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Drosophila fragile X mental retardation protein developmentally regulates activity-dependent axon pruning.
Drosophila fragile X mental retardation protein developmentally regulates activity-dependent axon pruning.
- 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.
- Subjects :
- Animals
Animals, Genetically Modified
Axons ultrastructure
Base Sequence
Brain growth & development
Brain metabolism
Brain ultrastructure
DNA Primers genetics
Drosophila genetics
Drosophila ultrastructure
Drosophila Proteins genetics
Fragile X Mental Retardation Protein genetics
Gene Expression Regulation, Developmental
Genes, Insect
Humans
Nerve Net growth & development
Nerve Net metabolism
RNA genetics
RNA metabolism
Axons metabolism
Drosophila growth & development
Drosophila metabolism
Drosophila Proteins metabolism
Fragile X Mental Retardation Protein metabolism
Subjects
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