作者
Akiko Maeda, Tadao Maeda, Yoshikazu Imanishi, Wenyu Sun, Beata Jastrzebska, Denise A Hatala, Huub J Winkens, Klaus Peter Hofmann, Jacques J Janssen, Wolfgang Baehr, Carola A Driessen, Krzysztof Palczewski
发表日期
2006/12/8
期刊
Journal of Biological Chemistry
卷号
281
期号
49
页码范围
37697-37704
出版商
Elsevier
简介
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 damage1.
引用总数
200720082009201020112012201320142015201620172018201920202021202220233991341358197577465
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A Maeda, T Maeda, Y Imanishi, W Sun, B Jastrzebska… - Journal of Biological Chemistry, 2006