{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T14:00:28Z","timestamp":1791295228739,"version":"4.1.0"},"reference-count":23,"publisher":"Wiley","license":[{"start":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T00:00:00Z","timestamp":1791244800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T00:00:00Z","timestamp":1791244800000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["anatomypubs.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Developmental Dynamics"],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Sclerostin domain\u2010containing 1 (SOSTDC1) deficiency enhances BMP and Wnt signaling, causing supernumerary teeth formation. Competing hypotheses of this formation at different sites have been proposed, and the underlying molecular mechanisms and cell types remain poorly understood.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      We examined\n                      <jats:italic>Sostdc1<\/jats:italic>\n                      knockout effects on spatiotemporal expression of Wnt pathway genes in developing maxillary incisors. In wild type mice, we observed an inverse spatiotemporal expression of\n                      <jats:italic>Sostdc1<\/jats:italic>\n                      and\n                      <jats:italic>Wnt10a<\/jats:italic>\n                      . The knockout caused ectopic expression of\n                      <jats:italic>Wnt10a, Wnt6<\/jats:italic>\n                      ,\n                      <jats:italic>Lrp4<\/jats:italic>\n                      ,\n                      <jats:italic>Dkk1<\/jats:italic>\n                      , and\n                      <jats:italic>Sost<\/jats:italic>\n                      and produced a distinct subpopulation of mesenchymal cells. Their spatial transcriptomic analysis and validation of marker genes by in situ mRNA hybridization suggested an unexpected mix of osteogenic and odontogenic differentiation. These cells evolved at the rudimentary labial incisor outgrowth at ~E15.5. At E16.5, their population expanded toward the lingual cervical loop, where a separate supernumerary tooth formed at E17.5.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>The loss of Wnt and BMP signaling inhibition by SOSTDC1 causes aberrant mixed osteogenic and odontogenic differentiation of mesenchymal cells, contributing first to the labial outgrowth and then to the formation of the lingual supernumerary tooth. We hypothesize that this aberrant differentiation underlies the pathology, yet its direct functional role in the supernumerary tooth formation remains to be established.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/dvdy.70190","type":"journal-article","created":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T13:20:58Z","timestamp":1791292858000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Molecular insights into the role of Wnt signaling in supernumerary maxillary incisor formation"],"prefix":"10.1002","author":[{"given":"Resmi","family":"Raju","sequence":"first","affiliation":[{"name":"Section on Craniofacial Genetic Disorders <i>Eunice Kennedy Shriver<\/i> National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH)  Bethesda Maryland USA"},{"name":"Center for Engineering in Medicine and Surgery, Department of Surgery Massachusetts General Hospital  Boston Massachusetts USA"},{"name":"Department of Surgery Harvard Medical School  Boston Massachusetts USA"},{"name":"Shriners Hospital for Children  Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeremie Oliver","family":"Pi\u00f1a","sequence":"additional","affiliation":[{"name":"Section on Craniofacial Genetic Disorders <i>Eunice Kennedy Shriver<\/i> National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH)  Bethesda Maryland USA"},{"name":"Craniofacial Anomalies and Regeneration Section, National Institute of Dental and Craniofacial Research (NIDCR) National Institutes of Health (NIH)  Bethesda Maryland USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aye Chan","family":"Myo","sequence":"additional","affiliation":[{"name":"Section on Craniofacial Genetic Disorders <i>Eunice Kennedy Shriver<\/i> National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH)  Bethesda Maryland USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Parna","family":"Chattaraj","sequence":"additional","affiliation":[{"name":"Section on Craniofacial Genetic Disorders <i>Eunice Kennedy Shriver<\/i> National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH)  Bethesda Maryland USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Taishi","family":"Komori","sequence":"additional","affiliation":[{"name":"Molecular Biology of Bones and Teeth Section, National Institute of Dental and Craniofacial Research (NIDCR) National Institutes of Health (NIH)  Bethesda Maryland USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mitsuaki","family":"Ono","sequence":"additional","affiliation":[{"name":"Department of Molecular Biology and Biochemistry Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences  Okayama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ziyi","family":"Wang","sequence":"additional","affiliation":[{"name":"Department of Molecular Biology and Biochemistry Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences  Okayama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Didier","family":"Portilla","sequence":"additional","affiliation":[{"name":"Division of Nephrology, Center for Immunity, Inflammation and Regenerative Medicine University of Virginia  Charlottesville Virginia USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rena N.","family":"D'Souza","sequence":"additional","affiliation":[{"name":"Section on Craniofacial Genetic Disorders <i>Eunice Kennedy Shriver<\/i> National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH)  Bethesda Maryland USA"},{"name":"National Institute of Dental and Craniofacial Research (NIDCR), National Institutes of Health (NIH)  Bethesda Maryland USA"},{"name":"New York University College of Dentistry  New York City New York 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