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We investigated multiple mouse models of human ciliopathies (including\n                    <jats:italic>Tctn2, Cc2d2a,<\/jats:italic>\n                    and\n                    <jats:italic>Tmem231<\/jats:italic>\n                    mutants) and discovered that each displays hypotelorism, a narrowing of the midface. As early in development as the end of gastrulation,\n                    <jats:italic>Tctn2<\/jats:italic>\n                    mutants displayed reduced activation of the Hedgehog (HH) pathway in the prechordal plate, the head organizer. This prechordal plate defect preceded a reduction of HH pathway activation and\n                    <jats:italic>Shh<\/jats:italic>\n                    expression in the adjacent neurectoderm. Concomitant with the reduction of HH pathway activity,\n                    <jats:italic>Tctn2<\/jats:italic>\n                    mutants exhibited increased cell death in the neurectoderm and facial ectoderm, culminating in a collapse of the facial midline. Enhancing HH signaling by decreasing the gene dosage of a negative regulator of the pathway,\n                    <jats:italic>Ptch1<\/jats:italic>\n                    , decreased cell death and rescued the midface defect in both\n                    <jats:italic>Tctn2<\/jats:italic>\n                    and\n                    <jats:italic>Cc2d2a<\/jats:italic>\n                    mutants. These results reveal that ciliary HH signaling mediates communication between the prechordal plate and the neurectoderm to provide cellular survival cues essential for development of the facial midline.\n                  <\/jats:p>","DOI":"10.7554\/elife.68558","type":"journal-article","created":{"date-parts":[[2021,10,21]],"date-time":"2021-10-21T08:00:23Z","timestamp":1634803223000},"update-policy":"https:\/\/doi.org\/10.7554\/elife.68558","source":"Crossref","is-referenced-by-count":12,"title":["Ciliary Hedgehog signaling regulates cell survival to build the facial midline"],"prefix":"10.7554","volume":"10","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1479-9322","authenticated-orcid":true,"given":"Shaun R","family":"Abrams","sequence":"first","affiliation":[{"name":"Department of Biochemistry and Biophysics, Cardiovascular Research Institute","place":["San Francisco, United States"]},{"name":"Oral and Craniofacial Sciences Program, School of Dentistry","place":["San 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