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It impaired tracheal cartilage formation, altered cell differentiation, abrogated lung branching and caused airway dilation similar to that seen in human congenital cystic lung diseases. The cystic lung phenotype in Yy1 mutants can be partly explained by the reduced expression of Shh, a transcriptional target of YY1, in lung endoderm, and the subsequent derepression of mesenchymal Fgf10 expression. Accordingly, SHH supplementation partially rescued the lung phenotype in vitro. Analysis of human lung tissues revealed decreased YY1 expression in children with pleuropulmonary blastoma (PPB), a rare pediatric lung tumor arising during fetal development and associated with DICER1 mutations. No evidence for a potential genetic interplay between murine Dicer and Yy1 genes during lung morphogenesis was observed. However, the cystic lung phenotype resulting from the epithelial inactivation of Dicer function mimics the Yy1 lung malformations with similar changes in Shh and Fgf10 expression. Together, our data demonstrate the crucial requirement for YY1 in lung morphogenesis and identify Yy1 mutant mice as a potential model for studying the genetic basis of PPB.<\/jats:p>","DOI":"10.1242\/dev.120469","type":"journal-article","created":{"date-parts":[[2015,9,1]],"date-time":"2015-09-01T11:23:32Z","timestamp":1441106612000},"page":"2981-2995","source":"Crossref","is-referenced-by-count":38,"title":["Epithelial inactivation of <i>Yy1<\/i> abrogates lung branching morphogenesis"],"prefix":"10.1242","volume":"142","author":[{"given":"Olivier","family":"Boucherat","sequence":"first","affiliation":[{"name":"Centre de Recherche sur le Cancer de l'Universit\u00e9 Laval; CRCHUQ, L'H\u00f4tel-Dieu de Qu\u00e9bec, Qu\u00e9bec, G1R 3S3, Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kim","family":"Landry-Truchon","sequence":"additional","affiliation":[{"name":"Centre de Recherche sur le Cancer de l'Universit\u00e9 Laval; CRCHUQ, L'H\u00f4tel-Dieu de Qu\u00e9bec, Qu\u00e9bec, G1R 3S3, Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"F\u00e9lix-Antoine","family":"B\u00e9rub\u00e9-Simard","sequence":"additional","affiliation":[{"name":"Centre de Recherche sur le Cancer de l'Universit\u00e9 Laval; CRCHUQ, L'H\u00f4tel-Dieu de Qu\u00e9bec, Qu\u00e9bec, G1R 3S3, Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nicolas","family":"Houde","sequence":"additional","affiliation":[{"name":"Centre de Recherche sur le Cancer de l'Universit\u00e9 Laval; CRCHUQ, L'H\u00f4tel-Dieu de Qu\u00e9bec, Qu\u00e9bec, G1R 3S3, Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Laurent","family":"Beuret","sequence":"additional","affiliation":[{"name":"Centre de Recherche sur le Cancer de l'Universit\u00e9 Laval; CRCHUQ, L'H\u00f4tel-Dieu de Qu\u00e9bec, Qu\u00e9bec, G1R 3S3, Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Guillaume","family":"Lezmi","sequence":"additional","affiliation":[{"name":"AP-HP, H\u00f4pital Necker-Enfants Malades, Service de Pneumologie P\u00e9diatrique, Universit\u00e9 Paris-Descartes, Paris, 75015, France"},{"name":"Inserm U955, IMRB, Equipe 04, Cr\u00e9teil, 94011, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"William D.","family":"Foulkes","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics, Lady Davis Institute and Segal Cancer Centre, Jewish General Hospital, Montr\u00e9al, H3G 1A4, Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Christophe","family":"Delacourt","sequence":"additional","affiliation":[{"name":"AP-HP, H\u00f4pital Necker-Enfants Malades, Service de Pneumologie P\u00e9diatrique, Universit\u00e9 Paris-Descartes, Paris, 75015, France"},{"name":"Inserm U955, IMRB, Equipe 04, Cr\u00e9teil, 94011, France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jean","family":"Charron","sequence":"additional","affiliation":[{"name":"Centre de Recherche sur le Cancer de l'Universit\u00e9 Laval; CRCHUQ, L'H\u00f4tel-Dieu de Qu\u00e9bec, Qu\u00e9bec, G1R 3S3, Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lucie","family":"Jeannotte","sequence":"additional","affiliation":[{"name":"Centre de Recherche sur le Cancer de l'Universit\u00e9 Laval; CRCHUQ, L'H\u00f4tel-Dieu de Qu\u00e9bec, Qu\u00e9bec, G1R 3S3, Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"237","reference":[{"key":"2021042608080506500_DEV120469C1","doi-asserted-by":"publisher","first-page":"3565","DOI":"10.1128\/MCB.26.9.3565-3581.2006","article-title":"Essential dosage-dependent functions of the transcription factor yin yang 1 in late embryonic development and cell cycle progression","volume":"26","author":"Affar","year":"2006","journal-title":"Mol. 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