{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,11,8]],"date-time":"2025-11-08T17:41:43Z","timestamp":1762623703373,"version":"3.44.0"},"reference-count":26,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2015,12,29]],"date-time":"2015-12-29T00:00:00Z","timestamp":1451347200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","award":["F30 DE021945"],"award-info":[{"award-number":["F30 DE021945"]}],"id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","award":["T32 DE017607"],"award-info":[{"award-number":["T32 DE017607"]}],"id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000576","name":"British Society for Oral and Dental Research","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000576","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100005685","name":"Department of Genetics, University of Alabama at Birmingham","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100005685","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Journal Cellular Physiology"],"published-print":{"date-parts":[[2016,5]]},"abstract":"<jats:sec><jats:label\/><jats:p>Overexpression of transforming growth factor\u2010\u03b21 (TGF\u2010\u03b21) has been shown to lead to mineralization defects in both the enamel and dentin layers of teeth. A <jats:italic>TGFB1<\/jats:italic> point mutation (H222D), derived from published cases of Camurati\u2013Engelmann disease (CED), has been shown to constitutively activate TGF\u2010\u03b21, leading to excess bone matrix production. Although CED has been well documented in clinical case reports, there are no published studies on the effect of CED on the dentition. The objective of this study was to determine the dental manifestations of hyperactivated TGF\u2010\u03b21 signaling using an established mouse model of CED\u2010derived TGF\u2010\u03b21 mutation. Murine dental tissues were studied via radiography, micro\u2010CT, immunohistochemistry, and qRT\u2010PCR. Results showed that initial decreased dental mineralized tissue density is resolved. Proliferation assays of incisor pulp and alveolar bone cell cultures revealed that cells from transgenic animals displayed a reduced rate of growth compared to alveolar bone cultures from wild\u2010type mice. TGF\u2010\u03b2 family gene expression analysis indicated significant fold changes in the expression of <jats:italic>Alpl, Bmp2\u20135<\/jats:italic>, <jats:italic>Col\u20101<\/jats:italic>, <jats:italic>\u20102<\/jats:italic>, <jats:italic>\u20104<\/jats:italic>, and \u2010<jats:italic>6<\/jats:italic>, <jats:italic>Fgf<\/jats:italic>, <jats:italic>Mmp<\/jats:italic>, <jats:italic>Runx2<\/jats:italic>, <jats:italic>Tgfb3<\/jats:italic>, <jats:italic>Tfgbr3<\/jats:italic>, and <jats:italic>Vdr<\/jats:italic> genes. Assessment of SIBLINGs revealed downregulation of <jats:italic>Ibsp<\/jats:italic>, <jats:italic>Dmp1<\/jats:italic>, <jats:italic>Dspp<\/jats:italic>, <jats:italic>Mepe<\/jats:italic>, and <jats:italic>Spp1<\/jats:italic>, as well as reduced staining for BMP\u20102 and VDR in mesenchymal\u2010derived pulp tissue in CED animals. Treatment of dental pulp cells with recombinant human TGF\u2010\u03b21 resulted in increased SIBLING gene expression. Conclusions: Our results provide in vivo evidence suggesting that TFG\u2010\u03b21 mediates expression of important dentin extracellular matrix components secreted by dental pulp, and when unbalanced, may contribute to abnormal dentin disorders. J. Cell. Physiol. 231: 1106\u20131113, 2016. \u00a9 2015 Wiley Periodicals, Inc.<\/jats:p><\/jats:sec>","DOI":"10.1002\/jcp.25207","type":"journal-article","created":{"date-parts":[[2015,10,1]],"date-time":"2015-10-01T12:30:45Z","timestamp":1443702645000},"page":"1106-1113","source":"Crossref","is-referenced-by-count":2,"title":["Reduced Dentin Matrix Protein Expression in Camurati\u2010Engelmann Disease Transgenic Mouse Model"],"prefix":"10.1002","volume":"231","author":[{"given":"Angela","family":"Gullard","sequence":"first","affiliation":[{"name":"Department of Pathology University of Alabama at Birmingham Birmingham Alabama"},{"name":"Institute of Oral Health Research University of Alabama at Birmingham Birmingham Alabama"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Christina M.","family":"Croney","sequence":"additional","affiliation":[{"name":"Institute of Oral Health Research University of Alabama at Birmingham Birmingham Alabama"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xiangwei","family":"Wu","sequence":"additional","affiliation":[{"name":"Department of Surgery School of Medicine, Shihezi University Shihezi Xinjiang China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Olga","family":"Mamaeva","sequence":"additional","affiliation":[{"name":"Institute of Oral Health Research University of Alabama at Birmingham Birmingham Alabama"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Philip","family":"Sohn","sequence":"additional","affiliation":[{"name":"Institute of Oral Health Research University of Alabama at Birmingham Birmingham Alabama"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xu","family":"Cao","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery Johns Hopkins University Baltimore Maryland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mary","family":"MacDougall","sequence":"additional","affiliation":[{"name":"Institute of Oral Health Research University of Alabama at Birmingham Birmingham Alabama"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2015,12,29]]},"reference":[{"key":"e_1_2_6_2_1","first-page":"662","article-title":"Di Uno raro caso di osteite simmetrica ereditaria degli arti inferiori","volume":"6","author":"Camurati M","year":"1922","journal-title":"Chir Organi Mov"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M709492200"},{"key":"e_1_2_6_4_1","first-page":"132","article-title":"Case for diagnosis","author":"Cockayne EA","year":"1920","journal-title":"Proc R Soc Med 13 (Sect Study Dis Child)"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.1038\/316701a0"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.3109\/03008209509013704"},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.1359\/jbmr.1997.12.12.2040"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1600-0722.1998.tb02174.x"},{"key":"e_1_2_6_9_1","first-page":"1101","article-title":"Ein fall von osteopathia hyperostotica (sclerotisans) multiplex infantilis","volume":"39","author":"Engelmann G","year":"1929","journal-title":"Fortschr Geb Roentgenstr Nukl"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1600-0722.2006.00276.x"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10735-007-9148-8"},{"key":"e_1_2_6_12_1","doi-asserted-by":"publisher","DOI":"10.1080\/10408360490460933"},{"key":"e_1_2_6_13_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.37.4.245"},{"key":"e_1_2_6_14_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M208857200"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.2005.033522"},{"key":"e_1_2_6_16_1","first-page":"3","article-title":"Transforming growth factor\u2010beta 1 knockout mice. 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