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A High-Throughput Assay for Congenital and Age-Related Eye Diseases in Zebrafish

Debilitating visual impairment caused by cataracts or microphthalmia is estimated to affect roughly 20 million people in the United States alone. According to the National Eye Institute, by 2050 that number is expected to more than double to roughly 50 million. The identification of candidate diseas...

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Autores principales: Brastrom, Lindy K., Scott, C. Anthony, Dawson, Deborah V., Slusarski, Diane C.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6631034/
https://www.ncbi.nlm.nih.gov/pubmed/30979021
http://dx.doi.org/10.3390/biomedicines7020028
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author Brastrom, Lindy K.
Scott, C. Anthony
Dawson, Deborah V.
Slusarski, Diane C.
author_facet Brastrom, Lindy K.
Scott, C. Anthony
Dawson, Deborah V.
Slusarski, Diane C.
author_sort Brastrom, Lindy K.
collection PubMed
description Debilitating visual impairment caused by cataracts or microphthalmia is estimated to affect roughly 20 million people in the United States alone. According to the National Eye Institute, by 2050 that number is expected to more than double to roughly 50 million. The identification of candidate disease-causing alleles for cataracts and microphthalmia has been accelerated with advanced sequencing technologies creating a need for verification of the pathophysiology of these genes. Zebrafish pose many advantages as a high-throughput model for human eye disease. By 5 days post-fertilization, zebrafish have quantifiable behavioral responses to visual stimuli. Their small size, many progeny, and external fertilization allows for rapid screening for vision defects. We have adapted the OptoMotor Response to assay visual impairment in zebrafish models of cataracts and microphthalmia. This research demonstrates an inexpensive, high-throughput method for analyzing candidate genes involved in visual impairment.
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spelling pubmed-66310342019-08-19 A High-Throughput Assay for Congenital and Age-Related Eye Diseases in Zebrafish Brastrom, Lindy K. Scott, C. Anthony Dawson, Deborah V. Slusarski, Diane C. Biomedicines Article Debilitating visual impairment caused by cataracts or microphthalmia is estimated to affect roughly 20 million people in the United States alone. According to the National Eye Institute, by 2050 that number is expected to more than double to roughly 50 million. The identification of candidate disease-causing alleles for cataracts and microphthalmia has been accelerated with advanced sequencing technologies creating a need for verification of the pathophysiology of these genes. Zebrafish pose many advantages as a high-throughput model for human eye disease. By 5 days post-fertilization, zebrafish have quantifiable behavioral responses to visual stimuli. Their small size, many progeny, and external fertilization allows for rapid screening for vision defects. We have adapted the OptoMotor Response to assay visual impairment in zebrafish models of cataracts and microphthalmia. This research demonstrates an inexpensive, high-throughput method for analyzing candidate genes involved in visual impairment. MDPI 2019-04-11 /pmc/articles/PMC6631034/ /pubmed/30979021 http://dx.doi.org/10.3390/biomedicines7020028 Text en © 2019 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (http://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Brastrom, Lindy K.
Scott, C. Anthony
Dawson, Deborah V.
Slusarski, Diane C.
A High-Throughput Assay for Congenital and Age-Related Eye Diseases in Zebrafish
title A High-Throughput Assay for Congenital and Age-Related Eye Diseases in Zebrafish
title_full A High-Throughput Assay for Congenital and Age-Related Eye Diseases in Zebrafish
title_fullStr A High-Throughput Assay for Congenital and Age-Related Eye Diseases in Zebrafish
title_full_unstemmed A High-Throughput Assay for Congenital and Age-Related Eye Diseases in Zebrafish
title_short A High-Throughput Assay for Congenital and Age-Related Eye Diseases in Zebrafish
title_sort high-throughput assay for congenital and age-related eye diseases in zebrafish
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6631034/
https://www.ncbi.nlm.nih.gov/pubmed/30979021
http://dx.doi.org/10.3390/biomedicines7020028
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