@article{36a9424662a3415dab70629701de5ce9,
title = "Drosophila fragile x-related gene regulates the MAP1B homolog Futsch to control synaptic structure and function",
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.",
author = "Zhang, {Yong Q.} and Bailey, {Adina M.} and Matthies, {Heinrich J.G.} and Renden, {Robert B.} and Smith, {Mark A.} and Speese, {Sean D.} and Rubin, {Gerald M.} and Kendal Broadie",
note = "Funding Information: We are indebted to the Berkeley Drosophila Genome Project for EP lines and clones. We are particular grateful to G. Dreyfuss for his gift of dFXR antibody. We thank G. Davis for futsch alleles; K. Zinsmaier, T. Littleton, D. Woods, and R. Jackson for anti-CSP, anti-syt, anti-DLG, and anti-dGAD1, respectively; B. Dickson for sev-GAL4; M. Bastiani for access to confocal imaging; S. Mullaney for germline transformation services; and J. Rehm for inverse PCR data. M. Evans, C. Suh, G. Tsang, and A. Wong participated in DNA sequencing. Y.Q.Z. was supported by a fellowship from the FRAXA Research Foundation. A.M.B. was a fellow of the Helen Hay Whitney Foundation. R.B.R. was supported by a National Institutes of Health training grant ST32 HD07491. G.M.R. is an Investigator of the Howard Hughes Medical Institute. K.B. is supported by an EJLB Scholarship and by National Institutes of Health grant HD40654. ",
year = "2001",
month = nov,
day = "30",
doi = "10.1016/S0092-8674(01)00589-X",
language = "English (US)",
volume = "107",
pages = "591--603",
journal = "Cell",
issn = "0092-8674",
publisher = "Cell Press",
number = "5",
}