The Drosophila fragile X gene negatively regulates neuronal elaboration and synaptic differentiation.

PubWeight™: 1.60‹?› | Rank: Top 4%

🔗 View Article (PMID 15498496)

Published in Curr Biol on October 26, 2004

Authors

Luyuan Pan1, Yong Q Zhang, Elvin Woodruff, Kendal Broadie

Author Affiliations

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

Articles citing this

Fragile X syndrome: loss of local mRNA regulation alters synaptic development and function. Neuron (2008) 6.26

Imbalance of neocortical excitation and inhibition and altered UP states reflect network hyperexcitability in the mouse model of fragile X syndrome. J Neurophysiol (2008) 2.46

The state of synapses in fragile X syndrome. Neuroscientist (2009) 2.07

Sleep and synaptic homeostasis: structural evidence in Drosophila. Science (2011) 2.07

Modeling spinal muscular atrophy in Drosophila. PLoS One (2008) 1.71

Drosophila fragile X mental retardation protein developmentally regulates activity-dependent axon pruning. Development (2008) 1.52

Drosophila melanogaster in the study of human neurodegeneration. CNS Neurol Disord Drug Targets (2010) 1.33

The Drosophila fragile X mental retardation gene regulates sleep need. J Neurosci (2009) 1.25

Nerve growth factor-induced formation of axonal filopodia and collateral branches involves the intra-axonal synthesis of regulators of the actin-nucleating Arp2/3 complex. J Neurosci (2012) 1.24

A dynamic deep sleep stage in Drosophila. J Neurosci (2013) 1.24

Temporal requirements of the fragile X mental retardation protein in the regulation of synaptic structure. Development (2008) 1.21

Genetic controls balancing excitatory and inhibitory synaptogenesis in neurodevelopmental disorder models. Front Synaptic Neurosci (2010) 1.18

Mechanistic relationships between Drosophila fragile X mental retardation protein and metabotropic glutamate receptor A signaling. Mol Cell Neurosci (2008) 1.15

Understanding neuronal connectivity through the post-transcriptional toolkit. Genes Dev (2010) 1.06

Independent role for presynaptic FMRP revealed by an FMR1 missense mutation associated with intellectual disability and seizures. Proc Natl Acad Sci U S A (2015) 1.06

Fragile X mental retardation protein has a unique, evolutionarily conserved neuronal function not shared with FXR1P or FXR2P. Dis Model Mech (2010) 1.02

Understanding the neurogenetics of sleep: progress from Drosophila. Trends Genet (2009) 1.02

Fragile X mental retardation protein regulates new neuron differentiation in the adult olfactory bulb. J Neurosci (2011) 1.00

The fragile X mental retardation protein in circadian rhythmicity and memory consolidation. Mol Neurobiol (2009) 0.99

Presynaptic translation: stepping out of the postsynaptic shadow. Front Neural Circuits (2009) 0.98

In vivo neuronal function of the fragile X mental retardation protein is regulated by phosphorylation. Hum Mol Genet (2011) 0.96

The Drosophila FMRP and LARK RNA-binding proteins function together to regulate eye development and circadian behavior. J Neurosci (2008) 0.95

RNA-binding proteins and translational regulation in axons and growth cones. Front Neurosci (2013) 0.95

Fragile X syndrome: a preclinical review on metabotropic glutamate receptor 5 (mGluR5) antagonists and drug development. Psychopharmacology (Berl) (2014) 0.95

From genetics to structure to function: exploring sleep in Drosophila. Int Rev Neurobiol (2011) 0.95

Limbic epileptogenesis in a mouse model of fragile X syndrome. Cereb Cortex (2008) 0.94

Matrix metalloproteinases and minocycline: therapeutic avenues for fragile X syndrome. Neural Plast (2012) 0.94

Neural circuit architecture defects in a Drosophila model of Fragile X syndrome are alleviated by minocycline treatment and genetic removal of matrix metalloproteinase. Dis Model Mech (2011) 0.94

Proteasome function is required to maintain muscle cellular architecture. Biol Cell (2007) 0.94

Activity-dependent modulation of neural circuit synaptic connectivity. Front Mol Neurosci (2009) 0.93

Bicaudal-D regulates fragile X mental retardation protein levels, motility, and function during neuronal morphogenesis. Curr Biol (2010) 0.93

