{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,3]],"date-time":"2026-10-03T20:46:30Z","timestamp":1791060390594,"version":"4.1.0"},"reference-count":51,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2011,4,1]],"date-time":"2011-04-01T00:00:00Z","timestamp":1301616000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Genesis"],"published-print":{"date-parts":[[2011,4]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Proper craniofacial development begins during gastrulation and requires the coordinated integration of each germ layer tissue (ectoderm, mesoderm, and endoderm) and its derivatives in concert with the precise regulation of cell proliferation, migration, and differentiation. Neural crest cells, which are derived from ectoderm, are a migratory progenitor cell population that generates most of the cartilage, bone, and connective tissue of the head and face. Neural crest cell development is regulated by a combination of intrinsic cell autonomous signals acquired during their formation, balanced with extrinsic signals from tissues with which the neural crest cells interact during their migration and differentiation. Although craniofacial anomalies are typically attributed to defects in neural crest cell development, the cause may be intrinsic or extrinsic. Therefore, we performed a phenotype\u2010driven ENU mutagenesis screen in mice with the aim of identifying novel alleles in an unbiased manner, that are critically required for early craniofacial development. Here we describe 10 new mutant lines, which exhibit phenotypes affecting frontonasal and pharyngeal arch patterning, neural and vascular development as well as sensory organ morphogenesis. Interestingly, our data imply that neural crest cells and endothelial cells may employ similar developmental programs and be interdependent during early embryogenesis, which collectively is critical for normal craniofacial morphogenesis. Furthermore our novel mutants that model human conditions such as exencephaly, craniorachischisis, DiGeorge, and Velocardiofacial sydnromes could be very useful in furthering our understanding of the complexities of specific human diseases. genesis 49:342\u2013359, 2011. \u00a9 2011 Wiley\u2010Liss, Inc.<\/jats:p>","DOI":"10.1002\/dvg.20727","type":"journal-article","created":{"date-parts":[[2011,2,9]],"date-time":"2011-02-09T11:47:53Z","timestamp":1297252073000},"page":"342-359","source":"Crossref","is-referenced-by-count":27,"title":["A phenotype\u2010driven ENU mutagenesis screen identifies novel alleles with functional roles in early mouse craniofacial development"],"prefix":"10.1002","volume":"49","author":[{"given":"Lisa L.","family":"Sandell","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Angelo","family":"Iulianella","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kristin R.","family":"Melton","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Megan","family":"Lynn","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Macie","family":"Walker","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kimberly E.","family":"Inman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shachi","family":"Bhatt","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Margot","family":"Leroux\u2010Berger","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michelle","family":"Crawford","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Natalie C.","family":"Jones","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jennifer F.","family":"Dennis","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul A.","family":"Trainor","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2011,4]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.1006\/dbio.2001.0156"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature03875"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.nutr.22.010402.102745E"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/aja.1001660406"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.20796"},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0603730103"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1097\/00005072-199011000-00001"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1038\/380439a0"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10456-008-9096-2"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.00175"},{"key":"e_1_2_6_12_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.200302047"},{"key":"e_1_2_6_13_1","volume-title":"Syndromes of the head and neck","author":"Gorlin RJ","year":"1990"},{"key":"e_1_2_6_14_1","doi-asserted-by":"publisher","DOI":"10.1172\/JCI30070"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.182296499"},{"key":"e_1_2_6_16_1","doi-asserted-by":"publisher","DOI":"10.1038\/nm1725"},{"key":"e_1_2_6_17_1","doi-asserted-by":"publisher","DOI":"10.1038\/383707a0"},{"key":"e_1_2_6_18_1","doi-asserted-by":"publisher","DOI":"10.1007\/s003350010094"},{"key":"e_1_2_6_19_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/8.10.1955"},{"key":"e_1_2_6_20_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.95.13.7485"},{"key":"e_1_2_6_21_1","doi-asserted-by":"crossref","first-page":"3229","DOI":"10.1242\/dev.122.10.3229","article-title":"Rhombencephalic