ArticleChild's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery2021
Ependymal ciliary motion and their role in congenital hydrocephalus.
Article in Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 22 papers.
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Who cites it
22 citing papers in PubMed, 34 citations in OpenAlex.
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- Patient-informed CRISPR Screen Identifies FLNB as a Novel Congenital Heart Disease and Ciliopathy Gene.bioRxiv : the preprint server for biology · 2025Article
- Emerging insights into primary ciliary dyskinesia-associated hydrocephalus: a scoping review.Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery · 2025Article
- Lineage trajectories and fate determinants of postnatal neural stem cells and ependymal cells in the developing ventricular zone.PLoS biology · 2025Article
- Age-related cerebral ventriculomegaly occurs in patients with primary ciliary dyskinesia.Fluids and barriers of the CNS · 2025Article
- LRRC56 deficiency cause motile ciliopathies in humans and mice.Frontiers in genetics · 2025Article
- Loss ofCells · 2024Article
- Cases of familial idiopathic normal pressure hydrocephalus implicate genetic factors in disease pathogenesis.Cerebral cortex (New York, N.Y. : 1991) · 2023Review
- Novel dominant-negativeTranslational pediatrics · 2023Article
- Distinct dynein complexes defined by DYNLRB1 and DYNLRB2 regulate mitotic and male meiotic spindle bipolarity.Nature communications · 2023Article
- Molecular genetics of human developmental neurocranial anomalies: towards "precision surgery".Cerebral cortex (New York, N.Y. : 1991) · 2023Article
- Congenital hydrocephalus: new Mendelian mutations and evidence for oligogenic inheritance.Human genomics · 2023Article
- Current Neurosurgery.Juntendo Iji zasshi = Juntendo medical journal · 2023Review
- History of research concerning the ependyma: a view from inside the human brain.Frontiers in cellular neuroscience · 2023Review
- The diagnostic yield, candidate genes, and pitfalls for a genetic study of intellectual disability in 118 middle eastern families.Scientific reports · 2022Article
- Laparoscopic bladder diverticulectomy in a child with situs inversus totalis: A case report and literature review.Frontiers in surgery · 2022Article
- Tsukushi proteoglycan maintains RNA splicing and developmental signaling network in GFAP-expressing subventricular zone neural stem/progenitor cells.Frontiers in cell and developmental biology · 2022Article
Corrections and comments
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Authors and funding
8 authors at 2 institutions in 1 country.
Funding
Abstract
purposeSince a case of hydrocephalus in humans considered to be caused by ciliary dysfunction was first reported by Greenstone et al. in 1984, numerous papers on the correlation between ciliary function and hydrocephalus have been published.
methodsWe reviewed the published literature on primary ciliary dyskinesia in humans causing hydrocephalus, focusing on articles specifically examining the relation between ciliary function and hydrocephalus and its treatment. In addition, the authors' experience is briefly discussed.
resultsFull texts of 16 articles reporting cases of human hydrocephalus (including ventriculomegaly) due to defects in ependymal ciliary function or primary ciliary dyskinesia observed in clinical practice were extracted. In recent years, studies on animal models, especially employing knockout mice, have revealed genetic mutations that cause hydrocephalus via ciliary dysfunction. However, a few reports on the onset of hydrocephalus in human patients with primary ciliary dyskinesia have confirmed that the incidence of this condition was extremely low compared to that in animal models.
conclusionIn humans, it is rare for hydrocephalus to develop solely because of abnormalities in the cilia, and it is highly likely that other factors are also involved along with ciliary dysfunction.
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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.