Reference Report

Reference
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. (Export to RIS)
FlyBase ID	FBrf0141416
Publication Type	Research paper
PubMed ID	11733059
PubMed 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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Language of Publication	English
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Publication Type	Journal
Abbreviation	Cell
Title	Cell
Publication Year	1974-
ISBN/ISSN	0092-8674
Data from Reference
Aberrations (1)
	Df(3R)by62
Alleles (15)
	Fmr1^9N Fmr1^EP3422 Fmr1^EP3517 Fmr1^{I307N.Z.Scer\UAS} Fmr1^Scer\UAS.cZa Fmr1^Δ50M Fmr1^Δ83M Fmr1^Δ113M Fmr1^Δ192N futsch^EP1419 futsch^N94 Scer\GAL4^elav-C155 Scer\GAL4^G7 Scer\GAL4^hs.2sev Scer\GAL4^Mhc.PW
Constructs (3)
	P{GAL4-Mhc.W} P{UAS-Fmr1.I307N.Z} P{UAS-Fmr1.Z}
Genes (6)
	Csp dlg1 Fmr1 futsch Scer\GAL4 Syt1
Insertions (5)
	P{EP}Fmr1^EP3422 P{EP}Fmr1^EP3517 P{EP}futsch^EP1419 P{GAL4}G7 P{GawB}elav^C155