ArticleNeuron2014
Loss of Dishevelleds disrupts planar polarity in ependymal motile cilia and results in hydrocephalus.
Article in Neuron, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 99 papers.
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Who cites it
99 citing papers in PubMed, 156 citations in OpenAlex.
- Dishevelled-mediated clustering stabilizes Frizzled6 and Vangl2 to establish planar cell polarity in the mammalian skin.Science advances · 2026Article
- Specialized Shh-sensing cell with unique cilia and basal body in the forebrain ventricular epithelium.bioRxiv : the preprint server for biology · 2026Article
- Cerebrospinal fluid-driven ependymal motile cilia defects are implicated in multiple sclerosis.Brain : a journal of neurology · 2026Article
- De NovoHuman mutation · 2026Article
- Evaluation of Interstitial Fluid Volume and Diffusivity in Patients With Idiopathic Normal Pressure Hydrocephalus Using Spectral Diffusion Analysis.Journal of magnetic resonance imaging : JMRI · 2025Article
- Ependymal and neural stem cells are close relatives.Stem cell reports · 2025Review
- Prdm16 regulates the postnatal fate of embryonic radial glia via Vcam1-dependent mechanisms.Nature communications · 2025Article
- Adherent junctions: Physiology, role in hydrocephalus and potential therapeutic targets.IBRO neuroscience reports · 2025Review
- Advances in Imaging Techniques for Mammalian/Human Ciliated Cell's Cilia: Insights into Structure, Function, and Dynamics.Biology · 2025Review
- A polarized multicomponent foundation upholds ciliary central microtubules.Journal of molecular cell biology · 2025Article
- Advancing Hydrocephalus Management: Pathogenesis Insights, Therapeutic Innovations, and Emerging Challenges.Aging and disease · 2024Review
- Directional ciliary beats across epithelia require Ccdc57-mediated coupling between axonemal orientation and basal body polarity.Nature communications · 2024Article
- Distinct roles of Kif6 and Kif9 in mammalian ciliary trafficking and motility.The Journal of cell biology · 2024Article
- Multiciliated ependymal cells: an update on biology and pathology in the adult brain.Acta neuropathologica · 2024Review
- Diffusion magnetic resonance imaging of cerebrospinal fluid dynamics: Current techniques and future advancements.NMR in biomedicine · 2024Review
- Pathogenic variants in autism geneProceedings of the National Academy of Sciences of the United States of America · 2024Article
- Modeling cerebrospinal fluid dynamics across the entire intracranial space through integration of four-dimensional flow and intravoxel incoherent motion magnetic resonance imaging.Fluids and barriers of the CNS · 2024Article
- Cerebral furin deficiency causes hydrocephalus in mice.Genes & diseases · 2024Article
- Intraventricular dimethyl sulfoxide (DMSO) induces hydrocephalus in a dose-dependent pattern.Heliyon · 2024Article
- Genetic and molecular mechanisms of hydrocephalus.Frontiers in molecular neuroscience · 2024Review
39 more citing papers are in PubMed but not listed here.
Corrections and comments
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Authors and funding
10 authors at 6 institutions in 3 countries.
Funding
Abstract
Defects in ependymal (E) cells, which line the ventricle and generate cerebrospinal fluid flow through ciliary beating, can cause hydrocephalus. Dishevelled genes (Dvls) are essential for Wnt signaling, and Dvl2 has been shown to localize to the rootlet of motile cilia. Using the hGFAP-Cre;Dvl1(-/-);2(flox/flox);3(+/-) mouse, we show that compound genetic ablation of Dvls causes hydrocephalus. In hGFAP-Cre;Dvl1(-/-);2(flox/flox);3(+/-) mutants, E cells differentiated normally, but the intracellular and intercellular rotational alignments of ependymal motile cilia were disrupted. As a consequence, the fluid flow generated by the hGFAP-Cre;Dvl1(-/-);2(flox/flox);3(+/-) E cells was significantly slower than that observed in control mice. Dvls were also required for the proper positioning of motile cilia on the apical surface. Tamoxifen-induced conditional removal of Dvls in adult mice also resulted in defects in intracellular rotational alignment and positioning of ependymal motile cilia. These results suggest that Dvls are continuously required for E cell planar polarity and may prevent hydrocephalus.
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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.