Literature DB >> 30851223

Oral levodopa rescues retinal morphology and visual function in a murine model of human albinism.

Helena Lee1,2, Jennifer Scott1, Helen Griffiths1, Jay E Self1,2, Andrew Lotery1,2.   

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.
© 2019 The Authors. Pigment Cell & Melanoma Research Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  albinism; levodopa; neuronal plasticity; ocular; retina; therapeutics; vision

Mesh:

Substances:

Year:  2019        PMID: 30851223      PMCID: PMC6766973          DOI: 10.1111/pcmr.12782

Source DB:  PubMed          Journal:  Pigment Cell Melanoma Res        ISSN: 1755-1471            Impact factor:   4.693


  43 in total

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Review 6.  Anatomical correlates to the bands seen in the outer retina by optical coherence tomography: literature review and model.

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7.  Characterization of mouse cortical spatial vision.

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8.  Structural grading of foveal hypoplasia using spectral-domain optical coherence tomography a predictor of visual acuity?

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Journal:  PLoS Biol       Date:  2008-09-30       Impact factor: 8.029

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Journal:  NPJ Genom Med       Date:  2022-01-13       Impact factor: 6.083

4.  The Dct-/- Mouse Model to Unravel Retinogenesis Misregulation in Patients with Albinism.

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5.  Non-syndromic Oculocutaneous Albinism: Novel Genetic Variants and Clinical Follow Up of a Brazilian Pediatric Cohort.

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  5 in total

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