The nonsense-mediated decay pathway maintains synapse architecture and synaptic vesicle cycle efficacy. J Cell Sci (2010) 0.92

Fruit flies and intellectual disability. Fly (Austin) (2009) 0.92

iPSC-derived forebrain neurons from FXS individuals show defects in initial neurite outgrowth. Stem Cells Dev (2014) 0.92

Argonaute2 suppresses Drosophila fragile X expression preventing neurogenesis and oogenesis defects. PLoS One (2009) 0.91

Metabotropic glutamate receptor-mediated use-dependent down-regulation of synaptic excitability involves the fragile X mental retardation protein. J Neurophysiol (2008) 0.90

The fragile X mental retardation protein developmentally regulates the strength and fidelity of calcium signaling in Drosophila mushroom body neurons. Neurobiol Dis (2010) 0.89

The development of cortical columns: role of Fragile X mental retardation protein. J Physiol (2009) 0.89

hnRNP Q regulates Cdc42-mediated neuronal morphogenesis. Mol Cell Biol (2012) 0.89

Rolling blackout is required for bulk endocytosis in non-neuronal cells and neuronal synapses. J Cell Sci (2008) 0.89

Temporal requirements of the fragile x mental retardation protein in modulating circadian clock circuit synaptic architecture. Front Neural Circuits (2009) 0.89

Receptor tyrosine phosphatases regulate birth order-dependent axonal fasciculation and midline repulsion during development of the Drosophila mushroom body. Mol Cell Neurosci (2008) 0.87

Identifying intrinsic and extrinsic determinants that regulate internal initiation of translation mediated by the FMR1 5' leader. BMC Mol Biol (2008) 0.87

Translational regulation of neuronal electrical properties. Invert Neurosci (2007) 0.86

GABAergic circuit dysfunction in the Drosophila Fragile X syndrome model. Neurobiol Dis (2014) 0.85

A non-canonical start codon in the Drosophila fragile X gene yields two functional isoforms. Neuroscience (2011) 0.85

Substitution of critical isoleucines in the KH domains of Drosophila fragile X protein results in partial loss-of-function phenotypes. Genetics (2006) 0.85

Learning and behavioral deficits associated with the absence of the fragile X mental retardation protein: what a fly and mouse model can teach us. Learn Mem (2014) 0.85

Fragile X mental retardation protein regulates trans-synaptic signaling in Drosophila. Dis Model Mech (2013) 0.84

Elevated levels of the vesicular monoamine transporter and a novel repetitive behavior in the Drosophila model of fragile X syndrome. PLoS One (2011) 0.83

The cyclic AMP phenotype of fragile X and autism. Neurosci Biobehav Rev (2008) 0.83

A novel fragile X syndrome mutation reveals a conserved role for the carboxy-terminus in FMRP localization and function. EMBO Mol Med (2015) 0.83

Molecular and genetic analysis of the Drosophila model of fragile X syndrome. Results Probl Cell Differ (2012) 0.83

Activity-dependent FMRP requirements in development of the neural circuitry of learning and memory. Development (2015) 0.83

A Drosophila model of Fragile X syndrome exhibits defects in phagocytosis by innate immune cells. J Cell Biol (2017) 0.82

Altered LARK expression perturbs development and physiology of the Drosophila PDF clock neurons. Mol Cell Neurosci (2009) 0.82

Increasing our understanding of human cognition through the study of Fragile X Syndrome. Dev Neurobiol (2013) 0.82

An assay for social interaction in Drosophila fragile X mutants. Fly (Austin) (2010) 0.81

Fragile X mental retardation protein: A paradigm for translational control by RNA-binding proteins. Biochimie (2015) 0.81

Two peptide transmitters co-packaged in a single neurosecretory vesicle. Peptides (2008) 0.81

Deletion of Fmr1 alters function and synaptic inputs in the auditory brainstem. PLoS One (2015) 0.80

The Drosophila gene RanBPM functions in the mushroom body to regulate larval behavior. PLoS One (2010) 0.80

Neuron class-specific requirements for Fragile X Mental Retardation Protein in critical period development of calcium signaling in learning and memory circuitry. Neurobiol Dis (2016) 0.80

