Hypercalcaemia after treatment with denosumab in children: bisphosphonates as an option for therapy and prevention?

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Názov: Hypercalcaemia after treatment with denosumab in children: bisphosphonates as an option for therapy and prevention?
Autori: Sydlik, Carmen, Dürr, Hans Roland, Bechtold-Dalla Pozza, Susanne, Weißenbacher, Claudia, Roeb, Julia, Schmidt, Heinrich
Zdroj: World J Pediatr
Informácie o vydavateľovi: Springer Science and Business Media LLC, 2020.
Rok vydania: 2020
Predmety: Giant Cell Tumor of Bone, Male, Adolescent, Diphosphonates, Brief Report, Diphosphonates/adverse effects [MeSH], Adolescent [MeSH], Female [MeSH], Giant cell tumor of the bone, Giant Cell Tumor of Bone/drug therapy [MeSH], Humans [MeSH], Bone Cysts, Aneurysmal/drug therapy [MeSH], Male [MeSH], Aneurysmatic bone cyst, Hypercalcemia/chemically induced [MeSH], Denosumab, Denosumab/adverse effects [MeSH], Bisphosphonates, Calcium homeostasis, Child [MeSH], 3. Good health, Bone Cysts, Aneurysmal, 03 medical and health sciences, 0302 clinical medicine, Hypercalcemia, Humans, Female, Child
Popis: Background Pharmacologic 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. Methods We 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. Results One 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. Conclusions There 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.
Druh dokumentu: Article
Other literature type
Jazyk: English
ISSN: 1867-0687
1708-8569
DOI: 10.1007/s12519-020-00378-w
Prístupová URL adresa: https://link.springer.com/content/pdf/10.1007/s12519-020-00378-w.pdf
https://pubmed.ncbi.nlm.nih.gov/32776272
https://link.springer.com/content/pdf/10.1007/s12519-020-00378-w.pdf
https://link.springer.com/article/10.1007/s12519-020-00378-w
https://www.ncbi.nlm.nih.gov/pubmed/32776272
https://paperity.org/p/256023730/hypercalcaemia-after-treatment-with-denosumab-in-children-bisphosphonates-as-an-option
https://epub.ub.uni-muenchen.de/73144/
https://pubmed.ncbi.nlm.nih.gov/32776272/
https://repository.publisso.de/resource/frl:6471940
https://epub.ub.uni-muenchen.de/73144/
Rights: CC BY
Prístupové číslo: edsair.doi.dedup.....ff143f171262ca6ed0b2920a767a19a9
Databáza: OpenAIRE
Popis
Abstrakt:Background Pharmacologic 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. Methods We 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. Results One 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. Conclusions There 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.
ISSN:18670687
17088569
DOI:10.1007/s12519-020-00378-w