Abstract
Vision is essential for vertebrates including humans. Sustained vision is accomplished by the retinoid metabolism, ‘visual cycle’, where all-trans retinol (atROL) is incorporated into the retinal pigment epithelium (RPE) from photoreceptors presumably through decade-long missing receptor(s). Here, we show that LDL-related receptor-5 (LRP5) protein is linked to retinol binding protein 1 (RBP1), the transporter of atROL in the visual cycle, by generating and analyzing the digenic eyes shut homolog+/-; lrp5+/- zebrafish, the same form of gene defects emerged from a human case report of a candidate inherited retinal degeneration. Global gene expression analysis followed by genetic study clarified that rbp1 played a role downstream of lrp5. RBP1 protein was colocalized with LRP5 protein at microvilli of RPE cells. Furthermore, RBP1 directly bound to C-terminal intracellular region of LRP5 in vitro. Collectively, these results strongly suggest that LRP5 is a potent candidate of the receptor of atROL in the visual cycle.
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