{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,3]],"date-time":"2026-09-03T10:17:53Z","timestamp":1788430673200,"version":"build-2803163510"},"reference-count":45,"publisher":"The Company of Biologists","issue":"17","license":[{"start":{"date-parts":[[2026,9,1]],"date-time":"2026-09-01T00:00:00Z","timestamp":1788220800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/www.biologists.com\/user-licence-1-2\/"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"crossref","award":["DE028918"],"award-info":[{"award-number":["DE028918"]}],"id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Eye, Ear, Nose and Throat Foundation","award":["EE240302"],"award-info":[{"award-number":["EE240302"]}]},{"DOI":"10.13039\/100000057","name":"National Institute of General Medical Sciences","doi-asserted-by":"crossref","award":["P30GM145498"],"award-info":[{"award-number":["P30GM145498"]}],"id":[{"id":"10.13039\/100000057","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,9,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Craniosynostosis is a congenital disorder characterized by premature fusion of cranial sutures. Lambdoid synostosis is a rare and severe form of the disease, but its developmental etiology remains poorly understood. Although ectopic cartilage has been associated with lambdoid suture fusion, whether abnormal chondrocyte development is causative has not been directly tested. Here, we have investigated the role of platelet-derived growth factor receptor alpha (PDGFR\u03b1) signaling in chondrocyte development and lambdoid suture morphogenesis. Mesoderm-specific expression of an autoactivated Pdgfra allele (Pdgfra+\/K) caused premature fusion of the lambdoid and occipitointerparietal sutures, preceded by excessive cartilage expansion during embryogenesis. Spatial transcriptomic analysis identified a PDGFR\u03b1-dependent transcriptional program characterized by increased Col2a1, Sox9 and Sfrp2 expression, indicating maintenance of chondrocytes in a proliferative progenitor state. Consistent with this, PDGFR\u03b1-activated chondrocytes exhibited increased proliferation. Moreover, expression of Pdgfra+\/K in the Col2a1Cre lineage selectively induced occipitointerparietal suture fusion. Together, these findings demonstrate that PDGFR\u03b1 signaling promotes proliferative cartilage and impairs chondrocyte maturation, identifying dysregulated chondrogenesis as a developmental mechanism underlying lambdoid craniosynostosis.<\/jats:p>","DOI":"10.1242\/dev.205550","type":"journal-article","created":{"date-parts":[[2026,8,12]],"date-time":"2026-08-12T07:01:47Z","timestamp":1786518107000},"source":"Crossref","is-referenced-by-count":0,"title":["Regulation of lambdoid suture morphogenesis by PDGFR\u03b1-dependent chondrocyte programs"],"prefix":"10.1242","volume":"153","author":[{"given":"Garrett","family":"Bartoletti","sequence":"first","affiliation":[{"name":"School of Science and Engineering, Tulane University 1 Department of Cell and Molecular Biology , , New Orleans, LA 70118 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ryan","family":"Ebright","sequence":"additional","affiliation":[{"name":"School of Science and Engineering, Tulane University 1 Department of Cell and Molecular Biology , , New Orleans, LA 70118 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0003-5246-8406","authenticated-orcid":false,"given":"Haoyang","family":"Liang","sequence":"additional","affiliation":[{"name":"Tulane University School of Medicine 2 Department of Pathology and Laboratory Medicine , , New Orleans, LA 70112 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Josue","family":"Jaramillo","sequence":"additional","affiliation":[{"name":"Tulane School of Medicine 3 Department of Surgery , , New Orleans, LA 70112 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mimi","family":"Sammarco","sequence":"additional","affiliation":[{"name":"Tulane School of Medicine 3 Department of Surgery , , New Orleans, LA 70112 ,","place":["USA"]},{"name":"Limb and Musculoskeletal Regenerative Research Laboratory, Mayo Clinic 4 , Rochester, MN 55905 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaojiang","family":"Xu","sequence":"additional","affiliation":[{"name":"Tulane University School of Medicine 2 Department of Pathology and Laboratory Medicine , , New Orleans, LA 70112 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4717-8001","authenticated-orcid":false,"given":"Fenglei","family":"He","sequence":"additional","affiliation":[{"name":"School of Science and Engineering, Tulane University 1 Department of Cell and Molecular Biology , , New Orleans, LA 70118 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2026,9,3]]},"reference":[{"key":"2026090305455288000_DEV205550C1","doi-asserted-by":"publisher","first-page":"2813","DOI":"10.1101\/gad.1017802","article-title":"The transcription factor Sox9 has essential roles in successive steps of the chondrocyte differentiation pathway and is required for expression of Sox5 and Sox6","volume":"16","author":"Akiyama","year":"2002","journal-title":"Genes Dev."},{"key":"2026090305455288000_DEV205550C2","doi-asserted-by":"publisher","first-page":"1072","DOI":"10.1101\/gad.1171104","article-title":"Interactions between Sox9 and beta-catenin control chondrocyte differentiation","volume":"18","author":"Akiyama","year":"2004","journal-title":"Genes Dev."},{"key":"2026090305455288000_DEV205550C3","doi-asserted-by":"publisher","first-page":"1276","DOI":"10.1101\/gad.1653708","article-title":"Role of platelet-derived growth factors in physiology and medicine","volume":"22","author":"Andrae","year":"2008","journal-title":"Genes Dev."},{"key":"2026090305455288000_DEV205550C4","doi-asserted-by":"publisher","first-page":"dev201017","DOI":"10.1242\/dev.201017","article-title":"Cranium growth, patterning and homeostasis","volume":"149","author":"Ang","year":"2022","journal-title":"Development"},{"key":"2026090305455288000_DEV205550C5","doi-asserted-by":"publisher","first-page":"36","DOI":"10.1016\/j.ydbio.2020.08.004","article-title":"Pdgfra regulates multipotent cell differentiation towards chondrocytes via inhibiting Wnt9a\/beta-catenin pathway during chondrocranial cartilage development","volume":"466","author":"Bartoletti","year":"2020","journal-title":"Dev. 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