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Publication Detail
Antioxidant and lipid supplementation improve the development of photoreceptor outer segments in pluripotent stem cell-derived retinal organoids
  • Publication Type:
    Journal article
  • Authors:
    West EL, Majunder P, Naeem A, Fernando M, O'Hara-Wright M, Lanning E, Kloc M, Ribeiro J, Ovando-Roche P, Shum IO, Jumbu N, Sampson R, Hayes M, Bainbridge JWB, Georgiadis A, Smith AJ, Gonzalez-Cordero A, Ali RR
  • Publisher:
    Elsevier BV
  • Publication date:
  • Journal:
    Stem Cell Reports
  • Status:
  • Country:
    United States
  • PII:
  • Language:
  • Keywords:
    RPGR, degeneration, disease modeling, docosahexaenoic acid, gene therapy, induced pluripotent stem cells, outer segments, photoreceptors, retinal organoids, retinitis pigmentosa
  • Notes:
    © The Author 2022. Original content in this thesis is licensed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International (CC BY-NC-ND 4.0) Licence (https://creativecommons.org/licenses/by-nc-nd/4.0/).
The generation of retinal organoids from human pluripotent stem cells (hPSC) is now a well-established process that in part recapitulates retinal development. However, hPSC-derived photoreceptors that exhibit well-organized outer segment structures have yet to be observed. To facilitate improved inherited retinal disease modeling, we determined conditions that would support outer segment development in maturing hPSC-derived photoreceptors. We established that the use of antioxidants and BSA-bound fatty acids promotes the formation of membranous outer segment-like structures. Using new protocols for hPSC-derived retinal organoid culture, we demonstrated improved outer segment formation for both rod and cone photoreceptors, including organized stacked discs. Using these enhanced conditions to generate iPSC-derived retinal organoids from patients with X-linked retinitis pigmentosa, we established robust cellular phenotypes that could be ameliorated following adeno-associated viral vector-mediated gene augmentation. These findings should aid both disease modeling and the development of therapeutic approaches for the treatment of photoreceptor disorders.
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