Drosophila fragile x-related gene regulates the MAP1B homolog Futsch to control synaptic structure and function

Yong Q. Zhang, Adina M. Bailey, Heinrich J G Matthies, Robert B. Renden, Mark A. Smith, Sean Speese, Gerald M. Rubin, Kendal Broadie

Research output: Contribution to journalArticle

475 Citations (Scopus)

Abstract

Fragile X mental retardation gene (FMR1) encodes an RNA binding protein that acts as a negative translational regulator. We have developed a Drosophila fragile X syndrome model using loss-of-function mutants and overexpression of the FMR1 homolog (dfxr). dfxr nulls display enlarged synaptic terminals, whereas neuronal overexpression results in fewer and larger synaptic boutons. Synaptic structural defects are accompanied by altered neurotransmission, with synapse type-specific regulation in central and peripheral synapses. These phenotypes mimic those observed in mutants of microtubule-associated Futsch. Immunoprecipitation of dFXR shows association with futsch mRNA, and Western analyses demonstrate that dFXR inversely regulates Futsch expression. dfxr futsch double mutants restore normal synaptic structure and function. We propose that dFXR acts as a translational repressor of Futsch to regulate microtubule-dependent synaptic growth and function.

Original languageEnglish (US)
Pages (from-to)591-603
Number of pages13
JournalCell
Volume107
Issue number5
DOIs
StatePublished - Nov 30 2001
Externally publishedYes

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Presynaptic Terminals
Microtubules
Synapses
Drosophila
Genes
Fragile X Syndrome
RNA-Binding Proteins
Immunoprecipitation
Synaptic Transmission
Intellectual Disability
Phenotype
Messenger RNA
Growth
Display devices
Defects
microtubule-associated protein 1B

ASJC Scopus subject areas

  • Cell Biology
  • Molecular Biology

Cite this

Zhang, Y. Q., Bailey, A. M., Matthies, H. J. G., Renden, R. B., Smith, M. A., Speese, S., ... Broadie, K. (2001). Drosophila fragile x-related gene regulates the MAP1B homolog Futsch to control synaptic structure and function. Cell, 107(5), 591-603. https://doi.org/10.1016/S0092-8674(01)00589-X

Drosophila fragile x-related gene regulates the MAP1B homolog Futsch to control synaptic structure and function. / Zhang, Yong Q.; Bailey, Adina M.; Matthies, Heinrich J G; Renden, Robert B.; Smith, Mark A.; Speese, Sean; Rubin, Gerald M.; Broadie, Kendal.

In: Cell, Vol. 107, No. 5, 30.11.2001, p. 591-603.

Research output: Contribution to journalArticle

Zhang, YQ, Bailey, AM, Matthies, HJG, Renden, RB, Smith, MA, Speese, S, Rubin, GM & Broadie, K 2001, 'Drosophila fragile x-related gene regulates the MAP1B homolog Futsch to control synaptic structure and function', Cell, vol. 107, no. 5, pp. 591-603. https://doi.org/10.1016/S0092-8674(01)00589-X
Zhang, Yong Q. ; Bailey, Adina M. ; Matthies, Heinrich J G ; Renden, Robert B. ; Smith, Mark A. ; Speese, Sean ; Rubin, Gerald M. ; Broadie, Kendal. / Drosophila fragile x-related gene regulates the MAP1B homolog Futsch to control synaptic structure and function. In: Cell. 2001 ; Vol. 107, No. 5. pp. 591-603.
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AU - Rubin, Gerald M.

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