{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,25]],"date-time":"2026-09-25T22:27:44Z","timestamp":1790375264606,"version":"4.1.0"},"reference-count":37,"publisher":"Elsevier BV","issue":"11","license":[{"start":{"date-parts":[[2022,11,1]],"date-time":"2022-11-01T00:00:00Z","timestamp":1667260800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2022,11,1]],"date-time":"2022-11-01T00:00:00Z","timestamp":1667260800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"}],"content-domain":{"domain":["aacejournalendocrinepractice.org","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Endocrine Practice"],"published-print":{"date-parts":[[2022,11]]},"DOI":"10.1016\/j.eprac.2022.07.012","type":"journal-article","created":{"date-parts":[[2022,8,8]],"date-time":"2022-08-08T18:00:46Z","timestamp":1659981646000},"page":"1196-1201","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":6,"title":["Denosumab Use in Adults With Fibrous Dysplasia: Case Reports and Review of the Literature"],"prefix":"10.1016","volume":"28","author":[{"given":"Chermaine","family":"Hung","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2405-8963","authenticated-orcid":false,"given":"Amal","family":"Shibli-Rahhal","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"issue":"3","key":"10.1016\/j.eprac.2022.07.012_bib1","first-page":"750","article-title":"An activating Gs alpha mutation is present in fibrous dysplasia of bone in the McCune-Albright syndrome","volume":"79","author":"Shenker","year":"1994","journal-title":"J\u00a0Clin Endocrinol Metab"},{"key":"10.1016\/j.eprac.2022.07.012_bib2","doi-asserted-by":"crossref","first-page":"727","DOI":"10.1056\/NEJM193704292161701","article-title":"Syndrome characterized by osteitis fibrosa disseminata, areas of pigmentation and endocrine dysfunction, with precocious puberty in females. Report of five cases","volume":"216","author":"Albright","year":"1937","journal-title":"N\u00a0Engl J Med"},{"issue":"6236","key":"10.1016\/j.eprac.2022.07.012_bib3","doi-asserted-by":"crossref","first-page":"692","DOI":"10.1038\/340692a0","article-title":"GTPase inhibiting mutations activate the alpha chain of Gs and stimulate adenylyl cyclase in human pituitary tumours","volume":"340","author":"Landis","year":"1989","journal-title":"Nature"},{"issue":"3","key":"10.1016\/j.eprac.2022.07.012_bib4","first-page":"981","article-title":"Cellular and molecular basis of fibrous dysplasia","volume":"16","author":"Marie","year":"2001","journal-title":"Histol Histopathol"},{"issue":"3","key":"10.1016\/j.eprac.2022.07.012_bib5","doi-asserted-by":"crossref","first-page":"E428","DOI":"10.1073\/pnas.1713710115","article-title":"Expression of an active G\u03b1 s mutant in skeletal stem cells is sufficient and necessary for fibrous dysplasia initiation and maintenance","volume":"115","author":"Zhao","year":"2018","journal-title":"Proc Natl Acad Sci U S A"},{"issue":"5","key":"10.1016\/j.eprac.2022.07.012_bib6","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\u00a0Bone Miner Res"},{"issue":"3","key":"10.1016\/j.eprac.2022.07.012_bib7","doi-asserted-by":"crossref","first-page":"E418","DOI":"10.1073\/pnas.1714313114","article-title":"Induced GnasR201H expression from the endogenous Gnas locus causes fibrous dysplasia by up-regulating Wnt\/\u03b2-catenin signaling","volume":"115","author":"Khan","year":"2018","journal-title":"Proc Natl Acad Sci U S A"},{"issue":"2","key":"10.1016\/j.eprac.2022.07.012_bib8","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\u00a0Bone