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Behavior in a Drosophila Model of Fragile X

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Modeling Fragile X Syndrome

Part of the book series: Results and Problems in Cell Differentiation ((RESULTS,volume 54))

Abstract

This chapter will briefly tie together a captivating string of scientific discoveries that began in the 1800s and catapulted us into the current state of the field where trials are under way in humans that have arisen directly from the discoveries made in model organisms such as Drosophila (fruit flies) and mice. The hope is that research efforts in the field of fragile X currently represent a roadmap that demonstrates the utility of identifying a mutant gene responsible for human disease, tracking down the molecular underpinnings of pathogenic phenotypes, and utilizing model organisms to identify and validate potential pharmacologic targets for testing in afflicted humans. Indeed, in fragile X this roadmap has already yielded successful trials in humans (J. Med. Genetic 46(4) 266–271; Jacquemont et al. Sci Transl Med 3(64):64ra61), although the work in studying these interventions in humans is just getting underway as the work in model organisms continues to generate new potential therapeutic targets.

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Authors and Affiliations

  1. Department of Psychiatry, University of Pennsylvania School of Medicine, Philadelphia, PA, USA

    Sean M. McBride

  2. Department of Genetics, University of Pennsylvania School of Medicine, Philadelphia, PA, USA

    Aaron J. Bell & Thomas A. Jongens

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  1. Sean M. McBride
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  2. Aaron J. Bell
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  3. Thomas A. Jongens
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Correspondence to Sean M. McBride .

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  1. New York State Institute for Basic Resea, Forest Hill Road 1050, Staten Island, 10314, USA

    Robert B. Denman

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McBride, S.M., Bell, A.J., Jongens, T.A. (2012). Behavior in a Drosophila Model of Fragile X. In: Denman, R. (eds) Modeling Fragile X Syndrome. Results and Problems in Cell Differentiation, vol 54. Springer, Berlin, Heidelberg. https://doi.org/10.1007/978-3-642-21649-7_6

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