Published in Neuron on June 13, 2002
A Drosophila fragile X protein interacts with components of RNAi and ribosomal proteins. Genes Dev (2002) 6.10
Molecular and comparative genetics of mental retardation. Genetics (2004) 2.38
Circadian organization of behavior and physiology in Drosophila. Annu Rev Physiol (2010) 2.36
Human disease models in Drosophila melanogaster and the role of the fly in therapeutic drug discovery. Pharmacol Rev (2011) 2.25
The state of synapses in fragile X syndrome. Neuroscientist (2009) 2.07
Identification of four highly conserved genes between breakpoint hotspots BP1 and BP2 of the Prader-Willi/Angelman syndromes deletion region that have undergone evolutionary transposition mediated by flanking duplicons. Am J Hum Genet (2003) 1.82
A signaling network for patterning of neuronal connectivity in the Drosophila brain. PLoS Biol (2006) 1.66
Posttranscriptional control of neuronal development by microRNA networks. Trends Neurosci (2007) 1.56
The steady-state level of the nervous-system-specific microRNA-124a is regulated by dFMR1 in Drosophila. J Neurosci (2008) 1.38
Netrin-1-induced local β-actin synthesis and growth cone guidance requires zipcode binding protein 1. J Neurosci (2011) 1.32
Fragile X-related proteins regulate mammalian circadian behavioral rhythms. Am J Hum Genet (2008) 1.32
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The Drosophila fragile X mental retardation gene regulates sleep need. J Neurosci (2009) 1.25
Altered hippocampal synaptic plasticity in the FMR1 gene family knockout mouse models. J Neurophysiol (2009) 1.24
Disruption of the MAP1B-related protein FUTSCH leads to changes in the neuronal cytoskeleton, axonal transport defects, and progressive neurodegeneration in Drosophila. Mol Biol Cell (2005) 1.24
Temporal requirements of the fragile X mental retardation protein in the regulation of synaptic structure. Development (2008) 1.21
Fragile X related protein 1 isoforms differentially modulate the affinity of fragile X mental retardation protein for G-quartet RNA structure. Nucleic Acids Res (2006) 1.19
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Pharmacological reversal of synaptic plasticity deficits in the mouse model of fragile X syndrome by group II mGluR antagonist or lithium treatment. Brain Res (2010) 1.14
Fragile X Mental Retardation Protein is Involved in Protein Synthesis-Dependent Collapse of Growth Cones Induced by Semaphorin-3A. Front Neural Circuits (2009) 1.08
Casein kinase II phosphorylates the fragile X mental retardation protein and modulates its biological properties. Mol Cell Biol (2002) 1.06
Drosophila as a genetic and cellular model for studies on axonal growth. Neural Dev (2007) 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
Glutamate receptor dysfunction and drug targets across models of autism spectrum disorders. Pharmacol Biochem Behav (2011) 1.01
Fragile X mental retardation protein regulates new neuron differentiation in the adult olfactory bulb. J Neurosci (2011) 1.00
Emerging pharmacotherapies for neurodevelopmental disorders. J Dev Behav Pediatr (2010) 0.99
The fragile X mental retardation protein in circadian rhythmicity and memory consolidation. Mol Neurobiol (2009) 0.99
Short- and long-term memory are modulated by multiple isoforms of the fragile X mental retardation protein. J Neurosci (2010) 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
Motility screen identifies Drosophila IGF-II mRNA-binding protein--zipcode-binding protein acting in oogenesis and synaptogenesis. PLoS Genet (2008) 0.95
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
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
iPSC-derived forebrain neurons from FXS individuals show defects in initial neurite outgrowth. Stem Cells Dev (2014) 0.92
Expression of three zebrafish orthologs of human FMR1-related genes and their phylogenetic relationships. Dev Genes Evol (2004) 0.92
Age-dependent cognitive impairment in a Drosophila fragile X model and its pharmacological rescue. Biogerontology (2009) 0.91
