Dysregulation and restoration of translational homeostasis in fragile X syndrome

Human MicroRNA Targets

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FMRP Stalls Ribosomal Translocation on mRNAs Linked to Synaptic Function and Autism

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The mGluR theory of fragile X mental retardation

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Regulation of translation initiation by FRAP/mTOR

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mTOR signaling: At the crossroads of plasticity, memory and disease

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CLIP Identifies Nova-Regulated RNA Networks in the Brain

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Microarray identification of FMRP-associated brain mRNAs and altered mRNA translational profiles in fragile X syndrome

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Correction of Fragile X Syndrome in Mice

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Fragile X mental retardation protein targets G quartet mRNAs important for neuronal function

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Mitogen‐activated protein kinases activate the serine/threonine kinases Mnk1 and Mnk2

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Hierarchical phosphorylation of the translation inhibitor 4E-BP1

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The Scanning Mechanism of Eukaryotic Translation Initiation

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Regulation of Synaptic Structure and Function by FMRP-Associated MicroRNAs miR-125b and miR-132

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FMRP targets distinct mRNA sequence elements to regulate protein expression

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The Fragile X Syndrome Protein FMRP Associates with BC1 RNA and Regulates the Translation of Specific mRNAs at Synapses

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A marker X chromosome.

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The Fragile X Syndrome Protein Represses Activity-Dependent Translation through CYFIP1, a New 4E-BP

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Biochemical and genetic interaction between the fragile X mental retardation protein and the microRNA pathway

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Mutations causing syndromic autism define an axis of synaptic pathophysiology

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Dysregulation of mTOR signaling in fragile X syndrome

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Molecular Mechanisms of Fragile X Syndrome: A Twenty-Year Perspective

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Suppression of two major Fragile X Syndrome mouse model phenotypes by the mGluR5 antagonist MPEP

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RNA cargoes associating with FMRP reveal deficits in cellular functioning in Fmr1 null mice

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The translation of translational control by FMRP: therapeutic targets for FXS

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Chronic Pharmacological mGlu5 Inhibition Corrects Fragile X in Adult Mice

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CPEB-Mediated Cytoplasmic Polyadenylation and the Regulation of Experience-Dependent Translation of α-CaMKII mRNA at Synapses

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A PEDIGREE OF MENTAL DEFECT SHOWING SEX-LINKAGE

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Phosphorylation of eucaryotic translation initiation factor 4B Ser422 is modulated by S6 kinases

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The fragile X mental retardation protein binds specifically to its mRNA via a purine quartet motif

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Dynamic Translational and Proteasomal Regulation of Fragile X Mental Retardation Protein Controls mGluR-Dependent Long-Term Depression

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Phosphorylation of the Cap-Binding Protein Eukaryotic Translation Initiation Factor 4E by Protein Kinase Mnk1 In Vivo

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Minocycline promotes dendritic spine maturation and improves behavioural performance in the fragile X mouse model

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Dysregulated Metabotropic Glutamate Receptor-Dependent Translation of AMPA Receptor and Postsynaptic Density-95 mRNAs at Synapses in a Mouse Model of Fragile X Syndrome

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The HMG-CoA Reductase Inhibitor Lovastatin Reverses the Learning and Attention Deficits in a Mouse Model of Neurofibromatosis Type 1

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Hypersensitivity to mGluR5 and ERK1/2 Leads to Excessive Protein Synthesis in the Hippocampus of a Mouse Model of Fragile X Syndrome

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Evidence for reassociation of RNA-binding proteins after cell lysis: Implications for the interpretation of immunoprecipitation analyses

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Functional characterization of IRESes by an inhibitor of the RNA helicase eIF4A

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A new function for the fragile X mental retardation protein in regulation of PSD-95 mRNA stability

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The Pathophysiology of Fragile X (and What It Teaches Us about Synapses)

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Reversible Inhibition of PSD-95 mRNA Translation by miR-125a, FMRP Phosphorylation, and mGluR Signaling

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Translational Suppression by Trinucleotide Repeat Expansion at FMR1

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A highly conserved protein family interacting with the fragile X mental retardation protein (FMRP) and displaying selective interactions with FMRP-related proteins FXR1P and FXR2P

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FMRP regulates neurotransmitter release and synaptic information transmission by modulating action potential duration via BK channels

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RNA and microRNAs in fragile X mental retardation

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Effects of STX209 (Arbaclofen) on Neurobehavioral Function in Children and Adults with Fragile X Syndrome: A Randomized, Controlled, Phase 2 Trial

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Phosphorylation influences the translation state of FMRP-associated polyribosomes

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CYFIP/Sra-1 controls neuronal connectivity in Drosophila and links the Rac1 GTPase pathway to the fragile X protein.

