Distinct models of induced hyperactivity in zebrafish larvae

Ellis, Lee; Seibert, Jake; Soanes, Kelly H.

doi:10.1016/j.brainres.2012.02.022

Cited by 83 publications

(96 citation statements)

References 45 publications

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“…Seizure behaviors were never observed in controls at any stage of development ( n = 36). In pools of mutant and sibling control larvae, scn1Lab mutants stay close to the sides of the petri dish, which is considered a form of thigmotaxis in fish 29 . These results reveal a striking epilepsy phenotype in scn1Lab mutant zebrafish.…”

Section: Resultsmentioning

confidence: 99%

Drug screening in Scn1a zebrafish mutant identifies clemizole as a potential Dravet syndrome treatment

2013

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Dravet syndrome (DS) is a catastrophic pediatric epilepsy with severe intellectual disability, impaired social development and persistent drug-resistant seizures. One of its primary monogenic causes are mutations in Nav1.1 (SCN1A), a voltage-gated sodium channel. Here we characterise zebrafish Nav1.1 (scn1Lab) mutants originally identified in a chemical mutagenesis screen. Mutants exhibit spontaneous abnormal electrographic activity, hyperactivity and convulsive behaviors. Although scn1Lab expression is reduced, microarray analysis is remarkable for the small fraction of differentially expressed genes (~3%) and lack of compensatory expression changes in other scn subunits. Ketogenic diet, diazepam, valproate, potassium bromide and stiripentol attenuate mutant seizure activity; seven other antiepileptic drugs have no effect. A phenotype-based screen of 320 compounds identifies a US Food and Drug Administration-approved compound (clemizole) that inhibits convulsive behaviors and electrographic seizures. This approach represents a new direction in modeling pediatric epilepsy and could be used to identify novel therapeutics for any monogenic epilepsy disorder.

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Section: Resultsmentioning

confidence: 99%

Drug screening in Scn1a zebrafish mutant identifies clemizole as a potential Dravet syndrome treatment

2013

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“…optogenetics) (Chen et al, 2016;Portugues et al, 2013). "Snapshots" of whole brain activity can also be taken using in situ hybridization of the immediate early gene c-fos, a marker of neuronal activity (Ellis et al, 2012), or with antibody staining for another activity marker, phosphorylated ERK (Randlett et al, 2015). Together, this collection of tools for the interrogation of neural development and function will enable in vivo dissection of the changes that occur when ASD risk genes are mutated.…”

Section: Unique Advantages Of Zebrafish In Asd Modelingmentioning

confidence: 99%

The zebrafish as a promising tool for modeling human brain disorders: A review based upon an IBNS Symposium

Shams

Rihel

Ortíz

et al. 2018

Neuroscience & Biobehavioral Reviews

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The zebrafish represents an excellent compromise between system complexity and practical simplicity, features that make it useful for modeling and mechanistic analysis of complex brain disorders. Also promising are screens for psychoactive drugs with effects on larval and adult zebrafish behavior. This review, based upon a recent symposium held at the 2016 IBNS Congress, provides different perspectives on how the zebrafish may be utilized to advance research into human central nervous system disorders. It starts with a discussion on an important bottleneck in zebrafish research, measuring the behavior of this species (specifically shoaling), and continues with examples on research on autism spectrum disorder in larval zebrafish, on screening natural products for compounds with psychoactive properties in adult zebrafish, and on the development of a zebrafish model of fetal alcohol spectrum disorders. By providing information on a broad spectrum of brain disorders, experimental methods, and scientific approaches using both larval and adult zebrafish, the review is intended to showcase this underutilized laboratory species for behavioral neuroscience and psychopharmacology research.

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“…In a model of hyperactivity, Ellis et al (2012) showed zebrafish larvae could be used to distinguish the effects of three compounds associated with hyperactivity. Here, pentylenetetrazole (PTZ) exposure reversed the species-typical swimming pattern seen during conditions of light and dark.…”

Section: Therapeutic Drugsmentioning

confidence: 99%

Zebrafish model systems for developmental neurobehavioral toxicology

Bailey

Oliveri

Levin

2013

Birth Defects Research Pt C

158

116

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Zebrafish offer many advantages that complement classic mammalian models for the study of normal development as well as for the teratogenic effects of exposure to hazardous compounds. The clear chorion and embryo of the zebrafish allow for continuous visualization of the anatomical changes associated with development, which, along with short maturation times and the capability of complex behavior, makes this model particularly useful for measuring changes to the developing nervous system. Moreover, the rich array of developmental, behavioral, and molecular benefits offered by the zebrafish have contributed to an increasing demand for the use of zebrafish in behavioral teratology. Essential for this endeavor has been the development of a battery of tests to evaluate a spectrum of behavior in zebrafish. Measures of sensorimotor plasticity, emotional function, cognition and social interaction have been used to characterize the persisting adverse effects of developmental exposure to a variety of chemicals including therapeutic drugs, drugs of abuse and environmental toxicants. In this review, we present and discuss such tests and data from a range of developmental neurobehavioral toxicology studies using zebrafish as a model. Zebrafish provide a key intermediate model between high throughput in vitro screens and the classic mammalian models as they have the accessibility of in vitro models and the complex functional capabilities of mammalian models.

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Distinct models of induced hyperactivity in zebrafish larvae

Cited by 83 publications

References 45 publications

Drug screening in Scn1a zebrafish mutant identifies clemizole as a potential Dravet syndrome treatment

Drug screening in Scn1a zebrafish mutant identifies clemizole as a potential Dravet syndrome treatment

The zebrafish as a promising tool for modeling human brain disorders: A review based upon an IBNS Symposium

Zebrafish model systems for developmental neurobehavioral toxicology

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