Literature DB >> 32434914

Altered photoreceptor metabolism in mouse causes late stage age-related macular degeneration-like pathologies.

Shun-Yun Cheng1, Joris Cipi1, Shan Ma1, Brian P Hafler2,3, Rahul N Kanadia4, Richard S Brush5,6,7,8, Martin-Paul Agbaga9,6,7,8, Claudio Punzo10.   

Abstract

Age-related macular degeneration (AMD) is the leading cause of blindness in the elderly. While the histopathology of the different disease stages is well characterized, the cause underlying the progression, from the early drusen stage to the advanced macular degeneration stage that leads to blindness, remains unknown. Here, we show that photoreceptors (PRs) of diseased individuals display increased expression of two key glycolytic genes, suggestive of a glucose shortage during disease. Mimicking aspects of this metabolic profile in PRs of wild-type mice by activation of the mammalian target of rapamycin complex 1 (mTORC1) caused early drusen-like pathologies, as well as advanced AMD-like pathologies. Mice with activated mTORC1 in PRs also displayed other early disease features, such as a delay in photoreceptor outer segment (POS) clearance and accumulation of lipofuscin in the retinal-pigmented epithelium (RPE) and of lipoproteins at the Bruch's membrane (BrM), as well as changes in complement accumulation. Interestingly, formation of drusen-like deposits was dependent on activation of mTORC1 in cones. Both major types of advanced AMD pathologies, including geographic atrophy (GA) and neovascular pathologies, were also seen. Finally, activated mTORC1 in PRs resulted in a threefold reduction in di-docosahexaenoic acid (DHA)-containing phospholipid species. Feeding mice a DHA-enriched diet alleviated most pathologies. The data recapitulate many aspects of the human disease, suggesting that metabolic adaptations in photoreceptors could contribute to disease progression in AMD. Identifying the changes downstream of mTORC1 that lead to advanced pathologies in mouse might present new opportunities to study the role of PRs in AMD pathogenesis.
Copyright © 2020 the Author(s). Published by PNAS.

Entities:  

Keywords:  AMD; geographic atrophy; photoreceptor metabolism; photoreceptors; wet AMD

Year:  2020        PMID: 32434914     DOI: 10.1073/pnas.2000339117

Source DB:  PubMed          Journal:  Proc Natl Acad Sci U S A        ISSN: 0027-8424            Impact factor:   11.205


  26 in total

1.  Elevated energy requirement of cone photoreceptors.

Authors:  Norianne T Ingram; Gordon L Fain; Alapakkam P Sampath
Journal:  Proc Natl Acad Sci U S A       Date:  2020-07-27       Impact factor: 11.205

2.  A complement factor H homolog, heparan sulfation, and syndecan maintain inversin compartment boundaries in C. elegans cilia.

Authors:  Natalie Acker; Harold Smith; Claire Devine; Sharon L Oltjen; Sofia Tsiropoulou; Zeljka Smit-McBride; Karen Lange; Oliver E Blacque; Joanne A Matsubara; Andrew Gordus; Andy Golden; Bruce E Vogel
Journal:  Proc Natl Acad Sci U S A       Date:  2021-04-20       Impact factor: 11.205

Review 3.  Light responses of mammalian cones.

Authors:  Gordon L Fain; Alapakkam P Sampath
Journal:  Pflugers Arch       Date:  2021-03-19       Impact factor: 4.458

Review 4.  Retina Metabolism and Metabolism in the Pigmented Epithelium: A Busy Intersection.

Authors:  James B Hurley
Journal:  Annu Rev Vis Sci       Date:  2021-06-08       Impact factor: 6.422

5.  Spaceflight decelerates the epigenetic clock orchestrated with a global alteration in DNA methylome and transcriptome in the mouse retina.

Authors:  Zhong Chen; Seta Stanbouly; Nina C Nishiyama; Xin Chen; Michael D Delp; Hongyu Qiu; Xiao W Mao; Charles Wang
Journal:  Precis Clin Med       Date:  2021-05-17

6.  Progressive Photoreceptor Dysfunction and Age-Related Macular Degeneration-Like Features in rp1l1 Mutant Zebrafish.

Authors:  Nicole C L Noel; Nathan J Nadolski; Jennifer C Hocking; Ian M MacDonald; W Ted Allison
Journal:  Cells       Date:  2020-09-30       Impact factor: 6.600

Review 7.  A Re-Appraisal of Pathogenic Mechanisms Bridging Wet and Dry Age-Related Macular Degeneration Leads to Reconsider a Role for Phytochemicals.

Authors:  Roberto Pinelli; Francesca Biagioni; Fiona Limanaqi; Miorica Bertelli; Elena Scaffidi; Maico Polzella; Carla Letizia Busceti; Francesco Fornai
Journal:  Int J Mol Sci       Date:  2020-08-03       Impact factor: 5.923

8.  An In-Vitro Cell Model of Intracellular Protein Aggregation Provides Insights into RPE Stress Associated with Retinopathy.

Authors:  Eloise Keeling; Annabelle J Culling; David A Johnston; David S Chatelet; Anton Page; David A Tumbarello; Andrew J Lotery; J Arjuna Ratnayaka
Journal:  Int J Mol Sci       Date:  2020-09-11       Impact factor: 5.923

Review 9.  Potential Therapeutic Benefit of NAD+ Supplementation for Glaucoma and Age-Related Macular Degeneration.

Authors:  Gloria Cimaglia; Marcela Votruba; James E Morgan; Helder André; Pete A Williams
Journal:  Nutrients       Date:  2020-09-19       Impact factor: 5.717

Review 10.  Photoreceptor metabolic reprogramming: current understanding and therapeutic implications.

Authors:  Warren W Pan; Thomas J Wubben; Cagri G Besirli
Journal:  Commun Biol       Date:  2021-02-24
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