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J Biol Chem. 2006 Dec 8;281(49):37697-704. Epub 2006 Oct 10.

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

Author information

1
Department of Pharmacology and Ophthalmology, Case Western Reserve University, Cleveland, Ohio 44106, USA.

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).

PMID:
17032653
PMCID:
PMC4124513
DOI:
10.1074/jbc.M608375200
[Indexed for MEDLINE]
Free PMC Article

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