{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,19]],"date-time":"2026-03-19T10:43:07Z","timestamp":1773916987423,"version":"3.50.1"},"reference-count":36,"publisher":"Elsevier BV","license":[{"start":{"date-parts":[[2026,4,1]],"date-time":"2026-04-01T00:00:00Z","timestamp":1775001600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2026,4,1]],"date-time":"2026-04-01T00:00:00Z","timestamp":1775001600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2027,1,16]],"date-time":"2027-01-16T00:00:00Z","timestamp":1800057600000},"content-version":"am","delay-in-days":290,"URL":"http:\/\/www.elsevier.com\/open-access\/userlicense\/1.0\/"}],"funder":[{"DOI":"10.13039\/100000054","name":"National Cancer Institute","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000054","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100002429","name":"Amgen","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100002429","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["clinicalkey.com","clinicalkey.com.au","clinicalkey.es","clinicalkey.fr","clinicalkey.jp","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Bone"],"published-print":{"date-parts":[[2026,4]]},"DOI":"10.1016\/j.bone.2026.117792","type":"journal-article","created":{"date-parts":[[2026,1,12]],"date-time":"2026-01-12T16:35:51Z","timestamp":1768235751000},"page":"117792","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":0,"special_numbering":"C","title":["G\u03b1s variant detection discerns skeletal cell types involved in fibrous dysplasia pathogenesis"],"prefix":"10.1016","volume":"205","author":[{"given":"Zachary","family":"Michel","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Vivian","family":"MacDonald","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Aleena","family":"Arif","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ibrahim I.","family":"Elbashir","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jonathan","family":"Freeman","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Zubeyir H.","family":"Gun","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Charles","family":"Osamor","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jocelyn","family":"Taylor","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michael T.","family":"Collins","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kelly L.","family":"Roszko","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Luis F.","family":"de Castro","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Vardit","family":"Kram","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Alison M.","family":"Boyce","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"78","reference":[{"issue":"2","key":"10.1016\/j.bone.2026.117792_bb0005","doi-asserted-by":"crossref","first-page":"345","DOI":"10.1210\/endrev\/bnz011","article-title":"Fibrous Dysplasia\/McCune-Albright Syndrome: A Rare, Mosaic Disease of G\u03b1s Activation","volume":"41","author":"Boyce","year":"2020","journal-title":"Endocr. Rev."},{"issue":"5","key":"10.1016\/j.bone.2026.117792_bb0010","doi-asserted-by":"crossref","first-page":"1482","DOI":"10.1210\/clinem\/dgab053","article-title":"Genotype-Phenotype Correlation in Fibrous Dysplasia\/McCune-Albright Syndrome","volume":"106","author":"Zhadina","year":"2021","journal-title":"J. Clin. Endocrinol. Metab."},{"issue":"9","key":"10.1016\/j.bone.2026.117792_bb0015","doi-asserted-by":"crossref","first-page":"660","DOI":"10.1097\/PAI.0000000000000242","article-title":"GNAS Mutations in Fibrous Dysplasia: A Comparative Study of Standard Sequencing and Locked Nucleic Acid PCR Sequencing on Decalcified and Nondecalcified Formalin-fixed Paraffin-embedded Tissues","volume":"24","author":"Jour","year":"2016","journal-title":"Appl. Immunohistochem. Mol. Morphol."