{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,16]],"date-time":"2026-06-16T01:15:55Z","timestamp":1781572555564,"version":"3.54.5"},"reference-count":31,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2025,7,28]],"date-time":"2025-07-28T00:00:00Z","timestamp":1753660800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2025,7,28]],"date-time":"2025-07-28T00:00:00Z","timestamp":1753660800000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["17K17792"],"award-info":[{"award-number":["17K17792"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["JP16H06280"],"award-info":[{"award-number":["JP16H06280"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["22K12780"],"award-info":[{"award-number":["22K12780"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Cytoskeleton"],"published-print":{"date-parts":[[2026,5]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    Primary ciliary dyskinesia (PCD) is a congenital disease caused by gene mutations linked to ciliary dysfunction. PCD causes different symptoms, including chronic sinusitis, infertility, situs inversus and hydrocephalus. Motile cilia on ventricular ependymal cells are a crucial factor in cerebrospinal fluid circulation, and dysfunction of these cells causes hydrocephalus. Deleted in primary ciliary dyskinesia (\n                    <jats:italic>Dpcd<\/jats:italic>\n                    ) is one genetic abnormality known to cause PCD, and its knockout leads to hydrocephalus in mice. PCD occurs in\n                    <jats:italic>Dpcd<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    mice because of the lack of an inner dynein arm (IDA) in the motile cilia. However, how this deficiency is associated with the motility of ventricular ependymal motile cilia in\n                    <jats:italic>Dpcd<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    mice has not been demonstrated. Herein, we show that\n                    <jats:italic>Dpcd<\/jats:italic>\n                    induces partial defects in dyneins and aberrant motility in ventricular ependymal cilia. In\n                    <jats:italic>Dpcd<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    mice, the ependymal cilia demonstrated decreased amplitude, abnormal waveforms and low cerebrospinal fluid flow velocity. In addition, the amount of dynein axonemal heavy chains in some IDAs decreased in the ependymal cilia. In wild\u2010type mice, Dpcd was localised in the cytoplasm and cilia of ependymal cells. Thus, abnormal ciliary movement in\n                    <jats:italic>Dpcd<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    mice is likely attributed to a defect in IDA assembly in the ependymal cilia.\n                  <\/jats:p>","DOI":"10.1002\/cm.70012","type":"journal-article","created":{"date-parts":[[2025,7,28]],"date-time":"2025-07-28T13:07:09Z","timestamp":1753708029000},"page":"237-249","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Dpcd Induces Hydrocephalus Because of Partial Defects in the Inner Dynein Arms, With Abnormal Ciliary Motility"],"prefix":"10.1002","volume":"83","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-9734-4931","authenticated-orcid":false,"given":"Taiki","family":"Yamamoto","sequence":"first","affiliation":[{"name":"Department of Neurosurgery Nagoya University  Nagoya Aichi Japan"},{"name":"Department of Neurosurgery Gifu Prefectural Tajimi Hospital  Tajimi Gifu Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kazuhito","family":"Takeuchi","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Nagoya University  Nagoya Aichi Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuichi","family":"Nagata","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Nagoya University  Nagoya Aichi Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Akihiro","family":"Mizuno","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Komaki City Hospital  Komaki Aichi Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hideyuki","family":"Harada","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Tosei General Hospital  Seto Aichi Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Takayuki","family":"Ishikawa","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Japanese Red Cross Aichi Medical Center Nagoya Daini Hospital  Nagoya Aichi Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sachi","family":"Maeda","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Nagoya University  Nagoya Aichi Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fumiharu","family":"Ohka","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Nagoya University  Nagoya Aichi Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9419-398X","authenticated-orcid":false,"given":"Ryuji","family":"Yanase","sequence":"additional","affiliation":[{"name":"Shimoda Marine Research Center University of Tsukuba  Shizuoka Japan"},{"name":"School of Life Sciences, Queen's Medical Centre University of Nottingham  Nottingham UK"},{"name":"Department of Genetics and Genome Biology, College of Life Sciences University of Leicester  Leicester UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7742-8158","authenticated-orcid":false,"given":"Kogiku","family":"Shiba","sequence":"additional","affiliation":[{"name":"Shimoda Marine Research Center University of Tsukuba  Shizuoka Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hironori","family":"Ueno","sequence":"additional","affiliation":[{"name":"Natural Science, Aichi University of Education  Kariya Aichi Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8848-9733","authenticated-orcid":false,"given":"Kazuo","family":"Inaba","sequence":"additional","affiliation":[{"name":"Shimoda Marine Research Center University of Tsukuba  Shizuoka Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2484-1360","authenticated-orcid":false,"given":"Ryuta","family":"Saito","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Nagoya University  Nagoya Aichi Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2025,7,28]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.154500"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.3390\/cells10081885"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2013.11.017"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/cm.970080110"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jmb.2022.167760"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nbd.2022.105913"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.aae0450"},{"key":"e_1_2_9_9_1","first-page":"271","article-title":"Electron Microscopic Studies of Spermatozoa","volume":"2","author":"Grigg G. 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