Literature DB >> 12086643

Drosophila fragile X protein, DFXR, regulates neuronal morphology and function in the brain.

Joannella Morales1, P Robin Hiesinger, Andrew J Schroeder, Kazuhiko Kume, Patrik Verstreken, F Rob Jackson, David L Nelson, Bassem A Hassan.   

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.

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Year:  2002        PMID: 12086643     DOI: 10.1016/s0896-6273(02)00731-6

Source DB:  PubMed          Journal:  Neuron        ISSN: 0896-6273            Impact factor:   17.173


  105 in total

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