},{"issue":"6","key":"10.1016\/j.bone.2026.117792_bb0020","doi-asserted-by":"crossref","first-page":"691","DOI":"10.1111\/j.1365-2559.2007.02676.x","article-title":"A sensitive mutation-specific screening technique for GNAS1 mutations in cases of fibrous dysplasia: the first report of a codon 227 mutation in bone","volume":"50","author":"Idowu","year":"2007","journal-title":"Histopathology"},{"issue":"3","key":"10.1016\/j.bone.2026.117792_bb0025","first-page":"1059","article-title":"Increased proliferation of osteoblastic cells expressing the activating Gs alpha mutation in monostotic and polyostotic fibrous dysplasia","volume":"150","author":"Marie","year":"1997","journal-title":"Am. J. Pathol."},{"issue":"6","key":"10.1016\/j.bone.2026.117792_bb0030","doi-asserted-by":"crossref","first-page":"1023","DOI":"10.1242\/dev.102210","article-title":"Skeletal stem cells","volume":"142","author":"Bianco","year":"2015","journal-title":"Development"},{"issue":"8","key":"10.1016\/j.bone.2026.117792_bb0035","doi-asserted-by":"crossref","first-page":"1737","DOI":"10.1172\/JCI2361","article-title":"Reproduction of human fibrous dysplasia of bone in immunocompromised mice by transplanted mosaics of normal and Gsalpha-mutated skeletal progenitor cells","volume":"101","author":"Bianco","year":"1998","journal-title":"J. Clin. Invest."},{"issue":"5","key":"10.1016\/j.bone.2026.117792_bb0040","doi-asserted-by":"crossref","first-page":"1103","DOI":"10.1359\/jbmr.091036","article-title":"Transfer, analysis, and reversion of the fibrous dysplasia cellular phenotype in human skeletal progenitors","volume":"25","author":"Piersanti","year":"2010","journal-title":"J. Bone Miner. Res."},{"issue":"9","key":"10.1016\/j.bone.2026.117792_bb0045","doi-asserted-by":"crossref","first-page":"2067","DOI":"10.1016\/j.ajhg.2025.07.018","article-title":"Single-cell analysis of human fibrous dysplasia bone reveals a fibrotic transcriptome and GNAS variants in endothelial, perivascular, and stromal cells","volume":"112","author":"Wentworth","year":"2025","journal-title":"Am. J. Hum. Genet."},{"issue":"2","key":"10.1016\/j.bone.2026.117792_bb0050","doi-asserted-by":"crossref","first-page":"111","DOI":"10.1007\/BF02893455","article-title":"Fibrocartilaginous dysplasia (fibrous dysplasia with extensive cartilaginous differentiation)","volume":"12","author":"Vargas-Gonzalez","year":"2006","journal-title":"Pathol. Oncol. Res."},{"issue":"1","key":"10.1016\/j.bone.2026.117792_bb0055","doi-asserted-by":"crossref","first-page":"51","DOI":"10.1007\/s00256-003-0718-x","article-title":"Fibrous dysplasia with cartilaginous differentiation (\u201cfibrocartilaginous dysplasia\u201d): a review, with an illustrative case followed for 18 years","volume":"33","author":"Kyriakos","year":"2004","journal-title":"Skeletal Radiol."},{"issue":"1","key":"10.1016\/j.bone.2026.117792_bb0060","doi-asserted-by":"crossref","first-page":"126","DOI":"10.1159\/000443476","article-title":"Fibrous Dysplasia with Massive Cartilaginous Differentiation (Fibrocartilaginous Dysplasia) in the Proximal Femur: A Case Report and Review of the Literature","volume":"9","author":"Morioka","year":"2016","journal-title":"Case Rep Oncol"},{"key":"10.1016\/j.bone.2026.117792_bb0065","doi-asserted-by":"crossref","DOI":"10.1136\/jcp-2024-209626","article-title":"Molecular confirmation that fibrocartilaginous dysplasia is a variant of fibrous dysplasia","author":"Zhou","year":"2024","journal-title":"J. Clin. Pathol."},{"issue":"1","key":"10.1016\/j.bone.2026.117792_bb0070","doi-asserted-by":"crossref","first-page":"10","DOI":"10.1038\/s41413-023-00311-7","article-title":"RANKL inhibition reduces lesional cellularity and G\u03b1(s) variant expression and enables osteogenic maturation in fibrous dysplasia","volume":"12","author":"de Castro","year":"2024","journal-title":"Bone Res"},{"issue":"4","key":"10.1016\/j.bone.2026.117792_bb0075","doi-asserted-by":"crossref","DOI":"10.1016\/j.isci.2023.106470","article-title":"An inducible explant model of osteoclast-osteoprogenitor coordination in exacerbated osteoclastogenesis","volume":"26","author":"Whitlock","year":"2023","journal-title":"iScience"},{"issue":"8","key":"10.1016\/j.bone.2026.117792_bb0080","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":"de Castro","year":"2023","journal-title":"N. Engl. J. Med."