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腹部脂肪肉瘤异位产生骨化三醇导致的恶性高钙血症。

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

作者信息

Figueiredo Ana, Pereira Teresa, Cabrera Rafael, Simões-Pereira Joana, Leite Valeriano

机构信息

Endocrinology, Instituto Português de Oncologia de Lisboa Francisco Gentil, Lisboa, PRT.

Pathology, Instituto Português de Oncologia de Lisboa Francisco Gentil, Lisboa, PRT.

出版信息

Cureus. 2023 Jan 6;15(1):e33446. doi: 10.7759/cureus.33446. eCollection 2023 Jan.

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.

摘要

恶性肿瘤高钙血症(HM)是一种常见的副肿瘤综合征,与疾病的不良预后相关。在实体瘤中,HM主要是由于甲状旁腺激素相关肽(PTHrP)的产生所致。我们报告一例60岁男性,患有25 cm的腹膜后脂肪肉瘤,术前血液检查诊断为严重高钙血症(16.8 mg/dL)。高钙血症检查显示甲状旁腺激素(PTH)受抑制、PTHrP正常以及血清1,25-二羟维生素D(1,25(OH)2D)水平升高。手术后,高钙血症和骨化三醇水平恢复正常。肿瘤的免疫组织化学分析显示肿瘤细胞表达1α-羟化酶。据我们所知,这是首例仅由骨化三醇异位产生引起的脂肪肉瘤相关性高钙血症。尽管高钙血症的这种病因罕见,但对于血清钙水平高、PTH受抑制且PTHrP正常的患者,在检查过程中应考虑检测1,25(OH)2D。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ccf6/9897700/7a327b7c5f9f/cureus-0015-00000033446-i01.jpg

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