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Each MCC contains dozens to hundreds of motile cilia that beat in a synchronized manner to drive fluid flow across epithelia, the dysfunction of which is associated with a group of human diseases referred to as motile ciliopathies, such as primary cilia dyskinesia. Given the dynamic and complex process of multiciliogenesis, the biological events essential for forming multiple motile cilia are comparatively unelucidated. Thanks to advancements in genetic tools, omics technologies, and structural biology, significant progress has been achieved in the past decade in understanding the molecular mechanism underlying the regulation of multiple motile cilia formation. In this review, we discuss recent studies with ex vivo culture MCC and animal models, summarize current knowledge of multiciliogenesis, and particularly highlight recent advances and their implications.<\/jats:p>","DOI":"10.1083\/jcb.202307150","type":"journal-article","created":{"date-parts":[[2023,11,15]],"date-time":"2023-11-15T02:21:31Z","timestamp":1700014891000},"update-policy":"https:\/\/doi.org\/10.1083\/jcb.crossmarkpolicy","source":"Crossref","is-referenced-by-count":68,"title":["Formation and function of multiciliated cells"],"prefix":"10.1083","volume":"223","author":[{"ORCID":"https:\/\/orcid.org\/0009-0002-0198-6433","authenticated-orcid":true,"given":"Qian","family":"Lyu","sequence":"first","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01wy3h363","id-type":"ROR","asserted-by":"publisher"}],"name":"Shandong Normal University 1 Center for Cell Structure and Function, Shandong Provincial Key Laboratory of Animal Resistance Biology, Collaborative Innovation Center of Cell Biology in Universities of Shandong, College of Life Sciences, , Jinan, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0307-0085","authenticated-orcid":true,"given":"Qingchao","family":"Li","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01wy3h363","id-type":"ROR","asserted-by":"publisher"}],"name":"Shandong Normal University 1 Center for Cell Structure and Function, Shandong Provincial Key Laboratory of Animal Resistance Biology, Collaborative Innovation Center of Cell Biology in Universities of Shandong, College of Life Sciences, , Jinan, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3131-7804","authenticated-orcid":true,"given":"Jun","family":"Zhou","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01wy3h363","id-type":"ROR","asserted-by":"publisher"}],"name":"Shandong Normal University 1 Center for Cell Structure and Function, Shandong Provincial Key Laboratory of Animal Resistance Biology, Collaborative Innovation Center of Cell Biology in Universities of Shandong, College of Life Sciences, , Jinan, China"},{"name":"Nankai University 2 State Key Laboratory of Medicinal Chemical Biology, Haihe Laboratory of Cell Ecosystem, College of Life Sciences, , Tianjin, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8595-8159","authenticated-orcid":true,"given":"Huijie","family":"Zhao","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01wy3h363","id-type":"ROR","asserted-by":"publisher"}],"name":"Shandong Normal University 1 Center for Cell Structure and Function, Shandong Provincial Key Laboratory of Animal Resistance Biology, Collaborative Innovation Center of Cell Biology in Universities of Shandong, College of Life Sciences, , Jinan, China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"291","published-online":{"date-parts":[[2023,11,30]]},"reference":[{"key":"2023113013595825400_bib1","doi-asserted-by":"publisher","first-page":"104","DOI":"10.1038\/nature13770","article-title":"Centriole amplification by mother and daughter centrioles differs in multiciliated cells","volume":"516","author":"Al Jord","year":"2014","journal-title":"Nature"},{"key":"2023113013595825400_bib2","doi-asserted-by":"publisher","first-page":"803","DOI":"10.1126\/science.aan8311","article-title":"Calibrated mitotic oscillator drives motile ciliogenesis","volume":"358","author":"Al Jord","year":"2017","journal-title":"Science"},{"key":"2023113013595825400_bib3","doi-asserted-by":"publisher","first-page":"10","DOI":"10.1083\/jcb.50.1.10","article-title":"The formation of basal bodies (centrioles) in the Rhesus monkey oviduct","volume":"50","author":"Anderson","year":"1971","journal-title":"J. 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