},{"issue":"3","key":"10.1016\/j.bone.2026.117792_bb0085","doi-asserted-by":"crossref","first-page":"434","DOI":"10.1016\/S8756-3282(03)00064-4","article-title":"Osteoclastogenesis in fibrous dysplasia of bone: in situ and in vitro analysis of IL-6 expression","volume":"33","author":"Riminucci","year":"2003","journal-title":"Bone"},{"issue":"9","key":"10.1016\/j.bone.2026.117792_bb0090","doi-asserted-by":"crossref","DOI":"10.3390\/cells13090774","article-title":"Transcriptomic Signature and Pro-Osteoclastic Secreted Factors of Abnormal Bone-Marrow Stromal Cells in Fibrous Dysplasia","volume":"13","author":"Michel","year":"2024","journal-title":"Cells"},{"issue":"1","key":"10.1016\/j.bone.2026.117792_bb0095","doi-asserted-by":"crossref","first-page":"139","DOI":"10.1186\/s13023-019-1102-9","article-title":"Best practice management guidelines for fibrous dysplasia\/McCune-Albright syndrome: a consensus statement from the FD\/MAS international consortium","volume":"14","author":"Javaid","year":"2019","journal-title":"Orphanet J. Rare Dis."},{"issue":"1","key":"10.1016\/j.bone.2026.117792_bb0100","doi-asserted-by":"crossref","DOI":"10.1038\/s41598-017-17204-5","article-title":"QuPath: Open source software for digital pathology image analysis","volume":"7","author":"Bankhead","year":"2017","journal-title":"Sci. Rep."},{"issue":"1","key":"10.1016\/j.bone.2026.117792_bb0105","doi-asserted-by":"crossref","DOI":"10.1038\/s41598-017-12651-6","article-title":"Small leucine rich proteoglycans, a novel link to osteoclastogenesis","volume":"7","author":"Kram","year":"2017","journal-title":"Sci. Rep."},{"issue":"3","key":"10.1016\/j.bone.2026.117792_bb0110","doi-asserted-by":"crossref","first-page":"443","DOI":"10.1002\/jbmr.4766","article-title":"Identification of GNAS Variants in Circulating Cell-Free DNA from Patients with Fibrous Dysplasia\/McCune Albright Syndrome","volume":"38","author":"Roszko","year":"2023","journal-title":"J. Bone Miner. Res."},{"key":"10.1016\/j.bone.2026.117792_bb0115","doi-asserted-by":"crossref","DOI":"10.1136\/jcp-2024-210007","article-title":"New evidence for fibrocartilaginous dysplasia representing a variant of fibrous dysplasia","author":"De Andrea","year":"2025","journal-title":"J. Clin. Pathol."},{"issue":"16","key":"10.1016\/j.bone.2026.117792_bb0120","doi-asserted-by":"crossref","first-page":"1921","DOI":"10.1093\/hmg\/ddm139","article-title":"GNAS transcripts in skeletal progenitors: evidence for random asymmetric allelic expression of Gs alpha","volume":"16","author":"Michienzi","year":"2007","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.bone.2026.117792_bb0125","doi-asserted-by":"crossref","first-page":"2683","DOI":"10.1038\/srep02683","article-title":"Hypoxia enhances chondrogenesis and prevents terminal differentiation through PI3K\/Akt\/FoxO dependent anti-apoptotic effect","volume":"3","author":"Lee","year":"2013","journal-title":"Sci. Rep."},{"issue":"2","key":"10.1016\/j.bone.2026.117792_bb0130","doi-asserted-by":"crossref","first-page":"458","DOI":"10.1016\/j.bbrc.2008.09.154","article-title":"Dynamic compression can inhibit chondrogenesis of mesenchymal stem cells","volume":"377","author":"Thorpe","year":"2008","journal-title":"Biochem. Biophys. Res. Commun."},{"issue":"5","key":"10.1016\/j.bone.2026.117792_bb0135","doi-asserted-by":"crossref","first-page":"509","DOI":"10.1007\/s002560050126","article-title":"Fibrocartilaginous dysplasia","volume":"25","author":"Hermann","year":"1996","journal-title":"Skeletal Radiol."