Regulation of RPE65 expression in human retinal pigment epithelium cells

Abstract The visual cycle is an important pathway in the retinal pigment epithelium (RPE) which regenerates 11-cis retinal chromophore for the retinal photoreceptors. The central enzyme in the visual cycle is RPE65 retinol isomerase. Expression of RPE65 mRNA and protein levels are significantly lowe...

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Main Authors: Olga A. Postnikova, Samuel William, Sheetal Uppal, Steven L. Bernstein, Eugenia Poliakov, Igor B. Rogozin, T. Michael Redmond
Format: Article
Language:English
Published: Nature Portfolio 2025-07-01
Series:Scientific Reports
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Online Access:https://doi.org/10.1038/s41598-025-12926-3
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author Olga A. Postnikova
Samuel William
Sheetal Uppal
Steven L. Bernstein
Eugenia Poliakov
Igor B. Rogozin
T. Michael Redmond
author_facet Olga A. Postnikova
Samuel William
Sheetal Uppal
Steven L. Bernstein
Eugenia Poliakov
Igor B. Rogozin
T. Michael Redmond
author_sort Olga A. Postnikova
collection DOAJ
description Abstract The visual cycle is an important pathway in the retinal pigment epithelium (RPE) which regenerates 11-cis retinal chromophore for the retinal photoreceptors. The central enzyme in the visual cycle is RPE65 retinol isomerase. Expression of RPE65 mRNA and protein levels are significantly lower in RPE cell culture models when compared to native RPE. This limits the use of these models to study the visual cycle. To determine the main drivers of RPE65 regulation we compared the transcriptional profiles of native and cell culture models of RPE with various levels of RPE65 expression. We also compared the levels of RPE65 expression between ARPE-19 cells grown in media supplemented with 1 mM pyruvate (PYR) or 10 mM nicotinamide (NAM). In addition, we performed experiments directed at transcriptional and translational regulation of RPE65. We show that RPE65 mRNA and protein expression is significantly higher in NAM media grown cells than PYR cells. Transfection of cells with a variety of different vectors containing RPE65 ORFs with different promoters, codon optimization, IRES, 3’ UTRs, suggest that translational effects are less important than transcriptional status. Importantly, we found that feeding with rod outer segments (ROS) decreases RPE65 expression in NAM grown cells, suggesting that certain primary functions of the RPE (here, visual cycle and phagocytosis) are not positively linked. Analysis of differentially regulated microRNAs (miRs) provides a basis for this downregulation. It appears that the regulation of RPE65 expression in ARPE-19 cells, in particular, is multifactorial, involving primarily metabolic and transcriptional status of the cells, with translation of RPE65 mRNA playing a smaller role.
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spelling doaj-art-f6de1ca559aa4a00a683573672870d172025-08-20T03:04:34ZengNature PortfolioScientific Reports2045-23222025-07-0115111510.1038/s41598-025-12926-3Regulation of RPE65 expression in human retinal pigment epithelium cellsOlga A. Postnikova0Samuel William1Sheetal Uppal2Steven L. Bernstein3Eugenia Poliakov4Igor B. Rogozin5T. Michael Redmond6Laboratory of Retinal Cell & Molecular Biology, National Eye Institute, NIHLaboratory of Retinal Cell & Molecular Biology, National Eye Institute, NIHLaboratory of Retinal Cell & Molecular Biology, National Eye Institute, NIHDepartments of Ophthalmology and Visual Sciences, and Anatomy and Neurobiology, School of Medicine, University of MarylandLaboratory of Retinal Cell & Molecular Biology, National Eye Institute, NIHNational Center for Biotechnology Information, National Library of Medicine, National Institutes of HealthLaboratory of Retinal Cell & Molecular Biology, National Eye Institute, NIHAbstract The visual cycle is an important pathway in the retinal pigment epithelium (RPE) which regenerates 11-cis retinal chromophore for the retinal photoreceptors. The central enzyme in the visual cycle is RPE65 retinol isomerase. Expression of RPE65 mRNA and protein levels are significantly lower in RPE cell culture models when compared to native RPE. This limits the use of these models to study the visual cycle. To determine the main drivers of RPE65 regulation we compared the transcriptional profiles of native and cell culture models of RPE with various levels of RPE65 expression. We also compared the levels of RPE65 expression between ARPE-19 cells grown in media supplemented with 1 mM pyruvate (PYR) or 10 mM nicotinamide (NAM). In addition, we performed experiments directed at transcriptional and translational regulation of RPE65. We show that RPE65 mRNA and protein expression is significantly higher in NAM media grown cells than PYR cells. Transfection of cells with a variety of different vectors containing RPE65 ORFs with different promoters, codon optimization, IRES, 3’ UTRs, suggest that translational effects are less important than transcriptional status. Importantly, we found that feeding with rod outer segments (ROS) decreases RPE65 expression in NAM grown cells, suggesting that certain primary functions of the RPE (here, visual cycle and phagocytosis) are not positively linked. Analysis of differentially regulated microRNAs (miRs) provides a basis for this downregulation. It appears that the regulation of RPE65 expression in ARPE-19 cells, in particular, is multifactorial, involving primarily metabolic and transcriptional status of the cells, with translation of RPE65 mRNA playing a smaller role.https://doi.org/10.1038/s41598-025-12926-3Retinal pigment epitheliumRetinaRPE65TranscriptionTranslationRibosome
spellingShingle Olga A. Postnikova
Samuel William
Sheetal Uppal
Steven L. Bernstein
Eugenia Poliakov
Igor B. Rogozin
T. Michael Redmond
Regulation of RPE65 expression in human retinal pigment epithelium cells
Scientific Reports
Retinal pigment epithelium
Retina
RPE65
Transcription
Translation
Ribosome
title Regulation of RPE65 expression in human retinal pigment epithelium cells
title_full Regulation of RPE65 expression in human retinal pigment epithelium cells
title_fullStr Regulation of RPE65 expression in human retinal pigment epithelium cells
title_full_unstemmed Regulation of RPE65 expression in human retinal pigment epithelium cells
title_short Regulation of RPE65 expression in human retinal pigment epithelium cells
title_sort regulation of rpe65 expression in human retinal pigment epithelium cells
topic Retinal pigment epithelium
Retina
RPE65
Transcription
Translation
Ribosome
url https://doi.org/10.1038/s41598-025-12926-3
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