{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,27]],"date-time":"2026-08-27T13:20:44Z","timestamp":1787836844074,"version":"build-2784847793"},"reference-count":44,"publisher":"SAGE Publications","license":[{"start":{"date-parts":[[2026,8,25]],"date-time":"2026-08-25T00:00:00Z","timestamp":1787616000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["The Cleft Palate Craniofacial Journal"],"abstract":"<jats:p>\n                    Cherubism is a rare autosomal dominant disorder characterized by proliferative fibro-osseous lesions of the maxilla and mandible. Surgical management varies by disease severity and symptom burden, often resulting in substantial operative burden in pediatric patients. Denosumab, a monoclonal antibody targeting RANKL, offers potential off-label therapy to suppress osseous overgrowth and reduce surgical morbidity. The authors previously reported early success with denosumab in 1 patient\n                    <jats:bold>;<\/jats:bold>\n                    however, during long-term treatment, the patient developed multiple long-bone fragility fractures and severe rebound hypercalcemia. This report updates that patient and describes treatment modifications in a second patient with cherubism, supporting denosumab as a disease-suppressive therapy with refined dosing and metabolic surveillance.\n                  <\/jats:p>","DOI":"10.1177\/10556656261480924","type":"journal-article","created":{"date-parts":[[2026,8,25]],"date-time":"2026-08-25T16:13:35Z","timestamp":1787674415000},"update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":0,"title":["Evolving Use of Denosumab in Severe Cherubism: Updates, Fragility Fractures, Rebound Hypercalcemia, and Lessons from Two Cases"],"prefix":"10.1177","author":[{"ORCID":"https:\/\/orcid.org\/0009-0004-5069-1257","authenticated-orcid":false,"given":"Julia","family":"Sharpe","sequence":"first","affiliation":[{"name":"University of Mississippi Medical Center"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ashlie A","family":"Elver","sequence":"additional","affiliation":[{"name":"University of Mississippi Medical Center"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laura","family":"Arnold","sequence":"additional","affiliation":[{"name":"University of Mississippi Medical Center"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Steele","family":"Liles","sequence":"additional","affiliation":[{"name":"University of Mississippi Medical Center"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emily","family":"Boothe","sequence":"additional","affiliation":[{"name":"University of Mississippi Medical Center"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ian C","family":"Hoppe","sequence":"additional","affiliation":[{"name":"University of Mississippi Medical Center"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2026,8,25]]},"reference":[{"key":"e_1_3_2_2_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijom.2020.05.021"},{"key":"e_1_3_2_3_2","doi-asserted-by":"publisher","DOI":"10.1186\/1750-1172-7-S1-S6"},{"key":"e_1_3_2_4_2","doi-asserted-by":"publisher","DOI":"10.1097\/01.sap.0000248141.36904.19"},{"key":"e_1_3_2_5_2","doi-asserted-by":"publisher","DOI":"10.7181\/acfs.2023.00577"},{"key":"e_1_3_2_6_2","doi-asserted-by":"publisher","DOI":"10.5037\/jomr.2010.1202"},{"key":"e_1_3_2_7_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.oooo.2025.01.728"},{"key":"e_1_3_2_8_2","doi-asserted-by":"publisher","DOI":"10.1097\/SCS.0000000000001444"},{"key":"e_1_3_2_9_2","doi-asserted-by":"publisher","DOI":"10.1002\/jbm4.10210"},{"key":"e_1_3_2_10_2","doi-asserted-by":"publisher","DOI":"10.1177\/10556656221113891"},{"key":"e_1_3_2_11_2","doi-asserted-by":"publisher","DOI":"10.4103\/jofs.jofs_169_24"},{"key":"e_1_3_2_12_2","doi-asserted-by":"publisher","DOI":"10.1186\/s12903-024-05067-y"},{"key":"e_1_3_2_13_2","doi-asserted-by":"publisher","DOI":"10.1177\/10556656231176879"},{"key":"e_1_3_2_14_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.joms.2023.04.007"},{"key":"e_1_3_2_15_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.bone.2010.11.020"},{"key":"e_1_3_2_16_2","unstructured":"US Food and Drug Administration. Prolia (denosumab) injection for subcutaneous use: prescribing information. Revised June 2024. Accessed August 11 2025. https:\/\/www.accessdata.fda.gov\/drugsatfda_docs\/label\/2024\/125320s159lbl.pdf."