Successful denosumab treatment for central giant cell granuloma in a 9‐year‐old child

Abstract

Background Denosumab is a nonsurgical treatment option for central giant cell granulomas (CGCG), especially in aggressive lesions. Case report We describe a 9‐year‐old girl with an aggressive maxillary CGCG successfully treated with denosumab, avoiding a mutilating surgery after intralesional corticosteroid injections failed, and the lesion continued to rapidly grow. During denosumab treatment, she developed a self‐limiting area of osteonecrosis in the maxillary alveolar bone, which rapidly resolved after antibiotic therapy. Six months after denosumab discontinuation, a maxillary surgical recontour was performed. Two weeks after surgery, the patient presented vomiting, pallor, dehydration, but no fever. Blood tests revealed severe hypercalcemia and acute renal dysfunction. After discarding thyroid, parathyroid, and adrenal alterations, a diagnosis of severe rebound hypercalcemia after denosumab treatment was made. Treatment consisted of hyperhydration, calcium pamidronate, and methylprednisolone, restoring calcium levels to normal. Conclusion After 2 years of follow‐up, she remains on orthodontic treatment, with no recurrences or other episodes of hypercalcemia.

Hypercalcemia in Children Following a Discontinuation of Denosumab Therapy: A Case Report and Literature Review

“…Among the 39 children, 25 experienced symptomatic hypercalcemia, with most of them presenting with nausea or vomiting. In severe cases, hypertension [17][18][19] and acute kidney inj ury 10,15,18,[20][21][22] were observed. The standard treatments included aggressive hydration and administration of diuretics, which were effective in correcting mild hypercalcemia.…”

“…This hypercalcemia was successfully normalized through the administration of bisphosphonates. Denosumab has been recently explored in the treatment of skeletal diseases such as CGCG, 7,13,14,16,17 OI, 5 ABC, 18-20 FD, 9 and GCTB 21 in immature patients. However, a rebound hypercalcemia effect induced by denosumab discontinuation has been reported in several cases in children.…”

Hypercalcemia following discontinuation of denosumab therapy: A systematic review

“…In most cases, treatments for hypercalcemia followed the standard protocol, consisting of hydration and administration of diuretics, corticosteroids, and/or calcitonin. However, in severe cases, repeated use of bisphosphonates or reinjection of denosumab was often required to repress the surge of calcium released into the circulation 14 , 16 , 17 , 19 , 22 , 24 , 29 , 30 , 31 , 32 , 33 , 34 . Although no established treatment modality exists to prevent this condition, tapering of the dosage of denosumab and prophylactic use of bisphosphonates were attempted in several cases 22 , 23 , 31 .…”

Denosumab in pediatric bone disorders and the role of RANKL blockade: a narrative review

“…The largest group of children studied with CGCG treated using denosumab was reported in Texas Children's Hospital in 2021 (75,76). In this study, 6 children aged 5 to 12 years were followed up for an average of 20 months.…”