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Both phenotypes could develop from defective migration and differentiation of osteoblasts in the skull bone primordia. The Wnt\/Planar cell polarity (Wnt\/PCP) signaling pathway regulates cell migration and movement in other tissues and led us to test the role of <jats:italic>Prickle1<\/jats:italic>, a core component of the Wnt\/PCP pathway, in the skull. For these studies, we used the missense allele of <jats:italic>Prickle1<\/jats:italic> named <jats:italic>Prickle1<\/jats:italic><jats:sup><jats:italic>Beetlejuice<\/jats:italic><\/jats:sup><jats:italic>(Prickle1<\/jats:italic><jats:sup><jats:italic>Bj<\/jats:italic><\/jats:sup>). The <jats:italic>Prickle1<\/jats:italic><jats:sup><jats:italic>Bj\/Bj<\/jats:italic><\/jats:sup> mutants are microcephalic and develop enlarged fontanelles between insufficient frontal bones, while the parietal bones are normal. <jats:italic>Prickle1<\/jats:italic><jats:sup><jats:italic>Bj\/Bj<\/jats:italic><\/jats:sup> mutants have several other craniofacial defects including a midline cleft lip, incompletely penetrant cleft palate, and decreased proximal-distal growth of the head. We observed decreased Wnt\/\u03b2-catenin and Hedgehog signaling in the frontal bone condensations of the <jats:italic>Prickle1<\/jats:italic><jats:sup><jats:italic>Bj\/Bj<\/jats:italic><\/jats:sup> mutants. Surprisingly, the smaller frontal bones do not result from defects in cell proliferation or death, but rather significantly delayed differentiation and decreased expression of migratory markers in the frontal bone osteoblast precursors. Our data suggests that <jats:italic>Prickle1<\/jats:italic> protein function contributes to both the migration and differentiation of osteoblast precursors in the frontal bone.<\/jats:p>","DOI":"10.1038\/s41598-018-36742-0","type":"journal-article","created":{"date-parts":[[2018,12,17]],"date-time":"2018-12-17T11:36:20Z","timestamp":1545046580000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":20,"title":["Prickle1 regulates differentiation of frontal bone osteoblasts"],"prefix":"10.1038","volume":"8","author":[{"given":"Yong","family":"Wan","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brandi","family":"Lantz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brian J.","family":"Cusack","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7921-8031","authenticated-orcid":false,"given":"Heather L.","family":"Szabo-Rogers","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2018,12,21]]},"reference":[{"key":"36742_CR1","doi-asserted-by":"publisher","first-page":"121","DOI":"10.1016\/j.ydbio.2008.07.016","volume":"322","author":"B McBratney-Owen","year":"2008","unstructured":"McBratney-Owen, B., Iseki, S., Bamforth, S. 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