Literature DB >> 31043342

AKT3 Gene Transfer Promotes Anabolic Reprogramming and Photoreceptor Neuroprotection in a Pre-clinical Model of Retinitis Pigmentosa.

Devin S McDougald1, Tyler E Papp1, Alexandra U Zezulin1, Shangzhen Zhou1, Jean Bennett2.   

Abstract

Mutations within over 250 known genes are associated with inherited retinal degeneration. Clinical success following gene-replacement therapy for congenital blindness due to RPE65 mutations establishes a platform for the development of downstream treatments targeting other forms of inherited ocular disease. Unfortunately, several challenges relevant to complex disease pathology and limitations of current gene-transfer technologies impede the development of related strategies for each specific form of inherited retinal degeneration. Here, we describe a gene-augmentation strategy that delays retinal degeneration by stimulating features of anabolic metabolism necessary for survival and structural maintenance of photoreceptors. We targeted two critical points of regulation in the canonical insulin/AKT/mammalian target of rapamycin (mTOR) pathway with AAV-mediated gene augmentation in a mouse model of retinitis pigmentosa. AAV vectors expressing the serine/threonine kinase, AKT3, promote dramatic preservation of photoreceptor numbers, structure, and partial visual function. This protective effect was associated with successful reprogramming of photoreceptor metabolism toward pathways associated with cell growth and survival. Collectively, these findings underscore the importance of AKT activity and downstream pathways associated with anabolic metabolism in photoreceptor survival and maintenance.
Copyright © 2019 The American Society of Gene and Cell Therapy. Published by Elsevier Inc. All rights reserved.

Entities:  

Keywords:  AAV; AKT; gene therapy; neuroprotection; photoreceptor; retinitis pigmentosa; rheb

Mesh:

Substances:

Year:  2019        PMID: 31043342      PMCID: PMC6612630          DOI: 10.1016/j.ymthe.2019.04.009

Source DB:  PubMed          Journal:  Mol Ther        ISSN: 1525-0016            Impact factor:   12.910


  47 in total

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Review 6.  AKT/PKB Signaling: Navigating the Network.

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8.  In vivo-directed evolution of a new adeno-associated virus for therapeutic outer retinal gene delivery from the vitreous.

Authors:  Deniz Dalkara; Leah C Byrne; Ryan R Klimczak; Meike Visel; Lu Yin; William H Merigan; John G Flannery; David V Schaffer
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9.  Stimulation of the insulin/mTOR pathway delays cone death in a mouse model of retinitis pigmentosa.

Authors:  Claudio Punzo; Karl Kornacker; Constance L Cepko
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Review 10.  Loss of daylight vision in retinal degeneration: are oxidative stress and metabolic dysregulation to blame?

Authors:  Claudio Punzo; Wenjun Xiong; Constance L Cepko
Journal:  J Biol Chem       Date:  2011-11-10       Impact factor: 5.157

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  3 in total

Review 1.  Endoplasmic reticulum stress: New insights into the pathogenesis and treatment of retinal degenerative diseases.

Authors:  Marina S Gorbatyuk; Christopher R Starr; Oleg S Gorbatyuk
Journal:  Prog Retin Eye Res       Date:  2020-04-06       Impact factor: 21.198

Review 2.  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

Review 3.  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
  3 in total

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