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ADIPOR1 is essential for vision and its RPE expression is lost in the Mfrprd6 mouse.
Sluch, Valentin M; Banks, Angela; Li, Hui; Crowley, Maura A; Davis, Vanessa; Xiang, Chuanxi; Yang, Junzheng; Demirs, John T; Vrouvlianis, Joanna; Leehy, Barrett; Hanks, Shawn; Hyman, Alexandra M; Aranda, Jorge; Chang, Bo; Bigelow, Chad E; Rice, Dennis S.
Afiliação
  • Sluch VM; Department of Ophthalmology, Novartis Institutes for BioMedical Research, Cambridge, Massachusetts, United States. valsluch@gmail.com.
  • Banks A; Department of Ophthalmology, Novartis Institutes for BioMedical Research, Cambridge, Massachusetts, United States.
  • Li H; Department of Ophthalmology, Novartis Institutes for BioMedical Research, Cambridge, Massachusetts, United States.
  • Crowley MA; Department of Ophthalmology, Novartis Institutes for BioMedical Research, Cambridge, Massachusetts, United States.
  • Davis V; Global Scientific Operations, Novartis Institutes for BioMedical Research, Cambridge, Massachusetts, United States.
  • Xiang C; Department of Ophthalmology, Novartis Institutes for BioMedical Research, Cambridge, Massachusetts, United States.
  • Yang J; Department of Ophthalmology, Novartis Institutes for BioMedical Research, Cambridge, Massachusetts, United States.
  • Demirs JT; Department of Ophthalmology, Novartis Institutes for BioMedical Research, Cambridge, Massachusetts, United States.
  • Vrouvlianis J; Department of Ophthalmology, Novartis Institutes for BioMedical Research, Cambridge, Massachusetts, United States.
  • Leehy B; Department of Ophthalmology, Novartis Institutes for BioMedical Research, Cambridge, Massachusetts, United States.
  • Hanks S; Department of Ophthalmology, Novartis Institutes for BioMedical Research, Cambridge, Massachusetts, United States.
  • Hyman AM; Department of Ophthalmology, Novartis Institutes for BioMedical Research, Cambridge, Massachusetts, United States.
  • Aranda J; Department of Ophthalmology, Novartis Institutes for BioMedical Research, Cambridge, Massachusetts, United States.
  • Chang B; The Jackson Laboratory, Bar Harbor, Maine, United States.
  • Bigelow CE; Department of Ophthalmology, Novartis Institutes for BioMedical Research, Cambridge, Massachusetts, United States.
  • Rice DS; Department of Ophthalmology, Novartis Institutes for BioMedical Research, Cambridge, Massachusetts, United States. dennis.rice@novartis.com.
Sci Rep ; 8(1): 14339, 2018 09 25.
Article em En | MEDLINE | ID: mdl-30254279
ABSTRACT
The knockout (KO) of the adiponectin receptor 1 (AdipoR1) gene causes retinal degeneration. Here we report that ADIPOR1 protein is primarily found in the eye and brain with little expression in other tissues. Further analysis of AdipoR1 KO mice revealed that these animals exhibit early visual system abnormalities and are depleted of RHODOPSIN prior to pronounced photoreceptor death. A KO of AdipoR1 post-development either in photoreceptors or the retinal pigment epithelium (RPE) resulted in decreased expression of retinal proteins, establishing a role for ADIPOR1 in supporting vision in adulthood. Subsequent analysis of the Mfrprd6 mouse retina demonstrated that these mice are lacking ADIPOR1 in their RPE layer alone, suggesting that loss of ADIPOR1 drives retinal degeneration in this model. Moreover, we found elevated levels of IRBP in both the AdipoR1 KO and the Mfrprd6 models. The spatial distribution of IRBP was also abnormal. This dysregulation of IRBP hypothesizes a role for ADIPOR1 in retinoid metabolism.
Assuntos

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Visão Ocular / Regulação da Expressão Gênica / Receptores de Adiponectina / Epitélio Pigmentado da Retina / Técnicas de Inativação de Genes Limite: Animals / Humans Idioma: En Revista: Sci Rep Ano de publicação: 2018 Tipo de documento: Article País de afiliação: Estados Unidos

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Visão Ocular / Regulação da Expressão Gênica / Receptores de Adiponectina / Epitélio Pigmentado da Retina / Técnicas de Inativação de Genes Limite: Animals / Humans Idioma: En Revista: Sci Rep Ano de publicação: 2018 Tipo de documento: Article País de afiliação: Estados Unidos