ArticleFrontiers in physiology2022
Changes in Blood Cell Deformability in Chorea-Acanthocytosis and Effects of Treatment With Dasatinib or Lithium.
Article in Frontiers in physiology, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 15 papers.
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15 citing papers in PubMed, 20 citations in OpenAlex.
- High-throughput mechanomic screening reveals novel regulators of single-cell mechanics.Biophysical journal · 2026Article
- Impact of red blood cell rigidity on in vivo flow dynamics and lingering in bifurcations.Biophysical journal · 2026Article
- Chorein Regulates Key Osteoblast Genes in UMR-106 Cells.Cell biochemistry and biophysics · 2026Article
- Red blood cell lipid distribution in the pathophysiology and laboratory evaluation of chorea-acanthocytosis and McLeod syndrome patients.Frontiers in physiology · 2025Article
- Physiological and Pathogenesis Significance of Chorein in Health and Disease.Physiological research · 2024Review
- Confinement effect on the microcapillary flow and shape of red blood cells.Biomicrofluidics · 2024Article
- Mechano-regulation by clathrin pit-formation and passive cholesterol-dependent tubules during de-adhesion.Cellular and molecular life sciences : CMLS · 2024Article
- A case of chorea-acanthocytosis with significant improvement of symptoms at one year with deep brain stimulation: case report and literature review.Frontiers in neurology · 2024Article
- Osmotic gradient ektacytometry - a novel diagnostic approach for neuroacanthocytosis syndromes.Frontiers in neuroscience · 2024Article
- Case report: Neuroacanthocytosis associated with novel variants in theFrontiers in neuroscience · 2024Article
- Article
- High-throughput mechano-cytometry as a method to detect apoptosis, necroptosis, and ferroptosis.Cell proliferation · 2023Article
- Competition between deformation and free volume quantified by 3D image analysis of red blood cell.Biophysical journal · 2023Article
- Red blood cell lingering modulates hematocrit distribution in the microcirculation.Biophysical journal · 2023Article
- Early tension regulation coupled to surface myomerger is necessary for the primary fusion of C2C12 myoblasts.Frontiers in physiology · 2022Article
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12 authors at 6 institutions in 2 countries.
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No grant is acknowledged in the PubMed record.
Abstract
Misshaped red blood cells (RBCs), characterized by thorn-like protrusions known as acanthocytes, are a key diagnostic feature in Chorea-Acanthocytosis (ChAc), a rare neurodegenerative disorder. The altered RBC morphology likely influences their biomechanical properties which are crucial for the cells to pass the microvasculature. Here, we investigated blood cell deformability of five ChAc patients compared to healthy controls during up to 1-year individual off-label treatment with the tyrosine kinase inhibitor dasatinib or several weeks with lithium. Measurements with two microfluidic techniques allowed us to assess RBC deformability under different shear stresses. Furthermore, we characterized leukocyte stiffness at high shear stresses. The results showed that blood cell deformability-including both RBCs and leukocytes - in general was altered in ChAc patients compared to healthy donors. Therefore, this study shows for the first time an impairment of leukocyte properties in ChAc. During treatment with dasatinib or lithium, we observed alterations in RBC deformability and a stiffness increase for leukocytes. The hematological phenotype of ChAc patients hinted at a reorganization of the cytoskeleton in blood cells which partly explains the altered mechanical properties observed here. These findings highlight the need for a systematic assessment of the contribution of impaired blood cell mechanics to the clinical manifestation of ChAc.
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