Behavior in a Drosophila model of fragile X

Sean M. McBride, Aaron J. Bell, Thomas A. Jongens

Research output: Chapter in Book/Report/Conference proceedingChapter

19 Scopus citations


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.

Original languageEnglish (US)
Title of host publicationModeling Fragile X Syndrome
EditorsRobert Denman
Number of pages35
StatePublished - 2012
Externally publishedYes

Publication series

NameResults and Problems in Cell Differentiation
ISSN (Print)0080-1844
ISSN (Electronic)1861-0412

All Science Journal Classification (ASJC) codes

  • Developmental Biology
  • Cell Biology


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