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Genetic Removal of p70 S6 Kinase 1 Corrects Molecular, Synaptic, and Behavioral Phenotypes in Fragile X Syndrome Mice

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Epigenetic Characterization of the FMR1 Gene and Aberrant Neurodevelopment in Human Induced Pluripotent Stem Cell Models of Fragile X Syndrome

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A Randomized Double-Blind, Placebo-Controlled Trial of Minocycline in Children and Adolescents with Fragile X Syndrome

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CYFIP1 Coordinates mRNA Translation and Cytoskeleton Remodeling to Ensure Proper Dendritic Spine Formation

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A Mechanism for Translationally Coupled mRNA Turnover

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Kissing complex RNAs mediate interaction between the Fragile-X mental retardation protein KH2 domain and brain polyribosomes

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Role for metabotropic glutamate receptor 5 (mGluR5) in the pathogenesis of fragile X syndrome

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Fragile X mental retardation protein is associated with translating polyribosomes in neuronal cells

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Fragile X mental retardation protein controls gating of the sodium-activated potassium channel Slack

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Rescue of fragile X syndrome phenotypes in Fmr1 KO mice by the small-molecule PAK inhibitor FRAX486

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Identification of small molecules rescuing fragile X syndrome phenotypes in Drosophila

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Fragile X Mental Retardation Protein Regulates Translation by Binding Directly to the Ribosome

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Disrupted Homer scaffolds mediate abnormal mGluR5 function in a mouse model of fragile X syndrome

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G-quadruplex RNA structure as a signal for neurite mRNA targeting

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Reversal of Disease-Related Pathologies in the Fragile X Mouse Model by Selective Activation of GABA B Receptors with Arbaclofen

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Targeting the endocannabinoid system in the treatment of fragile X syndrome

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Excess phosphoinositide 3-kinase subunit synthesis and activity as a novel therapeutic target in fragile X syndrome

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The fragile X mental retardation protein is associated with poly(A)+ mRNA in actively translating polyribosomes

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Lovastatin corrects excess protein synthesis and prevents epileptogenesis in a mouse model of fragile X syndrome

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FMRP Phosphorylation Reveals an Immediate-Early Signaling Pathway Triggered by Group I mGluR and Mediated by PP2A

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Cytoplasmic Polyadenylation Element Binding Proteins in Development, Health, and Disease

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Fragile X mental retardation protein is necessary for neurotransmitter-activated protein translation at synapses

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The Unstable Repeats—Three Evolving Faces of Neurological Disease

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Pharmacogenetic inhibition of eIF4E-dependent Mmp9 mRNA translation reverses fragile X syndrome-like phenotypes

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Altered mTOR signaling and enhanced CYFIP2 expression levels in subjects with fragile X syndrome

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Prolonged Epileptiform Discharges Induced by Altered Group I Metabotropic Glutamate Receptor-Mediated Synaptic Responses in Hippocampal Slices of a Fragile X Mouse Model

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Biochemical evidence for the association of fragile X mental retardation protein with brain polyribosomal ribonucleoparticles

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Quantitative proteomic analysis of primary neurons reveals diverse changes in synaptic protein content in fmr1 knockout mice

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iPSC-derived forebrain neurons from FXS individuals show defects in initial neurite outgrowth

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Fragile X mental retardation protein regulates protein expression and mRNA translation of the potassium channel Kv4.2

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Inhibition of GSK3 improves hippocampus-dependent learning and rescues neurogenesis in a mouse model of fragile X syndrome

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Genetic removal of matrix metalloproteinase 9 rescues the symptoms of fragile X syndrome in a mouse model

