{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,14]],"date-time":"2026-04-14T15:57:40Z","timestamp":1776182260314,"version":"3.50.1"},"reference-count":25,"publisher":"BMJ","issue":"4","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2026,3,31]]},"published-print":{"date-parts":[[2026,4]]},"abstract":"<jats:p>Hypercalcaemia of malignancy (HM) can result from excess parathyroid hormone-related peptide (PTHrP), osteolysis from bone metastasis or overproduction of 1,25 dihydroxyvitamin D (1,25[OH]2D) by the enzyme 1-\u03b1 hydroxylase. HM among sarcomas is rare, with the most common mechanism being overproduction of PTHrP. There are only three published cases of sarcoma-related HM caused by overproduction of 1,25[OH]2D. We herein report a patient who presented with abdominal pain and generalised weakness and was diagnosed with a large multifocal retroperitoneal dedifferentiated liposarcoma. His bloodwork showed severe hypercalcaemia and extensive workup revealed 1,25[OH]2D-related HM. He was treated with intravenous fluids, bisphosphonates, glucocorticoids and cytotoxic chemotherapy with improvement in his pain and normalisation of his calcium levels. We describe the unique features of his case and review existing reports of 1,25[OH]2D and PTHrP-associated HM in sarcomas.<\/jats:p>","DOI":"10.1136\/bcr-2025-269928","type":"journal-article","created":{"date-parts":[[2026,4,14]],"date-time":"2026-04-14T14:55:19Z","timestamp":1776178519000},"page":"e269928","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":0,"title":["Dedifferentiated liposarcoma and 1,25 dihydroxyvitamin D-induced paraneoplastic hypercalcaemia"],"prefix":"10.1136","volume":"19","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1263-6570","authenticated-orcid":false,"given":"Meher Binte","family":"Ali","sequence":"first","affiliation":[{"name":"Internal Medicine, University of Maryland Medical Center, Baltimore, Maryland, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Aaron","family":"Ciner","sequence":"additional","affiliation":[{"name":"University of Maryland Marlene and Stewart Greenebaum Comprehensive Cancer Center, Baltimore, Maryland, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2026,4,14]]},"reference":[{"key":"2026041407551179000_19.4.e269928.1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMcp042806"},{"key":"2026041407551179000_19.4.e269928.2","doi-asserted-by":"publisher","DOI":"10.2147\/TCRM.S83681"},{"key":"2026041407551179000_19.4.e269928.3","first-page":"2686","article-title":"Increasing incidence of liposarcoma: a population-based study of national surveillance databases, 2001\u20132016","volume":"26","author":"Crago","year":"2019","journal-title":"Ann Surg Oncol"},{"key":"2026041407551179000_19.4.e269928.4","doi-asserted-by":"crossref","DOI":"10.1016\/j.ctrv.2024.102846","article-title":"Diagnosis and management of dedifferentiated liposarcoma: A multidisciplinary position statement","volume":"131","author":"Haddox","year":"2024","journal-title":"Cancer Treat Rev"},{"key":"2026041407551179000_19.4.e269928.5","doi-asserted-by":"publisher","DOI":"10.1016\/S1470-2045(17)30622-8"},{"key":"2026041407551179000_19.4.e269928.6","first-page":"39","article-title":"Hypercalcemia in clear cell sarcoma","volume":"73","author":"Arakawa","year":"2014","journal-title":"Hawaii J Med Public Health"},{"key":"2026041407551179000_19.4.e269928.7","doi-asserted-by":"crossref","DOI":"10.1002\/pbc.26778","article-title":"Hypercalcaemia secondary to ectopic parathyroid hormone expression in an adolescent with metastatic alveolar rhabdomyosarcoma","volume":"65","author":"Brooks","year":"2018","journal-title":"Pediatr Blood Cancer"},{"key":"2026041407551179000_19.4.e269928.8","doi-asserted-by":"publisher","DOI":"10.1136\/jcp.42.3.330"},{"key":"2026041407551179000_19.4.e269928.9","first-page":"e131","article-title":"Double trouble: a case of primary hyperparathyroidism and humoral hypercalcemia of malignancy secondary to epithelioid angiosarcoma occurring in a single patient","volume":"18","author":"Jensen","year":"2012","journal-title":"Endocr Pract"},{"key":"2026041407551179000_19.4.e269928.10","doi-asserted-by":"crossref","first-page":"125","DOI":"10.1002\/(SICI)1096-9098(199610)63:2<125::AID-JSO10>3.0.CO;2-5","article-title":"Tumor-induced hypercalcemia in a patient with extensive soft tissue sarcoma: effects of bisphosphonate therapy and surgery","volume":"63","author":"Oleffe","year":"1996","journal-title":"J Surg Oncol"},{"key":"2026041407551179000_19.4.e269928.11","doi-asserted-by":"crossref","first-page":"263","DOI":"10.1111\/jog.12136","article-title":"Humoral hypercalcemia caused by uterine corpus carcinosarcoma consisting of squamous cell carcinoma in its epithelial component","volume":"40","author":"Takamatsu","year":"2014","journal-title":"J Obstet Gynaecol