Life is sweet! A novel role for N-glycans in Drosophila lifespan. Fly (Austin) (2011) 0.79

Synergistic interactions between Drosophila orthologues of genes spanned by de novo human CNVs support multiple-hit models of autism. PLoS Genet (2015) 0.78

Inability to activate Rac1-dependent forgetting contributes to behavioral inflexibility in mutants of multiple autism-risk genes. Proc Natl Acad Sci U S A (2016) 0.78

Hyperactive locomotion in a Drosophila model is a functional readout for the synaptic abnormalities underlying fragile X syndrome. Sci Signal (2017) 0.76

Fmrp Interacts with Adar and Regulates RNA Editing, Synaptic Density and Locomotor Activity in Zebrafish. PLoS Genet (2015) 0.76

It takes a village to raise a branch: Cellular mechanisms of the initiation of axon collateral branches. Mol Cell Neurosci (2017) 0.75

Astrocytes and developmental plasticity in fragile X. Neural Plast (2012) 0.75

Splice form-dependent regulation of axonal arbor complexity by FMRP. Dev Neurobiol (2016) 0.75

Conserved properties of Drosophila insomniac link sleep regulation and synaptic function. PLoS Genet (2017) 0.75

Using Drosophila as a tool to identify Pharmacological Therapies for Fragile X Syndrome. Drug Discov Today Technol (2012) 0.75

New X-chromosomal interactors of dFMRP regulate axonal and synaptic morphology of brain neurons in Drosophila melanogaster. Biotechnol Biotechnol Equip (2014) 0.75

ESCRT-III Membrane Trafficking Misregulation Contributes To Fragile X Syndrome Synaptic Defects. Sci Rep (2017) 0.75

Articles by these authors

AP-3 directs the intracellular trafficking of HIV-1 Gag and plays a key role in particle assembly. Cell (2005) 2.68

The hereditary spastic paraplegia gene, spastin, regulates microtubule stability to modulate synaptic structure and function. Curr Biol (2004) 2.06

Living synaptic vesicle marker: synaptotagmin-GFP. Genesis (2002) 1.99

The ubiquitin proteasome system acutely regulates presynaptic protein turnover and synaptic efficacy. Curr Biol (2003) 1.91

Spartin regulates synaptic growth and neuronal survival by inhibiting BMP-mediated microtubule stabilization. Neuron (2013) 1.83

CNS-derived glia ensheath peripheral nerves and mediate motor root development. Nat Neurosci (2008) 1.73

Motor deficit in a Drosophila model of mucolipidosis type IV due to defective clearance of apoptotic cells. Cell (2008) 1.69

Wrestling with pleiotropy: genomic and topological analysis of the yeast gene expression network. Bioessays (2002) 1.58

Drosophila fragile X mental retardation protein developmentally regulates activity-dependent axon pruning. Development (2008) 1.52

An essential Drosophila glutamate receptor subunit that functions in both central neuropil and neuromuscular junction. J Neurosci (2005) 1.36

Electrophysiological analysis of synaptic transmission in central neurons of Drosophila larvae. J Neurophysiol (2002) 1.33

Developmental regulation of glutamate receptor field size by nonvesicular glutamate release. Nat Neurosci (2002) 1.26

Ceramidase regulates synaptic vesicle exocytosis and trafficking. J Neurosci (2004) 1.24

Lipid regulation of the synaptic vesicle cycle. Nat Rev Neurosci (2005) 1.22

Temporal requirements of the fragile X mental retardation protein in the regulation of synaptic structure. Development (2008) 1.21

Roles of ubiquitination at the synapse. Biochim Biophys Acta (2008) 1.19

Genetic controls balancing excitatory and inhibitory synaptogenesis in neurodevelopmental disorder models. Front Synaptic Neurosci (2010) 1.18

Mechanistic relationships between Drosophila fragile X mental retardation protein and metabotropic glutamate receptor A signaling. Mol Cell Neurosci (2008) 1.15

Presynaptic establishment of the synaptic cleft extracellular matrix is required for post-synaptic differentiation. Genes Dev (2007) 1.07

Synaptic Drosophila UNC-13 is regulated by antagonistic G-protein pathways via a proteasome-dependent degradation mechanism. J Neurobiol (2003) 1.05

