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Here, we provide an overview of normal embryonic morphogenesis during the critical window for HPE induction, focusing on the morphology and positional relationship of the developing brain and subjacent prechordal plate and prechordal mesoderm cell populations. Subsequent morphogenesis of the HPE spectrum is then examined in selected teratogenesis mouse models. The temporal profile of <jats:italic>Sonic Hedgehog<\/jats:italic> expression in rostral embryonic cell populations and evidence for direct or indirect perturbation of the Hedgehog pathway by teratogenic agents in the genesis of HPE is highlighted. Emerging opportunities based on recent insights and new techniques to further characterize the mechanisms and pathogenesis of HPE are discussed. \u00a9 2010 Wiley\u2010Liss, Inc.<\/jats:p>","DOI":"10.1002\/ajmg.c.30239","type":"journal-article","created":{"date-parts":[[2010,1,26]],"date-time":"2010-01-26T22:17:46Z","timestamp":1264544266000},"page":"29-42","source":"Crossref","is-referenced-by-count":58,"title":["Genesis of teratogen\u2010induced holoprosencephaly in mice"],"prefix":"10.1002","volume":"154C","author":[{"given":"Robert J.","family":"Lipinski","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elizabeth A.","family":"Godin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shonagh K.","family":"O'leary\u2010Moore","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Scott 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