Zebrafish models for translational neuroscience research: from tank to bedside

• The zebrafish is a popular novel model in translational neuroscience research.
• Both larval and adult zebrafish contribute to modeling complex brain disorders.
• Zebrafish models can be used to study CNS cancer, epilepsy, anxiety, and autism.
• Zebrafish are highly sensitive to major classes of neurotropic drugs.
• Zebrafish are a useful tool for high-throughput genetic and small molecule screening.

The zebrafish (Danio rerio) is emerging as a new important species for studying mechanisms of brain function and dysfunction. Focusing on selected central nervous system (CNS) disorders (brain cancer, epilepsy, and anxiety) and using them as examples, we discuss the value of zebrafish models in translational neuroscience. We further evaluate the contribution of zebrafish to neuroimaging, circuit level, and drug discovery research. Outlining the role of zebrafish in modeling a wide range of human brain disorders, we also summarize recent applications and existing challenges in this field. Finally, we emphasize the potential of zebrafish models in behavioral phenomics and high-throughput genetic/small molecule screening, which is critical for CNS drug discovery and identifying novel candidate genes.