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Herein, we have uncovered the genome-wide redistribution of accessible chromatin regions and extensive alterations of expression of cilia genes during Ellis\u2013van Creveld syndrome (EVC) ciliopathy pathogenesis. Mechanistically, the distinct EVC ciliopathy-activated accessible regions (CAAs) are shown to positively regulate robust changes in flanking cilia genes, which are a key requirement for cilia transcription in response to developmental signals. Moreover, a single transcription factor, ETS1, can be recruited to CAAs, leading to prominent chromatin accessibility reconstruction in EVC ciliopathy patients. In zebrafish, the collapse of CAAs driven by ets1 suppression subsequently causes defective cilia proteins, resulting in body curvature and pericardial oedema. Our results depict a dynamic landscape of chromatin accessibility in EVC ciliopathy patients, and uncover an insightful role for ETS1 in controlling the global transcriptional program of cilia genes by reprogramming the widespread chromatin state.<\/jats:p>","DOI":"10.1093\/nar\/gkad506","type":"journal-article","created":{"date-parts":[[2023,6,16]],"date-time":"2023-06-16T09:46:29Z","timestamp":1686908789000},"page":"6684-6701","source":"Crossref","is-referenced-by-count":10,"title":["Recruitment of transcription factor ETS1 to activated accessible regions promotes the transcriptional program of cilia genes"],"prefix":"10.1093","volume":"51","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6208-6984","authenticated-orcid":false,"given":"Donghui","family":"Zhang","sequence":"first","affiliation":[{"name":"Zhanjiang Institute of Clinical Medicine, Central People's Hospital of Zhanjiang, Guangdong Medical University Zhanjiang Central Hospital , Zhanjiang 524045, PR China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chong","family":"Zhang","sequence":"additional","affiliation":[{"name":"Zhanjiang Institute of Clinical Medicine, Central People's Hospital of Zhanjiang, Guangdong Medical University Zhanjiang Central Hospital , Zhanjiang 524045, PR China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yanmei","family":"Zhu","sequence":"additional","affiliation":[{"name":"Zhanjiang Institute of Clinical Medicine, Central People's Hospital of Zhanjiang, Guangdong Medical University Zhanjiang Central Hospital , Zhanjiang 524045, PR China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Haixia","family":"Xie","sequence":"additional","affiliation":[{"name":"Precision Clinical Laboratory, Central People's Hospital of Zhanjiang, Guangdong Medical University Zhanjiang Central Hospital , Zhanjiang 524045, PR China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Caifeng","family":"Yue","sequence":"additional","affiliation":[{"name":"Precision Clinical Laboratory, Central People's Hospital of Zhanjiang, Guangdong Medical University Zhanjiang Central Hospital , Zhanjiang 524045, PR China"},{"name":"Department of Laboratory Medicine, Central People's Hospital of Zhanjiang, Guangdong Medical University Zhanjiang Central Hospital , Zhanjiang 524045, PR China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mingfeng","family":"Li","sequence":"additional","affiliation":[{"name":"Zhanjiang Institute of Clinical Medicine, Central People's Hospital of Zhanjiang, Guangdong Medical University Zhanjiang Central Hospital , Zhanjiang 524045, PR China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wenlu","family":"Wei","sequence":"additional","affiliation":[{"name":"Zhanjiang Institute of Clinical Medicine, Central People's Hospital of Zhanjiang, Guangdong Medical University Zhanjiang Central Hospital , Zhanjiang 524045, PR China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yu","family":"Peng","sequence":"additional","affiliation":[{"name":"Pediatric Intensive Care Unit Central, People's Hospital of Zhanjiang, Guangdong Medical University Zhanjiang Central Hospital , Zhanjiang 524045, PR China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Guibin","family":"Yin","sequence":"additional","affiliation":[{"name":"Department of Orthopedics, Central People's Hospital of Zhanjiang, Guangdong Medical University Zhanjiang Central Hospital , Zhanjiang 524045, PR China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yunmiao","family":"Guo","sequence":"additional","affiliation":[{"name":"Zhanjiang Institute of Clinical Medicine, Central People's Hospital of Zhanjiang, Guangdong Medical University Zhanjiang Central Hospital , Zhanjiang 524045, PR China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5158-0226","authenticated-orcid":false,"given":"Yiting","family":"Guan","sequence":"additional","affiliation":[{"name":"Zhanjiang Institute of Clinical Medicine, Central People's Hospital of Zhanjiang, Guangdong Medical University Zhanjiang Central Hospital , Zhanjiang 524045, PR China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2023,6,16]]},"reference":[{"key":"2023072104322072700_B1","doi-asserted-by":"crossref","first-page":"1039","DOI":"10.1007\/s00467-010-1731-7","article-title":"Ciliopathies: an expanding disease spectrum","volume":"26","author":"Waters","year":"2011","journal-title":"Pediatr. 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