Neuronal loss of Drosophila NPC1a causes cholesterol aggregation and age-progressive neurodegeneration. J Neurosci (2008) 1.04

Rolling blackout, a newly identified PIP2-DAG pathway lipase required for Drosophila phototransduction. Nat Neurosci (2004) 1.04

Fragile X mental retardation protein has a unique, evolutionarily conserved neuronal function not shared with FXR1P or FXR2P. Dis Model Mech (2010) 1.02

Protein expression profiling of the drosophila fragile X mutant brain reveals up-regulation of monoamine synthesis. Mol Cell Proteomics (2005) 1.02

Cellular bases of activity-dependent paralysis in Drosophila stress-sensitive mutants. J Neurobiol (2004) 1.02

Cellular bases of behavioral plasticity: establishing and modifying synaptic circuits in the Drosophila genetic system. J Neurobiol (2003) 1.01

RIM promotes calcium channel accumulation at active zones of the Drosophila neuromuscular junction. J Neurosci (2012) 1.00

The Drosophila metabotropic glutamate receptor DmGluRA regulates activity-dependent synaptic facilitation and fine synaptic morphology. J Neurosci (2004) 1.00

The Cdc42-selective GAP rich regulates postsynaptic development and retrograde BMP transsynaptic signaling. J Cell Biol (2010) 1.00

Rolling blackout is required for synaptic vesicle exocytosis. J Neurosci (2006) 1.00

A targeted glycan-related gene screen reveals heparan sulfate proteoglycan sulfation regulates WNT and BMP trans-synaptic signaling. PLoS Genet (2012) 1.00

The fragile X mental retardation protein in circadian rhythmicity and memory consolidation. Mol Neurobiol (2009) 0.99

The Drosophila fragile X-related gene regulates axoneme differentiation during spermatogenesis. Dev Biol (2004) 0.98

Harvesting and preparing Drosophila embryos for electrophysiological recording and other procedures. J Vis Exp (2009) 0.97

Integrins regulate DLG/FAS2 via a CaM kinase II-dependent pathway to mediate synapse elaboration and stabilization during postembryonic development. Development (2002) 0.97

Presynaptic calcium channel localization and calcium-dependent synaptic vesicle exocytosis regulated by the Fuseless protein. J Neurosci (2008) 0.97

The ubiquitin-proteasome system postsynaptically regulates glutamatergic synaptic function. Mol Cell Neurosci (2007) 0.96

Fathoming fragile X in fruit flies. Trends Genet (2005) 0.96

Anterograde Jelly belly ligand to Alk receptor signaling at developing synapses is regulated by Mind the gap. Development (2010) 0.96

In vivo neuronal function of the fragile X mental retardation protein is regulated by phosphorylation. Hum Mol Genet (2011) 0.96

Glycosylated synaptomatrix regulation of trans-synaptic signaling. Dev Neurobiol (2012) 0.95

Matrix metalloproteinases and minocycline: therapeutic avenues for fragile X syndrome. Neural Plast (2012) 0.94

Distinct presynaptic and postsynaptic dismantling processes of Drosophila neuromuscular junctions during metamorphosis. J Neurosci (2010) 0.94

Neural circuit architecture defects in a Drosophila model of Fragile X syndrome are alleviated by minocycline treatment and genetic removal of matrix metalloproteinase. Dis Model Mech (2011) 0.94

Drosophila sec10 is required for hormone secretion but not general exocytosis or neurotransmission. Traffic (2002) 0.94

Proteasome function is required to maintain muscle cellular architecture. Biol Cell (2007) 0.94

Mutation and activation of Galpha s similarly alters pre- and postsynaptic mechanisms modulating neurotransmission. J Neurophysiol (2003) 0.93

Drosophila cyfip regulates synaptic development and endocytosis by suppressing filamentous actin assembly. PLoS Genet (2013) 0.93

Activity-dependent modulation of neural circuit synaptic connectivity. Front Mol Neurosci (2009) 0.93

Mutational analysis establishes a critical role for the N terminus of fragile X mental retardation protein FMRP. J Neurosci (2008) 0.93

miR-153 regulates SNAP-25, synaptic transmission, and neuronal development. PLoS One (2013) 0.92

Drosophila FMRP regulates microtubule network formation and axonal transport of mitochondria. Hum Mol Genet (2010) 0.92

