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To better delineate neural crest defects in CHARGE syndrome, we generated induced pluripotent stem cells (iPSCs) from two patients with typical syndrome manifestations, and characterized neural crest cells differentiated in vitro from these iPSCs (iPSC-NCCs). We found that expression of genes associated with cell migration was altered in CHARGE iPSC-NCCs compared to control iPSC-NCCs. Consistently, CHARGE iPSC-NCCs showed defective delamination, migration and motility in vitro, and their transplantation\n                    <jats:italic>in ovo<\/jats:italic>\n                    revealed overall defective migratory activity in the chick embryo. These results support the historical inference that CHARGE syndrome patients exhibit defects in neural crest migration, and provide the first successful application of patient-derived iPSCs in modeling craniofacial disorders.\n                  <\/jats:p>","DOI":"10.7554\/elife.21114","type":"journal-article","created":{"date-parts":[[2017,11,27]],"date-time":"2017-11-27T19:00:20Z","timestamp":1511809220000},"update-policy":"https:\/\/doi.org\/10.7554\/elife.21114","source":"Crossref","is-referenced-by-count":64,"title":["CHARGE syndrome modeling using patient-iPSCs reveals defective migration of neural crest cells harboring CHD7 mutations"],"prefix":"10.7554","volume":"6","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1932-9482","authenticated-orcid":true,"given":"Hironobu","family":"Okuno","sequence":"first","affiliation":[{"name":"Department of Physiology, Keio University School of Medicine, Tokyo, 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