{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,3]],"date-time":"2026-07-03T18:03:26Z","timestamp":1783101806706,"version":"3.54.6"},"reference-count":89,"publisher":"The Company of Biologists","issue":"19","license":[{"start":{"date-parts":[[2025,10,13]],"date-time":"2025-10-13T00:00:00Z","timestamp":1760313600000},"content-version":"vor","delay-in-days":12,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["R01DE030596"],"award-info":[{"award-number":["R01DE030596"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["R01DE027677"],"award-info":[{"award-number":["R01DE027677"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100007277","name":"Icahn School of Medicine at Mount Sinai","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100007277","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,10,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Craniofacial development depends on the formation of fibrous joints, or sutures, between skull bones. Premature fusion of sutures, or craniosynostosis, is a common human pathology. Ectopic Hedgehog (HH) signaling is one cause of craniosynostosis. Hhip encodes an inhibitor of HH ligands, and we previously identified coronal suture dysgenesis in embryonic Hhip\u2212\/\u2212 mice, in which suture mesenchyme was depleted between closely opposed but unfused osteogenic fronts at E18.5. Here, we report that the lambdoid suture fuses in Hhip\u2212\/\u2212 mice by E18.5. RNA-seq analysis of the Hhip\u2212\/\u2212 coronal and lambdoid sutures show that HH target gene expression, including Pthlh, is upregulated. Paradoxically, expression of Ihh is downregulated. We hypothesized that PTHLH, a negative regulator of Ihh expression, may reduce HH signaling to promote coronal suture patency and prevent fusion of the Hhip\u2212\/\u2212 coronal suture. We generated Hhip\u2212\/\u2212;Pthlh\u2212\/\u2212 embryos and found that coronal sutures are fusing by E18.5. Our results reveal a previously undescribed role for Pthlh in suture development and demonstrate suture-specific roles for HH inhibitors in maintaining suture patency.<\/jats:p>","DOI":"10.1242\/dev.204875","type":"journal-article","created":{"date-parts":[[2025,9,26]],"date-time":"2025-09-26T09:09:36Z","timestamp":1758877776000},"source":"Crossref","is-referenced-by-count":1,"title":["Differential regulation of coronal and lambdoid suture patency by PTHLH and HHIP activity in mice"],"prefix":"10.1242","volume":"152","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6677-5675","authenticated-orcid":false,"given":"Madrikha D.","family":"Saturne","sequence":"first","affiliation":[{"name":"Icahn School of Medicine at Mount Sinai 1 Department of Genetics and Genomic Sciences , , New York, NY 10029 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3412-221X","authenticated-orcid":false,"given":"Susan M.","family":"Motch Perrine","sequence":"additional","affiliation":[{"name":"Pennsylvania State University 2 Department of Anthropology , , University Park, PA 16802 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Qingyang","family":"Li","sequence":"additional","affiliation":[{"name":"Icahn School of Medicine at Mount Sinai 1 Department of Genetics and Genomic Sciences , , New York, NY 10029 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0239-5822","authenticated-orcid":false,"given":"Joan T.","family":"Richtsmeier","sequence":"additional","affiliation":[{"name":"Pennsylvania State University 2 Department of Anthropology , , University Park, PA 16802 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8983-5466","authenticated-orcid":false,"given":"Ethylin Wang","family":"Jabs","sequence":"additional","affiliation":[{"name":"Icahn School of Medicine at Mount Sinai 1 Department of Genetics and Genomic Sciences , , New York, NY 10029 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1376-6916","authenticated-orcid":false,"given":"Harm","family":"van Bakel","sequence":"additional","affiliation":[{"name":"Icahn School of Medicine at Mount Sinai 1 Department of Genetics and Genomic Sciences , , New York, NY 10029 ,","place":["USA"]},{"name":"Icahn Genomics Institute, Icahn School of Medicine at Mount Sinai 3 , New York, NY 10029 ,","place":["USA"]},{"name":"Icahn School of Medicine at Mount Sinai 4 Department of Microbiology , , New York, NY 10029 ,","place":["USA"]},{"name":"Icahn School of Medicine at Mount Sinai 5 Windreich Department of Artificial Intelligence and Human Health , , New York, NY 10029 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0717-6722","authenticated-orcid":false,"given":"Greg","family":"Holmes","sequence":"additional","affiliation":[{"name":"Icahn School of Medicine at Mount Sinai 1 Department of Genetics and Genomic Sciences , , New York, NY 10029 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2025,10,13]]},"reference":[{"key":"2025101305040572500_DEV204875C1","doi-asserted-by":"publisher","first-page":"3133","DOI":"10.1242\/dev.002709","article-title":"Regulation of skeletogenic differentiation in cranial dermal bone","volume":"134","author":"Abzhanov","year":"2007","journal-title":"Development"},{"key":"2025101305040572500_DEV204875C2","doi-asserted-by":"publisher","first-page":"29513","DOI":"10.1074\/jbc.M803681200","article-title":"MSX2 stimulates chondrocyte maturation by controlling Ihh expression","volume":"283","author":"Amano","year":"2008","journal-title":"J. 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