Come FLY with us: toward understanding fragile X syndrome

Genes Brain Behav. 2005 Aug;4(6):385-92. doi: 10.1111/j.1601-183X.2005.00136.x.


The past few years have seen an increased number of articles using Drosophila as a model system to study fragile X syndrome. Phenotypic analyses have demonstrated an array of neuronal and behavioral defects similar to the phenotypes reported in mouse models as well as human patients. The availability of both cellular and molecular tools along with the power of genetics makes the tiny fruit fly a premiere model in elucidating the molecular basis of fragile X syndrome. Here, we summarize the advances made in recent years in the characterization of fragile X Drosophila models and the identification of new molecular partners in neural development.

Publication types

  • Review

MeSH terms

  • Animals
  • Disease Models, Animal
  • Drosophila Proteins / genetics*
  • Drosophila Proteins / metabolism
  • Drosophila melanogaster / embryology
  • Drosophila melanogaster / genetics*
  • Drosophila melanogaster / metabolism
  • Fragile X Mental Retardation Protein
  • Fragile X Syndrome / genetics*
  • Fragile X Syndrome / metabolism
  • Fragile X Syndrome / physiopathology
  • Gametogenesis / genetics
  • Gonads / cytology
  • Gonads / embryology
  • Gonads / metabolism
  • Humans
  • MicroRNAs / metabolism
  • Nervous System / embryology*
  • Nervous System / metabolism
  • RNA, Messenger / metabolism
  • RNA-Binding Proteins / genetics*
  • RNA-Binding Proteins / metabolism
  • Synaptic Transmission / genetics*


  • Drosophila Proteins
  • FMR1 protein, Drosophila
  • MicroRNAs
  • RNA, Messenger
  • RNA-Binding Proteins
  • Fragile X Mental Retardation Protein