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多发异位甲状旁腺癌致持续性高钙血症危象和复发性急性胰腺炎:纵隔甲状旁腺癌病例报告及文献复习。

Persistent Hypercalcemia Crisis and Recurrent Acute Pancreatitis Due to Multiple Ectopic Parathyroid Carcinomas: Case Report and Literature Review of Mediastinal Parathyroid Carcinoma.

机构信息

Key Laboratory of Endocrinology, Department of Endocrinology, National Commission of Health, Peking Union Medical College Hospital, Chinese Academy of Medical Science, Beijing, China.

出版信息

Front Endocrinol (Lausanne). 2020 Sep 10;11:647. doi: 10.3389/fendo.2020.00647. eCollection 2020.

Abstract

Mediastinal parathyroid carcinoma (PC) is a rare entity in primary hyperparathyroidism. The aim of this report is to demonstrate a case of mediastinal PC, and to provide a systemic literature review of this rare condition. A 34-year-old woman who had already undergone two cervical operations for hyperparathyroidism suffered from another recurrence, presenting with recurrent acute pancreatitis and persistent hypercalcemic crisis. Technetium-99 methoxyisobutylisonitrile imaging (MIBI) and computed tomography scanning (CT) identified three possible parathyroid tumors, one of which was the recurrence of residual tumor locating in the thyroid region, while the other two were ectopic tumors locating in the suprasternal fossa and thymus region, respectively. Pathological examination confirmed the diagnosis of PC. We conducted a systemic literature review by searching the PubMed MEDLINE from 1951 to 2019 for studies of all types in the English language only, using terms "mediastinal, mediastinum, parathyroid, carcinoma." Including our reported case, a total of 21 cases with ectopic mediastinal PCs were assessed for demographic data, tumor location and size, biochemical findings, and symptomatology, etc. Two thirds of the patients were men, with a mean age of 44 years old, a mean serum calcium of 14.2 mg/dl, and a mean serum intact parathyroid hormone of 1,216 pg/ml. We identified 89.5% of carcinomas in the anterosuperior mediastinum, and 10.5% in the middle mediastinum, with a mean diameter of 54 mm, and a mean weight of 216 g. MIBI and CT were the most commonly used methods to localize these mediastinal tumors, with 69.2 and 100% sensitivity, respectively. Half of the patients underwent more than one operation. Diagnosis and treatment of mediastinal PCs represent a challenge. Early suspicion, appropriate preoperative localization studies, and the cooperation of endocrinologists and surgeons are crucial in the effective management.

摘要

纵隔甲状旁腺癌(PC)在原发性甲状旁腺功能亢进症中较为罕见。本报告旨在介绍 1 例纵隔 PC 病例,并对这种罕见疾病进行系统的文献复习。1 例 34 岁女性,曾因甲状旁腺功能亢进症行 2 次颈部手术,此次因反复发作性急性胰腺炎和持续性高钙血症危象而就诊。锝-99 甲氧异丁基异腈(MIBI)显像和计算机断层扫描(CT)确定了 3 个可能的甲状旁腺肿瘤,其中 1 个为位于甲状腺区域的残留肿瘤复发,另 2 个为位于胸骨上窝和胸腺区域的异位肿瘤。病理检查证实为 PC。我们通过检索 1951 年至 2019 年在 PubMed MEDLINE 数据库中发表的英文文献,使用“纵隔、胸腔、甲状旁腺、癌”等术语进行系统的文献复习。包括我们报告的病例在内,共有 21 例异位纵隔 PC 患者的人口统计学数据、肿瘤位置和大小、生化发现和症状等进行了评估。2/3 的患者为男性,平均年龄为 44 岁,平均血清钙为 14.2mg/dl,平均血清全段甲状旁腺激素为 1216pg/ml。我们发现 89.5%的癌位于前上纵隔,10.5%位于中纵隔,平均直径为 54mm,平均重量为 216g。MIBI 和 CT 是定位这些纵隔肿瘤最常用的方法,敏感性分别为 69.2%和 100%。一半的患者接受了不止一次手术。纵隔 PCs 的诊断和治疗是一个挑战。早期怀疑、适当的术前定位研究以及内分泌科医生和外科医生的合作对有效管理至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7376/7511513/1c1e7ac742ab/fendo-11-00647-g0001.jpg

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