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

[摘要] Parathyroid carcinoma is a rare disease leading to severe hypercalcemia due to hyperparathyroidism. Surgery is theprimary treatment option. A more progressive form of the disease is characterized by parathyrotoxicosis, and subsequenthypercalcemia is the most common cause of death. We report a case presenting with severe hypercalcemia due toparathyrotoxicosis from parathyroid carcinoma treated for the first time using the monoclonal antibody denosumab as arescue therapy and present long-term follow-up data. The 71-year-old patient presented with severe hypercalcemia due tometastatic parathyroid carcinoma. Despite undergoing treatment with bisphosphonates, cinacalcet hydrochloride, andforced diuresis, the patient‘s condition deteriorated rapidly due to resistant hypercalcemia. Surgery performed becauseof spinal metastasis and forced diuresis lowered calcium levels, albeit they remained in the hypercalcemic range andsignificantly increased when forced diuresis was stopped. Considering a palliative situation to overcome hypercalcemia, wedecided to administer denosumab, a monoclonal antibody that binds to the receptor activator of nuclear factor-kappa Bligand. 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 risingparathyroid 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 patientswith advanced stages of parathyroid cancer.

[学科分类] 血液学

[关键词] Adult;Male;White;Germany;Parathyroid;Endocrine disruptors;PTH;Hypercalcaemia;Parathyroid carcinoma;Metastatic carcinoma;Hypercalcaemia;Bony metastases;Abdominal pain;Back pain;Cachexia;Appetite reduction/loss;Nausea;Weight loss;Constipation;Bowel obstruction;Calcium (serum);PTH;PET scan;CT scan;Resection of tumour;Denosumab;Zoledronic acid;Furosemide;Cinacalcet;Calcimimetics;Diuretics;Sodium chloride;Bisphosphonates;General practice;Nephrology;Oncology;Surgery;Novel treatment;November;2015