Res"},{"key":"2026041407551179000_19.4.e269928.12","doi-asserted-by":"crossref","DOI":"10.1093\/jscr\/rjae022","article-title":"Metastatic multicentric epithelioid angiosarcoma of bone. A case report with pitfalls. Tumor seeding after percutaneous biopsy and hypercalcemia","volume":"2025","author":"Fuentes-S\u00e1nchez","year":"2025","journal-title":"J Surg Case Rep"},{"key":"2026041407551179000_19.4.e269928.13","doi-asserted-by":"crossref","first-page":"22","DOI":"10.12659\/AJCR.900088","article-title":"Humoral Hypercalcemia in Uterine Cancers: A Case Report and Literature Review","volume":"18","author":"Motilal Nehru","year":"2017","journal-title":"Am J Case Rep"},{"key":"2026041407551179000_19.4.e269928.14","doi-asserted-by":"crossref","first-page":"246","DOI":"10.1007\/s00223-023-01099-8","article-title":"A Case of Hypercalcemia from PTHrP-Producing Fibromyxoid Sarcoma Responsive to Glucocorticoid Therapy","volume":"113","author":"Niu","year":"2023","journal-title":"Calcif Tissue Int"},{"key":"2026041407551179000_19.4.e269928.15","doi-asserted-by":"crossref","first-page":"643","DOI":"10.1111\/j.1349-7006.1989.tb01691.x","article-title":"A case of leiomyosarcoma associated with humoral hypercalcemia of malignancy: demonstration of biological and immunological activities of parathyroid hormone-related protein in the tumor extract","volume":"80","author":"Nagata","year":"1989","journal-title":"Jpn J Cancer Res"},{"key":"2026041407551179000_19.4.e269928.16","doi-asserted-by":"crossref","first-page":"184","DOI":"10.4158\/EP.11.3.184","article-title":"Hypercalcemia and local production of parathyroid hormone-related protein by a perisellar rhabdomyosarcoma after remote pituitary irradiation","volume":"11","author":"Florez","year":"2005","journal-title":"Endocr Pract"},{"key":"2026041407551179000_19.4.e269928.17","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2014-4250"},{"key":"2026041407551179000_19.4.e269928.18","doi-asserted-by":"crossref","first-page":"e181","DOI":"10.5858\/2003-127-e181-APHPME","article-title":"A parathyroid hormone-related protein-secreting metastatic epithelioid leiomyosarcoma. A case report and review of the literature","volume":"127","author":"Tang","year":"2003","journal-title":"Arch Pathol Lab Med"},{"key":"2026041407551179000_19.4.e269928.19","article-title":"Malignant Hypercalcemia Due to the Ectopic Production of Calcitriol by an Abdominal Liposarcoma","volume":"15","author":"Figueiredo","year":"2023","journal-title":"Cureus"},{"key":"2026041407551179000_19.4.e269928.20","first-page":"169","article-title":"Hypercalcemia of Malignancy: Simultaneous Elevation in Parathyroid Hormone-Related Peptide and 1,25 Dihydroxyvitamin D in Sarcoma","volume":"7","author":"Kim","year":"2021","journal-title":"AACE Clin Case Rep"},{"key":"2026041407551179000_19.4.e269928.21","unstructured":"The WHO Classification of Tumors Editorial Board . WHO classification of tumours of soft tissue and bone. 5th edn. Lyon: IARC Press, 2020:36\u201348."},{"key":"2026041407551179000_19.4.e269928.22","doi-asserted-by":"crossref","first-page":"22","DOI":"10.1007\/s11864-020-0705-7","article-title":"Dedifferentiated Liposarcoma: Systemic Therapy Options","volume":"21","author":"Gahvari","year":"2020","journal-title":"Curr Treat Options in Oncol"},{"key":"2026041407551179000_19.4.e269928.23","doi-asserted-by":"crossref","DOI":"10.1016\/j.bonr.2021.101148","article-title":"Hypercalcemia following discontinuation of denosumab therapy: A systematic review","volume":"15","author":"Horiuchi","year":"2021","journal-title":"Bone Rep"},{"key":"2026041407551179000_19.4.e269928.24","unstructured":"Goltzman D . Approach to hypercalcemia. In: Endotext. South Dartmouth [MA]: MDText.com, Inc, 2000. Available: https:\/\/pubmed.ncbi.nlm.nih.gov\/25905352\/"},{"key":"2026041407551179000_19.4.e269928.25","doi-asserted-by":"crossref","first-page":"249","DOI":"10.11005\/jbm.2017.24.4.249","article-title":"Clinical Characteristics, Causes and Survival in 115 Cancer Patients with Parathyroid Hormone Related Protein-mediated Hypercalcemia","volume":"24","author":"Jin","year":"2017","journal-title":"J Bone Metab"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2025-269928","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,14]],"date-time":"2026-04-14T14:55:22Z","timestamp":1776178522000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2025-269928"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,4]]},"references-count":25,"journal-issue":{"issue":"4","published-online":{"date-parts":[[2026,4,14]]},"published-print":{"date-parts":[[2026,4]]}},"alternative-id":["10.1136\/bcr-2025-269928"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2025-269928","relation":{},"ISSN":["1757-790X"],"issn-type":[{"value":"1757-790X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,4]]}}}