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

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.

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Publication Details

Journal
Children
Published
2025-11-11
DOI
https://doi.org/10.3390/children12111524
Primary Topic
Bone health and treatments
Type
article
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article

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

Hallie Coltin, Sebastiano A. G. Lava, Abha A. Gupta, Sherif Elbaz et al.
Children
Bone health and treatments
article

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

Hallie Coltin, Sebastiano A. G. Lava, Abha A. Gupta, Sherif Elbaz, Dardye Eugène, Christina Coleman, Sayanthen Sathyakumar, Samuele Renzi, Laurence Allain, William Le Gallou
article en

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.

ChildrenVol. 12(11)
University Health Network (CA), Montreal Children's Hospital (CA), Centre Hospitalier Universitaire Vaudois (CH), Centre Hospitalier Universitaire Sainte-Justine (CA), Princess Margaret Cancer Centre (CA), Ente Ospedaliero Cantonale (CH), Princess Margaret Hospital (NZ), Université Laval (CA)
Openalex Percentile: Top 54%
Bone health and treatments
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