@article{ea0aad36b8ff401d8b0de4e1ad85a208,
title = "Drosophila fragile X protein DFXR regulates neuronal morphology and function in the brain",
abstract = "Mental retardation is a pervasive societal problem 25 times more common than blindness for example. Fragile X syndrome the most common form of inherited mental retardation is caused by mutations in the FMR1 gene. Fragile X patients display neurite morphology defects in the brain suggesting that this may be the basis of their mental retardation. Drosophila contains a single homolog of FMR1 dfxr (also called dfmr1). We analyzed the role of dfxr in neurite development in three distinct neuronal classes. We find that DFXR is required for normal neurite extension guidance and branching. dfxr mutants also display strong eclosion failure and circadian rhythm defects. Interestingly distinct neuronal cell types show different phenotypes suggesting that dfxr differentially regulates diverse targets in the brain.",
author = "Joannella Morales and Hiesinger, {P. Robin} and Schroeder, {Andrew J.} and Kazuhiko Kume and Patrik Verstreken and Jackson, {F. Rob} and Nelson, {David L.} and Hassan, {Bassem A.}",
note = "Funding Information: We thank Mary Roberts for invaluable technical assistance in behavioral studies, Michael Greenbaum for help with ERG studies, and Joel Levine and Jeff Hall for help with determining the “strength” of locomotor activity rhythms. We thank Adina Bailey and Gerald Rubin for providing dfxr Drosophila lines that made this study possible, as well as Gideon Dreyfuss for providing α-DFXR antibody (mAb6A15) and Isaac Edery for α-PER antibodies. We also thank Hugo Bellen, Bart De Strooper, and Wim Annaert for comments on the manuscript. Funding from NIH R01 grants HL59873 (to F.R.J.) and HD29256 (to D.L.N.) contributed to this work. B.H. was funded by an NRSA postdoctoral fellowship during the initial parts of this project, and by the VIB for the later parts.",
year = "2002",
month = jun,
day = "13",
doi = "10.1016/S0896-6273(02)00731-6",
language = "English (US)",
volume = "34",
pages = "961--972",
journal = "Neuron",
issn = "0896-6273",
publisher = "Cell Press",
number = "6",
}