{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T01:39:16Z","timestamp":1791164356434,"version":"4.1.0"},"reference-count":62,"publisher":"Wiley","issue":"7","license":[{"start":{"date-parts":[[2013,6,3]],"date-time":"2013-06-03T00:00:00Z","timestamp":1370217600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Developmental Dynamics"],"published-print":{"date-parts":[[2013,7]]},"abstract":"<jats:p><jats:styled-content>Background:<\/jats:styled-content> The regulatory mechanisms underpinning facial development are conserved between diverse species. Therefore, results from model systems provide insight into the genetic causes of human craniofacial defects. Previously, we generated a comprehensive dataset examining gene expression during development and fusion of the mouse facial prominences. Here, we used this resource to identify genes that have dynamic expression patterns in the facial prominences, but for which only limited information exists concerning developmental function. <jats:styled-content>Results:<\/jats:styled-content> This set of \u223c80 genes was used for a high\u2010throughput functional analysis in the zebrafish system using Morpholino gene knockdown technology. This screen revealed three classes of cranial cartilage phenotypes depending upon whether knockdown of the gene affected the neurocranium, viscerocranium, or both. The targeted genes that produced consistent phenotypes encoded proteins linked to transcription (<jats:italic>meis1, meis2a, tshz2, vgll4l<\/jats:italic>), signaling (<jats:italic>pkdcc, vlk, macc1, wu:fb16h09<\/jats:italic>), and extracellular matrix function (<jats:italic>smoc2<\/jats:italic>). The majority of these phenotypes were not altered by reduction of p53 levels, demonstrating that both p53\u2010dependent and \u2010independent mechanisms were involved in the craniofacial abnormalities. <jats:styled-content>Conclusions:<\/jats:styled-content> This Morpholino\u2010based screen highlights new genes involved in development of the zebrafish craniofacial skeleton with wider relevance to formation of the face in other species, particularly mouse and human. <jats:italic>Developmental Dynamics 242:817\u2013831, 2013<\/jats:italic>. \u00a9 2013 Wiley Periodicals, Inc.<\/jats:p>","DOI":"10.1002\/dvdy.23969","type":"journal-article","created":{"date-parts":[[2013,4,5]],"date-time":"2013-04-05T05:32:45Z","timestamp":1365139965000},"page":"817-831","source":"Crossref","is-referenced-by-count":56,"title":["A morpholino\u2010based screen to identify novel genes involved in craniofacial morphogenesis"],"prefix":"10.1002","volume":"242","author":[{"given":"Vida Senkus","family":"Melvin","sequence":"first","affiliation":[{"name":"Department of Craniofacial Biology School of Dental Medicine University of Colorado Denver Colorado"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Weiguo","family":"Feng","sequence":"additional","affiliation":[{"name":"Department of Craniofacial Biology School of Dental Medicine University of Colorado Denver Colorado"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laura","family":"Hernandez\u2010Lagunas","sequence":"additional","affiliation":[{"name":"Department of Craniofacial Biology School of Dental Medicine University of Colorado Denver Colorado"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kristin Bruk","family":"Artinger","sequence":"additional","affiliation":[{"name":"Department of Craniofacial Biology School of Dental Medicine University of Colorado Denver Colorado"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Trevor","family":"Williams","sequence":"additional","affiliation":[{"name":"Department of Craniofacial Biology School of Dental Medicine University of Colorado Denver Colorado"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2013,6,3]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2010.12.002"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1101\/gad.13.20.2713"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0406252102"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2011.04.026"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.201007008"},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2011.11.002"},{"key":"e_1_2_6_8_1","doi-asserted-by":"crossref","first-page":"129","DOI":"10.1242\/dev.123.1.129","article-title":"Mutations affecting development of the midline and general body shape during zebrafish embryogenesis","volume":"123","author":"Brand