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Photoreceptor and retinal pigment epithelium (RPE) dysfunction in inherited retinal degenerations (IRDs) and age-related macular degeneration (AMD) necessitate innovative therapies to preserve vision. Vision impairment incurs a substantial global economic burden, with the World Health Organization reporting an annual global productivity loss of approximately $411 billion. Current treatments are limited, underscoring the urgency for novel solutions. Leveraging new screening techniques, novel drugs restoring vision can be uncovered. Here, a workflow is described utilising orthogonal pooling to screen randomised library compounds for drug hits restoring vision and assessing the optokinetic response (OKR) in the atp6v0e1−/− zebrafish model of inherited blindness.

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Uncovering Novel Drugs that Restore Vision Using Orthogonal Pooling in Zebrafish

  • 会议论文

  • 发表日期：

    2025年

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  • Justine O’Brien,
  • Patrizia Colucci,
  • Yolanda Alvarez,
  • Breandán N. Kennedy

摘要

Photoreceptor and retinal pigment epithelium (RPE) dysfunction in inherited retinal degenerations (IRDs) and age-related macular degeneration (AMD) necessitate innovative therapies to preserve vision. Vision impairment incurs a substantial global economic burden, with the World Health Organization reporting an annual global productivity loss of approximately $411 billion. Current treatments are limited, underscoring the urgency for novel solutions. Leveraging new screening techniques, novel drugs restoring vision can be uncovered. Here, a workflow is described utilising orthogonal pooling to screen randomised library compounds for drug hits restoring vision and assessing the optokinetic response (OKR) in the atp6v0e1−/− zebrafish model of inherited blindness.