},{"key":"e_1_3_2_17_2","doi-asserted-by":"publisher","DOI":"10.20945\/2359-4292-2024-0035"},{"key":"e_1_3_2_18_2","doi-asserted-by":"publisher","DOI":"10.21037\/tp-22-276"},{"key":"e_1_3_2_19_2","doi-asserted-by":"publisher","DOI":"10.1007\/s40520-025-02991-z"},{"key":"e_1_3_2_20_2","doi-asserted-by":"publisher","DOI":"10.1530\/EJE-18-0056"},{"issue":"3","key":"e_1_3_2_21_2","first-page":"183","article-title":"First use of the RANKL antibody denosumab in osteogenesis imperfecta type VI","volume":"12","author":"Semler O","year":"2012","unstructured":"Semler O, Netzer C, Hoyer-Kuhn H, Becker J, Eysel P, Schoenau E. First use of the RANKL antibody denosumab in osteogenesis imperfecta type VI. J Musculoskelet Neuronal Interact. 2012;12(3):183-188.","journal-title":"J Musculoskelet Neuronal Interact"},{"key":"e_1_3_2_22_2","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-014-0145-1"},{"issue":"1","key":"e_1_3_2_23_2","first-page":"24","article-title":"Safety and efficacy of denosumab in children with osteogenesis imperfect\u2013a first prospective trial","volume":"16","author":"Hoyer-Kuhn H","year":"2016","unstructured":"Hoyer-Kuhn H, Franklin J, Allo G, Kron M, Netzer C, Eysel P, Hero B, Schoenau E, Semler O. Safety and efficacy of denosumab in children with osteogenesis imperfect\u2013a first prospective trial. J Musculoskelet Neuronal Interact. 2016;16(1):24-32.","journal-title":"J Musculoskelet Neuronal Interact"},{"issue":"1","key":"e_1_3_2_24_2","first-page":"76","article-title":"Hypercalcemia and hypercalciuria during denosumab treatment in children with osteogenesis imperfecta type VI","volume":"18","author":"Trejo P","year":"2018","unstructured":"Trejo P, Rauch F, Ward L. Hypercalcemia and hypercalciuria during denosumab treatment in children with osteogenesis imperfecta type VI. J Musculoskelet Neuronal Interact. 2018;18(1):76-80.","journal-title":"J Musculoskelet Neuronal Interact"},{"key":"e_1_3_2_25_2","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-019-1197-z"},{"key":"e_1_3_2_26_2","doi-asserted-by":"publisher","DOI":"10.1210\/clinem\/dgad732"},{"key":"e_1_3_2_27_2","doi-asserted-by":"publisher","DOI":"10.3389\/fendo.2025.1445093"},{"key":"e_1_3_2_28_2","doi-asserted-by":"publisher","DOI":"10.1002\/jbmr.1603"},{"key":"e_1_3_2_29_2","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2013-3081"},{"key":"e_1_3_2_30_2","doi-asserted-by":"publisher","DOI":"10.1002\/jbmr.2999"},{"key":"e_1_3_2_31_2","doi-asserted-by":"publisher","DOI":"10.1007\/s00198-021-05895-6"},{"key":"e_1_3_2_32_2","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.2012.46.4255"},{"key":"e_1_3_2_33_2","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.25393"},{"key":"e_1_3_2_34_2","doi-asserted-by":"publisher","DOI":"10.1097\/MPH.0000000000001654"},{"key":"e_1_3_2_35_2","doi-asserted-by":"publisher","DOI":"10.1007\/s00586-013-2715-7"},{"key":"e_1_3_2_36_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.trsl.2014.03.005"},{"key":"e_1_3_2_37_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.otsr.2019.04.028"},{"key":"e_1_3_2_38_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.joms.2020.06.016"},{"key":"e_1_3_2_39_2","doi-asserted-by":"publisher","DOI":"10.1515\/jpem-2019-0581"},{"key":"e_1_3_2_40_2","doi-asserted-by":"publisher","DOI":"10.1210\/jendso\/bvab048.442"},{"key":"e_1_3_2_41_2","doi-asserted-by":"publisher","DOI":"10.1038\/s41598-019-56151-1"},{"key":"e_1_3_2_42_2","doi-asserted-by":"publisher","DOI":"10.1038\/s41598-023-37944-x"},{"key":"e_1_3_2_43_2","doi-asserted-by":"publisher","DOI":"10.7759\/cureus.85295"},{"key":"e_1_3_2_44_2","doi-asserted-by":"publisher","DOI":"10.2106\/JBJS.25.01058"},{"key":"e_1_3_2_45_2","doi-asserted-by":"publisher","DOI":"10.1007\/s00247-006-0261-8"}],"container-title":["The Cleft Palate Craniofacial Journal"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/10556656261480924","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1177\/10556656261480924","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/10556656261480924","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,27]],"date-time":"2026-08-27T12:48:45Z","timestamp":1787834925000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1177\/10556656261480924"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,8,25]]},"references-count":44,"alternative-id":["10.1177\/10556656261480924"],"URL":"https:\/\/doi.org\/10.1177\/10556656261480924","relation":{},"ISSN":["1055-6656","1545-1569"],"issn-type":[{"value":"1055-6656","type":"print"},{"value":"1545-1569","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,8,25]]},"article-number":"10556656261480924"}}