ArticleDevelopmental dynamics : an official publication of the American Association of Anatomists2016
Automated, high-throughput, in vivo analysis of visual function using the zebrafish.
Article in Developmental dynamics : an official publication of the American Association of Anatomists, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 25 papers.
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Who cites it
25 citing papers in PubMed.
- Comparing the Behavioral Impacts of Heavy Metals and Rare Earth Elements on Black Soldier Fly (Toxics · 2026Article
- Pharmaco-behavioral profiling identifies suppressors of autism gene-associated phenotypes in zebrafish.Proceedings of the National Academy of Sciences of the United States of America · 2026Article
- Estradiol modulates neuronal network hyperexcitability in select NDD risk genes.bioRxiv : the preprint server for biology · 2026Article
- Behavioral Vision Testing Using an Automated Observational Tracking System (VisioBox) to Obtain a Measure of Visual Acuity in Larval Zebrafish.Methods in molecular biology (Clifton, N.J.) · 2026Article
- Synaptic Gpr85 Influences Cerebellar-Granule-Cell Electrical Properties and Light-Induced Behavior in Zebrafish.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2025Article
- A Blueprint for Translational Precision Medicine in Autism Spectrum Disorder and Related Neurogenetic Syndromes.Journal of child and adolescent psychopharmacology · 2025Review
- Natterin bridges IFN-φ1 and non-canonical inflammasome pathways viaFrontiers in cellular and infection microbiology · 2025Article
- Bi-allelic ACBD6 variants lead to a neurodevelopmental syndrome with progressive and complex movement disorders.Brain : a journal of neurology · 2024Article
- Evaluating the association of biallelic OGDHL variants with significant phenotypic heterogeneity.Genome medicine · 2023Article
- High-throughput functional analysis of autism genes in zebrafish identifies convergence in dopaminergic and neuroimmune pathways.Cell reports · 2023Article
- Zebrafish Larvae Behavior Models as a Tool for Drug Screenings and Pre-Clinical Trials: A Review.International journal of molecular sciences · 2022Review
- Development and biological characterization of a clinical gene transfer vector for the treatment of MAK-associated retinitis pigmentosa.Gene therapy · 2022Article
- Article
- The pursuit of precision mitochondrial medicine: Harnessing preclinical cellular and animal models to optimize mitochondrial disease therapeutic discovery.Journal of inherited metabolic disease · 2021Review
- Functional Role of the RNA-Binding Protein Rbm24a and Its TargetBiomedicines · 2021Article
- The αScientific reports · 2019Article
- Genome-wide suppressor screen identifies USP35/USP38 as therapeutic candidates for ciliopathies.JCI insight · 2019Article
- Let's get small (and smaller): Combining zebrafish and nanomedicine to advance neuroregenerative therapeutics.Advanced drug delivery reviews · 2019Review
- A High-Throughput Assay for Congenital and Age-Related Eye Diseases in Zebrafish.Biomedicines · 2019Article
- Mutations in the Glycosyltransferase Domain of GLT8D1 Are Associated with Familial Amyotrophic Lateral Sclerosis.Cell reports · 2019Article
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3 authors.
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Abstract
backgroundModern genomics has enabled the identification of an unprecedented number of genetic variants, which in many cases are extremely rare, associated with blinding disorders. A significant challenge will be determining the pathophysiology of each new variant. The Zebrafish is an excellent model for the study of inherited diseases of the eye. By 5 days post-fertilization (dpf), they have quantifiable behavioral responses to visual stimuli. However, visual behavior assays can take several hours to perform or can only be assessed one fish at a time.
resultsTo increase the throughput for vision assays, we used the Viewpoint Zebrabox to automate the visual startle response and created software, Visual Interrogation of Zebrafish Manipulations (VIZN), to automate data analysis. This process allows 96 Zebrafish larvae to be tested and resultant data to be analyzed in less than 35 minutes. We validated this system by disrupting function of a gene necessary for photoreceptor differentiation and observing decreased response to visual stimuli.
conclusionsThis automated method along with VIZN allows rapid, high-throughput, in vivo testing of Zebrafish's ability to respond to light/dark stimuli. This allows the rapid analysis of novel genes involved in visual function by morpholino, CRISPRS, or small-molecule drug screens. Developmental Dynamics 245:605-613, 2016. © 2016 Wiley Periodicals, Inc.
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