title: Function Over Form: Modeling Groups of Inherited Neurological Conditions in Zebrafish.
abstract: 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.
authors.affiliation: Department of Biology, University of Miami Coral Gables, FL, USA.
authors.forename: Robert A
authors.initials: RA
authors.lastname: Kozol
authors.affiliation: Department of Human Genetics, John P. Hussman Institute for Human Genomics, Dr. John T. Macdonald Foundation, University of Miami Miami, FL, USA.
authors.forename: Alexander J
authors.initials: AJ
authors.lastname: Abrams
authors.affiliation: Department of Biology, University of Miami Coral Gables, FL, USA.
authors.forename: David M
authors.initials: DM
authors.lastname: James
authors.affiliation: Department of Human Genetics, John P. Hussman Institute for Human Genomics, Dr. John T. Macdonald Foundation, University of Miami Miami, FL, USA.
authors.forename: Elena
authors.initials: E
authors.lastname: Buglo
authors.affiliation: Department of Biology, University of Miami Coral Gables, FL, USA.
authors.forename: Qing
authors.initials: Q
authors.lastname: Yan
authors.affiliation: Department of Biology, University of Miami Coral Gables, FL, USA.
authors.forename: Julia E
authors.initials: JE
authors.lastname: Dallman
keywords: Charcot-Marie tooth; ataxia; autism spectrum disorder; disease modeling; hereditary spastic paraplegia; intellectual disability; schizophrenia; zebrafish