{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,2]],"date-time":"2026-06-02T08:08:50Z","timestamp":1780387730289,"version":"3.54.1"},"reference-count":50,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2026,1,30]],"date-time":"2026-01-30T00:00:00Z","timestamp":1769731200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,1,30]],"date-time":"2026-01-30T00:00:00Z","timestamp":1769731200000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100000027","name":"National Institute on Alcohol Abuse and Alcoholism","doi-asserted-by":"publisher","award":["AA026068"],"award-info":[{"award-number":["AA026068"]}],"id":[{"id":"10.13039\/100000027","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000027","name":"National Institute on Alcohol Abuse and Alcoholism","doi-asserted-by":"publisher","award":["AA031346"],"award-info":[{"award-number":["AA031346"]}],"id":[{"id":"10.13039\/100000027","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Birth Defects Research"],"published-print":{"date-parts":[[2026,2]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Prenatal alcohol exposure can cause fetal alcohol syndrome, but genetic variation can account for some of the observed differential susceptibility to alcohol\u2010induced birth defects. Here we investigate an inbred mouse strain, 129S1\/SvImJ (129S1), for its potential susceptibility to alcohol\u2010 and Sonic hedgehog pathway inhibitor\u2010induced craniofacial birth defects and the ways complex genetic factors work to alter susceptibility compared to the highly alcohol\u2010susceptible strain, the C57BL\/6J (B6J).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>129S1 pregnant females were treated with alcohol (two doses of 2.9\u2009g\/kg), CP\u201055,940 (2.0\u2009mg\/kg), vismodegib (40 or 80\u2009mg\/kg), or smoothened agonist (12\u2009mg\/kg) during gastrulation or neurulation, then fetuses were assessed for defects. Gastrulation\u2010 and neurulation\u2010stage embryo gene expression and apoptosis were also analyzed.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      129S1 mice are completely resistant to alcohol\u2010induced craniofacial birth defects despite high blood alcohol concentrations compared to other alcohol\u2010susceptible mouse strains. This strain is also resistant to craniofacial birth defects after exposure to a synthetic cannabinoid, CP\u201055,940, and vismodegib. All three drugs inhibit the Sonic hedgehog (Shh) pathway. 129S1 embryos have higher basal expression of two key Shh\u2010related mRNAs,\n                      <jats:italic>Smo<\/jats:italic>\n                      and\n                      <jats:italic>Efcab7<\/jats:italic>\n                      , and lower apoptosis after alcohol compared to B6J embryos. 129S1 embryos are more susceptible to birth defects after treatment with a Shh pathway agonist, smoothened agonist (SAG), than B6J embryos.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>129S1 embryos have higher expression of key Shh pathway genes and low apoptosis after alcohol, which may contribute to the alcohol\u2010resistant phenotype seen in the 129S1\/SvImJ strain, adding to the understanding of protective genetic factors in alcohol teratogenesis.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/bdr2.70020","type":"journal-article","created":{"date-parts":[[2026,1,30]],"date-time":"2026-01-30T11:40:36Z","timestamp":1769773236000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["The\n                    <scp>129S1<\/scp>\n                    \/\n                    <scp>SvImJ<\/scp>\n                    Mouse Strain Is Resistant to the Effects of Early Embryonic Alcohol and Other Sonic Hedgehog Inhibitors"],"prefix":"10.1002","volume":"118","author":[{"given":"Ruby L.","family":"Lapham","sequence":"first","affiliation":[{"name":"Bowles Center for Alcohol Studies University of North Carolina  Chapel Hill North Carolina USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6598-5654","authenticated-orcid":false,"given":"Eric W.","family":"Fish","sequence":"additional","affiliation":[{"name":"Bowles Center for Alcohol Studies University of North Carolina  Chapel Hill North Carolina USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2038-6548","authenticated-orcid":false,"given":"Scott E.","family":"Parnell","sequence":"additional","affiliation":[{"name":"Bowles Center for Alcohol Studies University of North Carolina  Chapel Hill North Carolina USA"},{"name":"Department of Cell Biology and Physiology University of North Carolina  Chapel Hill North Carolina USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,1,30]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.162356199"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdr2.2466"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdr2.1509"},{"key":"e_1_2_10_5_1","first-page":"839","article-title":"Chronologic Evaluation of Teratogenicity in Sheep Fed \n                     Veratrum californicum","volume":"147","author":"Binns W.","year":"1965","journal-title":"Journal of the American Veterinary Medical