Zebrafish as a new animal model for movement disorders

Flinn, Laura; Bretaud, Sandrine; Lo, Christine; Ingham, Philip W.; Bandmann, Oliver

doi:10.1111/j.1471-4159.2008.05463.x

Cited by 125 publications

(76 citation statements)

References 40 publications

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“…T is generally recognized that studies on genetically tractable model organisms can provide information that is relevant for human disorders (Bilen and Bonini 2005;Mackay and Anholt 2006;Crabbe 2008;Flinn et al 2008;Woodruff-Pak 2008). Nonetheless, attempts to identify risk alleles for human diseases have relied almost exclusively on linkage studies in human families or association analyses in human populations without reliance on translational approaches based on model systems.…”

mentioning

confidence: 99%

Alcohol Sensitivity in Drosophila: Translational Potential of Systems Genetics

et al. 2009

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Identification of risk alleles for human behavioral disorders through genomewide association studies (GWAS) has been hampered by a daunting multiple testing problem. This problem can be circumvented for some phenotypes by combining genomewide studies in model organisms with subsequent candidate gene association analyses in human populations. Here, we characterized genetic networks that underlie the response to ethanol exposure in Drosophila melanogaster by measuring ethanol knockdown time in 40 wildderived inbred Drosophila lines. We associated phenotypic variation in ethanol responses with genomewide variation in gene expression and identified modules of correlated transcripts associated with a first and second exposure to ethanol vapors as well as the induction of tolerance. We validated the computational networks and assessed their robustness by transposon-mediated disruption of focal genes within modules in a laboratory inbred strain, followed by measurements of transcript abundance of connected genes within the module. Many genes within the modules have human orthologs, which provides a stepping stone for the identification of candidate genes associated with alcohol drinking behavior in human populations. We demonstrated the potential of this translational approach by identifying seven intronic single nucleotide polymorphisms of the Malic Enzyme 1 (ME1) gene that are associated with cocktail drinking in 1687 individuals of the Framingham Offspring cohort, implicating that variation in levels of cytoplasmic malic enzyme may contribute to variation in alcohol consumption.

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mentioning

confidence: 99%

Alcohol Sensitivity in Drosophila: Translational Potential of Systems Genetics

et al. 2009

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“…And zebrafish physiology is very similar to ours (Bakkers, 2011;Gestri et al, 2012). Therefore, by using zebrafish we can comprehend almost every human disease (Chan and Mably, 2011;Chico et al, 2008;Flinn et al, 2008;Meijer and Spaink, 2011;Rocke et al, 2009;Terriente and Pujades, 2013). To this end, the genetic tools generated for zebrafish developmental biology research are helping to build zebrafish human disease models.…”

Section: Switching To Zebrafish Obsessive-compulsive Disorder Modelsmentioning

confidence: 95%

Switching to zebrafish neurobehavioral models: The obsessive–compulsive disorder paradigm

D’Amico

Estivill

Terriente

2015

European Journal of Pharmacology

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“…Several zebrafish embryo assays can help to predict drug safety in humans (Berghmans et al, 2008;Redfern et al, 2008), and therefore zebrafish disease models have been developed (Flinn et al, 2008;Tanguay and Reimers, 2008;Brittijn et al, 2009).…”

Section: The Zebrafish Embryo In Drug Screeningmentioning

confidence: 99%

Zebrafish embryos and larvae: A new generation of disease models and drug screens

Ali

Champagne

Spaink

et al. 2011

Birth Defects Research Part C: Embryo Today: Reviews

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135

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Technological innovation has helped the zebrafish embryo gain ground as a disease model and an assay system for drug screening. Here, we review the use of zebrafish embryos and early larvae in applied biomedical research, using selected cases. We look at the use of zebrafish embryos as disease models, taking fetal alcohol syndrome and tuberculosis as examples. We discuss advances in imaging, in culture techniques (including microfluidics), and in drug delivery (including new techniques for the robotic injection of compounds into the egg). The use of zebrafish embryos in early stages of drug safety-screening is discussed. So too are the new behavioral assays that are being adapted from rodent research for use in zebrafish embryos, and which may become relevant in validating the effects of neuroactive compounds such as anxiolytics and antidepressants. Readouts, such as morphological screening and cardiac function, are examined. There are several drawbacks in the zebrafish model. One is its very rapid development, which means that screening with zebrafish is analogous to "screening on a run-away train." Therefore, we argue that zebrafish embryos need to be precisely staged when used in acute assays, so as to ensure a consistent window of developmental exposure. We believe that zebrafish embryo screens can be used in the pre-regulatory phases of drug development, although more validation studies are needed to overcome industry scepticism. Finally, the zebrafish poses no challenge to the position of rodent models: it is complementary to them, especially in early stages of drug research.

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Zebrafish as a new animal model for movement disorders

Cited by 125 publications

References 40 publications

Alcohol Sensitivity in Drosophila: Translational Potential of Systems Genetics

Alcohol Sensitivity in Drosophila: Translational Potential of Systems Genetics

Switching to zebrafish neurobehavioral models: The obsessive–compulsive disorder paradigm

Zebrafish embryos and larvae: A new generation of disease models and drug screens

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