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Loop-tail (Lp) mice carry a mutation in the Vangl2 gene, a member of the Wnt-planar cell polarity pathway. In Vangl2+\/Lp embryos, the mutation induces a failure in the completion of caudal neural tube closure, but only a small percentage of embryos develop open spina bifida. Here, we show that the majority of Vangl2+\/Lp embryos developed caudal closed NTDs and presented cellular aggregates that may facilitate the sealing of these defects. The cellular aggregates expressed neural crest cell markers and, using these as a readout, we describe a systematic method to assess the severity of the neural tube dorsal fusion failure. We observed that this defect worsened in combination with other NTD mutants, Daam1 and Grhl3. Besides, we found that in Vangl2+\/Lp embryos, these NTDs were resistant to maternal folic acid and inositol supplementation. Loop-tail mice provide a useful model for research on the molecular interactions involved in the development of open and closed NTDs and for the design of prevention strategies for these diseases.<\/jats:p>","DOI":"10.1242\/dmm.050175","type":"journal-article","created":{"date-parts":[[2023,8,17]],"date-time":"2023-08-17T14:02:31Z","timestamp":1692280951000},"source":"Crossref","is-referenced-by-count":3,"title":["The <i>loop-tail<\/i> mouse model displays open and closed caudal neural tube defects"],"prefix":"10.1242","volume":"16","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1447-8115","authenticated-orcid":false,"given":"Beatriz","family":"Fern\u00e1ndez-Santos","sequence":"first","affiliation":[{"name":"Institute of Biomedicine of Seville (IBiS)\/Virgen del Roc\u00edo University Hospital\/CSIC\/University of Seville , 41013 Seville , Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5490-0624","authenticated-orcid":false,"given":"Marta","family":"Reyes-Corral","sequence":"additional","affiliation":[{"name":"Institute of Biomedicine of Seville (IBiS)\/Virgen del Roc\u00edo University Hospital\/CSIC\/University of Seville , 41013 Seville , Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4795-3485","authenticated-orcid":false,"given":"Jos\u00e9 Manuel","family":"Caro-Vega","sequence":"additional","affiliation":[{"name":"Institute of Biomedicine of Seville (IBiS)\/Virgen del Roc\u00edo University Hospital\/CSIC\/University of Seville , 41013 Seville , Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6707-6825","authenticated-orcid":false,"given":"Miguel","family":"Lao-P\u00e9rez","sequence":"additional","affiliation":[{"name":"Institute of Biomedicine of Seville (IBiS)\/Virgen del Roc\u00edo University Hospital\/CSIC\/University of Seville , 41013 Seville , Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0008-3008-6446","authenticated-orcid":false,"given":"Claudia","family":"Vallejo-Grijalba","sequence":"additional","affiliation":[{"name":"Institute of Biomedicine of Seville (IBiS)\/Virgen del Roc\u00edo University Hospital\/CSIC\/University of Seville , 41013 Seville , Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0751-8593","authenticated-orcid":false,"given":"Cristina","family":"Mesa-Cruz","sequence":"additional","affiliation":[{"name":"Institute of Biomedicine of Seville (IBiS)\/Virgen del Roc\u00edo University Hospital\/CSIC\/University of Seville , 41013 Seville , Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1961-2723","authenticated-orcid":false,"given":"Francisco J.","family":"Mor\u00f3n","sequence":"additional","affiliation":[{"name":"Institute of Biomedicine of Seville (IBiS)\/Virgen del Roc\u00edo University Hospital\/CSIC\/University of Seville , 41013 Seville , Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7836-2659","authenticated-orcid":false,"given":"Patricia","family":"Ybot-Gonz\u00e1lez","sequence":"additional","affiliation":[{"name":"Institute of Biomedicine of Seville (IBiS)\/Virgen del Roc\u00edo University Hospital\/CSIC\/University of Seville , 41013 Seville , Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"237","published-online":{"date-parts":[[2023,8,29]]},"reference":[{"key":"2023082908034832600_DMM050175C1","doi-asserted-by":"publisher","first-page":"66","DOI":"10.1101\/gad.186601","article-title":"The transcription factor Sox10 is a key regulator of peripheral glial development","volume":"15","author":"Britsch","year":"2001","journal-title":"Genes Dev."},{"key":"2023082908034832600_DMM050175C2","doi-asserted-by":"publisher","first-page":"2244","DOI":"10.1002\/ajmg.a.37168","article-title":"When folic acid fails: insights from 20 years of neural tube defect surveillance in South Carolina","volume":"167","author":"Bupp","year":"2015","journal-title":"Am. 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