{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,5]],"date-time":"2026-09-05T14:17:03Z","timestamp":1788617823736,"version":"build-2803163510"},"reference-count":33,"publisher":"The Company of Biologists","issue":"2","license":[{"start":{"date-parts":[[2024,2,29]],"date-time":"2024-02-29T00:00:00Z","timestamp":1709164800000},"content-version":"vor","delay-in-days":28,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"name":"Eunice Kennedy Shriver National Institute of Child Health and Human Development","award":["R01HD085901"],"award-info":[{"award-number":["R01HD085901"]}]},{"DOI":"10.13039\/100000050","name":"National Heart, Lung, and Blood Institute","doi-asserted-by":"crossref","award":["R01HL117164"],"award-info":[{"award-number":["R01HL117164"]}],"id":[{"id":"10.13039\/100000050","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"crossref"}]},{"name":"The University of Texas at Austin"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,2,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Motile cilia on ependymal cells that line brain ventricular walls beat in concert to generate a flow of laminar cerebrospinal fluid (CSF). Dyneins and kinesins are ATPase microtubule motor proteins that promote the rhythmic beating of cilia axonemes. Despite common consensus about the importance of axonemal dynein motor proteins, little is known about how kinesin motors contribute to cilia motility. Here, we show that Kif6 is a slow processive motor (12.2\u00b12.0\u2005nm\/s) on microtubules in vitro and localizes to both the apical cytoplasm and the axoneme in ependymal cells, although it does not display processive movement in vivo. Using a mouse mutant that models a human Kif6 mutation in a proband displaying macrocephaly, hypotonia and seizures, we found that loss of Kif6 function causes decreased ependymal cilia motility and, subsequently, decreases fluid flow on the surface of brain ventricular walls. Disruption of Kif6 also disrupts orientation of cilia, formation of robust apical actin networks and stabilization of basal bodies at the apical surface. This suggests a role for the Kif6 motor protein in the maintenance of ciliary homeostasis within ependymal cells.<\/jats:p>","DOI":"10.1242\/dmm.050137","type":"journal-article","created":{"date-parts":[[2024,1,18]],"date-time":"2024-01-18T04:18:23Z","timestamp":1705551503000},"source":"Crossref","is-referenced-by-count":16,"title":["Motor protein Kif6 regulates cilia motility and polarity in brain ependymal cells"],"prefix":"10.1242","volume":"17","author":[{"given":"Maki","family":"Takagishi","sequence":"first","affiliation":[{"name":"The University of Texas at Austin 1 Department of Molecular Biosciences, Patterson Labs , , TX 78712 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yang","family":"Yue","sequence":"additional","affiliation":[{"name":"University of Michigan Medical School 2 Department of Cell and Developmental Biology , , Ann Arbor, MI 48109 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9668-6497","authenticated-orcid":false,"given":"Ryan S.","family":"Gray","sequence":"additional","affiliation":[{"name":"Dell Pediatric Research Institute, The University of Texas at Austin, Dell Medical School 3 Departments of Nutrition and Pediatrics , , Austin, TX 78712 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9329-4981","authenticated-orcid":false,"given":"Kristen J.","family":"Verhey","sequence":"additional","affiliation":[{"name":"University of Michigan Medical School 2 Department of Cell and Developmental Biology , , Ann Arbor, MI 48109 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6280-8625","authenticated-orcid":false,"given":"John B.","family":"Wallingford","sequence":"additional","affiliation":[{"name":"The University of Texas at Austin 1 Department of Molecular Biosciences, Patterson Labs , , TX 78712 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2024,2,29]]},"reference":[{"key":"2024022914354377200_DMM050137C1","doi-asserted-by":"publisher","first-page":"1552","DOI":"10.1016\/j.jacc.2010.06.022","article-title":"Lack of association between the Trp719Arg polymorphism in kinesin-like protein-6 and coronary artery disease in 19 case-control studies","volume":"56","author":"Assimes","year":"2010","journal-title":"AJ. 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