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Drosophila Fatty Acid Transport Protein Regulates Rhodopsin-1 Metabolism and Is Required for Photoreceptor Neuron Survival
Tight regulation of the visual response is essential for photoreceptor function and survival. Visual response dysregulation often leads to photoreceptor cell degeneration, but the causes of such cell death are not well understood. In this study, we investigated a fatty acid transport protein (fatp)...
Autores principales: | , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Public Library of Science
2012
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3405995/ https://www.ncbi.nlm.nih.gov/pubmed/22844251 http://dx.doi.org/10.1371/journal.pgen.1002833 |
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author | Dourlen, Pierre Bertin, Benjamin Chatelain, Gilles Robin, Marion Napoletano, Francesco Roux, Michel J. Mollereau, Bertrand |
author_facet | Dourlen, Pierre Bertin, Benjamin Chatelain, Gilles Robin, Marion Napoletano, Francesco Roux, Michel J. Mollereau, Bertrand |
author_sort | Dourlen, Pierre |
collection | PubMed |
description | Tight regulation of the visual response is essential for photoreceptor function and survival. Visual response dysregulation often leads to photoreceptor cell degeneration, but the causes of such cell death are not well understood. In this study, we investigated a fatty acid transport protein (fatp) null mutation that caused adult-onset and progressive photoreceptor cell death. Consistent with fatp having a role in the retina, we showed that fatp is expressed in adult photoreceptors and accessory cells and that its re-expression in photoreceptors rescued photoreceptor viability in fatp mutants. The visual response in young fatp-mutant flies was abnormal with elevated electroretinogram amplitudes associated with high levels of Rhodopsin-1 (Rh1). Reducing Rh1 levels in rh1 mutants or depriving flies of vitamin A rescued photoreceptor cell death in fatp mutant flies. Our results indicate that fatp promotes photoreceptor survival by regulating Rh1 abundance. |
format | Online Article Text |
id | pubmed-3405995 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2012 |
publisher | Public Library of Science |
record_format | MEDLINE/PubMed |
spelling | pubmed-34059952012-07-27 Drosophila Fatty Acid Transport Protein Regulates Rhodopsin-1 Metabolism and Is Required for Photoreceptor Neuron Survival Dourlen, Pierre Bertin, Benjamin Chatelain, Gilles Robin, Marion Napoletano, Francesco Roux, Michel J. Mollereau, Bertrand PLoS Genet Research Article Tight regulation of the visual response is essential for photoreceptor function and survival. Visual response dysregulation often leads to photoreceptor cell degeneration, but the causes of such cell death are not well understood. In this study, we investigated a fatty acid transport protein (fatp) null mutation that caused adult-onset and progressive photoreceptor cell death. Consistent with fatp having a role in the retina, we showed that fatp is expressed in adult photoreceptors and accessory cells and that its re-expression in photoreceptors rescued photoreceptor viability in fatp mutants. The visual response in young fatp-mutant flies was abnormal with elevated electroretinogram amplitudes associated with high levels of Rhodopsin-1 (Rh1). Reducing Rh1 levels in rh1 mutants or depriving flies of vitamin A rescued photoreceptor cell death in fatp mutant flies. Our results indicate that fatp promotes photoreceptor survival by regulating Rh1 abundance. Public Library of Science 2012-07-26 /pmc/articles/PMC3405995/ /pubmed/22844251 http://dx.doi.org/10.1371/journal.pgen.1002833 Text en Dourlen et al. http://creativecommons.org/licenses/by/4.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are properly credited. |
spellingShingle | Research Article Dourlen, Pierre Bertin, Benjamin Chatelain, Gilles Robin, Marion Napoletano, Francesco Roux, Michel J. Mollereau, Bertrand Drosophila Fatty Acid Transport Protein Regulates Rhodopsin-1 Metabolism and Is Required for Photoreceptor Neuron Survival |
title |
Drosophila Fatty Acid Transport Protein Regulates Rhodopsin-1 Metabolism and Is Required for Photoreceptor Neuron Survival |
title_full |
Drosophila Fatty Acid Transport Protein Regulates Rhodopsin-1 Metabolism and Is Required for Photoreceptor Neuron Survival |
title_fullStr |
Drosophila Fatty Acid Transport Protein Regulates Rhodopsin-1 Metabolism and Is Required for Photoreceptor Neuron Survival |
title_full_unstemmed |
Drosophila Fatty Acid Transport Protein Regulates Rhodopsin-1 Metabolism and Is Required for Photoreceptor Neuron Survival |
title_short |
Drosophila Fatty Acid Transport Protein Regulates Rhodopsin-1 Metabolism and Is Required for Photoreceptor Neuron Survival |
title_sort | drosophila fatty acid transport protein regulates rhodopsin-1 metabolism and is required for photoreceptor neuron survival |
topic | Research Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3405995/ https://www.ncbi.nlm.nih.gov/pubmed/22844251 http://dx.doi.org/10.1371/journal.pgen.1002833 |
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