Abstract
BackgroundPharmacologic options for treatment of osteolytic diseases especially in children are limited. Although not licensed for use, denosumab, a fully humanized antibody to RANKL, is used in children with good effects. Among others, one possible indication are giant cell tumors and aneurysmatic bone cysts. However, there are reports of severe hypercalcemia during weeks to months after termination of denosumab, that are rarely seen in adults.MethodsWe collected data of four patients, aged 6–17 years, who experienced severe hypercalcemia after completion of treatment with denosumab for unresectable giant cell tumors of bone or aneurysmal bone cysts and methods of their treatment. The detailed case information were described.ResultsOne patient was treated with long-term, high-dose steroid therapy, leading to typical Cushing’s syndrome. Another patient was restarted on denosumab repeatedly due to relapses of hypercalcemia after every stop. Finally, in two patients, hypercalcemia ceased definitely after treatment with bisphosphonates. However, several applications were necessary to stabilize calcium levels.ConclusionsThere is a considerable risk of hypercalcemia as an adverse effect after denosumab treatment in children. Therapeutic and, preferably, preventive strategies are needed. Bisphosphonates seem to be an option for both, but effective proceedings still remain to be established.
Highlights
Treatment of osteolytic diseases is known to be difficult as pharmacological possibilities are limited, in children
Surgical treatment was difficult owing to the localisation and extension of the tumor; offlabel-use of denosumab was started in November 2014 at a dose of 60 mg on days 1, 8, 15, 28, and once a month
Few reports exist on hypercalcemia after treatment with denosumab, as seen in the four patients presented here
Summary
Treatment of osteolytic diseases is known to be difficult as pharmacological possibilities are limited, in children. Denosumab, a fully humanized antibody to RANKL, is available for treatment of postmenopausal osteoporosis and skeletal related events caused by bone metastases of certain solid tumors in adults [1]. It is an effective and quite frequently used therapeutic. Along with the abovementioned notions of reactively increased osteoclast activity after cessation of denosumab, most of the reports on children mention hypercalcaemic episodes [4, 9, 14, 15, 18, 19], which are often difficult to treat These sequelae have not yet been systematically addressed in a clinical study. Bisphosphonates seem to be an option for both, but effective proceedings still remain to be established
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