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We established a reproducible ex\u2009vivo model of lung development using transgene-free human induced pluripotent stem cells generated from fetuses and infants with Bochdalek congenital diaphragmatic hernia (CDH), a polygenic disorder associated with fetal lung compression and pulmonary hypoplasia at birth. Molecular and cellular comparisons of CDH LOs revealed impaired generation of NKX2.1+ progenitors, type\u2009II alveolar epithelial cells, and PDGFR\u03b1+ myofibroblasts. We then subjected these LOs to disease relevant mechanical cues through ex\u2009vivo compression and observed significant changes in genes associated with pulmonary progenitors, alveolar epithelial cells, and mesenchymal fibroblasts. Collectively, these data suggest both primary cell-intrinsic and secondary mechanical causes of CDH lung hypoplasia and support the use of this stem cell-based approach for disease modeling in CDH.<\/jats:p>","DOI":"10.1002\/sctm.20-0199","type":"journal-article","created":{"date-parts":[[2020,9,19]],"date-time":"2020-09-19T09:43:54Z","timestamp":1600508634000},"page":"98-114","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":46,"title":["Human induced pluripotent stem cell-derived lung organoids in an ex\u2009vivo model of the congenital diaphragmatic hernia fetal lung"],"prefix":"10.1093","volume":"10","author":[{"given":"Shaun M.","family":"Kunisaki","sequence":"first","affiliation":[{"name":"Department of Surgery Johns Hopkins University, Baltimore, Maryland, USA"},{"name":"Institute for Cell Engineering, Johns Hopkins University, Baltimore, Maryland, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Guihua","family":"Jiang","sequence":"additional","affiliation":[{"name":"Department of Surgery University of Michigan, Ann Arbor, Michigan, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Juan C.","family":"Biancotti","sequence":"additional","affiliation":[{"name":"Department of Surgery Johns Hopkins University, Baltimore, Maryland, USA"},{"name":"Institute for Cell Engineering, Johns Hopkins University, Baltimore, Maryland, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kenneth K. 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