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Bidirectional Regulation of Dendritic Voltage-Gated Potassium Channels by the Fragile X Mental Retardation Protein

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Evidence for a Fragile X Mental Retardation Protein-Mediated Translational Switch in Metabotropic Glutamate Receptor-Triggered Arc Translation and Long-Term Depression

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MOV10 and FMRP Regulate AGO2 Association with MicroRNA Recognition Elements

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Increased Expression of the PI3K Enhancer PIKE Mediates Deficits in Synaptic Plasticity and Behavior in Fragile X Syndrome

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The bantam microRNA is associated with drosophila fragile X mental retardation protein and regulates the fate of germline stem cells

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Glycogen Synthase Kinase-3 Inhibitors Reverse Deficits in Long-term Potentiation and Cognition in Fragile X Mice

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The Fragile X mental retardation protein regulates matrix metalloproteinase 9 mRNA at synapses.

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Reversal of Fragile X Phenotypes by Manipulation of AβPP/Aβ Levels in Fmr1KO Mice

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Impaired Dendritic Expression and Plasticity of h-Channels in the fmr1 Mouse Model of Fragile X Syndrome

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Genetic manipulation of STEP reverses behavioral abnormalities in a fragile X syndrome mouse model

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The Steady-State Level of the Nervous-System-Specific MicroRNA-124a Is Regulated by dFMR1 inDrosophila

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A Molecular Circuit Composed of CPEB-1 and c-Jun Controls Growth Hormone-Mediated Synaptic Plasticity in the Mouse Hippocampus

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Molecular Mechanisms Regulating the Defects in Fragile X Syndrome Neurons Derived from Human Pluripotent Stem Cells

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Fragile X mental retardation protein interactions with the microtubule associated protein 1B RNA

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BDNF stimulation of protein synthesis in cortical neurons requires the MAP kinase-interacting kinase MNK1

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Insights into GABAAergic system deficits in fragile X syndrome lead to clinical trials

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Translational control from head to tail

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Loss of Functional A-Type Potassium Channels in the Dendrites of CA1 Pyramidal Neurons from a Mouse Model of Fragile X Syndrome

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Selective Role of the Catalytic PI3K Subunit p110β in Impaired Higher Order Cognition in Fragile X Syndrome

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Excess Protein Synthesis in FXS Patient Lymphoblastoid Cells Can Be Rescued with a p110β-Selective Inhibitor

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Glycogen Synthase Kinase-3: A Promising Therapeutic Target for Fragile X Syndrome

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SnapShot: FMRP mRNA Targets and Diseases

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Protein Expression Profiling of the Fragile X Mutant Brain Reveals Up-regulation of Monoamine Synthesis

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Identification of Fragile X Syndrome Specific Molecular Markers in Human Fibroblasts: A Useful Model to Test the Efficacy of Therapeutic Drugs

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Meet the players: local translation at the synapse

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G quadruplex RNA structures in PSD-95 mRNA: potential regulators of miR-125a seed binding site accessibility

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GABAergic circuit dysfunction in the Drosophila Fragile X syndrome model

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Homer Protein–Metabotropic Glutamate Receptor Binding Regulates Endocannabinoid Signaling and Affects Hyperexcitability in a Mouse Model of Fragile X Syndrome

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Unmasking the messenger

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FMRP regulates miR196a-mediated repression of HOXB8 via interaction with the AGO2 MID domain

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Local translation in neurons: visualization and function

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Excitation/Inhibition Imbalance in Animal Models of Autism Spectrum Disorders

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Local protein synthesis is a ubiquitous feature of neuronal pre- and postsynaptic compartments

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Balancing translation and Rac1 signaling

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Genetic and acute CPEB1 depletion ameliorate fragile X pathophysiology

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m⁶A-dependent FMRP control of DGKκ translation underlies core Fragile X phenotypesPreprint

Cakil, O., Zámbó, B., Maroilley, T., Drouot, N., Petrova, A., Négroni, L., Gógl, G., Piton, A., & Moine, H. (2026) · bioRxiv (Cold Spring Harbor Laboratory) · doi:10.64898/2026.01.20.700356