Malignant Hypercalcemia Due to the Ectopic Production of Calcitriol by an Abdominal Liposarcoma

Overview

Journal Cureus

Specialty Biomedical Engineering

Date 2023 Feb 8

PMID 36751194

Authors

Ana Figueiredo

Teresa Pereira

Rafael Cabrera

Joana Simoes-Pereira

Valeriano Leite

Affiliations

Soon will be listed here.

Abstract

Hypercalcemia of malignancy (HM) is a common form of paraneoplastic syndrome associated with a poor prognosis of the disease. In solid tumors, HM occurs mainly due to the production of parathyroid hormone-related peptide (PTHrP). We present a case of a 60-year-old male with a 25 cm retroperitoneal liposarcoma diagnosed with severe hypercalcemia (16.8 mg/dL) by a preoperative blood sampling. Hypercalcemia workup showed suppressed parathyroid hormone (PTH), normal PTHrP, and high 1,25-dihydroxyvitamin D (1,25(OH)2D) serum levels. After surgery, hypercalcemia and calcitriol levels normalized. Immunohistochemical analysis of the tumor showed 1α-hydroxylase expression by tumor cells. To our knowledge, this is the first case of liposarcoma-associated hypercalcemia caused exclusively by the ectopic production of calcitriol. Despite being a rare cause of hypercalcemia, measuring 1,25(OH)2D should be considered in the workup of a patient with high serum calcium levels, suppressed PTH, and normal PTHrP.

References

Zagzag J, Hu M, Fisher S, Perrier N . Hypercalcemia and cancer: Differential diagnosis and treatment. CA Cancer J Clin. 2018; 68(5):377-386. DOI: 10.3322/caac.21489. View

Hu M, Glezerman I, Leboulleux S, Insogna K, Gucalp R, Misiorowski W . Denosumab for treatment of hypercalcemia of malignancy. J Clin Endocrinol Metab. 2014; 99(9):3144-52. PMC: 4154084. DOI: 10.1210/jc.2014-1001. View

Clines G . Mechanisms and treatment of hypercalcemia of malignancy. Curr Opin Endocrinol Diabetes Obes. 2011; 18(6):339-46. DOI: 10.1097/MED.0b013e32834b4401. View

Brooks R, Lord C, Davies J, Gray J . Hypercalcaemia secondary to ectopic parathyroid hormone expression in an adolescent with metastatic alveolar rhabdomyosarcoma. Pediatr Blood Cancer. 2017; 65(1). DOI: 10.1002/pbc.26778. View

Kaukinen A, Pelkonen J, Harvima I . Mast cells express CYP27A1 and CYP27B1 in epithelial skin cancers and psoriasis. Eur J Dermatol. 2015; 25(6):548-55. DOI: 10.1684/ejd.2015.2645. View

Goldner W . Cancer-Related Hypercalcemia. J Oncol Pract. 2016; 12(5):426-32. DOI: 10.1200/JOP.2016.011155. View

Evans K, Taylor H, Zehnder D, Kilby M, Bulmer J, Shah F . Increased expression of 25-hydroxyvitamin D-1alpha-hydroxylase in dysgerminomas: a novel form of humoral hypercalcemia of malignancy. Am J Pathol. 2004; 165(3):807-13. PMC: 1618616. DOI: 10.1016/s0002-9440(10)63343-3. View

Hewison M, Kantorovich V, Liker H, Herle A, Cohan P, Zehnder D . Vitamin D-mediated hypercalcemia in lymphoma: evidence for hormone production by tumor-adjacent macrophages. J Bone Miner Res. 2003; 18(3):579-82. DOI: 10.1359/jbmr.2003.18.3.579. View

Herrera-Martinez Y, Contreras Gonzalez M, Pedraza-Arevalo S, Guerrero Martinez M, Rodrigo Martinez A, Gonzalez Menchen A . Calcitriol-Mediated Hypercalcemia, Somatostatin Receptors Expression and 25-Hydroxyvitamin D-1α- Hydroxylase in GIST Tumors. Front Endocrinol (Lausanne). 2022; 12:812385. PMC: 8826724. DOI: 10.3389/fendo.2021.812385. View

10.

Kim D, Miller A, Li A, Hardy N, Silver K . Hypercalcemia of Malignancy: Simultaneous Elevation in Parathyroid Hormone-Related Peptide and 1,25 Dihydroxyvitamin D in Sarcoma. AACE Clin Case Rep. 2021; 7(3):169-173. PMC: 8165116. DOI: 10.1016/j.aace.2020.11.037. View

11.

Rodriguez-Gutierrez R, Zapata-Rivera M, Quintanilla-Flores D, Camara-Lemarroy C, Lavalle-Gonzalez F, Gonzalez-Gonzalez J . 1,25-dihydroxyvitamin D and PTHrP mediated malignant hypercalcemia in a seminoma. BMC Endocr Disord. 2014; 14:32. PMC: 3991903. DOI: 10.1186/1472-6823-14-32. View

12.

Lumachi F, Brunello A, Roma A, Basso U . Cancer-induced hypercalcemia. Anticancer Res. 2009; 29(5):1551-5. View