Fatp1 deficiency affects retinal light response and dark adaptation, and induces age-related alterations.

FATP1 is involved in lipid transport into cells and in intracellular lipid metabolism. We showed previously that this protein interacts with and inhibits the limiting-step isomerase of the visual cycle RPE65. Here, we aimed to analyze the effect of Fatp1-deficiency in vivo on the visual cycle, struc...

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Main Authors: Karim Chekroud, Laurent Guillou, Stephane Grégoire, Gilles Ducharme, Emilie Brun, Chantal Cazevieille, Lionel Bretillon, Christian P Hamel, Philippe Brabet, Marie O Pequignot
Format: Article
Language:English
Published: Public Library of Science (PLoS) 2012-01-01
Series:PLoS ONE
Online Access:http://europepmc.org/articles/PMC3500375?pdf=render
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author Karim Chekroud
Laurent Guillou
Stephane Grégoire
Gilles Ducharme
Emilie Brun
Chantal Cazevieille
Lionel Bretillon
Christian P Hamel
Philippe Brabet
Marie O Pequignot
author_facet Karim Chekroud
Laurent Guillou
Stephane Grégoire
Gilles Ducharme
Emilie Brun
Chantal Cazevieille
Lionel Bretillon
Christian P Hamel
Philippe Brabet
Marie O Pequignot
author_sort Karim Chekroud
collection DOAJ
description FATP1 is involved in lipid transport into cells and in intracellular lipid metabolism. We showed previously that this protein interacts with and inhibits the limiting-step isomerase of the visual cycle RPE65. Here, we aimed to analyze the effect of Fatp1-deficiency in vivo on the visual cycle, structure and function, and on retinal aging. Among the Fatp family members, we observed that only Fatp1 and 4 are expressed in the control retina, in both the neuroretina and the retinal pigment epithelium. In the neuroretina, Fatp1 is mostly expressed in photoreceptors. In young adult Fatp1(-/-) mice, Fatp4 expression was unchanged in retinal pigment epithelium and reduced two-fold in the neuroretina as compared to Fatp1(+/+) mice. The Fatp1(-/-) mice had a preserved retinal structure but a decreased electroretinogram response to light. These mice also displayed a delayed recovery of the b-wave amplitude after bleaching, however, visual cycle speed was unchanged, and both retinal pigment epithelium and photoreceptors presented the same fatty acid pattern compared to controls. In 2 year-old Fatp1(-/-) mice, transmission electron microscopy studies showed specific abnormalities in the retinas comprising choroid vascularization anomalies and thickening of the Bruch membrane with material deposits, and sometimes local disorganization of the photoreceptor outer segments. These anomalies lead us to speculate that the absence of FATP1 accelerates the aging process.
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spelling doaj.art-3cc7cd44e72b4dfaac9cce08d26b078e2022-12-21T20:29:18ZengPublic Library of Science (PLoS)PLoS ONE1932-62032012-01-01711e5023110.1371/journal.pone.0050231Fatp1 deficiency affects retinal light response and dark adaptation, and induces age-related alterations.Karim ChekroudLaurent GuillouStephane GrégoireGilles DucharmeEmilie BrunChantal CazevieilleLionel BretillonChristian P HamelPhilippe BrabetMarie O PequignotFATP1 is involved in lipid transport into cells and in intracellular lipid metabolism. We showed previously that this protein interacts with and inhibits the limiting-step isomerase of the visual cycle RPE65. Here, we aimed to analyze the effect of Fatp1-deficiency in vivo on the visual cycle, structure and function, and on retinal aging. Among the Fatp family members, we observed that only Fatp1 and 4 are expressed in the control retina, in both the neuroretina and the retinal pigment epithelium. In the neuroretina, Fatp1 is mostly expressed in photoreceptors. In young adult Fatp1(-/-) mice, Fatp4 expression was unchanged in retinal pigment epithelium and reduced two-fold in the neuroretina as compared to Fatp1(+/+) mice. The Fatp1(-/-) mice had a preserved retinal structure but a decreased electroretinogram response to light. These mice also displayed a delayed recovery of the b-wave amplitude after bleaching, however, visual cycle speed was unchanged, and both retinal pigment epithelium and photoreceptors presented the same fatty acid pattern compared to controls. In 2 year-old Fatp1(-/-) mice, transmission electron microscopy studies showed specific abnormalities in the retinas comprising choroid vascularization anomalies and thickening of the Bruch membrane with material deposits, and sometimes local disorganization of the photoreceptor outer segments. These anomalies lead us to speculate that the absence of FATP1 accelerates the aging process.http://europepmc.org/articles/PMC3500375?pdf=render
spellingShingle Karim Chekroud
Laurent Guillou
Stephane Grégoire
Gilles Ducharme
Emilie Brun
Chantal Cazevieille
Lionel Bretillon
Christian P Hamel
Philippe Brabet
Marie O Pequignot
Fatp1 deficiency affects retinal light response and dark adaptation, and induces age-related alterations.
PLoS ONE
title Fatp1 deficiency affects retinal light response and dark adaptation, and induces age-related alterations.
title_full Fatp1 deficiency affects retinal light response and dark adaptation, and induces age-related alterations.
title_fullStr Fatp1 deficiency affects retinal light response and dark adaptation, and induces age-related alterations.
title_full_unstemmed Fatp1 deficiency affects retinal light response and dark adaptation, and induces age-related alterations.
title_short Fatp1 deficiency affects retinal light response and dark adaptation, and induces age-related alterations.
title_sort fatp1 deficiency affects retinal light response and dark adaptation and induces age related alterations
url http://europepmc.org/articles/PMC3500375?pdf=render
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