ArticleNature communications2023
Linoleic acid improves PIEZO2 dysfunction in a mouse model of Angelman Syndrome.
Article in Nature communications, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 41 papers.
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Who cites it
41 citing papers in PubMed, 53 citations in OpenAlex.
- Loss of DrosophilaFly · 2026Article
- Cold-gated potentiation of mechanotransduction supports touch sensation below 5°C in a mammalian hibernator.Current biology : CB · 2026Article
- Piezo channels in physical field therapy: From mechanotransduction mechanisms to bioelectromagnetic modulation and biomedical applications.Mechanobiology in medicine · 2026Review
- An inherited hypomorphic variant in PIEZO2 reveals structural features of mechanotransduction.Neuron · 2026Article
- Acbd7 is essential for preserving hair cell-mediated auditory and vestibular function.Experimental & molecular medicine · 2026Article
- Piezo2 Mediates a Vicious Cycle of "Mechanical Homeostasis Imbalance-Inflammation" in Sensory Nerves and the Cartilage Endplate.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2026Article
- PIEZO1-mediated mechanosensation links aging to bladder dysfunction.bioRxiv : the preprint server for biology · 2026Article
- Piezo2-Initiated Ultrafast Signaling and Its Acquired Channelopathy in Light of Quantum Gravity Theory.International journal of molecular sciences · 2026Review
- Ether phospholipids modulate somatosensory responses by tuning multiple receptor functions iniScience · 2026Article
- MDFIC2 is a sensory neuron-specific PIEZO channel auxiliary subunit.Proceedings of the National Academy of Sciences of the United States of America · 2026Article
- Structures of invertebrate PEZO-1 isoforms with a compact architecture and a dispensable pore-distal N-terminal blade.Cell reports · 2026Article
- Biomolecules involved in the metabolism of Escherichia coli affected by photodynamics: A calorimetry study.PloS one · 2026Article
- Article
- PIEZO2 in tumors: from mechanobiological switches to activity-targeted therapies.Journal of experimental & clinical cancer research : CR · 2025Review
- MDFIC2 is a PIEZO channel modulator that can alleviate mechanical allodynia associated with neuropathic pain.Proceedings of the National Academy of Sciences of the United States of America · 2025Article
- Cofilin Inhibition Ameliorates PIEZO2 and AMPA Dysfunction in a Mouse Model of Angelman Syndrome.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2025Article
- Enhanced PIEZO1 function contributes to the pathogenesis of sickle cell disease.Proceedings of the National Academy of Sciences of the United States of America · 2025Article
- Courtship and distress ultrasonic vocalizations are altered in a mouse model of Angelman syndrome.Journal of neurodevelopmental disorders · 2025Article
- Influence of Olive Oil Components on Ion Channels.Molecules (Basel, Switzerland) · 2025Review
- Profiling human iPSC-derived sensory neurons for analgesic drug screening using a multi-electrode array.Cell reports methods · 2025Article
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Authors and funding
12 authors at 5 institutions in 3 countries.
Funding
Abstract
Angelman syndrome (AS) is a neurogenetic disorder characterized by intellectual disability and atypical behaviors. AS results from loss of expression of the E3 ubiquitin-protein ligase UBE3A from the maternal allele in neurons. Individuals with AS display impaired coordination, poor balance, and gait ataxia. PIEZO2 is a mechanosensitive ion channel essential for coordination and balance. Here, we report that PIEZO2 activity is reduced in Ube3a deficient male and female mouse sensory neurons, a human Merkel cell carcinoma cell line and female human iPSC-derived sensory neurons with UBE3A knock-down, and de-identified stem cell-derived neurons from individuals with AS. We find that loss of UBE3A decreases actin filaments and reduces PIEZO2 expression and function. A linoleic acid (LA)-enriched diet increases PIEZO2 activity, mechano-excitability, and improves gait in male AS mice. Finally, LA supplementation increases PIEZO2 function in stem cell-derived neurons from individuals with AS. We propose a mechanism whereby loss of UBE3A expression reduces PIEZO2 function and identified a fatty acid that enhances channel activity and ameliorates AS-associated mechano-sensory deficits.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.