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Palliative treatment of uncontrollable hypercalcemia due to parathyrotoxicosis: denosumab as rescue therapy

Parathyroid carcinoma is a rare disease leading to severe hypercalcemia due to hyperparathyroidism. Surgery is the primary treatment option. A more progressive form of the disease is characterized by parathyrotoxicosis, and subsequent hypercalcemia is the most common cause of death. We report a case...

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Autores principales: Jumpertz von Schwartzenberg, Reiner, Elbelt, Ulf, Ventz, Manfred, Mai, Knut, Kienitz, Tina, Maurer, Lukas, Rose, Thomas, Rückert, Jens C, Strasburger, Christian J, Spranger, Joachim
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Bioscientifica Ltd 2015
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4653612/
https://www.ncbi.nlm.nih.gov/pubmed/26605043
http://dx.doi.org/10.1530/EDM-15-0082
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author Jumpertz von Schwartzenberg, Reiner
Elbelt, Ulf
Ventz, Manfred
Mai, Knut
Kienitz, Tina
Maurer, Lukas
Rose, Thomas
Rückert, Jens C
Strasburger, Christian J
Spranger, Joachim
author_facet Jumpertz von Schwartzenberg, Reiner
Elbelt, Ulf
Ventz, Manfred
Mai, Knut
Kienitz, Tina
Maurer, Lukas
Rose, Thomas
Rückert, Jens C
Strasburger, Christian J
Spranger, Joachim
author_sort Jumpertz von Schwartzenberg, Reiner
collection PubMed
description Parathyroid carcinoma is a rare disease leading to severe hypercalcemia due to hyperparathyroidism. Surgery is the primary treatment option. A more progressive form of the disease is characterized by parathyrotoxicosis, and subsequent hypercalcemia is the most common cause of death. We report a case presenting with severe hypercalcemia due to parathyrotoxicosis from parathyroid carcinoma treated for the first time using the monoclonal antibody denosumab as a rescue therapy and present long-term follow-up data. The 71-year-old patient presented with severe hypercalcemia due to metastatic parathyroid carcinoma. Despite undergoing treatment with bisphosphonates, cinacalcet hydrochloride, and forced diuresis, the patient`s condition deteriorated rapidly due to resistant hypercalcemia. Surgery performed because of spinal metastasis and forced diuresis lowered calcium levels, albeit they remained in the hypercalcemic range and significantly increased when forced diuresis was stopped. Considering a palliative situation to overcome hypercalcemia, we decided to administer denosumab, a monoclonal antibody that binds to the receptor activator of nuclear factor-kappa B ligand. After a single subcutaneous administration of 60 mg denosumab, calcium levels normalized within one day. Subsequent denosumab injections led to permanent control of serum calcium for more than 2 years despite rising parathyroid hormone levels and repeated surgeries. Together with recent cases in the literature supporting our observation, we believe that denosumab is relevant for future trials and represents an effective tool to control hypercalcemia in patients with advanced stages of parathyroid cancer. LEARNING POINTS: Severe hypercalcemia is the most common cause of death in patients with parathyroid carcinoma. The monoclonal antibody denosumab rapidly lowered severely elevated serum calcium levels due to parathyrotoxicosis. Denosumab was effective in the long-term treatment of hypercalcemia despite progression of parathyroid carcinoma.
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spelling pubmed-46536122015-11-24 Palliative treatment of uncontrollable hypercalcemia due to parathyrotoxicosis: denosumab as rescue therapy Jumpertz von Schwartzenberg, Reiner Elbelt, Ulf Ventz, Manfred Mai, Knut Kienitz, Tina Maurer, Lukas Rose, Thomas Rückert, Jens C Strasburger, Christian J Spranger, Joachim Endocrinol Diabetes Metab Case Rep Novel Treatment Parathyroid carcinoma is a rare disease leading to severe hypercalcemia due to hyperparathyroidism. Surgery is the primary treatment option. A more progressive form of the disease is characterized by parathyrotoxicosis, and subsequent hypercalcemia is the most common cause of death. We report a case presenting with severe hypercalcemia due to parathyrotoxicosis from parathyroid carcinoma treated for the first time using the monoclonal antibody denosumab as a rescue therapy and present long-term follow-up data. The 71-year-old patient presented with severe hypercalcemia due to metastatic parathyroid carcinoma. Despite undergoing treatment with bisphosphonates, cinacalcet hydrochloride, and forced diuresis, the patient`s condition deteriorated rapidly due to resistant hypercalcemia. Surgery performed because of spinal metastasis and forced diuresis lowered calcium levels, albeit they remained in the hypercalcemic range and significantly increased when forced diuresis was stopped. Considering a palliative situation to overcome hypercalcemia, we decided to administer denosumab, a monoclonal antibody that binds to the receptor activator of nuclear factor-kappa B ligand. After a single subcutaneous administration of 60 mg denosumab, calcium levels normalized within one day. Subsequent denosumab injections led to permanent control of serum calcium for more than 2 years despite rising parathyroid hormone levels and repeated surgeries. Together with recent cases in the literature supporting our observation, we believe that denosumab is relevant for future trials and represents an effective tool to control hypercalcemia in patients with advanced stages of parathyroid cancer. LEARNING POINTS: Severe hypercalcemia is the most common cause of death in patients with parathyroid carcinoma. The monoclonal antibody denosumab rapidly lowered severely elevated serum calcium levels due to parathyrotoxicosis. Denosumab was effective in the long-term treatment of hypercalcemia despite progression of parathyroid carcinoma. Bioscientifica Ltd 2015-10-29 2015 /pmc/articles/PMC4653612/ /pubmed/26605043 http://dx.doi.org/10.1530/EDM-15-0082 Text en © 2015 The authors This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivs 3.0 Unported License (http://creativecommons.org/licenses/by-nc-nd/3.0/deed.en_GB) .
spellingShingle Novel Treatment
Jumpertz von Schwartzenberg, Reiner
Elbelt, Ulf
Ventz, Manfred
Mai, Knut
Kienitz, Tina
Maurer, Lukas
Rose, Thomas
Rückert, Jens C
Strasburger, Christian J
Spranger, Joachim
Palliative treatment of uncontrollable hypercalcemia due to parathyrotoxicosis: denosumab as rescue therapy
title Palliative treatment of uncontrollable hypercalcemia due to parathyrotoxicosis: denosumab as rescue therapy
title_full Palliative treatment of uncontrollable hypercalcemia due to parathyrotoxicosis: denosumab as rescue therapy
title_fullStr Palliative treatment of uncontrollable hypercalcemia due to parathyrotoxicosis: denosumab as rescue therapy
title_full_unstemmed Palliative treatment of uncontrollable hypercalcemia due to parathyrotoxicosis: denosumab as rescue therapy
title_short Palliative treatment of uncontrollable hypercalcemia due to parathyrotoxicosis: denosumab as rescue therapy
title_sort palliative treatment of uncontrollable hypercalcemia due to parathyrotoxicosis: denosumab as rescue therapy
topic Novel Treatment
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4653612/
https://www.ncbi.nlm.nih.gov/pubmed/26605043
http://dx.doi.org/10.1530/EDM-15-0082
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