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Citation
Zhang, Y.Q., Bailey, A.M., Matthies, H.J.G., Renden, R.B., Smith, M.A., Speese, S.D., Rubin, G.M., Broadie, K. (2001). Drosophila fragile X-related gene regulates the MAP1B homolog Futsch to control synaptic structure and function.  Cell 107(5): 591--603.
FlyBase ID
FBrf0141416
Publication Type
Research paper
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.

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Note

RNA targets of the fragile X protein.
Kaytor and Orr, 2001, Cell 107(5): 555--557 [FBrf0141414]

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Secondary IDs
    Language of Publication
    English
    Additional Languages of Abstract
    Parent Publication
    Publication Type
    Journal
    Abbreviation
    Cell
    Title
    Cell
    Publication Year
    1974-
    ISBN/ISSN
    0092-8674
    Data From Reference
    Aberrations (1)
    Alleles (15)
    Genes (6)
    Human Disease Models (1)
    Physical Interactions (2)
    Insertions (5)
    Experimental Tools (1)
    Transgenic Constructs (3)