ArticleProceedings of the National Academy of Sciences of the United States of America2013
Xerocytosis is caused by mutations that alter the kinetics of the mechanosensitive channel PIEZO1.
Article in Proceedings of the National Academy of Sciences of the United States of America, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 187 papers.
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Who cites it
187 citing papers in PubMed, 313 citations in OpenAlex.
- State-dependent binding of the wedge domain controls inactivation of the mechanosensitive ion channel PIEZO1.Nature communications · 2026Article
- Sono-Mechanogenetics: Linking Ultrasound Physics With Cellular Mechanobiology.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2026Review
- Yoda molecules agonize PIEZO2.bioRxiv : the preprint server for biology · 2026Article
- Stretch, Piezo channels, and store operated calcium entry in developing human airway smooth muscle.Pediatric research · 2026Article
- Deciphering gain-of-function from loss-of-function variants with AlphaMissense: A case study with the mechanosensitive PIEZO1 ion channel protein.Biochemistry and biophysics reports · 2026Article
- Structures of invertebrate PEZO-1 isoforms with a compact architecture and a dispensable pore-distal N-terminal blade.Cell reports · 2026Article
- PIEZO1 in Immune Cells: From Force to Function.Results and problems in cell differentiation · 2026Review
- Blood Mechanical Intelligence: From Force Sensing to Precision Mechanomedicine.Research (Washington, D.C.) · 2026Review
- 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
- Insulin facilitates entry of calcium ions into human and murine erythrocytes via Piezo1: a newly identified mechanism with implications for type 2 diabetes.The FEBS journal · 2025Article
- Piezo1 is an essential player in volume regulation of human glioblastoma cells.The Journal of physiology · 2025Article
- Mechanical memory of excitable cells induced by mechanosensitive channel inactivation.Biophysical journal · 2025Article
- Mechanical stretch promotes sustained proliferation and inflammation in developing human airway smooth muscle.American journal of physiology. Lung cellular and molecular physiology · 2025Article
- Article
- Visualizing PIEZO1 localization and activity in hiPSC-derived single cells and organoids with HaloTag technology.Nature communications · 2025Article
- Review
- Targeted elimination of mesenchymal-like cancer cells through cyclic stretch activation of Piezo1 channels: the physical aspects.Biophysical reviews · 2025Review
- PIEZO channels as multimodal mechanotransducers.Biochemical Society transactions · 2025Review
- Visualizing PIEZO1 Localization and Activity in hiPSC-Derived Single Cells and Organoids with HaloTag Technology.bioRxiv : the preprint server for biology · 2025Article
- RAS signaling pathway is essential in regulating PIEZO1-mediated hepatic iron overload in dehydrated hereditary stomatocytosis.American journal of hematology · 2025Article
127 more citing papers are in PubMed but not listed here.
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Authors and funding
5 authors at 2 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Familial xerocytosis (HX) in humans is an autosomal disease that causes dehydration of red blood cells resulting in hemolytic anemia which has been traced to two individual mutations in the mechanosensitive ion channel, PIEZO1. Each mutation alters channel kinetics in ways that can explain the clinical presentation. Both mutations slowed inactivation and introduced a pronounced latency for activation. A conservative substitution of lysine for arginine (R2456K) eliminated inactivation and also slowed deactivation, indicating that this mutant's loss of charge is not responsible for HX. Fitting the current vs. pressure data to Boltzmann distributions showed that the half-activation pressure, P1/2, for M2225R was similar to that of WT, whereas mutations at position 2456 were left shifted. The absolute stress sensitivity was calibrated by cotransfection and comparison with MscL, a well-characterized mechanosensitive channel from bacteria that is driven by bilayer tension. The slope sensitivity of WT and mutant human PIEZO1 (hPIEZO1) was similar to that of MscL implying that the in-plane area increased markedly, by ∼6-20 nm(2) during opening. In addition to the behavior of individual channels, groups of hPIEZO1 channels could undergo simultaneous changes in kinetics including a loss of inactivation and a long (∼200 ms), silent latency for activation. These observations suggest that hPIEZO1 exists in spatial domains whose global properties can modify channel gating. The mutations that create HX affect cation fluxes in two ways: slow inactivation increases the cation flux, and the latency decreases it. These data provide a direct link between pathology and mechanosensitive channel dysfunction in nonsensory cells.
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