},{"issue":"7","key":"10.1016\/j.bone.2026.117792_bb0140","doi-asserted-by":"crossref","first-page":"1462","DOI":"10.1002\/jbmr.1603","article-title":"Denosumab treatment for fibrous dysplasia","volume":"27","author":"Boyce","year":"2012","journal-title":"J. Bone Miner. Res."},{"issue":"2","key":"10.1016\/j.bone.2026.117792_bb0145","doi-asserted-by":"crossref","first-page":"290","DOI":"10.1002\/jbmr.3602","article-title":"Activation of RANK\/RANKL\/OPG Pathway Is Involved in the Pathophysiology of Fibrous Dysplasia and Associated With Disease Burden","volume":"34","author":"de Castro","year":"2019","journal-title":"J. Bone Miner. Res."},{"issue":"1","key":"10.1016\/j.bone.2026.117792_bb0150","doi-asserted-by":"crossref","first-page":"65","DOI":"10.1002\/jbmr.2599","article-title":"Wnt Signaling Inhibits Osteoclast Differentiation by Activating Canonical and Noncanonical cAMP\/PKA Pathways","volume":"31","author":"Weivoda","year":"2016","journal-title":"J. Bone Miner. Res."},{"key":"10.1016\/j.bone.2026.117792_bb0155","doi-asserted-by":"crossref","DOI":"10.1038\/srep45140","article-title":"Gs\u03b1 Controls Cortical Bone Quality by Regulating Osteoclast Differentiation via cAMP\/PKA and \u03b2-Catenin Pathways","volume":"7","author":"Ramaswamy","year":"2017","journal-title":"Sci. Rep."},{"issue":"12","key":"10.1016\/j.bone.2026.117792_bb0160","doi-asserted-by":"crossref","first-page":"1472","DOI":"10.1038\/nm.3733","article-title":"Age-related mutations associated with clonal hematopoietic expansion and malignancies","volume":"20","author":"Xie","year":"2014","journal-title":"Nat. Med."},{"key":"10.1016\/j.bone.2026.117792_bb0165","doi-asserted-by":"crossref","first-page":"14","DOI":"10.1016\/j.exphem.2017.09.004","article-title":"The GNAS(R201C) mutation associated with clonal hematopoiesis supports transplantable hematopoietic stem cell activity","volume":"57","author":"Ostrander","year":"2018","journal-title":"Exp. Hematol."},{"issue":"1","key":"10.1016\/j.bone.2026.117792_bb0170","doi-asserted-by":"crossref","first-page":"237","DOI":"10.1007\/s00198-017-4217-7","article-title":"Bone marrow failure and extramedullary hematopoiesis in McCune-Albright syndrome","volume":"29","author":"Robinson","year":"2018","journal-title":"Osteoporos. Int."},{"issue":"1","key":"10.1016\/j.bone.2026.117792_bb0175","doi-asserted-by":"crossref","first-page":"40","DOI":"10.1159\/000507442","article-title":"McCune Albright Syndrome: Gastrointestinal Polyps and Platelet Dysfunction over 12 Years","volume":"93","author":"Grob","year":"2020","journal-title":"Horm. Res. Paediatr."},{"key":"10.1016\/j.bone.2026.117792_bb0180","doi-asserted-by":"crossref","DOI":"10.1177\/20420188251347350","article-title":"Update on the medical management of fibrous dysplasia of the bone","volume":"16","author":"Wentworth","year":"2025","journal-title":"Ther. Adv. Endocrinol. Metab."}],"container-title":["Bone"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S8756328226000189?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S8756328226000189?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2026,3,19]],"date-time":"2026-03-19T05:28:54Z","timestamp":1773898134000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S8756328226000189"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,4]]},"references-count":36,"alternative-id":["S8756328226000189"],"URL":"https:\/\/doi.org\/10.1016\/j.bone.2026.117792","relation":{},"ISSN":["8756-3282"],"issn-type":[{"value":"8756-3282","type":"print"}],"subject":[],"published":{"date-parts":[[2026,4]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"G\u03b1s variant detection discerns skeletal cell types involved in fibrous dysplasia pathogenesis","name":"articletitle","label":"Article Title"},{"value":"Bone","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.bone.2026.117792","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"Published by Elsevier Inc.","name":"copyright","label":"Copyright"}],"article-number":"117792"}}