{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,4]],"date-time":"2026-10-04T16:28:58Z","timestamp":1791131338191,"version":"4.1.0"},"reference-count":38,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2011,2,18]],"date-time":"2011-02-18T00:00:00Z","timestamp":1297987200000},"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":[[2011,4]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p><jats:italic>Vangl2<\/jats:italic> forms part of the planar cell polarity signalling pathway and is the gene defective in the <jats:italic>Looptail<\/jats:italic> (<jats:italic>Lp<\/jats:italic>) mouse mutant. Two previously described alleles, <jats:italic>Lp<\/jats:italic> and <jats:italic>Lp<jats:sup>m1Jus<\/jats:sup><\/jats:italic>, segregate in a semi\u2010dominant fashion, with heterozygotes displaying the looped\u2010tail appearance, while homozygotes show the neural tube defect called craniorachischisis. Here, we report a novel experimentally induced allele, <jats:italic>Lp<jats:sup>m2Jus<\/jats:sup><\/jats:italic>, that carries a missense mutation, R259L, in <jats:italic>Vangl2<\/jats:italic>. This mutation was specific to the <jats:italic>Lp<\/jats:italic> phenotype and absent from both parental strains and 28 other inbred strains. Notably, this mutation segregates in a recessive manner with all heterozygotes appearing normal and 47% of homozygotes showing a looped\u2010tail. Homozygous <jats:italic>Lp<jats:sup>m2Jus<\/jats:sup><\/jats:italic> embryos showed spina bifida in 12%. <jats:italic>Lp<jats:sup>m2Jus<\/jats:sup><\/jats:italic> genetically interacts with <jats:italic>Lp<\/jats:italic> with 77% of compound heterozygotes displaying craniorachischisis. <jats:italic>Vangl2<jats:sup>R259L<\/jats:sup><\/jats:italic> behaved like the wild\u2010type allele in overexpression and morpholino knockdown\/rescue assays in zebrafish embryos. These data suggest that <jats:italic>Lp<jats:sup>m2Jus<\/jats:sup><\/jats:italic> represents a new hypomorphic allele of <jats:italic>Lp<\/jats:italic>. Developmental Dynamics 240:839\u2013849, 2011. \u00a9 2011 Wiley\u2010Liss, Inc.<\/jats:p>","DOI":"10.1002\/dvdy.22577","type":"journal-article","created":{"date-parts":[[2011,2,18]],"date-time":"2011-02-18T10:40:44Z","timestamp":1298025644000},"page":"839-849","source":"Crossref","is-referenced-by-count":17,"title":["A novel hypomorphic <i>Looptail<\/i> allele at the planar cell polarity <i>Vangl2<\/i> gene"],"prefix":"10.1002","volume":"240","author":[{"given":"Marie\u2010Claude","family":"Guyot","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ciprian 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