HDAC6 mutations rescue human tau-induced microtubule defects in Drosophila. Proc Natl Acad Sci U S A (2013) 0.92

The nonsense-mediated decay pathway maintains synapse architecture and synaptic vesicle cycle efficacy. J Cell Sci (2010) 0.92

Drosophila Tubulin-specific chaperone E functions at neuromuscular synapses and is required for microtubule network formation. Development (2009) 0.91

Brain tumor regulates neuromuscular synapse growth and endocytosis in Drosophila by suppressing mad expression. J Neurosci (2013) 0.90

Metabotropic glutamate receptor-mediated use-dependent down-regulation of synaptic excitability involves the fragile X mental retardation protein. J Neurophysiol (2008) 0.90

The fragile X mental retardation protein developmentally regulates the strength and fidelity of calcium signaling in Drosophila mushroom body neurons. Neurobiol Dis (2010) 0.89

Rolling blackout is required for bulk endocytosis in non-neuronal cells and neuronal synapses. J Cell Sci (2008) 0.89

Drosophila FMRP participates in the DNA damage response by regulating G2/M cell cycle checkpoint and apoptosis. Hum Mol Genet (2012) 0.89

Temporal requirements of the fragile x mental retardation protein in modulating circadian clock circuit synaptic architecture. Front Neural Circuits (2009) 0.89

Drosophila Acyl-CoA synthetase long-chain family member 4 regulates axonal transport of synaptic vesicles and is required for synaptic development and transmission. J Neurosci (2011) 0.88

dAcsl, the Drosophila ortholog of acyl-CoA synthetase long-chain family member 3 and 4, inhibits synapse growth by attenuating bone morphogenetic protein signaling via endocytic recycling. J Neurosci (2014) 0.87

Drosophila twinfilin is required for cell migration and synaptic endocytosis. J Cell Sci (2010) 0.87

Microtubule-severing protein Katanin regulates neuromuscular junction development and dendritic elaboration in Drosophila. Development (2014) 0.87

Presynaptic secretion of mind-the-gap organizes the synaptic extracellular matrix-integrin interface and postsynaptic environments. Dev Dyn (2009) 0.86

Defective HIV-1 particle assembly in AP-3-deficient cells derived from patients with Hermansky-Pudlak syndrome type 2. J Virol (2012) 0.85

Fragile X mental retardation protein regulates trans-synaptic signaling in Drosophila. Dis Model Mech (2013) 0.84

Drosophila modeling of heritable neurodevelopmental disorders. Curr Opin Neurobiol (2011) 0.84

Jelly Belly trans-synaptic signaling to anaplastic lymphoma kinase regulates neurotransmission strength and synapse architecture. Dev Neurobiol (2012) 0.84

Fragile X mental retardation protein is required for programmed cell death and clearance of developmentally-transient peptidergic neurons. Dev Biol (2011) 0.83

Structure-function analysis of endogenous lectin mind-the-gap in synaptogenesis. Dev Neurobiol (2012) 0.83

Molecular and genetic analysis of the Drosophila model of fragile X syndrome. Results Probl Cell Differ (2012) 0.83

In vivo assay of presynaptic microtubule cytoskeleton dynamics in Drosophila. J Neurosci Methods (2007) 0.82

N-glycosylation requirements in neuromuscular synaptogenesis. Development (2013) 0.82

Two peptide transmitters co-packaged in a single neurosecretory vesicle. Peptides (2008) 0.81

The cell polarity scaffold Lethal Giant Larvae regulates synapse morphology and function. J Cell Sci (2013) 0.81

Drosophila CG10527 mutants are resistant to juvenile hormone and its analog methoprene. Biochem Biophys Res Commun (2010) 0.81

Electrophysiological recording in the Drosophila embryo. J Vis Exp (2009) 0.80

Techniques to dissect cellular and subcellular function in the Drosophila nervous system. Methods Cell Biol (2003) 0.80

Response: meaningless minis? Trends Neurosci (2002) 0.79

Expression and function of scalloped during Drosophila development. Dev Dyn (2013) 0.77

Drosophila rolling blackout displays lipase domain-dependent and -independent endocytic functions downstream of dynamin. Traffic (2010) 0.77