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J Biol Chem. 2008 Sep 5;283(36):25091-102. doi: 10.1074/jbc.M802981200. Epub 2008 Jul 7.

Retinyl ester homeostasis in the adipose differentiation-related protein-deficient retina.

Author information

1
Department of Pharmacology, Case Western Reserve University, Cleveland, Ohio 44106, USA. yxi19@case.edu

Abstract

The retinal pigmented epithelium (RPE) plays an essential role in vision, including storing and converting retinyl esters of the visual chromophore, 11-cis-retinal. Retinyl ester storage structures (RESTs), specialized lipid droplets within the RPE, take up retinyl esters synthesized in the endoplasmic reticulum. Here we report studies of mice lacking exons 2 and 3 of the gene encoding adipose differentiation-related protein (Adfp), a structural component of RESTs. We found that dark adaptation was slower in Adfp(Delta2-3/Delta2-3) than in Adfp+/+ mice and that Adfp(Delta2-3/Delta2-3) mice had consistently delayed clearances of all-trans-retinal and all-trans-retinol from rod photoreceptor cells. Two-photon microscopy revealed aberrant trafficking of all-trans-retinyl esters in the RPE of Adfp(Delta2-3/Delta2-3) mice, a problem caused by abnormal maintenance of RESTs in the dark-adapted state. Retinyl ester accumulation was also reduced in Adfp(Delta2-3/Delta2-3) as compared with Adfp+/+ mice. These observations suggest that Adfp plays a unique role in vision by maintaining proper storage and trafficking of retinoids within the eye.

PMID:
18606814
PMCID:
PMC2529016
DOI:
10.1074/jbc.M802981200
[Indexed for MEDLINE]
Free PMC Article

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