Oral Levodopa rescues retinal morphology and visual function in a murine model of human albinism
Oral Levodopa rescues retinal morphology and visual function in a murine model of human albinism
Albinism is a group of disorders characterized by pigment deficiency and abnormal retinal development. Despite being a common cause for visual impairment worldwide, there is a paucity of treatments and patients typically suffer lifelong visual disability. Residual plasticity of the developing retina in young children with albinism has been demonstrated, suggesting a post-natal window for therapeutic rescue. L-3, 4 dihydroxyphenylalanine (L-DOPA), a key signalling molecule which is essential for normal retinal development, is known to be deficient in albinism. In this study, we demonstrate for the first time that post-natal L-DOPA supplementation can rescue retinal development, morphology and visual function in a murine model of human albinism, but only if administered from birth or 15 days post-natal age.
Albinism, Ocular, Vision, Levodopa, Therapeutics, Retina, Neuronal Plasticity
657-671
Lee, Helena
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Scott, Jennifer
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Griffiths, Helen
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Self, James
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Lotery, Andrew
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Lee, Helena
5d36fd1e-9334-4db5-b201-034d147133fb
Scott, Jennifer
bdc803de-3082-4727-a4ca-f5a1cf3fcfcc
Griffiths, Helen
a097fdaa-d3d6-49a9-9c69-0e6e5a5d518b
Self, James
0f6efc58-ae24-4667-b8d6-6fafa849e389
Lotery, Andrew
5ecc2d2d-d0b4-468f-ad2c-df7156f8e514
Lee, Helena, Scott, Jennifer, Griffiths, Helen, Self, James and Lotery, Andrew
(2019)
Oral Levodopa rescues retinal morphology and visual function in a murine model of human albinism.
Pigment Cell & Melanoma Research, 32 (5), .
(doi:10.1111/pcmr.12782).
Abstract
Albinism is a group of disorders characterized by pigment deficiency and abnormal retinal development. Despite being a common cause for visual impairment worldwide, there is a paucity of treatments and patients typically suffer lifelong visual disability. Residual plasticity of the developing retina in young children with albinism has been demonstrated, suggesting a post-natal window for therapeutic rescue. L-3, 4 dihydroxyphenylalanine (L-DOPA), a key signalling molecule which is essential for normal retinal development, is known to be deficient in albinism. In this study, we demonstrate for the first time that post-natal L-DOPA supplementation can rescue retinal development, morphology and visual function in a murine model of human albinism, but only if administered from birth or 15 days post-natal age.
Text
Oral Levodopa rescues retinal morphology and visual function in a murine model of human albinism
- Accepted Manuscript
More information
Accepted/In Press date: 5 March 2019
e-pub ahead of print date: 9 March 2019
Keywords:
Albinism, Ocular, Vision, Levodopa, Therapeutics, Retina, Neuronal Plasticity
Identifiers
Local EPrints ID: 428618
URI: http://eprints.soton.ac.uk/id/eprint/428618
ISSN: 1755-1471
PURE UUID: 6a7191b2-1b7a-4b6f-9d1c-45927a809547
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Date deposited: 05 Mar 2019 17:30
Last modified: 06 Jun 2024 04:15
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Author:
Jennifer Scott
Author:
Helen Griffiths
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