Artigo Acesso aberto Revisado por pares

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

2002; Cell Press; Volume: 34; Issue: 6 Linguagem: Inglês

10.1016/s0896-6273(02)00731-6

ISSN

1097-4199

Autores

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

Tópico(s)

Epigenetics and DNA Methylation

Resumo

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.

Referência(s)
Altmetric
PlumX