Miner Res"},{"issue":"1","key":"10.1016\/j.eprac.2022.07.012_bib9","doi-asserted-by":"crossref","first-page":"30","DOI":"10.1172\/JCI118773","article-title":"Increased IL-6-production by cells isolated from the fibrous bone dysplasia tissues in patients with McCune-Albright syndrome","volume":"98","author":"Yamamoto","year":"1996","journal-title":"J\u00a0Clin Invest"},{"issue":"3","key":"10.1016\/j.eprac.2022.07.012_bib10","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\u00a0vitro analysis of IL-6 expression","volume":"33","author":"Riminucci","year":"2003","journal-title":"Bone"},{"issue":"23","key":"10.1016\/j.eprac.2022.07.012_bib11","doi-asserted-by":"crossref","first-page":"1546","DOI":"10.1056\/NEJM199506083322304","article-title":"Increased expression of the c-fos proto-oncogene in bone from patients with fibrous dysplasia","volume":"332","author":"Candeliere","year":"1995","journal-title":"N\u00a0Engl J Med"},{"issue":"2","key":"10.1016\/j.eprac.2022.07.012_bib12","doi-asserted-by":"crossref","first-page":"249","DOI":"10.1002\/(SICI)1096-9896(199901)187:2<249::AID-PATH222>3.0.CO;2-J","article-title":"The histopathology of fibrous dysplasia of bone in patients with activating mutations of the Gs alpha gene: site-specific patterns and recurrent histological hallmarks","volume":"187","author":"Riminucci","year":"1999","journal-title":"J\u00a0Pathol"},{"issue":"5","key":"10.1016\/j.eprac.2022.07.012_bib13","doi-asserted-by":"crossref","first-page":"R83","DOI":"10.1530\/EJE-19-0969","article-title":"Diagnosis of endocrine disease: mosaic disorders of FGF23 excess: fibrous dysplasia\/McCune-Albright syndrome and cutaneous skeletal hypophosphatemia syndrome","volume":"182","author":"de Castro","year":"2020","journal-title":"Eur J Endocrinol"},{"issue":"4","key":"10.1016\/j.eprac.2022.07.012_bib14","doi-asserted-by":"crossref","first-page":"571","DOI":"10.1359\/JBMR.0301262","article-title":"Fracture incidence in polyostotic fibrous dysplasia and the McCune-Albright syndrome","volume":"19","author":"Leet","year":"2004","journal-title":"J\u00a0Bone Miner Res"},{"issue":"Suppl 1","key":"10.1016\/j.eprac.2022.07.012_bib15","doi-asserted-by":"crossref","first-page":"S1","DOI":"10.1186\/1750-1172-7-S1-S1","article-title":"The surgical management of fibrous dysplasia of bone","volume":"7","author":"Stanton","year":"2012","journal-title":"Orphanet J Rare Dis"},{"issue":"1","key":"10.1016\/j.eprac.2022.07.012_bib16","doi-asserted-by":"crossref","first-page":"57","DOI":"10.1007\/s00198-007-0425-x","article-title":"Pain in fibrous dysplasia of bone: age-related changes and the anatomical distribution of skeletal lesions","volume":"19","author":"Kelly","year":"2008","journal-title":"Osteoporos Int"},{"issue":"4","key":"10.1016\/j.eprac.2022.07.012_bib17","doi-asserted-by":"crossref","first-page":"401","DOI":"10.1080\/17453674.2019.1608117","article-title":"Pain in fibrous dysplasia: relationship with anatomical and clinical features","volume":"90","author":"Majoor","year":"2019","journal-title":"Acta Orthop"},{"issue":"Suppl 1","key":"10.1016\/j.eprac.2022.07.012_bib18","doi-asserted-by":"crossref","first-page":"S3","DOI":"10.1186\/1750-1172-7-S1-S3","article-title":"Pathophysiology and medical treatment of pain in fibrous dysplasia of bone","volume":"7","author":"Chapurlat","year":"2012","journal-title":"Orphanet J Rare