Argonaute2 suppresses Drosophila fragile X expression preventing neurogenesis and oogenesis defects. PLoS One (2009) 0.91
Temporal requirements of the fragile x mental retardation protein in modulating circadian clock circuit synaptic architecture. Front Neural Circuits (2009) 0.89
Conservation of the RNA Transport Machineries and Their Coupling to Translation Control across Eukaryotes. Comp Funct Genomics (2012) 0.88
The drosophila fragile X protein dFMR1 is required during early embryogenesis for pole cell formation and rapid nuclear division cycles. Genetics (2006) 0.87
A presynaptic role for FMRP during protein synthesis-dependent long-term plasticity in Aplysia. Learn Mem (2010) 0.86
Substitution of critical isoleucines in the KH domains of Drosophila fragile X protein results in partial loss-of-function phenotypes. Genetics (2006) 0.85
Fragile X syndrome and model organisms: identifying potential routes of therapeutic intervention. Dis Model Mech (2010) 0.85
A non-canonical start codon in the Drosophila fragile X gene yields two functional isoforms. Neuroscience (2011) 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
A feed-forward mechanism involving Drosophila fragile X mental retardation protein triggers a replication stress-induced DNA damage response. Hum Mol Genet (2014) 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
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
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
Modeling brain dynamics using computational neurogenetic approach. Cogn Neurodyn (2008) 0.83
Molecular and genetic analysis of the Drosophila model of fragile X syndrome. Results Probl Cell Differ (2012) 0.83
Drosophila as an In Vivo Model for Human Neurodegenerative Disease. Genetics (2015) 0.83
Context-based FISH localization of genomic rearrangements within chromosome 15q11.2q13 duplicons. Mol Cytogenet (2011) 0.82
Neurodegeneration and microtubule dynamics: death by a thousand cuts. Front Cell Neurosci (2015) 0.82
Altered LARK expression perturbs development and physiology of the Drosophila PDF clock neurons. Mol Cell Neurosci (2009) 0.82
Cellular requirements for LARK in the Drosophila circadian system. J Biol Rhythms (2012) 0.81
Characterization of fragile X mental retardation protein recruitment and dynamics in Drosophila stress granules. PLoS One (2013) 0.81
The Little Fly that Could: Wizardry and Artistry of Drosophila Genomics. Genes (Basel) (2014) 0.81
Fragile X mental retardation protein: A paradigm for translational control by RNA-binding proteins. Biochimie (2015) 0.81
PDE-4 inhibition rescues aberrant synaptic plasticity in Drosophila and mouse models of fragile X syndrome. J Neurosci (2015) 0.81
From FMRP function to potential therapies for fragile X syndrome. Neurochem Res (2013) 0.80
The Drosophila gene RanBPM functions in the mushroom body to regulate larval behavior. PLoS One (2010) 0.80
Epigenetic regulation of neuronal dendrite and dendritic spine development. Front Biol (Beijing) (2010) 0.79
Lithium: a promising treatment for fragile X syndrome. ACS Chem Neurosci (2014) 0.78
Insulin signaling misregulation underlies circadian and cognitive deficits in a Drosophila fragile X model. Mol Psychiatry (2016) 0.78
Synergistic interactions between Drosophila orthologues of genes spanned by de novo human CNVs support multiple-hit models of autism. PLoS Genet (2015) 0.78
Neural progenitor cells from an adult patient with fragile X syndrome. BMC Med Genet (2005) 0.78
The role of the actin cytoskeleton in regulating Drosophila behavior. Rev Neurosci (2013) 0.78
Rasputin functions as a positive regulator of orb in Drosophila oogenesis. PLoS One (2013) 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
Multiple Drug Treatments That Increase cAMP Signaling Restore Long-Term Memory and Aberrant Signaling in Fragile X Syndrome Models. Front Behav Neurosci (2016) 0.76
Transcriptome Profiling Following Neuronal and Glial Expression of ALS-Linked SOD1 in Drosophila. G3 (Bethesda) (2013) 0.76
