Bone Tumor Diagnosis and Treatments
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Treatment modifications in a second patient with cherubism are described, supporting denosumab as a disease-suppressive therapy with refined dosing and metabolic surveillance, supporting denosumab as a disease-suppressive therapy with refined dosing and metabolic surveillance.
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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 ; 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.
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@article{SHARPE2026Evolving,
title = {Evolving Use of Denosumab in Severe Cherubism: Updates, Fragility Fractures, Rebound Hypercalcemia, and Lessons from Two Cases},
author = {JULIA SHARPE and Ashlie A. Elver and Laura Arnold and Steele Liles and Emily Boothe and Ian C. Hoppe},
journal = {The Cleft Palate-Craniofacial Journal},
year = {2026},
doi = {10.1177/10556656261480924},
url = {https://doi.org/10.1177/10556656261480924}
}FAQ
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