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Ectopic parathyroid hormone as a rare aetiology of hypercalcemia with rhabdomyosarcoma: a new treatment strategy with zoledronic acid and Denosumab

  • Fatih Kilci ORCID logo EMAIL logo , Uğur Demirsoy ORCID logo , Jeremy Huw Jones ORCID logo , Özgür Çakır ORCID logo and Filiz Mine Çizmecioğlu-Jones ORCID logo
Published/Copyright: May 10, 2022

Abstract

Objectives

Ectopic parathyroid hormone (PTH) secretion is rare in children with rhabdomyosarcoma, and only a few pediatric cases have been reported to date. Reports of the use of zoledronic acid (ZA) and Denosumab are limited for the treatment of hypercalcemia of malignancy (HCM) in the pediatric population. The aim of presenting this pediatric case of rhabdomyosarcoma accompanied by HCM, secondary to ectopic PTH secretion, was to highlight the benefits of ZA as a first-choice bisphosphonate in this situation with Denosumab as an alternative therapy.

Case presentation

The patient was diagnosed at 13 years with alveolar rhabdomyosarcoma. Multiple bone metastases first appeared at 15 years, but he remained normocalcemic until 17 years old when serum calcium was 15.1 mg/dL and PTH 249 pg/mL. While serum calcium responded well after ZA and Denosumab cycles, PTH remained elevated, reaching a peak value of 1851 pg/mL during treatment cycles.

Conclusions

We report a patient with rhabdomyosarcoma accompanied by HCM, secondary to ectopic PTH, in whom the HCM was successfully managed with ZA and Denosumab. We believe that ZA should be the bisphosphonate of choice in pediatric HCM with rhabdomyosarcoma, while Denosumab may be another option in ZA-refractory cases.


Corresponding author: Dr. Fatih Kilci, Division of Pediatric Endocrinology, Department of Pediatrics, School of Medicine, Kocaeli University, 41001, İzmit, Kocaeli, Turkey, Phone: +90 262 303 7500, E-mail:

  1. Research funding: None declared.

  2. Author contribution: All authors have accepted responsibility for the entire content of this manuscript and approved its submission.

  3. Competing interests: Authors state no conflict of interest.

  4. Informed consent: Informed consent was obtained from all individuals included in this study.

  5. Ethical approval: The local Institutional Review Board deemed the study exempt from review.

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Received: 2022-02-05
Accepted: 2022-04-19
Published Online: 2022-05-10
Published in Print: 2022-08-26

© 2022 Walter de Gruyter GmbH, Berlin/Boston

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