS | - |
dc.subject.keywordPlus | LIGHT-INDUCED DEGENERATION | - |
dc.subject.keywordPlus | FALSE DISCOVERY RATE | - |
dc.subject.keywordPlus | VISUAL CYCLE | - |
dc.subject.keywordAuthor | All-trans retinol | - |
dc.subject.keywordAuthor | Congenital Amaurosis | - |
dc.subject.keywordAuthor | Lipofuscin | - |
dc.subject.keywordAuthor | Nrl | - |
dc.subject.keywordAuthor | RDH12 | - |
dc.subject.keywordAuthor | RPE65 | - |
dc.subject.keywordAuthor | Retinoic Acid | - |
dc.subject.keywordAuthor | Short Chain Dehydrogenase/Reductase | - |
dc.identifier.url | https://www.sciencedirect.com/science/article/pii/S0021925818494999?via%3Dihub | - |
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