Ependymal ciliary motion and their role in congenital hydrocephalus View Full Text


Ontology type: schema:ScholarlyArticle      Open Access: True


Article Info

DATE

2021-05-17

AUTHORS

Koichiro Sakamoto, Madoka Nakajima, Kaito Kawamura, Eri Nakamura, Norihiro Tada, Akihide Kondo, Hajime Arai, Masakazu Miyajima

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. More... »

PAGES

3355-3364

Identifiers

URI

http://scigraph.springernature.com/pub.10.1007/s00381-021-05194-9

DOI

http://dx.doi.org/10.1007/s00381-021-05194-9

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1138109610

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/33999288


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