Dis"},{"issue":"6","key":"10.1016\/j.eprac.2022.07.012_bib19","doi-asserted-by":"crossref","first-page":"e2258","DOI":"10.1210\/clinem\/dgac120","article-title":"Neuropathic-like pain in fibrous dysplasia\/McCune-Albright syndrome","volume":"107","author":"Spencer","year":"2022","journal-title":"J\u00a0Clin Endocrinol Metab"},{"issue":"1","key":"10.1016\/j.eprac.2022.07.012_bib20","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"},{"key":"10.1016\/j.eprac.2022.07.012_bib21","doi-asserted-by":"crossref","DOI":"10.1016\/j.bone.2020.115784","article-title":"Bisphosphonates for the treatment of fibrous dysplasia of bone","volume":"143","author":"Chapurlat","year":"2021","journal-title":"Bone"},{"issue":"11","key":"10.1016\/j.eprac.2022.07.012_bib22","doi-asserted-by":"crossref","first-page":"4133","DOI":"10.1210\/jc.2014-1371","article-title":"A\u00a0randomized, double blind, placebo-controlled trial of alendronate treatment for fibrous dysplasia of bone","volume":"99","author":"Boyce","year":"2014","journal-title":"J\u00a0Clin Endocrinol Metab"},{"issue":"12","key":"10.1016\/j.eprac.2022.07.012_bib23","doi-asserted-by":"crossref","first-page":"2171","DOI":"10.1002\/jbmr.3828","article-title":"RANKL inhibition in fibrous dysplasia of bone: a preclinical study in a mouse model of the human disease","volume":"34","author":"Palmisano","year":"2019","journal-title":"J\u00a0Bone Miner Res"},{"key":"10.1016\/j.eprac.2022.07.012_bib24","doi-asserted-by":"crossref","DOI":"10.1016\/j.bone.2021.116301","article-title":"RANKL inhibition halts lesion progression and promotes bone remineralization in mice with fibrous dysplasia","volume":"156","author":"Liu","year":"2022","journal-title":"Bone"},{"issue":"9","key":"10.1016\/j.eprac.2022.07.012_bib25","doi-asserted-by":"crossref","first-page":"1729","DOI":"10.1002\/jbmr.4380","article-title":"Safety of therapy with and withdrawal from denosumab in fibrous dysplasia and McCune-Albright syndrome: an observational study","volume":"36","author":"Meier","year":"2021","journal-title":"J\u00a0Bone Miner Res"},{"issue":"12","key":"10.1016\/j.eprac.2022.07.012_bib26","doi-asserted-by":"crossref","first-page":"6069","DOI":"10.1210\/jc.2018-02543","article-title":"Denosumab in patients with fibrous dysplasia previously treated with bisphosphonates","volume":"104","author":"Majoor","year":"2019","journal-title":"J\u00a0Clin Endocrinol Metab"},{"issue":"11","key":"10.1016\/j.eprac.2022.07.012_bib27","doi-asserted-by":"crossref","first-page":"e4179","DOI":"10.1210\/clinem\/dgaa091","article-title":"Denosumab for fibrous dysplasia: promising, but questions remain","volume":"105","author":"Collins","year":"2020","journal-title":"J\u00a0Clin Endocrinol Metab"},{"issue":"9","key":"10.1016\/j.eprac.2022.07.012_bib28","doi-asserted-by":"crossref","first-page":"1889","DOI":"10.1007\/s00198-021-05895-6","article-title":"Denosumab for craniofacial fibrous dysplasia: duration of efficacy and post-treatment effects","volume":"32","author":"Raborn","year":"2021","journal-title":"Osteoporos Int"},{"issue":"4","key":"10.1016\/j.eprac.2022.07.012_bib29","doi-asserted-by":"crossref","first-page":"369","DOI":"10.1016\/j.jbspin.2020.03.007","article-title":"A\u00a0case of recalcitrant fibrous dysplasia treated with denosumab","volume":"87","author":"Gautam","year":"2020","journal-title":"Joint Bone Spine"},{"key":"10.1016\/j.eprac.2022.07.012_bib30","article-title":"Regression of fibrous dysplasia in response to denosumab therapy: a report of two cases","volume":"14","author":"Meier","year":"2021","journal-title":"Bone