{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,25]],"date-time":"2026-09-25T18:55:33Z","timestamp":1790362533432,"version":"4.1.0"},"reference-count":20,"publisher":"Wiley","issue":"7","license":[{"start":{"date-parts":[[2016,6,28]],"date-time":"2016-06-28T00:00:00Z","timestamp":1467072000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100001695","name":"Japan Science and Technology Corporation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100001695","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001700","name":"Ministry of Education, Culture, Sports, Science and Technology","doi-asserted-by":"publisher","award":["13J04993"],"award-info":[{"award-number":["13J04993"]}],"id":[{"id":"10.13039\/501100001700","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001700","name":"Ministry of Education, Culture, Sports, Science and Technology","doi-asserted-by":"publisher","award":["24113004"],"award-info":[{"award-number":["24113004"]}],"id":[{"id":"10.13039\/501100001700","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Genes to Cells"],"published-print":{"date-parts":[[2016,7]]},"abstract":"<jats:p>Lrrc6 encodes a cytoplasmic protein that is expressed specifically in cells with motile cilia including the node, trachea and testes of the mice. A mutation of Lrrc6 has been identified in human patients with primary ciliary dyskinesia (<jats:styled-content style=\"fixed-case\">PCD<\/jats:styled-content>). Mutant mice lacking Lrrc6 show typical <jats:styled-content style=\"fixed-case\">PCD<\/jats:styled-content> defects such as hydrocephalus and laterality defects. We found that in the absence of Lrrc6, the morphology of motile cilia remained normal, but their motility was completely lost. The 9\u00a0+\u00a02 arrangement of microtubules remained normal in <jats:italic>Lrrc6<jats:sup>\u2010\/\u2010<\/jats:sup><\/jats:italic> mice, but the outer dynein arms (<jats:styled-content style=\"fixed-case\">ODA<\/jats:styled-content>s), the structures essential for the ciliary beating, were absent from the cilia. In the absence of Lrrc6, <jats:styled-content style=\"fixed-case\">ODA<\/jats:styled-content> proteins such as <jats:styled-content style=\"fixed-case\">DNAH<\/jats:styled-content>5, <jats:styled-content style=\"fixed-case\">DNAH<\/jats:styled-content>9 and <jats:styled-content style=\"fixed-case\">IC<\/jats:styled-content>2, which are assembled in the cytoplasm and transported to the ciliary axoneme, remained in the cytoplasm and were not transported to the ciliary axoneme. The <jats:styled-content style=\"fixed-case\">IC<\/jats:styled-content>2\u2013<jats:styled-content style=\"fixed-case\">IC<\/jats:styled-content>1 interaction, which is the first step of <jats:styled-content style=\"fixed-case\">ODA<\/jats:styled-content> assembly, was normal in <jats:italic>Lrrc6<\/jats:italic><jats:sup>\u2212\/\u2212<\/jats:sup> mice testes. Our results suggest that <jats:styled-content style=\"fixed-case\">ODA<\/jats:styled-content> proteins may be transported from the cytoplasm to the cilia by an Lrrc6\u2010dependent mechanism.<\/jats:p>","DOI":"10.1111\/gtc.12380","type":"journal-article","created":{"date-parts":[[2016,6,29]],"date-time":"2016-06-29T02:26:48Z","timestamp":1467167208000},"page":"728-739","source":"Crossref","is-referenced-by-count":35,"title":["Transport of the outer dynein arm complex to cilia requires a cytoplasmic protein Lrrc6"],"prefix":"10.1111","volume":"21","author":[{"given":"Yasuko","family":"Inaba","sequence":"first","affiliation":[{"name":"Developmental Genetics Group Graduate School of Frontier Biosciences Osaka University, and CREST Japan Science and Technology Corporation (JST) 1\u20103 Yamada\u2010oka Suita Osaka 565\u20100871 Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kyosuke","family":"Shinohara","sequence":"additional","affiliation":[{"name":"Developmental Genetics Group Graduate School of Frontier Biosciences Osaka University, and CREST Japan Science and Technology Corporation (JST) 1\u20103 Yamada\u2010oka Suita Osaka 565\u20100871 Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yanick","family":"Botilde","sequence":"additional","affiliation":[{"name":"Developmental Genetics Group Graduate School of Frontier Biosciences Osaka University, and CREST Japan Science and Technology Corporation (JST) 1\u20103 Yamada\u2010oka Suita Osaka 565\u20100871 Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ryo","family":"Nabeshima","sequence":"additional","affiliation":[{"name":"Developmental Genetics Group Graduate School of Frontier Biosciences Osaka University, and CREST Japan Science and Technology Corporation (JST) 1\u20103 Yamada\u2010oka Suita Osaka 565\u20100871 Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katsuyoshi","family":"Takaoka","sequence":"additional","affiliation":[{"name":"Developmental Genetics Group Graduate School of Frontier Biosciences Osaka University, and CREST Japan Science and Technology Corporation (JST) 1\u20103 Yamada\u2010oka Suita Osaka 565\u20100871 Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rieko","family":"Ajima","sequence":"additional","affiliation":[{"name":"Developmental Genetics Group Graduate School of Frontier Biosciences Osaka University, and CREST Japan Science and Technology Corporation (JST) 1\u20103 Yamada\u2010oka Suita Osaka 565\u20100871 Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lynda","family":"Lamri","sequence":"additional","affiliation":[{"name":"Developmental Genetics Group Graduate School of Frontier Biosciences Osaka University, and CREST Japan Science and Technology Corporation (JST) 1\u20103 Yamada\u2010oka Suita Osaka 565\u20100871 Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiroyuki","family":"Takeda","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences Graduate School of Science University of Tokyo Hongo Bunkyo\u2010ku Tokyo 113\u20100033 Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yumiko","family":"Saga","sequence":"additional","affiliation":[{"name":"Division of Mammalian Development Genetic Strains Research Center National Institute of Genetics Yata 1111 Mishima Shizuoka 411\u20108540 Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tetsuya","family":"Nakamura","sequence":"additional","affiliation":[{"name":"Developmental Genetics Group Graduate School of Frontier Biosciences Osaka University, and CREST Japan Science and Technology Corporation (JST) 1\u20103 Yamada\u2010oka Suita Osaka 565\u20100871 Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiroshi","family":"Hamada","sequence":"additional","affiliation":[{"name":"Developmental Genetics Group Graduate School of Frontier Biosciences Osaka University, and CREST Japan Science and Technology Corporation (JST) 1\u20103 Yamada\u2010oka Suita Osaka 565\u20100871 Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2016,6,28]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2013.08.015"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pgen.1004577"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.201304076"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.1091\/mbc.9.9.2337"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1101\/cshperspect.a000802"},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrm3085"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2008.03.010"},{"key":"e_1_2_6_9_1","first-page":"L736","article-title":"Mutations in SPAG1 cause primary ciliary dyskinesia associated with defective outer and inner dynein arms","volume":"304","author":"Knowles M.R.","year":"2013","journal-title":"Am. J. Physiol. Lung Cell. Mol. Physiol."},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2009.10.018"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1152\/ajplung.00425.2012"},{"key":"e_1_2_6_12_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.1106"},{"key":"e_1_2_6_13_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature00849"},{"key":"e_1_2_6_14_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature07471"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.2277"},{"key":"e_1_2_6_16_1","doi-asserted-by":"publisher","DOI":"10.1101\/gad.13.3.259"},{"key":"e_1_2_6_17_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.01240"},{"key":"e_1_2_6_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2008.03.020"},{"key":"e_1_2_6_19_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.2707"},{"key":"e_1_2_6_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2013.06.007"},{"key":"e_1_2_6_21_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1300968110"}],"container-title":["Genes to Cells"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fgtc.12380","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fgtc.12380","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/gtc.12380","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,3]],"date-time":"2023-10-03T11:31:08Z","timestamp":1696332668000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/gtc.12380"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2016,6,28]]},"references-count":20,"journal-issue":{"issue":"7","published-print":{"date-parts":[[2016,7]]}},"alternative-id":["10.1111\/gtc.12380"],"URL":"https:\/\/doi.org\/10.1111\/gtc.12380","archive":["Portico"],"relation":{},"ISSN":["1356-9597","1365-2443"],"issn-type":[{"value":"1356-9597","type":"print"},{"value":"1365-2443","type":"electronic"}],"subject":[],"published":{"date-parts":[[2016,6,28]]}}}