Back to Search Start Over

Drosophila fragile X mental retardation protein developmentally regulates activity-dependent axon pruning.

Drosophila fragile X mental retardation protein developmentally regulates activity-dependent axon pruning.

Authors :
Tessier CR
Broadie K
Source :
Development (Cambridge, England) [Development] 2008 Apr; Vol. 135 (8), pp. 1547-57. Date of Electronic Publication: 2008 Mar 05.
Publication Year :
2008

Abstract

Fragile X Syndrome (FraX) is a broad-spectrum neurological disorder with symptoms ranging from hyperexcitability to mental retardation and autism. Loss of the fragile X mental retardation 1 (fmr1) gene product, the mRNA-binding translational regulator FMRP, causes structural over-elaboration of dendritic and axonal processes, as well as functional alterations in synaptic plasticity at maturity. It is unclear, however, whether FraX is primarily a disease of development, a disease of plasticity or both: a distinction that is vital for engineering intervention strategies. To address this crucial issue, we have used the Drosophila FraX model to investigate the developmental function of Drosophila FMRP (dFMRP). dFMRP expression and regulation of chickadee/profilin coincides with a transient window of late brain development. During this time, dFMRP is positively regulated by sensory input activity, and is required to limit axon growth and for efficient activity-dependent pruning of axon branches in the Mushroom Body learning/memory center. These results demonstrate that dFMRP has a primary role in activity-dependent neural circuit refinement during late brain development.

Details

Language :
English
ISSN :
0950-1991
Volume :
135
Issue :
8
Database :
MEDLINE
Journal :
Development (Cambridge, England)
Publication Type :
Academic Journal
Accession number :
18321984
Full Text :
https://doi.org/10.1242/dev.015867