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

被引:190
作者
Morales, J
Hiesinger, PR
Schroeder, AJ
Kume, K
Verstreken, P
Jackson, FR
Nelson, DL
Hassan, BA [1 ]
机构
[1] Baylor Coll Med, Dept Mol & Human Genet, Houston, TX 77030 USA
[2] Baylor Coll Med, Program Dev Biol, Houston, TX 77030 USA
[3] Tufts Univ, Sch Med, Dept Neurosci, Boston, MA 02111 USA
[4] Flanders Interuniv Inst Biotechnol VIB, Neurogenet Lab, Ctr Human Genet, B-3000 Louvain, Belgium
关键词
D O I
10.1016/S0896-6273(02)00731-6
中图分类号
Q189 [神经科学];
学科分类号
071006 ;
摘要
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.
引用
收藏
页码:961 / 972
页数:12
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