{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,9]],"date-time":"2026-07-09T10:11:42Z","timestamp":1783591902309,"version":"3.55.0"},"reference-count":64,"publisher":"Portland Press Ltd.","issue":"6","content-domain":{"domain":["portlandpress.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2016,12,15]]},"abstract":"<jats:p>Defects in the development of the mandible can lead to micrognathia, or small jaw, which manifests in ciliopathic conditions, such as orofaciodigital syndrome, Meckel\u2013Gruber syndrome, and Bardet\u2013Biedl syndrome. Although micrognathia occurs frequently in human and mouse ciliopathies, it has been difficult to pinpoint the underlying cellular causes. In this mini-review, we shed light on the tissue-specific contributions to ciliary dysfunction in the development of the mandible. First, we outline the steps involved in setting up the jaw primordium and subsequent steps in the outgrowth of the mandibular skeleton. We then determine the critical tissue interactions using mice carrying a conditional mutation in the cilia gene Ofd1. Our studies highlight the usefulness of the Ofd1 mouse model and illustrate long-term possibilities for understanding the cellular and biochemical events underlying micrognathia.<\/jats:p>","DOI":"10.1042\/bst20160241","type":"journal-article","created":{"date-parts":[[2016,12,2]],"date-time":"2016-12-02T17:30:14Z","timestamp":1480699814000},"page":"1753-1759","update-policy":"https:\/\/doi.org\/10.1042\/crossmark_policy","source":"Crossref","is-referenced-by-count":19,"title":["Micrognathia in mouse models of ciliopathies"],"prefix":"10.1042","volume":"44","author":[{"given":"Hadeel","family":"Adel Al-lami","sequence":"first","affiliation":[{"name":"Department of Craniofacial Development and Stem Cell Biology, King's College London Dental Institute, Guy's Hospital, London SE1 9RT, U.K."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"William B.","family":"Barrell","sequence":"additional","affiliation":[{"name":"Department of Craniofacial Development and Stem Cell Biology, King's College London Dental Institute, Guy's Hospital, London SE1 9RT, U.K."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karen J.","family":"Liu","sequence":"additional","affiliation":[{"name":"Department of Craniofacial Development and Stem Cell Biology, King's College London Dental Institute, Guy's Hospital, London SE1 9RT, U.K."}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"288","published-online":{"date-parts":[[2016,12,2]]},"reference":[{"key":"2021111720013933300_BST-2016-0241C1","doi-asserted-by":"publisher","first-page":"345","DOI":"10.1146\/annurev.cellbio.23.090506.123249","article-title":"Cilia and developmental signaling","volume":"23","author":"Eggenschwiler","year":"2007","journal-title":"Annu. 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