M","year":"1996","journal-title":"Development"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.022442"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.1765-10.2010"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.20833"},{"key":"e_1_2_6_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2007.05.038"},{"key":"e_1_2_6_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33375"},{"key":"e_1_2_6_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.mod.2010.11.003"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrg2933"},{"key":"e_1_2_6_16_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.02281"},{"key":"e_1_2_6_17_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.82"},{"key":"e_1_2_6_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2009.07.008"},{"key":"e_1_2_6_19_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0008066"},{"key":"e_1_2_6_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2011.08.005"},{"key":"e_1_2_6_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2010.11.030"},{"key":"e_1_2_6_22_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.123.1.143"},{"key":"e_1_2_6_23_1","doi-asserted-by":"publisher","DOI":"10.1128\/MCB.23.24.9189-9207.2003"},{"key":"e_1_2_6_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2008.10.026"},{"key":"e_1_2_6_25_1","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.21822"},{"key":"e_1_2_6_26_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2011.06.034"},{"key":"e_1_2_6_27_1","doi-asserted-by":"publisher","DOI":"10.1038\/nm1725"},{"key":"e_1_2_6_28_1","doi-asserted-by":"publisher","DOI":"10.1006\/dbio.2001.0201"},{"key":"e_1_2_6_29_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.026435"},{"key":"e_1_2_6_30_1","doi-asserted-by":"publisher","DOI":"10.1186\/1471-2202-11-137"},{"key":"e_1_2_6_31_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2011.06.007"},{"key":"e_1_2_6_32_1","doi-asserted-by":"publisher","DOI":"10.1091\/mbc.E07-05-0510"},{"key":"e_1_2_6_33_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.gene.2008.12.010"},{"key":"e_1_2_6_34_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1002447107"},{"key":"e_1_2_6_35_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.yexcr.2008.05.020"},{"key":"e_1_2_6_36_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2005.10.032"},{"key":"e_1_2_6_37_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0037877"},{"key":"e_1_2_6_38_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.123.1.357"},{"key":"e_1_2_6_39_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2010.11.012"},{"key":"e_1_2_6_40_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.123.1.345"},{"key":"e_1_2_6_41_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1097-2765(00)00027-7"},{"key":"e_1_2_6_42_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pgen.1002114"},{"key":"e_1_2_6_43_1","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.22248"},{"key":"e_1_2_6_44_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pgen.0030078"},{"key":"e_1_2_6_45_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M513463200"},{"key":"e_1_2_6_46_1","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.22228"},{"key":"e_1_2_6_47_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.120.3.483"},{"key":"e_1_2_6_48_1","doi-asserted-by":"publisher","DOI":"10.1186\/jbiol205"},{"key":"e_1_2_6_49_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.123.1.329"},{"key":"e_1_2_6_50_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.128.18.3543"},{"key":"e_1_2_6_51_1","doi-asserted-by":"publisher","DOI":"10.1038\/nm.1889"},{"key":"e_1_2_6_52_1","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.22713"},{"key":"e_1_2_6_53_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2009.11.021"},{"key":"e_1_2_6_54_1","unstructured":"ThisseB PflumioS F\u00fcrthauerM LoppinB HeyerV DegraveA WoehlR LuxA T. S CharbonnierXQ ThisseC.2001Expression of the zebrafish genome during embryogenesis (NIH R01 RR15402). ZFIN Direct Data Submission.http:\/\/zfin.org\/."},{"key":"e_1_2_6_55_1","unstructured":"ThisseB ThisseC.2004.Fast Release Clones: A High Throughput Expression Analysis. ZFIN Direct Data Submission.http:\/\/zfin.org\/ZDB\u2010PUB\u2010040907\u20101."},{"key":"e_1_2_6_56_1","unstructured":"ThisseC ThisseB.2005.High throughput expression analysis of ZF\u2010models consortium clones. ZFIN Direct Data Submission.http:\/\/zfin.org\/ZDB\u2010PUB\u2010051025\u20101."},{"key":"e_1_2_6_57_1","doi-asserted-by":"publisher","DOI":"10.1042\/BJ20030532"},{"key":"e_1_2_6_58_1","doi-asserted-by":"publisher","DOI":"10.1038\/sj.hdy.6800635"},{"key":"e_1_2_6_59_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.01943"},{"key":"e_1_2_6_60_1","doi-asserted-by":"publisher","DOI":"10.1080\/10520290701333558"},{"key":"e_1_2_6_61_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0705502104"},{"key":"e_1_2_6_62_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0029574"},{"key":"e_1_2_6_63_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.01674"}],"container-title":["Developmental Dynamics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fdvdy.23969","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/anatomypubs.onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/dvdy.23969","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,13]],"date-time":"2025-10-13T11:36:59Z","timestamp":1760355419000},"score":1,"resource":{"primary":{"URL":"https:\/\/anatomypubs.onlinelibrary.wiley.com\/doi\/10.1002\/dvdy.23969"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2013,6,3]]},"references-count":62,"journal-issue":{"issue":"7","published-print":{"date-parts":[[2013,7]]}},"alternative-id":["10.1002\/dvdy.23969"],"URL":"https:\/\/doi.org\/10.1002\/dvdy.23969","archive":["Portico"],"relation":{},"ISSN":["1058-8388","1097-0177"],"issn-type":[{"value":"1058-8388","type":"print"},{"value":"1097-0177","type":"electronic"}],"subject":[],"published":{"date-parts":[[2013,6,3]]}}}