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

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 aneurysmati...

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Autores principales: Sydlik, Carmen, Dürr, Hans Roland, Pozza, Susanne Bechtold-Dalla, Weißenbacher, Claudia, Roeb, Julia, Schmidt, Heinrich
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Springer Singapore 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7515853/
https://www.ncbi.nlm.nih.gov/pubmed/32776272
http://dx.doi.org/10.1007/s12519-020-00378-w
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author Sydlik, Carmen
Dürr, Hans Roland
Pozza, Susanne Bechtold-Dalla
Weißenbacher, Claudia
Roeb, Julia
Schmidt, Heinrich
author_facet Sydlik, Carmen
Dürr, Hans Roland
Pozza, Susanne Bechtold-Dalla
Weißenbacher, Claudia
Roeb, Julia
Schmidt, Heinrich
author_sort Sydlik, Carmen
collection PubMed
description 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.
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spelling pubmed-75158532020-10-07 Hypercalcaemia after treatment with denosumab in children: bisphosphonates as an option for therapy and prevention? Sydlik, Carmen Dürr, Hans Roland Pozza, Susanne Bechtold-Dalla Weißenbacher, Claudia Roeb, Julia Schmidt, Heinrich World J Pediatr Brief Report 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. Springer Singapore 2020-08-10 2020 /pmc/articles/PMC7515853/ /pubmed/32776272 http://dx.doi.org/10.1007/s12519-020-00378-w Text en © The Author(s) 2020 Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/.
spellingShingle Brief Report
Sydlik, Carmen
Dürr, Hans Roland
Pozza, Susanne Bechtold-Dalla
Weißenbacher, Claudia
Roeb, Julia
Schmidt, Heinrich
Hypercalcaemia after treatment with denosumab in children: bisphosphonates as an option for therapy and prevention?
title Hypercalcaemia after treatment with denosumab in children: bisphosphonates as an option for therapy and prevention?
title_full Hypercalcaemia after treatment with denosumab in children: bisphosphonates as an option for therapy and prevention?
title_fullStr Hypercalcaemia after treatment with denosumab in children: bisphosphonates as an option for therapy and prevention?
title_full_unstemmed Hypercalcaemia after treatment with denosumab in children: bisphosphonates as an option for therapy and prevention?
title_short Hypercalcaemia after treatment with denosumab in children: bisphosphonates as an option for therapy and prevention?
title_sort hypercalcaemia after treatment with denosumab in children: bisphosphonates as an option for therapy and prevention?
topic Brief Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7515853/
https://www.ncbi.nlm.nih.gov/pubmed/32776272
http://dx.doi.org/10.1007/s12519-020-00378-w
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