Function over form: Modeling groups of inherited neurological conditions in zebrafish

Robert A. Kozol, Alexander J. Abrams, David M. James, Elena Buglo, Qing Yan, Julia E. Dallman

Research output: Contribution to journalReview articlepeer-review

41 Scopus citations


Zebrafish are a unique cell to behavior model for studying the basic biology of human inherited neurological conditions. Conserved vertebrate genetics and optical transparency provide in vivo access to the developing nervous system as well as high-throughput approaches for drug screens. Here we review zebrafish modeling for two broad groups of inherited conditions that each share genetic and molecular pathways and overlap phenotypically: Neurodevelopmental disorders such as Autism Spectrum Disorders (ASD), Intellectual Disability (ID) and Schizophrenia (SCZ), and neurodegenerative diseases, such as Cerebellar Ataxia (CATX), Hereditary Spastic Paraplegia (HSP) and Charcot-Marie Tooth Disease (CMT). We also conduct a small meta-analysis of zebrafish orthologs of high confidence neurodevelopmental disorder and neurodegenerative disease genes by looking at duplication rates and relative protein sizes. In the past zebrafish genetic models of these neurodevelopmental disorders and neurodegenerative diseases have provided insight into cellular, circuit and behavioral level mechanisms contributing to these conditions. Moving forward, advances in genetic manipulation, live imaging of neuronal activity and automated high-throughput molecular screening promise to help delineate the mechanistic relationships between different types of neurological conditions and accelerate discovery of therapeutic strategies.

Original languageEnglish (US)
Article number55
JournalFrontiers in Molecular Neuroscience
Issue numberJUL
StatePublished - Jul 7 2016


  • Ataxia
  • Autism spectrum disorder
  • Charcot-marie tooth
  • Disease modeling
  • Hereditary spastic paraplegia
  • Intellectual disability
  • Schizophrenia
  • Zebrafish

ASJC Scopus subject areas

  • Molecular Biology
  • Cellular and Molecular Neuroscience


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