Circadian rhythm-dependent alterations of gene expression in Drosophila brain lacking fragile X mental retardation protein. PLoS One (2012) 0.76
Interference of the complex between NCS-1 and Ric8a with phenothiazines regulates synaptic function and is an approach for fragile X syndrome. Proc Natl Acad Sci U S A (2017) 0.75
Using Drosophila as a tool to identify Pharmacological Therapies for Fragile X Syndrome. Drug Discov Today Technol (2012) 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
Drosophila melanogaster As a Model Organism to Study RNA Toxicity of Repeat Expansion-Associated Neurodegenerative and Neuromuscular Diseases. Front Cell Neurosci (2017) 0.75
Progress toward therapeutic potential for AFQ056 in Fragile X syndrome. J Exp Pharmacol (2013) 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
Reduced Lateral Inhibition Impairs Olfactory Computations and Behaviors in a Drosophila Model of Fragile X Syndrome. Curr Biol (2017) 0.75
Evolutionary and biomedical insights from the rhesus macaque genome. Science (2007) 16.21
Annotation of the Drosophila melanogaster euchromatic genome: a systematic review. Genome Biol (2002) 8.59
The BDGP gene disruption project: single transposon insertions associated with 40% of Drosophila genes. Genetics (2004) 8.40
Comparative genome sequencing of Drosophila pseudoobscura: chromosomal, gene, and cis-element evolution. Genome Res (2005) 8.38
The DNA sequence of the human X chromosome. Nature (2005) 6.97
Revisiting the protein-coding gene catalog of Drosophila melanogaster using 12 fly genomes. Genome Res (2007) 4.73
Biochemical and genetic interaction between the fragile X mental retardation protein and the microRNA pathway. Nat Neurosci (2004) 4.69
Atomoxetine increases extracellular levels of norepinephrine and dopamine in prefrontal cortex of rat: a potential mechanism for efficacy in attention deficit/hyperactivity disorder. Neuropsychopharmacology (2002) 3.79
Dopamine is a regulator of arousal in the fruit fly. J Neurosci (2005) 3.65
RNA-binding proteins hnRNP A2/B1 and CUGBP1 suppress fragile X CGG premutation repeat-induced neurodegeneration in a Drosophila model of FXTAS. Neuron (2007) 3.30
A role for CK2 in the Drosophila circadian oscillator. Nat Neurosci (2003) 3.09
The v-ATPase V0 subunit a1 is required for a late step in synaptic vesicle exocytosis in Drosophila. Cell (2005) 3.05
Drosophila parkin mutants have decreased mass and cell size and increased sensitivity to oxygen radical stress. Development (2004) 3.03
Synaptojanin is recruited by endophilin to promote synaptic vesicle uncoating. Neuron (2003) 2.96
Shar-pei mediates cell proliferation arrest during imaginal disc growth in Drosophila. Development (2002) 2.82
Drosophila NMNAT maintains neural integrity independent of its NAD synthesis activity. PLoS Biol (2006) 2.70
Vitamin K2 is a mitochondrial electron carrier that rescues pink1 deficiency. Science (2012) 2.65
Exome sequencing identifies mutation in CNOT3 and ribosomal genes RPL5 and RPL10 in T-cell acute lymphoblastic leukemia. Nat Genet (2012) 2.64
Rescue of behavioral phenotype and neuronal protrusion morphology in Fmr1 KO mice. Neurobiol Dis (2008) 2.52
LRRK2 controls an EndoA phosphorylation cycle in synaptic endocytosis. Neuron (2012) 2.49
Dap160/intersectin acts as a stabilizing scaffold required for synaptic development and vesicle endocytosis. Neuron (2004) 2.47
Thirty-one flavors of Drosophila rab proteins. Genetics (2007) 2.46
Mutations in Drosophila sec15 reveal a function in neuronal targeting for a subset of exocyst components. Neuron (2005) 2.33
Expanded GGGGCC repeat RNA associated with amyotrophic lateral sclerosis and frontotemporal dementia causes neurodegeneration. Proc Natl Acad Sci U S A (2013) 2.30
Epidermal progenitors give rise to Merkel cells during embryonic development and adult homeostasis. J Cell Biol (2009) 2.24
Drosophila ebony activity is required in glia for the circadian regulation of locomotor activity. Neuron (2007) 2.17
NAD synthase NMNAT acts as a chaperone to protect against neurodegeneration. Nature (2008) 2.01
