Research Paper Volume 5, Issue 6 pp 427—444
Deficiency in the metabolite receptor SUCNR1 (GPR91) leads to outer retinal lesions
- 1 Department of Ophthalmology, Hopital Maisonneuve-Rosemont Research Centre, University of Montreal, Montreal, Quebec, H1T 2M4, Canada
- 2 Department of Biochemistry, Hopital Maisonneuve-Rosemont Research Centre, University of Montreal, Montreal, Quebec, H1T 2M4, Canada
- 3 Department of Immunology, Hopital Maisonneuve-Rosemont Research Centre, University of Montreal, Montreal, Quebec, H1T 2M4, Canada
- 4 Novartis Institutes for Biomedical Research, Basel CH-4002, Switzerland
- 5 Université Pierre et Marie Curie et Université Descartes, Paris, France; 2 INSERM, CRC UMRS872 team 17, Paris, France
- 6 INSERM, U 968, Paris, F-75012, France CNRS, UMR_7210 & UPMC Univ Paris 06, UMR_S 968, Institut de la Vision, Paris, F-75012, France
- 7 AP-HP, Hôtel Dieu, Service d'Ophtalmologie, Centre de Recherche ophtalmologique, 75006 Paris
- 8 Clinical Trials Branch and Clinical Research, National Eye Institute, Bethesda, MD 20892
Received: May 2, 2013 Accepted: June 15, 2013 Published: June 17, 2013
https://doi.org/10.18632/aging.100563How to Cite
Abstract
Age-related macular degeneration (AMD) is a prominent cause of blindness in the Western world. To date, its molecular pathogenesis as well as the sequence of events leading to retinal degeneration remain largely ill-defined. While the invasion of choroidal neovasculature in the retina is the primary mechanism that precipitates loss of sight, an earlier dry form may accompany it. Here we provide the first evidence for the protective role of the Retinal Pigment Epithelium (RPE)-resident metabolite receptor, succinate receptor 1 (SUCNR1; G-Protein coupled Receptor-91 (GPR91), in preventing dry AMD-like lesions of the outer retina. Genetic analysis of 925 patients with geographic atrophy and 1199 AMD-free peers revealed an increased risk of developing geographic atrophy associated with intronic variants in the SUCNR1 gene. In mice, outer retinal expression of SUCNR1 is observed in the RPE as well as microglial cells and decreases progressively with age. Accordingly, Sucnr1−/− mice show signs of premature sub-retinal dystrophy with accumulation of oxidized-LDL, abnormal thickening of Bruch's membrane and a buildup of subretinal microglia. The accumulation of microglia in Sucnr1-deficient mice is likely triggered by the inefficient clearance of oxidized lipids by the RPE as bone marrow transfer of wild-type microglia into Sucnr1−/− mice did not salvage the patho-phenotype and systemic lipolysis was equivalent between wild-type and control mice. Our findings suggest that deficiency in SUCNR1 is a possible contributing factor to the pathogenesis of dry AMD and thus broaden our understanding of this clinically unmet need.