Rep"},{"issue":"2","key":"10.1016\/j.eprac.2022.07.012_bib31","doi-asserted-by":"crossref","first-page":"777","DOI":"10.1007\/s00198-013-2585-1","article-title":"Rapid biochemical response to denosumab in fibrous dysplasia of bone: report of two cases","volume":"25","author":"Ganda","year":"2014","journal-title":"Osteoporos Int"},{"issue":"7","key":"10.1016\/j.eprac.2022.07.012_bib32","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\u00a0Bone Miner Res"},{"issue":"6","key":"10.1016\/j.eprac.2022.07.012_bib33","doi-asserted-by":"crossref","first-page":"549","DOI":"10.1016\/j.jbspin.2014.04.013","article-title":"Transient improvement of severe pain from fibrous dysplasia of bone with denosumab treatment","volume":"81","author":"Benhamou","year":"2014","journal-title":"Joint Bone Spine"},{"issue":"3","key":"10.1016\/j.eprac.2022.07.012_bib34","first-page":"253","article-title":"Prompt clinical and biochemical response to denosumab in a young adult patient with craniofacial fibrous dysplasia","volume":"13","author":"Eller-Vainicher","year":"2016","journal-title":"Clin Cases Miner Bone Metab"},{"issue":"49","key":"10.1016\/j.eprac.2022.07.012_bib35","doi-asserted-by":"crossref","DOI":"10.1097\/MD.0000000000028138","article-title":"Successful treatment with denosumab for pelvic fibrous dysplasia: a case report and review of the literature","volume":"100","author":"Ikuta","year":"2021","journal-title":"Med (Baltim)"},{"issue":"8","key":"10.1016\/j.eprac.2022.07.012_bib36","doi-asserted-by":"crossref","first-page":"e2980","DOI":"10.1210\/clinem\/dgab212","article-title":"Denosumab reduces lesional fluoride skeletal burden on Na[18F]F PET-CT in patients with fibrous dysplasia\/McCune-Albright syndrome","volume":"106","author":"van der Bruggen","year":"2021","journal-title":"J\u00a0Clin Endocrinol Metab"},{"issue":"2","key":"10.1016\/j.eprac.2022.07.012_bib37","doi-asserted-by":"crossref","first-page":"222","DOI":"10.1016\/j.bone.2008.04.007","article-title":"Effect of denosumab on bone density and turnover in postmenopausal women with low bone mass after long-term continued, discontinued, and restarting of therapy: a randomized blinded phase 2 clinical trial","volume":"43","author":"Miller","year":"2008","journal-title":"Bone"}],"container-title":["Endocrine Practice"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1530891X22005717?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1530891X22005717?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,10,26]],"date-time":"2025-10-26T22:14:03Z","timestamp":1761516843000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S1530891X22005717"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,11]]},"references-count":37,"journal-issue":{"issue":"11","published-print":{"date-parts":[[2022,11]]}},"alternative-id":["S1530891X22005717"],"URL":"https:\/\/doi.org\/10.1016\/j.eprac.2022.07.012","relation":{},"ISSN":["1530-891X"],"issn-type":[{"value":"1530-891X","type":"print"}],"subject":[],"published":{"date-parts":[[2022,11]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Denosumab Use in Adults With Fibrous Dysplasia: Case Reports and Review of the Literature","name":"articletitle","label":"Article Title"},{"value":"Endocrine Practice","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.eprac.2022.07.012","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2022 AACE. Published by Elsevier Inc. All rights reserved.","name":"copyright","label":"Copyright"}]}}