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Recently, phosphoinositides were shown to play an important role in cilia assembly and disassembly. However, the precise molecular and cellular mechanisms underlying this process remain unknown. Here, we report that suppression of apical phosphatidylinositol 4,5\u2010\n                    <jats:italic>bis<\/jats:italic>\n                    phosphate [PtdIns(4,5)P\n                    <jats:sub>2<\/jats:sub>\n                    ], by overexpressing apically targeted PtdIns(4,5)P\n                    <jats:sub>2<\/jats:sub>\n                    phosphatase or by knocking down type I phosphatidylinositol 4\u2010phosphate 5\u2010kinase (Pip5k1), leads to ciliogenesis defects and polycystic kidney disease (PKD) in zebrafish embryos that phenocopied\n                    <jats:italic>inpp5e<\/jats:italic>\n                    mutant, a Joubert syndrome model. We further demonstrate that decreased expression of apical PtdIns(4,5)P\n                    <jats:sub>2<\/jats:sub>\n                    disrupted apical ezrin recruitment, F\u2010actin organization, and basal body docking. Moreover, the ciliogenesis and polycystic kidney defects in PtdIns(4,5)P\n                    <jats:sub>2<\/jats:sub>\n                    \u2010depleted embryos can be rescued by overexpression of ezrin. Finally, Pip5k1a overexpression rescued the ciliogenesis defects and PKD phenotypes in Inpp5e\u2010depleted embryos. Taken together, our results reveal that apical PtdIns(4,5)P\n                    <jats:sub>2<\/jats:sub>\n                    is essential for ciliogenesis and the prevention of PKD and suggest a novel possibility for treating PKD and other human ciliopathies.\u2014Xu, W., Jin, M., Huang, W., Wang, H., Hu, R., Li, J., Cao, Y. Apical PtdIns(4,5)P\n                    <jats:sub>2<\/jats:sub>\n                    is required for ciliogenesis and suppression of polycystic kidney disease. FASEB J. 33, 2848\u20132857 (2019).\n                    <jats:ext-link xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" xlink:href=\"http:\/\/www.fasebj.org\">www.fasebj.org<\/jats:ext-link>\n                  <\/jats:p>","DOI":"10.1096\/fj.201800385rrr","type":"journal-article","created":{"date-parts":[[2018,10,15]],"date-time":"2018-10-15T14:08:58Z","timestamp":1539612538000},"page":"2848-2857","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":17,"title":["Apical PtdIns(4,5)P\n                    <sub>2<\/sub>\n                    is required for ciliogenesis and suppression of polycystic kidney disease"],"prefix":"10.1096","volume":"33","author":[{"given":"Wenyan","family":"Xu","sequence":"first","affiliation":[{"name":"Clinical and Translational Research Center of Shanghai First Maternity and Infant Hospital School of Life Sciences and Technology Tongji University Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Miaomiao","family":"Jin","sequence":"additional","affiliation":[{"name":"Clinical and Translational Research Center of Shanghai First Maternity and Infant Hospital School of Life Sciences and Technology Tongji University Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Weilai","family":"Huang","sequence":"additional","affiliation":[{"name":"Clinical and Translational Research Center of Shanghai First Maternity and Infant Hospital School of Life Sciences and Technology Tongji University Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hong","family":"Wang","sequence":"additional","affiliation":[{"name":"Clinical and Translational Research Center of Shanghai First Maternity and Infant Hospital School of Life Sciences and Technology Tongji University Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ruikun","family":"Hu","sequence":"additional","affiliation":[{"name":"Clinical and Translational Research Center of Shanghai First Maternity and Infant Hospital School of Life Sciences and Technology Tongji University Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jingyu","family":"Li","sequence":"additional","affiliation":[{"name":"Clinical and Translational Research Center of Shanghai First Maternity and Infant Hospital School of Life Sciences and Technology Tongji University Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ying","family":"Cao","sequence":"additional","affiliation":[{"name":"Clinical and Translational Research Center of Shanghai First Maternity and Infant Hospital School of Life Sciences and Technology Tongji University Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2018,10,15]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrg2774"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrm.2017.60"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1152\/physrev.00028.2012"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2008.12.006"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature05185"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1242\/jcs.024927"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/0014-4800(92)90047-F"},{"key":"e_1_2_8_9_1","doi-asserted-by":"crossref","first-page":"1183","DOI":"10.1093\/jn\/125.5.1183","article-title":"Diet and disease alter phosphoinositide composition and metabolism in murine polycystic kidneys","volume":"125","author":"Aukema H. 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