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Retinol dehydrogenase (RDH12) protects photoreceptors from light-induced degeneration in mice.


ABSTRACT: 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).

SUBMITTER: Maeda A 

PROVIDER: S-EPMC4124513 | biostudies-literature | 2006 Dec

REPOSITORIES: biostudies-literature

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Retinol dehydrogenase (RDH12) protects photoreceptors from light-induced degeneration in mice.

Maeda Akiko A   Maeda Tadao T   Imanishi Yoshikazu Y   Sun Wenyu W   Jastrzebska Beata B   Hatala Denise A DA   Winkens Huub J HJ   Hofmann Klaus Peter KP   Janssen Jacques J JJ   Baehr Wolfgang W   Driessen Carola A CA   Palczewski Krzysztof K  

The Journal of biological chemistry 20061010 49


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  ...[more]

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