Association"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/0006\u20102952(92)90214\u20104"},{"key":"e_1_2_10_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/acer.12417"},{"key":"e_1_2_10_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdr2.2292"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1242\/dmm.049012"},{"key":"e_1_2_10_10_1","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.270"},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1530\u20100277.2011.01465.x"},{"key":"e_1_2_10_12_1","doi-asserted-by":"publisher","DOI":"10.1002\/tera.1420220110"},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.280.5369.1603"},{"key":"e_1_2_10_14_1","first-page":"515","article-title":"Fetal Alcohol Syndrome and Fetal Alcohol Spectrum Disorders","volume":"96","author":"Denny L.","year":"2017","journal-title":"American Family Physician"},{"key":"e_1_2_10_15_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1221386110"},{"key":"e_1_2_10_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1530\u20100277.2009.00949.x"},{"key":"e_1_2_10_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1530-0277.2001.tb02156.x"},{"key":"e_1_2_10_18_1","doi-asserted-by":"publisher","DOI":"10.1111\/acer.13066"},{"key":"e_1_2_10_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.reprotox.2024.108729"},{"key":"e_1_2_10_20_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdr2.2009"},{"key":"e_1_2_10_21_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41598\u2010019\u201052336\u2010w"},{"key":"e_1_2_10_22_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.23571"},{"key":"e_1_2_10_23_1","doi-asserted-by":"publisher","DOI":"10.1111\/acer.14688"},{"key":"e_1_2_10_24_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdr2.1508"},{"key":"e_1_2_10_25_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20833"},{"key":"e_1_2_10_26_1","doi-asserted-by":"publisher","DOI":"10.1111\/jnc.12464"},{"key":"e_1_2_10_27_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ntt.2015.12.004"},{"key":"e_1_2_10_28_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1530\u20100277.1989.tb00402.x"},{"key":"e_1_2_10_29_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1530\u20100277.2009.01071.x"},{"key":"e_1_2_10_30_1","doi-asserted-by":"publisher","DOI":"10.15585\/mmwr.mm7101a2"},{"key":"e_1_2_10_31_1","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.21048"},{"key":"e_1_2_10_32_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.diff.2022.04.001"},{"key":"e_1_2_10_33_1","doi-asserted-by":"publisher","DOI":"10.24105\/apr.2019.5.23"},{"key":"e_1_2_10_34_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0120517"},{"key":"e_1_2_10_35_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20367"},{"key":"e_1_2_10_36_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pgen.1002999"},{"key":"e_1_2_10_37_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.reprotox.2024.108747"},{"key":"e_1_2_10_38_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.125.18.3553"},{"key":"e_1_2_10_39_1","doi-asserted-by":"publisher","DOI":"10.1101\/gad.1190304"},{"key":"e_1_2_10_40_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0089448"},{"key":"e_1_2_10_41_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320440210"},{"key":"e_1_2_10_42_1","doi-asserted-by":"publisher","DOI":"10.1016\/0736\u20105748(92)90016\u2010S"},{"key":"e_1_2_10_43_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00213\u2010023\u201006421\u20108"},{"key":"e_1_2_10_44_1","doi-asserted-by":"publisher","DOI":"10.3389\/fnins.2023.1154621"},{"key":"e_1_2_10_45_1","doi-asserted-by":"publisher","DOI":"10.1128\/MCB.26.9.3365\u20103377.2006"},{"key":"e_1_2_10_46_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.freeradbiomed.2013.05.049"},{"key":"e_1_2_10_47_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320470612"},{"key":"e_1_2_10_48_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.6795717"},{"key":"e_1_2_10_49_1","doi-asserted-by":"publisher","DOI":"10.3389\/fphar.2022.975020"},{"key":"e_1_2_10_50_1","doi-asserted-by":"publisher","DOI":"10.1007\/978-3-642-88418-4"},{"key":"e_1_2_10_51_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.expneurol.2017.10.020"}],"container-title":["Birth Defects Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/bdr2.70020","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/bdr2.70020","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/bdr2.70020","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,6,2]],"date-time":"2026-06-02T07:40:06Z","timestamp":1780386006000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/bdr2.70020"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,1,30]]},"references-count":50,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2026,2]]}},"alternative-id":["10.1002\/bdr2.70020"],"URL":"https:\/\/doi.org\/10.1002\/bdr2.70020","archive":["Portico"],"relation":{},"ISSN":["2472-1727","2472-1727"],"issn-type":[{"value":"2472-1727","type":"print"},{"value":"2472-1727","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,1,30]]},"assertion":[{"value":"2025-10-03","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-01-16","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-01-30","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e70020"}}