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It can progress from abnormal epithelial cysts to an aggressive sarcoma with poor survival. PPB is difficult to diagnose as it can be confounded with other cystic lung disorders, such as congenital pulmonary airway malformation (CPAM). PPB is associated with mutations in DICER1 that perturb the microRNA (miRNA) profile in lung. How DICER1 and miRNAs act during PPB pathogenesis remains unsolved. Lung epithelial deletion of the Yin Yang1 (Yy1) gene in mice causes a phenotype mimicking the cystic form of PPB and affects the expression of key regulators of lung development. Similar changes in expression were observed in PPB but not in CPAM lung biopsies, revealing a distinctive PPB molecular signature. Deregulation of molecules promoting epithelial\u2013mesenchymal transition (EMT) was detected in PPB specimens, suggesting that EMT might participate in tumor progression. Changes in miRNA expression also occurred in PPB lung biopsies. miR-125a-3p, a candidate to regulate YY1 expression and lung branching, was abnormally highly expressed in PPB samples. Together, these findings support the concept that reduced expression of YY1, due to the abnormal miRNA profile resulting from DICER1 mutations, contributes to PPB development via its impact on the expression of key lung developmental genes.<\/jats:p>\n               <jats:p>This article has an associated First Person interview with the joint first authors of the paper.<\/jats:p>","DOI":"10.1242\/dmm.045989","type":"journal-article","created":{"date-parts":[[2020,11,6]],"date-time":"2020-11-06T15:40:15Z","timestamp":1604677215000},"source":"Crossref","is-referenced-by-count":10,"title":["Deletion of <i>Yy1<\/i> in mouse lung epithelium unveils molecular mechanisms governing pleuropulmonary blastoma pathogenesis"],"prefix":"10.1242","volume":"13","author":[{"given":"Kim","family":"Landry-Truchon","sequence":"first","affiliation":[{"name":"Centre de recherche sur le cancer de l'Universit\u00e9 Laval, Centre de recherche du CHU de Qu\u00e9bec-Universit\u00e9 Laval 1 (Oncology Axis), Qu\u00e9bec , Canada G1R 3S3"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicolas","family":"Houde","sequence":"additional","affiliation":[{"name":"Centre de recherche sur le cancer de l'Universit\u00e9 Laval, Centre de recherche du CHU de Qu\u00e9bec-Universit\u00e9 Laval 1 (Oncology Axis), Qu\u00e9bec , Canada G1R 3S3"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Micka\u00ebl","family":"Lhuillier","sequence":"additional","affiliation":[{"name":"Inserm U1151, Institut Necker-Enfants Malades, Universit\u00e9 de Paris 2 , 75743 Paris, Cedex15, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Louis","family":"Charron","sequence":"additional","affiliation":[{"name":"Centre de recherche sur le cancer de l'Universit\u00e9 Laval, Centre de recherche du CHU de Qu\u00e9bec-Universit\u00e9 Laval 1 (Oncology Axis), Qu\u00e9bec , Canada G1R 3S3"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7451-9890","authenticated-orcid":false,"given":"Alice","family":"Hadchouel","sequence":"additional","affiliation":[{"name":"Inserm U1151, Institut Necker-Enfants Malades, Universit\u00e9 de Paris 2 , 75743 Paris, Cedex15, France"},{"name":"AP-HP, H\u00f4pital Necker-Enfants Malades 3 , 75743 Paris, Cedex15, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0007-7150","authenticated-orcid":false,"given":"Christophe","family":"Delacourt","sequence":"additional","affiliation":[{"name":"Inserm U1151, Institut Necker-Enfants Malades, Universit\u00e9 de Paris 2 , 75743 Paris, Cedex15, France"},{"name":"AP-HP, H\u00f4pital Necker-Enfants Malades 3 , 75743 Paris, Cedex15, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7427-4651","authenticated-orcid":false,"given":"William D.","family":"Foulkes","sequence":"additional","affiliation":[{"name":"Lady Davis Institute and Segal Cancer Centre, Jewish General Hospital 4 Department of Medical Genetics , , Montr\u00e9al , Canada H3T 1E2"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7081-0437","authenticated-orcid":false,"given":"Louise","family":"Galmiche-Rolland","sequence":"additional","affiliation":[{"name":"AP-HP, H\u00f4pital Necker-Enfants Malades 3 , 75743 Paris, Cedex15, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5578-420X","authenticated-orcid":false,"given":"Lucie","family":"Jeannotte","sequence":"additional","affiliation":[{"name":"Centre de recherche sur le cancer de l'Universit\u00e9 Laval, Centre de recherche du CHU de Qu\u00e9bec-Universit\u00e9 Laval 1 (Oncology Axis), Qu\u00e9bec , Canada G1R 3S3"},{"name":"Universit\u00e9 Laval 5 Department of Molecular Biology, Medical Biochemistry & Pathology , , Qu\u00e9bec , Canada G1V 0A6"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2020,12,29]]},"reference":[{"key":"2024061302364389500_DMM045989C1","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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