ArticleNature communications2020
FMRP(1-297)-tat restores ion channel and synaptic function in a model of Fragile X syndrome.
Article in Nature communications, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 22 papers.
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Who cites it
22 citing papers in PubMed, 45 citations in OpenAlex.
- Kv4.2The Journal of neuroscience : the official journal of the Society for Neuroscience · 2026Article
- Autophagy controls the hippocampal postsynaptic organization and affects cognition in a mouse model of Fragile X syndrome.Molecular psychiatry · 2026Article
- Autophagy controls the hippocampal postsynaptic organization and affects cognition in a mouse model of Fragile X syndrome.Research square · 2025Article
- Neuronal potassium channel activity triggers initiation of mRNA translation through binding of translation regulators.Science advances · 2025Article
- Oxygen concentration modulates HDAC1-Mediated regulation of osteogenic signaling pathways in dental pulp cells.Frontiers in cell and developmental biology · 2025Article
- Molecular Profiling of Mouse Models of Loss or Gain of Function of the KCNT1 (Slack) Potassium Channel and Antisense Oligonucleotide Treatment.Biomolecules · 2024Article
- Cage effects on synaptic plasticity and its modulation in a mouse model of fragile X syndrome.Philosophical transactions of the Royal Society of London. Series B, Biological sciences · 2024Article
- State-of-the-art therapies for fragile X syndrome.Developmental medicine and child neurology · 2024Review
- Calcium-Dependent Regulation of Neuronal Excitability Is Rescued in Fragile X Syndrome by a Tat-Conjugated N-Terminal Fragment of FMRP.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2024Article
- The effects of NMolecular and cellular biochemistry · 2023Review
- Tau reduction attenuates autism-like features in Fmr1 knockout mice.Molecular autism · 2023Article
- Efficient Delivery ofGenes · 2023Article
- Cellular distribution of the Fragile X mental retardation protein in the inner ear: a developmental and comparative study in the mouse, rat, gerbil, and chicken.The Journal of comparative neurology · 2023Article
- Function of FMRP Domains in Regulating Distinct Roles of Neuronal Protein Synthesis.Molecular neurobiology · 2022Article
- Article
- The role of altered translation in intellectual disability and epilepsy.Progress in neurobiology · 2022Review
- FMRP regulates GABACell reports · 2022Article
- Identification of microRNAs associated with human fragile X syndrome using next-generation sequencing.Scientific reports · 2022Article
- Channelopathies in fragile X syndrome.Nature reviews. Neuroscience · 2021Review
- The life cycle of voltage-gated CaNeuronal signaling · 2021Review
Corrections and comments
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Authors and funding
9 authors at 1 institution in 1 country.
Funding
Abstract
Fragile X Syndrome results from a loss of Fragile X Mental Retardation Protein (FMRP). We now show that FMRP is a member of a Cav3-Kv4 ion channel complex that is known to regulate A-type potassium current in cerebellar granule cells to produce mossy fiber LTP. Mossy fiber LTP is absent in Fmr1 knockout (KO) mice but is restored by FMRP(1-297)-tat peptide. This peptide further rapidly permeates the blood-brain barrier to enter cells across the cerebellar-cortical axis that restores the balance of protein translation for at least 24 h and transiently reduces elevated levels of activity of adult Fmr1 KO mice in the Open Field Test. These data reveal that FMRP(1-297)-tat can improve function from the levels of protein translation to synaptic efficacy and behaviour in a model of Fragile X syndrome, identifying a potential therapeutic strategy for this genetic disorder.
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Registered trials
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