Systematic discovery of Rab GTPases with synaptic functions in Drosophila. Curr Biol (2011) 1.99
The nicotinic acetylcholine receptor Dalpha7 is required for an escape behavior in Drosophila. PLoS Biol (2006) 1.97
Mapping Drosophila mutations with molecularly defined P element insertions. Proc Natl Acad Sci U S A (2003) 1.94
Targeted ablation of CCAP neuropeptide-containing neurons of Drosophila causes specific defects in execution and circadian timing of ecdysis behavior. Development (2003) 1.93
Parkinson's disease mutations in PINK1 result in decreased Complex I activity and deficient synaptic function. EMBO Mol Med (2009) 1.89
Intestinal stem cells lacking the Math1 tumour suppressor are refractory to Notch inhibitors. Nat Commun (2010) 1.89
Reexamining the role of TACI coding variants in common variable immunodeficiency and selective IgA deficiency. Nat Genet (2007) 1.87
Gender dimorphism in the role of cycle (BMAL1) in rest, rest regulation, and longevity in Drosophila melanogaster. J Biol Rhythms (2003) 1.85
ELP3 controls active zone morphology by acetylating the ELKS family member Bruchpilot. Neuron (2011) 1.83
Glial cells physiologically modulate clock neurons and circadian behavior in a calcium-dependent manner. Curr Biol (2011) 1.80
Mutations of the Wiskott-Aldrich Syndrome Protein (WASP): hotspots, effect on transcription, and translation and phenotype/genotype correlation. Blood (2004) 1.78
Identification of a dopamine pathway that regulates sleep and arousal in Drosophila. Nat Neurosci (2012) 1.76
Atonal homolog 1 is a tumor suppressor gene. PLoS Biol (2009) 1.76
Genetically encoded dendritic marker sheds light on neuronal connectivity in Drosophila. Proc Natl Acad Sci U S A (2010) 1.71
Decreased expression of the GABAA receptor in fragile X syndrome. Brain Res (2006) 1.70
Behavioral consequences of dopamine deficiency in the Drosophila central nervous system. Proc Natl Acad Sci U S A (2010) 1.67
A signaling network for patterning of neuronal connectivity in the Drosophila brain. PLoS Biol (2006) 1.66
Successful determination of lower inflection point and maximal compliance in a population of patients with acute respiratory distress syndrome. Crit Care Med (2002) 1.60
Amyloid precursor protein promotes post-developmental neurite arborization in the Drosophila brain. EMBO J (2005) 1.56
Locomotor activity is regulated by D2-like receptors in Drosophila: an anatomic and functional analysis. Dev Neurobiol (2007) 1.54
The Drosophila homologue of the amyloid precursor protein is a conserved modulator of Wnt PCP signaling. PLoS Biol (2013) 1.54
Ectopic expression of CGG containing mRNA is neurotoxic in mammals. Hum Mol Genet (2009) 1.54
Aberrant lysosomal carbohydrate storage accompanies endocytic defects and neurodegeneration in Drosophila benchwarmer. J Cell Biol (2005) 1.52
Activity-independent prespecification of synaptic partners in the visual map of Drosophila. Curr Biol (2006) 1.50
Loss of skywalker reveals synaptic endosomes as sorting stations for synaptic vesicle proteins. Cell (2011) 1.48
Ablation of Fmrp in adult neural stem cells disrupts hippocampus-dependent learning. Nat Med (2011) 1.47
Altered leukocyte response to CXCL12 in patients with warts hypogammaglobulinemia, infections, myelokathexis (WHIM) syndrome. Blood (2004) 1.46
Variants of the elongator protein 3 (ELP3) gene are associated with motor neuron degeneration. Hum Mol Genet (2008) 1.46
Inactivation of clathrin heavy chain inhibits synaptic recycling but allows bulk membrane uptake. J Cell Biol (2008) 1.45
Axonal injury and regeneration in the adult brain of Drosophila. J Neurosci (2008) 1.42
Recombineering-mediated tagging of Drosophila genomic constructs for in vivo localization and acute protein inactivation. Nucleic Acids Res (2008) 1.39
Fxr1 knockout mice show a striated muscle phenotype: implications for Fxr1p function in vivo. Hum Mol Genet (2004) 1.39
Suppression of neurodegeneration and increased neurotransmission caused by expanded full-length huntingtin accumulating in the cytoplasm. Neuron (2008) 1.38
