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Drosophila Fragile X Protein, DFXR, Regulates Neuronal Morphology and Function in the Brain

Authors :
Andrew J. Schroeder
Kazuhiko Kume
Joannella Morales
Bassem A. Hassan
David L. Nelson
Patrik Verstreken
P. Robin Hiesinger
F. Rob Jackson
Source :
Neuron. 34(6):961-972
Publication Year :
2002
Publisher :
Elsevier BV, 2002.

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.

Details

ISSN :
08966273
Volume :
34
Issue :
6
Database :
OpenAIRE
Journal :
Neuron
Accession number :
edsair.doi.dedup.....238ff6e1ffe7f1fac1a22b24a5b9fa66
Full Text :
https://doi.org/10.1016/s0896-6273(02)00731-6