{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T14:32:21Z","timestamp":1786977141256,"version":"build-2736575974"},"reference-count":70,"publisher":"Wiley","issue":"10","license":[{"start":{"date-parts":[[2014,9,25]],"date-time":"2014-09-25T00:00:00Z","timestamp":1411603200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100000024","name":"Canadian Institutes of Health Research","doi-asserted-by":"publisher","award":["MOP-74557"],"award-info":[{"award-number":["MOP-74557"]}],"id":[{"id":"10.13039\/501100000024","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Birth Defects Research"],"published-print":{"date-parts":[[2014,10]]},"abstract":"<jats:p>\n                    Background: The heritable multifactorial etiology of human nonsyndromic cleft lip with or without cleft palate (CL\u2009\u00b1\u2009P) is not understood. CL\u2009\u00b1\u2009P occurs in 15% of neonates in the homozygous A\/WySn mouse strain, with a multifactorial genetic etiology, the\n                    <jats:italic>clf1<\/jats:italic>\n                    and\n                    <jats:italic>clf2<\/jats:italic>\n                    variant genes.\n                    <jats:italic>Clf1<\/jats:italic>\n                    acts as a mutant allele of\n                    <jats:italic>Wnt9b<\/jats:italic>\n                    but its coding sequence is normal. An IAP (intracisternal A particle) retrotransposon inserted near the\n                    <jats:italic>Wnt9b<\/jats:italic>\n                    gene is associated with\n                    <jats:italic>clf1<\/jats:italic>\n                    . Methods: Transcription of noncoding sequence between the IAP and the\n                    <jats:italic>Wnt9b<\/jats:italic>\n                    gene was examined in A\/WySn embryos. The levels of\n                    <jats:italic>Wnt9b<\/jats:italic>\n                    transcript and of an \u201cIAP antisense\u201d transcript initiated in the IAP and extending into the noncoding interval were assayed in A\/WySn and C57BL\/6J whole embryos or heads across embryonic days 8 to 12. Methylation of the 5\u2032 LTR of the IAP was examined in E12 A\/WySn embryo heads. Results: Mean\n                    <jats:italic>Wnt9b<\/jats:italic>\n                    transcript levels were lower in A\/WySn than in C57BL\/6J at all ages examined and lower in CL\u2009\u00b1\u2009P embryos than in their normal littermates. The \u201cIAP antisense\u201d transcript was found in all A\/WySn embryos and was highest in CL\u2009\u00b1\u2009P embryos. The IAP at\n                    <jats:italic>Wnt9b<\/jats:italic>\n                    was generally unmethylated in CL\u2009\u00b1\u2009P embryos and approximately 50% methylated in normal littermates. Conclusion: The\n                    <jats:italic>clf1<\/jats:italic>\n                    mutation in A\/WySn is a \u201cmetastable epiallele\u201d, in which stochastic deficiency in some individuals of DNA methylation of a retrotransposon uniquely inserted near the\n                    <jats:italic>Wnt9b<\/jats:italic>\n                    gene allows transcriptional activity of the retrotransposon and interference with transcription from\n                    <jats:italic>Wnt9b<\/jats:italic>\n                    . Methylation of metastable epialleles should be investigated in human nonsyndromic CL\u2009\u00b1\u2009P. Birth Defects Research (Part A) 100:772\u2013788, 2014. \u00a9 2014 Wiley Periodicals, Inc.\n                  <\/jats:p>","DOI":"10.1002\/bdra.23320","type":"journal-article","created":{"date-parts":[[2014,9,25]],"date-time":"2014-09-25T02:51:01Z","timestamp":1411613461000},"page":"772-788","source":"Crossref","is-referenced-by-count":39,"title":["Epigenetic mechanism causes Wnt9b deficiency and nonsyndromic cleft lip and palate in the A\/WySn mouse strain"],"prefix":"10.1002","volume":"100","author":[{"given":"Diana M.","family":"Juriloff","sequence":"first","affiliation":[{"name":"Department of Medical Genetics University of British Columbia Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Muriel J.","family":"Harris","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics University of British Columbia Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dixie L.","family":"Mager","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics University of British Columbia Vancouver British Columbia Canada"},{"name":"Terry Fox Laboratory British Columbia Cancer Agency Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Liane","family":"Gagnier","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics University of British Columbia Vancouver British Columbia Canada"},{"name":"Terry Fox Laboratory British Columbia Cancer Agency Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2014,9,25]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20685"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2005.05.016"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddn121"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.1128\/MCB.8.3.1093"},{"key":"e_1_2_6_6_1","first-page":"369","article-title":"A comparative study of development during primary palate formation in A\/WySn, C57BL\/6, and their F1 crosses","volume":"6","author":"Ciriani D","year":"1986","journal-title":"J Craniofac Genet Dev Biol"},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.1002\/tera.1420020411"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/tera.1420410603"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1177\/10454411920040010201"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrg2933"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1203\/pdr.0b013e31804575f7"},{"key":"e_1_2_6_12_1","doi-asserted-by":"publisher","DOI":"10.1093\/nar\/gkh914"},{"key":"e_1_2_6_13_1","doi-asserted-by":"publisher","DOI":"10.1186\/1471-2164-14-48"},{"key":"e_1_2_6_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1369-5266(02)00233-9"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pgen.1002331"},{"key":"e_1_2_6_16_1","doi-asserted-by":"publisher","DOI":"10.1597\/13\u2010146"},{"key":"e_1_2_6_17_1","first-page":"336","article-title":"The genetics of cleft lip and cleft palate [review]","volume":"22","author":"Fraser FC","year":"1970","journal-title":"Am J Hum Genet"},{"key":"e_1_2_6_18_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-9926(199802)57:2<93::AID-TERA8>3.0.CO;2-7"},{"key":"e_1_2_6_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.alcohol.2011.02.308"},{"key":"e_1_2_6_20_1","doi-asserted-by":"publisher","DOI":"10.1597\/1545-1569(2001)038<0486:PAMAOA>2.0.CO;2"},{"key":"e_1_2_6_21_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.2009.069385"},{"key":"e_1_2_6_22_1","doi-asserted-by":"publisher","DOI":"10.4161\/epi.23438"},{"key":"e_1_2_6_23_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddp409"},{"key":"e_1_2_6_24_1","doi-asserted-by":"publisher","DOI":"10.1002\/mrd.20269"},{"key":"e_1_2_6_25_1","doi-asserted-by":"publisher","DOI":"10.1002\/dvdy.20646"},{"key":"e_1_2_6_26_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.075796"},{"key":"e_1_2_6_27_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20430"},{"key":"e_1_2_6_28_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20041"},{"key":"e_1_2_6_29_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20106"},{"key":"e_1_2_6_30_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.20302"},{"key":"e_1_2_6_31_1","doi-asserted-by":"publisher","DOI":"10.1002\/tera.1420140106"},{"key":"e_1_2_6_32_1","doi-asserted-by":"publisher","DOI":"10.1002\/tera.1420010208"},{"key":"e_1_2_6_33_1","unstructured":"KirkmanTW.1996. 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