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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
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
1755-1471
Lee, Helena
5d36fd1e-9334-4db5-b201-034d147133fb
Scott, Jennifer
bdc803de-3082-4727-a4ca-f5a1cf3fcfcc
Griffiths, Helen
a097fdaa-d3d6-49a9-9c69-0e6e5a5d518b
Self, James
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Lotery, Andrew
5ecc2d2d-d0b4-468f-ad2c-df7156f8e514
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. (doi:10.1111/pcmr.12782).

Record type: Article

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.

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Oral Levodopa rescues retinal morphology and visual function in a murine model of human albinism - Accepted Manuscript
Restricted to Repository staff only until 9 March 2020.
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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: https://eprints.soton.ac.uk/id/eprint/428618
ISSN: 1755-1471
PURE UUID: 6a7191b2-1b7a-4b6f-9d1c-45927a809547
ORCID for Helena Lee: ORCID iD orcid.org/0000-0002-2573-9536
ORCID for James Self: ORCID iD orcid.org/0000-0002-1030-9963
ORCID for Andrew Lotery: ORCID iD orcid.org/0000-0001-5541-4305

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Date deposited: 05 Mar 2019 17:30
Last modified: 24 Apr 2019 00:34

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Contributors

Author: Helena Lee ORCID iD
Author: Jennifer Scott
Author: Helen Griffiths
Author: James Self ORCID iD
Author: Andrew Lotery ORCID iD

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