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Citation
Bhogal, B., Jepson, J.E., Savva, Y.A., Pepper, A.S., Reenan, R.A., Jongens, T.A. (2011). Modulation of dADAR-dependent RNA editing by the Drosophila fragile X mental retardation protein.  Nat. Neurosci. 14(12): 1517--1524.
FlyBase ID
FBrf0216747
Publication Type
Research paper
Abstract

Loss of FMR1 gene function results in fragile X syndrome, the most common heritable form of intellectual disability. The protein encoded by this locus (FMRP) is an RNA-binding protein that is thought to primarily act as a translational regulator; however, recent studies have implicated FMRP in other mechanisms of gene regulation. We found that the Drosophila fragile X homolog (dFMR1) biochemically interacted with the adenosine-to-inosine RNA-editing enzyme dADAR. Adar and Fmr1 mutant larvae exhibited distinct morphological neuromuscular junction (NMJ) defects. Epistasis experiments based on these phenotypic differences revealed that Adar acts downstream of Fmr1 and that dFMR1 modulates dADAR activity. Furthermore, sequence analyses revealed that a loss or overexpression of dFMR1 affects editing efficiency on certain dADAR targets with defined roles in synaptic transmission. These results link dFMR1 with the RNA-editing pathway and suggest that proper NMJ synaptic architecture requires modulation of dADAR activity by dFMR1.

PubMed ID
PubMed Central ID
PMC3225737 (PMC) (EuropePMC)
Related Publication(s)
Note

Fragile balance: RNA editing tunes the synapse.
Bassell, 2011, Nat. Neurosci. 14(12): 1492--1494 [FBrf0217268]

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    Language of Publication
    English
    Additional Languages of Abstract
    Parent Publication
    Publication Type
    Journal
    Abbreviation
    Nat. Neurosci.
    Title
    Nature Neuroscience
    Publication Year
    1998-
    ISBN/ISSN
    1097-6256
    Data From Reference
    Alleles (14)
    Genes (9)
    Human Disease Models (1)
    Physical Interactions (3)
    Cell Lines (1)
    Natural transposons (1)
    Insertions (3)
    Experimental Tools (1)
    Transgenic Constructs (8)