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Case Report

Significant Response to Denosumab Yet with Severe Rebound Hypercalcemia in a 9-Year-Old Boy with Aneurysmal Bone Cyst: A Case Report

1
Faculty of Medicine, Laval University, Quebec City, QC G1V 0A6, Canada
2
Division of Pediatric Hematology Oncology, CHUL-Laval, Quebec City, QC G1V 0A6, Canada
3
Montreal Children’s Hospital, Montréal, QC H4A 3H9, Canada
4
Department of Pediatric Hematology/Oncology, CHU Sainte Justine, Université de Montréal, Montréal, QC H3C 3A7, Canada
5
Division of Pediatric Endocrinology, CHUL-Laval, Quebec City, QC G1V 0A6, Canada
6
Department of Pediatrics, Laval University, Quebec City, QC G1V 0A6, Canada
7
Princess Margaret Cancer Centre, University Health Network, Toronto, ON M5G 1X6, Canada
8
Adolescent & Young Adult (AYA) Oncology Program, Princess Margaret Hospital, Toronto, ON M5G 1X6, Canada
9
Paediatric Cardiology Unit, Department of Pediatrics, Centre Hospitalier Universitaire Vaudois and University of Lausanne, Rue du Bugnon 50, 1011 Lausanne, Switzerland
10
Division of Clinical Pharmacology and Toxicology, Institute of Pharmacological Sciences of Southern Switzerland, Ente Ospedaliero Cantonale, 6900 Lugano, Switzerland
*
Author to whom correspondence should be addressed.
These authors contributed equally to this work.
Children 2025, 12(11), 1524; https://doi.org/10.3390/children12111524
Submission received: 25 September 2025 / Revised: 30 October 2025 / Accepted: 6 November 2025 / Published: 11 November 2025
(This article belongs to the Section Pediatric Hematology & Oncology)

Abstract

A 9-year-old boy presented with edema of the left cheek. He was diagnosed with a large aneurysmal bone cyst of the mandibular bone. Off-label treatment with denosumab for 17 months resulted in a significant reduction in the lesion. Five months after discontinuing denosumab, the patient developed severe rebound hypercalcemia and acute kidney injury, which resolved with corticosteroids. Two years after treatment discontinuation, the lesion continues to decrease in size. This case highlights the efficacy of denosumab as an off-label treatment for aneurysmal bone cysts, though it can be associated with hypercalcemia and acute kidney injury. We conclude that the use of denosumab should be considered on a case-by-case basis, with regular monitoring for side effects.
Keywords: aneurysmal bone cyst; tumor; rare pediatric tumors; bones; denosumab; targeted therapy; hypercalcemia; acute kidney injury; toxicity aneurysmal bone cyst; tumor; rare pediatric tumors; bones; denosumab; targeted therapy; hypercalcemia; acute kidney injury; toxicity

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MDPI and ACS Style

Allain, L.; Elbaz, S.; Sathyakumar, S.; Le Gallou, W.; Coleman, C.; Coltin, H.; Eugène, D.; Gupta, A.; Lava, S.A.G.; Renzi, S. Significant Response to Denosumab Yet with Severe Rebound Hypercalcemia in a 9-Year-Old Boy with Aneurysmal Bone Cyst: A Case Report. Children 2025, 12, 1524. https://doi.org/10.3390/children12111524

AMA Style

Allain L, Elbaz S, Sathyakumar S, Le Gallou W, Coleman C, Coltin H, Eugène D, Gupta A, Lava SAG, Renzi S. Significant Response to Denosumab Yet with Severe Rebound Hypercalcemia in a 9-Year-Old Boy with Aneurysmal Bone Cyst: A Case Report. Children. 2025; 12(11):1524. https://doi.org/10.3390/children12111524

Chicago/Turabian Style

Allain, Laurence, Sarah Elbaz, Sayanthen Sathyakumar, William Le Gallou, Christina Coleman, Hallie Coltin, Dardye Eugène, Abha Gupta, Sebastiano A. G. Lava, and Samuele Renzi. 2025. "Significant Response to Denosumab Yet with Severe Rebound Hypercalcemia in a 9-Year-Old Boy with Aneurysmal Bone Cyst: A Case Report" Children 12, no. 11: 1524. https://doi.org/10.3390/children12111524

APA Style

Allain, L., Elbaz, S., Sathyakumar, S., Le Gallou, W., Coleman, C., Coltin, H., Eugène, D., Gupta, A., Lava, S. A. G., & Renzi, S. (2025). Significant Response to Denosumab Yet with Severe Rebound Hypercalcemia in a 9-Year-Old Boy with Aneurysmal Bone Cyst: A Case Report. Children, 12(11), 1524. https://doi.org/10.3390/children12111524

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