neural crest segmentation is preserved throughout craniofacial ontogeny","volume":"122","author":"Kontges G","year":"1996","journal-title":"Development"},{"key":"e_1_2_6_22_1","doi-asserted-by":"publisher","DOI":"10.1101\/gad.14.11.1343"},{"key":"e_1_2_6_23_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.00887"},{"key":"e_1_2_6_24_1","doi-asserted-by":"publisher","DOI":"10.1017\/CBO9780511897948"},{"key":"e_1_2_6_25_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10456-008-9100-x"},{"key":"e_1_2_6_26_1","doi-asserted-by":"publisher","DOI":"10.1006\/mcne.1999.0762"},{"key":"e_1_2_6_27_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng0797-243"},{"key":"e_1_2_6_28_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.21-05-01538.2001"},{"key":"e_1_2_6_29_1","doi-asserted-by":"publisher","DOI":"10.1006\/dbio.1999.9355"},{"key":"e_1_2_6_30_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-7580.2005.00475.x"},{"key":"e_1_2_6_31_1","volume-title":"Manipulating the mouse embryo","author":"Nagy A","year":"2003"},{"key":"e_1_2_6_32_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0168-9525(02)02820-2"},{"key":"e_1_2_6_33_1","first-page":"167","article-title":"Patterns and organization of craniofacial skeletogenic and myogenic mesenchyme: A perspective","volume":"101","author":"Noden DM","year":"1982","journal-title":"Prog Clin Biol Res"},{"key":"e_1_2_6_34_1","doi-asserted-by":"publisher","DOI":"10.1016\/0012-1606(83)90318-4"},{"key":"e_1_2_6_35_1","doi-asserted-by":"publisher","DOI":"10.1002\/aja.1001680302"},{"key":"e_1_2_6_36_1","first-page":"192","article-title":"Cell movements and control of patterned tissue assembly during craniofacial development","volume":"11","author":"Noden DM","year":"1991","journal-title":"J Craniofac Genet Dev Biol"},{"key":"e_1_2_6_37_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng0797-235"},{"key":"e_1_2_6_38_1","doi-asserted-by":"publisher","DOI":"10.1002\/1096-9861(20001002)425:4<479::AID-CNE2>3.0.CO;2-3"},{"key":"e_1_2_6_39_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng0298-171"},{"key":"e_1_2_6_40_1","doi-asserted-by":"publisher","DOI":"10.1101\/gad.1533407"},{"key":"e_1_2_6_41_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.015412"},{"key":"e_1_2_6_42_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng0198-60"},{"key":"e_1_2_6_43_1","doi-asserted-by":"crossref","first-page":"526","DOI":"10.1542\/peds.85.4.526","article-title":"Di George anomaly and velocardiofacial syndrome","volume":"85","author":"Stevens CA","year":"1990","journal-title":"Pediatrics"},{"key":"e_1_2_6_44_1","doi-asserted-by":"publisher","DOI":"10.1096\/fj.04-1991fje"},{"key":"e_1_2_6_45_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.120.9.2397"},{"key":"e_1_2_6_46_1","doi-asserted-by":"crossref","first-page":"2569","DOI":"10.1242\/dev.121.8.2569","article-title":"Cranial paraxial mesoderm and neural crest of the mouse embryo\u2010codistribution in the craniofacial mesenchyme but distinct segregation in the branchial arches","volume":"121","author":"Trainor PA","year":"1995","journal-title":"Development"},{"key":"e_1_2_6_47_1","doi-asserted-by":"publisher","DOI":"10.1038\/35039056"},{"key":"e_1_2_6_48_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0955-0674(00)00273-8"},{"key":"e_1_2_6_49_1","first-page":"247","article-title":"Monoclonal antibodies specific for endothelial cells of mouse blood vessels. Their application in the identification of adult and embryonic endothelium","volume":"63","author":"Vecchi A","year":"1994","journal-title":"Eur J Cell Biol"},{"key":"e_1_2_6_50_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0925-4773(01)00469-5"},{"key":"e_1_2_6_51_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0092-8674(00)81436-1"},{"key":"e_1_2_6_52_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1432-1033.1996.0015h.x"}],"container-title":["genesis"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fdvg.20727","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/dvg.20727","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,12]],"date-time":"2023-09-12T15:58:53Z","timestamp":1694534333000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/dvg.20727"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2011,4]]},"references-count":51,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2011,4]]}},"alternative-id":["10.1002\/dvg.20727"],"URL":"https:\/\/doi.org\/10.1002\/dvg.20727","archive":["Portico"],"relation":{},"ISSN":["1526-954X","1526-968X"],"issn-type":[{"value":"1526-954X","type":"print"},{"value":"1526-968X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2011,4]]}}}