{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,3]],"date-time":"2026-07-03T07:28:14Z","timestamp":1783063694564,"version":"3.54.6"},"reference-count":29,"publisher":"MDPI AG","issue":"17","license":[{"start":{"date-parts":[[2024,8,27]],"date-time":"2024-08-27T00:00:00Z","timestamp":1724716800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000002","name":"Division of Intramural Research, NIDCR via the Intramural Research Program of the National Institutes of Health, Department of Health and Human Services","doi-asserted-by":"publisher","award":["ZIA DE000649"],"award-info":[{"award-number":["ZIA DE000649"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"Division of Intramural Research, NIDCR via the Intramural Research Program of the National Institutes of Health, Department of Health and Human Services","doi-asserted-by":"publisher","award":["ZIADE000758"],"award-info":[{"award-number":["ZIADE000758"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"name":"NIH Medical Research Scholars Program","award":["ZIA DE000649"],"award-info":[{"award-number":["ZIA DE000649"]}]},{"name":"NIH Medical Research Scholars Program","award":["ZIADE000758"],"award-info":[{"award-number":["ZIADE000758"]}]},{"name":"NIH","award":["ZIA DE000649"],"award-info":[{"award-number":["ZIA DE000649"]}]},{"name":"NIH","award":["ZIADE000758"],"award-info":[{"award-number":["ZIADE000758"]}]},{"name":"Doris Duke Charitable Foundation","award":["ZIA DE000649"],"award-info":[{"award-number":["ZIA DE000649"]}]},{"name":"Doris Duke Charitable Foundation","award":["ZIADE000758"],"award-info":[{"award-number":["ZIADE000758"]}]},{"name":"Genentech","award":["ZIA DE000649"],"award-info":[{"award-number":["ZIA DE000649"]}]},{"name":"Genentech","award":["ZIADE000758"],"award-info":[{"award-number":["ZIADE000758"]}]},{"name":"American Association for Dental Research","award":["ZIA DE000649"],"award-info":[{"award-number":["ZIA DE000649"]}]},{"name":"American Association for Dental Research","award":["ZIADE000758"],"award-info":[{"award-number":["ZIADE000758"]}]},{"name":"Colgate-Palmolive Company","award":["ZIA DE000649"],"award-info":[{"award-number":["ZIA DE000649"]}]},{"name":"Colgate-Palmolive Company","award":["ZIADE000758"],"award-info":[{"award-number":["ZIADE000758"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Cells"],"abstract":"<jats:p>Fibrous dysplasia (FD) is a mosaic skeletal disorder involving the development of benign, expansile fibro-osseous lesions during childhood that cause deformity, fractures, pain, and disability. There are no well-established treatments for FD. Fibroblast activation protein (FAP\u03b1) is a serine protease expressed in pathological fibrotic tissues that has promising clinical applications as a biomarker and local pro-drug activator in several pathological conditions. In this study, we explored the expression of FAP in FD tissue and cells through published genetic expression datasets and measured circulating FAP\u03b1 in plasma samples from patients with FD and healthy donors. We found that FAP genetic expression was increased in FD tissue and cells, and present at higher concentrations in plasma from patients with FD compared to healthy donors. Moreover, FAP\u03b1 levels were correlated with skeletal disease burden in patients with FD. These findings support further investigation of FAP\u03b1 as a potential imaging and\/or biomarker of FD, as well as a pro-drug activator specific to FD tissue.<\/jats:p>","DOI":"10.3390\/cells13171434","type":"journal-article","created":{"date-parts":[[2024,8,27]],"date-time":"2024-08-27T06:19:01Z","timestamp":1724739541000},"page":"1434","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Fibroblast Activation Protein Is Expressed by Altered Osteoprogenitors and Associated to Disease Burden in Fibrous Dysplasia"],"prefix":"10.3390","volume":"13","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6925-2677","authenticated-orcid":false,"given":"Layne N.","family":"Raborn","sequence":"first","affiliation":[{"name":"Skeletal Disorders and Mineral Homeostasis Section, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5220-4046","authenticated-orcid":false,"given":"Zachary","family":"Michel","sequence":"additional","affiliation":[{"name":"Metabolic Bone Disorders Unit, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael T.","family":"Collins","sequence":"additional","affiliation":[{"name":"Skeletal Disorders and Mineral Homeostasis Section, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alison M.","family":"Boyce","sequence":"additional","affiliation":[{"name":"Metabolic Bone Disorders Unit, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3194-9780","authenticated-orcid":false,"given":"Luis F.","family":"de Castro","sequence":"additional","affiliation":[{"name":"Skeletal Disorders and Mineral Homeostasis Section, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2024,8,27]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"10","DOI":"10.1038\/s41413-023-00311-7","article-title":"RANKL inhibition reduces lesional cellularity and G\u03b1s variant expression and enables osteogenic maturation in fibrous dysplasia","volume":"12","author":"Whitlock","year":"2024","journal-title":"Bone Res."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"766","DOI":"10.1056\/NEJMc2214862","article-title":"Safety and Efficacy of Denosumab for Fibrous Dysplasia of Bone","volume":"388","author":"Michel","year":"2023","journal-title":"N. 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