PUBLICATION

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

Authors: Brastrom, L.K., Scott, C.A., Dawson, D.V., Slusarski, D.C.
ID: ZDB-PUB-190414-1
Date: 2019
Source: Biomedicines 7(2): (Journal)
Registered Authors: Slusarski, Diane C.
Keywords: cataracts, crim1, microphthalmia, optomotor response, rbm24a, vision, visual assay, visual impairment disorders, zebrafish
MeSH Terms: none
PubMed: 30979021 Full text @ Biomedicines

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.

Genes / Markers

Figures

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Expression

Gene	Fish	Conditions	Stage	Anatomy	Assay	Figure
crim1	WT	standard conditions	Protruding-mouth	optic choroid	ISH	Fig. 4
crim1	WT	standard conditions	Protruding-mouth	retinal ganglion cell layer	ISH	Fig. 4
rbm24a	WT	standard conditions	Protruding-mouth	lens	ISH	Fig. 4

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Phenotype

Fish	Conditions	Stage	Phenotype	Figure
WT + MO1-crim1	standard conditions	Protruding-mouth	eye decreased size, abnormal	Fig. 7
WT + MO1-crim1	standard conditions	Protruding-mouth	eye decreased size, abnormal	Fig. 4
WT + MO1-crim1	standard conditions	Protruding-mouth	lens nucleus increased amount, abnormal	Fig. 7
WT + MO1-crim1	standard conditions	Protruding-mouth	optomotor response behavioral quality of a process, normal	Fig. 4
WT + MO1-crim1	standard conditions	Protruding-mouth	retina layer formation disrupted, abnormal	Fig. 7
WT + MO1-crim1	standard conditions	Protruding-mouth	visual behavior process quality, normal	Fig. 4
WT + MO1-crim1	standard conditions	Protruding-mouth	whole organism shape, normal	Fig. 4
WT + MO6-rbm24a	standard conditions	Protruding-mouth	eye decreased size, abnormal	Fig. 7
WT + MO6-rbm24a	standard conditions	Protruding-mouth	eye decreased size, abnormal	Fig. 4
WT + MO6-rbm24a	standard conditions	Protruding-mouth	lens nucleus increased amount, abnormal	Fig. 7

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Mutations / Transgenics

Allele	Construct	Type	Affected Genomic Region
tm213		Point Mutation	axin1

Human Disease / Model

Human Disease	Fish	Conditions	Evidence
cataract	WT + MO1-crim1	standard conditions	TAS
cataract	WT + MO6-rbm24a	standard conditions	TAS
microphthalmia	WT + MO1-crim1	standard conditions	TAS
microphthalmia	WT + MO6-rbm24a	standard conditions	TAS

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Sequence Targeting Reagents

Target	Reagent	Reagent Type
crim1	MO1-crim1	MRPHLNO
rbm24a	MO6-rbm24a	MRPHLNO

Fish

Fish
axin1^tm213/tm213
WT
WT + MO1-crim1
WT + MO6-rbm24a

Antibodies

Orthology

Engineered Foreign Genes

Mapping

Brastrom et al., 2019 ZDB-PUB-190414-1 PMID:30979021

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

Brastrom et al., 2019

ZDB-PUB-190414-1
PMID:30979021