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First published online 5 March 2008
doi: 10.1242/dev.015867

This Article Figures Only Full Text Full Text (PDF) All Versions of this Article: dev.015867v1 135/8/1547    most recent Alert me when this article is cited Alert me if a correction is posted Services Email this article to a friend Related articles in Development Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Download to citation manager Citing Articles Citing Articles via HighWire Google Scholar Articles by Tessier, C. R. Articles by Broadie, K. PubMed PubMed Citation Articles by Tessier, C. R. Articles by Broadie, K.

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

Department of Biological Sciences, Kennedy Center for Research on Human Development, Vanderbilt University, Nashville, TN 37232, USA.

^* Author for correspondence (e-mail: kendal.broadie{at}vanderbilt.edu)

Accepted 4 February 2008

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.

Key words: Fragile X Syndrome, Mental retardation, Autism, Neural development, Translation, Synaptogenesis, Synaptic pruning

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				C. R. Tessier and K. Broadie Drosophila fragile x mental retardation protein developmentally regulates activity-dependent axon pruning J. Cell Sci., April 15, 2008; 121(8): e807 - e807. [Full Text]