ArticlePLoS biology2023
Ependymal polarity defects coupled with disorganized ciliary beating drive abnormal cerebrospinal fluid flow and spine curvature in zebrafish.
Article in PLoS biology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.
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Who cites it
18 citing papers in PubMed, 42 citations in OpenAlex.
- The Role of Microfilaments in Cilia Formation and Function.Cytoskeleton (Hoboken, N.J.) · 2026Review
- RGS22 is a metazoa-specific radial spoke component required for coordinated ciliary beating.Nature communications · 2026Article
- UFL1-mediated UFMylation antagonizes IFT88 ubiquitination and degradation to maintain ciliary homeostasis.Cell death and differentiation · 2026Article
- Ccdc57 regulates cilia and left-right patterning in Xenopus.Biology open · 2026Article
- Tulp3 deficiency results in ciliopathy phenotypes during zebrafish embryogenesis.Scientific reports · 2025Article
- C-mannosyltransferase DPY19L1L-mediated Reissner Fiber formation is critical for zebrafish (Science advances · 2025Article
- Toxic endpoints or ubiquitous expression?Toxicology research · 2025Article
- Directional ciliary beats across epithelia require Ccdc57-mediated coupling between axonemal orientation and basal body polarity.Nature communications · 2024Article
- Review
- Article
- Multiciliated ependymal cells: an update on biology and pathology in the adult brain.Acta neuropathologica · 2024Review
- Decoding pulsatile patterns of cerebrospinal fluid dynamics through enhancing interpretability in machine learning.Scientific reports · 2024Article
- Pyriproxyfen, villain or good guy? A brief review.Archives of endocrinology and metabolism · 2024Review
- SCO-spondin knockout mice exhibit small brain ventricles and mild spine deformation.Fluids and barriers of the CNS · 2023Article
- The role of cilia during organogenesis in zebrafish.Open biology · 2023Review
- Article
- Cerebrospinal fluid-contacting neurons: multimodal cells with diverse roles in the CNS.Nature reviews. Neuroscience · 2023Review
- Loss of CSF-contacting neuron sensory function is associated with a hyper-kyphosis of the spine reminiscent of Scheuermann's disease.Scientific reports · 2023Article
Corrections and comments
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Authors and funding
18 authors at 6 institutions in 2 countries.
Funding
Abstract
Idiopathic scoliosis (IS) is the most common spinal deformity diagnosed in childhood or early adolescence, while the underlying pathogenesis of this serious condition remains largely unknown. Here, we report zebrafish ccdc57 mutants exhibiting scoliosis during late development, similar to that observed in human adolescent idiopathic scoliosis (AIS). Zebrafish ccdc57 mutants developed hydrocephalus due to cerebrospinal fluid (CSF) flow defects caused by uncoordinated cilia beating in ependymal cells. Mechanistically, Ccdc57 localizes to ciliary basal bodies and controls the planar polarity of ependymal cells through regulating the organization of microtubule networks and proper positioning of basal bodies. Interestingly, ependymal cell polarity defects were first observed in ccdc57 mutants at approximately 17 days postfertilization, the same time when scoliosis became apparent and prior to multiciliated ependymal cell maturation. We further showed that mutant spinal cord exhibited altered expression pattern of the Urotensin neuropeptides, in consistent with the curvature of the spine. Strikingly, human IS patients also displayed abnormal Urotensin signaling in paraspinal muscles. Altogether, our data suggest that ependymal polarity defects are one of the earliest sign of scoliosis in zebrafish and disclose the essential and conserved roles of Urotensin signaling during scoliosis progression.
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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.