A dual function of V0-ATPase a1 provides an endolysosomal degradation mechanism in Drosophila melanogaster photoreceptors. J Cell Biol (2010) 1.38
Exaggerated behavioral phenotypes in Fmr1/Fxr2 double knockout mice reveal a functional genetic interaction between Fragile X-related proteins. Hum Mol Genet (2006) 1.38
The Drosophila fragile X mental retardation protein controls actin dynamics by directly regulating profilin in the brain. Curr Biol (2005) 1.38
Chromosomal microarray analysis (CMA) detects a large X chromosome deletion including FMR1, FMR2, and IDS in a female patient with mental retardation. Am J Med Genet A (2007) 1.37
Huntingtin-interacting protein 14, a palmitoyl transferase required for exocytosis and targeting of CSP to synaptic vesicles. J Cell Biol (2007) 1.37
Endophilin promotes a late step in endocytosis at glial invaginations in Drosophila photoreceptor terminals. J Neurosci (2003) 1.37
Mitochondria at the synapse. Neuroscientist (2006) 1.35
Role for Msh5 in the regulation of Ig class switch recombination. Proc Natl Acad Sci U S A (2007) 1.35
Expression of the GABAergic system in animal models for fragile X syndrome and fragile X associated tremor/ataxia syndrome (FXTAS). Brain Res (2008) 1.35
AGG interruptions within the maternal FMR1 gene reduce the risk of offspring with fragile X syndrome. Genet Med (2012) 1.33
Interruptions in the expanded ATTCT repeat of spinocerebellar ataxia type 10: repeat purity as a disease modifier? Am J Hum Genet (2005) 1.32
PINK1 loss-of-function mutations affect mitochondrial complex I activity via NdufA10 ubiquinone uncoupling. Science (2014) 1.32
Fragile X-related proteins regulate mammalian circadian behavioral rhythms. Am J Hum Genet (2008) 1.32
Conditional mutagenesis in Drosophila. Science (2009) 1.30
Differentiation of mouse and human embryonic stem cells into hepatic lineages. Genes Cells (2008) 1.27
Pharmacologic characterization of the cloned human trace amine-associated receptor1 (TAAR1) and evidence for species differences with the rat TAAR1. J Pharmacol Exp Ther (2006) 1.26
Haplotype and linkage disequilibrium architecture for human cancer-associated genes. Genome Res (2002) 1.26
Drosophila atonal fully rescues the phenotype of Math1 null mice: new functions evolve in new cellular contexts. Curr Biol (2002) 1.25
Altered hippocampal synaptic plasticity in the FMR1 gene family knockout mouse models. J Neurophysiol (2009) 1.24
Vibrio cholerae infection of Drosophila melanogaster mimics the human disease cholera. PLoS Pathog (2005) 1.24
Bmal1 and β-cell clock are required for adaptation to circadian disruption, and their loss of function leads to oxidative stress-induced β-cell failure in mice. Mol Cell Biol (2013) 1.22
Social behavior in Fmr1 knockout mice carrying a human FMR1 transgene. Behav Neurosci (2008) 1.21
Cytosolic phospholipase A2alpha-deficient mice are resistant to collagen-induced arthritis. J Exp Med (2003) 1.21
Guided differentiation of embryonic stem cells into Pdx1-expressing regional-specific definitive endoderm. Stem Cells (2008) 1.21
straightjacket is required for the synaptic stabilization of cacophony, a voltage-gated calcium channel alpha1 subunit. J Cell Biol (2008) 1.20
Positive selection of a pre-expansion CAG repeat of the human SCA2 gene. PLoS Genet (2005) 1.19
Evidence for RNA-mediated toxicity in the fragile X-associated tremor/ataxia syndrome. Future Neurol (2009) 1.17
Preparation of developing and adult Drosophila brains and retinae for live imaging. J Vis Exp (2010) 1.14
Robust target gene discovery through transcriptome perturbations and genome-wide enhancer predictions in Drosophila uncovers a regulatory basis for sensory specification. PLoS Biol (2010) 1.13
Enhanced expression of PDX-1 and Ngn3 by exendin-4 during beta cell regeneration in STZ-treated mice. Biochem Biophys Res Commun (2005) 1.13
Synaptic mitochondria in synaptic transmission and organization of vesicle pools in health and disease. Front Synaptic Neurosci (2010) 1.12