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

1
Department of Biology, University of Iowa, Iowa City, IA 52245, USA
2
Department of Pediatrics, Baylor College of Medicine, Houston, TX 77030, USA
3
Departments of Biostatistics and Pediatric Dentistry, University of Iowa, Iowa City, IA 52245, USA
*
Author to whom correspondence should be addressed.
Biomedicines 2019, 7(2), 28; https://doi.org/10.3390/biomedicines7020028
Received: 28 February 2019 / Revised: 2 April 2019 / Accepted: 8 April 2019 / Published: 11 April 2019
(This article belongs to the Special Issue Zebrafish Models for Development and Disease)
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Abstract

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. View Full-Text
Keywords: vision; visual assay; optomotor response; visual impairment disorders; microphthalmia; cataracts; rbm24a; crim1; zebrafish vision; visual assay; optomotor response; visual impairment disorders; microphthalmia; cataracts; rbm24a; crim1; zebrafish
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This is an open access article distributed under the Creative Commons Attribution License which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited (CC BY 4.0).

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Brastrom, L.K.; Scott, C.A.; Dawson, D.V.; Slusarski, D.C. A High-Throughput Assay for Congenital and Age-Related Eye Diseases in Zebrafish. Biomedicines 2019, 7, 28.

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