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      The Clinical Features of Cystic Parathyroid Adenoma in Chinese Population: A Single-Center Experience

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          Abstract

          Objective

          Cystic parathyroid adenoma is a rare cause of primary hyperparathyroidism, but its preoperative diagnosis and management remain inconclusive.

          Method

          We retrospectively identified patients with cystic parathyroid adenomas who underwent surgery at Peking Union Medical College Hospital.

          Results

          Patients with cystic parathyroid adenomas had higher serum intact parathyroid hormone and calcium levels, larger maximum tumor diameter, and lower serum inorganic phosphorus level than did those with solid adenomas. Patients with cystic adenomas were predominantly male, and hypercalcemic crisis and atypical adenomas were common. The accuracy of preoperative localization methodologies was lower in patients with cystic adenomas than in patients with solid adenomas. US-guided fine-needle aspiration was performed in 11 patients. In all patients, the iPTH level in cystic fluid was much higher than that in serum. No sign of recurrence was observed after a median follow-up of 39 months.

          Conclusions

          Cystic parathyroid adenomas may not be as rare as previously reported. FNA may be a safe and feasible localization methodology for patients with inconclusive preoperative localization methodologies. Close follow-up is necessary for patients with cystic parathyroid adenomas, which account for a substantial proportion of atypical adenoma cases.

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          Most cited references36

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          A single-institution 25-year review of true parathyroid cysts.

          Parathyroid cysts (PCs) are rare, and their origin is a subject of debate. They have been described as either functional, causing hyperparathyroidism, or non-functional in eucalcaemic patients. We have performed a 25-year departmental review of PCs. Features studied included the clinical presentation and intra-operative findings, and a histological review was performed. Cases of cystic degeneration of parathyroid adenomas and pseudocystic change were excluded. Over 25 years, 22,009 thyroidectomies and 2,505 parathyroidectomies were performed in our department. Amongst these, 38 non-functional PCs were documented in 37 patients. The mode of presentation included incidental findings on routine chest x-ray, compressive symptoms or an asymptomatic palpable neck mass. Aspiration was the initial treatment in 14 patients and was curative in 10 of these. Four out of 14 patients underwent surgical procedures for recurrence of the cyst that occurred 6 to 48 months after aspiration. In 27 patients, surgery was performed and all identified PCs were localized in the inferior parathyroid glands. Histologically, the cyst wall consisted in associations of lymphoid, muscular, thymic, salivary, adipose and mesenchymal tissues. PCs are rare but should be included within the differential diagnosis of a neck lump. True PCs are non-functional. Pathological and immunohistochemical findings are suggestive of a branchial origin. Fine-needle aspiration may be curative and is diagnostic due to the characteristic appearance of the fluid and high PTH levels on assay.
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            Cystic parathyroid lesions: functional and nonfunctional parathyroid cysts.

            Functional parathyroid cysts (FPCs) and nonfunctional parathyroid cysts (NPCs) are 2 distinct clinical and histologic entities. Review of prospective clinical database records. Tertiary academic center. Patients enrolled in a prospective surgical database between January 1, 1990, and May 31, 2007. Cervical exploration for primary hyperparathyroidism or cervical mass. Age, sex, morbidity, imaging results, pathologic findings, cyst characteristics (size, location, and fluid), and perioperative calcium and parathyroid hormone levels. Cystic parathyroid lesions were found in 48 of 1769 patients (3%) studied. Functional parathyroid cysts were more common than NPCs, arising in 41 of 48 patients (85%), and showed no predisposition for sex or embryologic origin. Single-photon emission computed tomographic imaging failed to localize FPCs in 12 of 37 patients (32%). The fluid in FPCs was clear or colorless in 9 of 15 characterized specimens (60%). Rupture of cystic parathyroid lesions during resection was associated with prolonged elevation of intraoperative parathyroid hormone levels (P =.045). Cystic parathyroid lesions weighing 4 g or more were associated with the development of postoperative symptomatic hypocalcemia (P =.03). Functional parathyroid cysts occurred exclusively in adenomas with cystic change, whereas NPCs (with 1 exception) were without associated adenoma on final histologic examination. Cystic parathyroid lesions often contain turbid or colored fluid, and FPCs are more common than NPCs. Neck cysts of uncertain origin should be diagnostically aspirated for parathyroid hormone content. During resection, cyst rupture should be avoided, and patients with large cysts should be managed expectantly to forestall the development of symptomatic hypocalcemia. Functional parathyroid cysts and NPCs are likely 2 separate clinical and histologic entities.
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              Negative parafibromin staining predicts malignant behavior in atypical parathyroid adenomas.

              The histopathological criteria for carcinoma proposed by the World Health Organization (WHO) are imperfect predictors of the malignant potential of parathyroid tumors. Negative parafibromin (PF) and positive protein gene product 9.5 (PGP9.5) staining are markers of CDC73 mutation and occur commonly in carcinoma but rarely in adenomas. We investigated whether PF and PGP9.5 staining could be used to predict the behavior of atypical parathyroid adenomas--tumors with atypical features that do not fulfill WHO criteria for malignancy.

                Author and article information

                Contributors
                Journal
                Int J Endocrinol
                Int J Endocrinol
                IJE
                International Journal of Endocrinology
                Hindawi
                1687-8337
                1687-8345
                2018
                15 July 2018
                : 2018
                : 3745239
                Affiliations
                1Department of General Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100730, China
                2Department of Ultrasound, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100730, China
                Author notes

                Academic Editor: Carlo Cappelli

                Author information
                http://orcid.org/0000-0001-8770-4585
                http://orcid.org/0000-0003-1324-6213
                http://orcid.org/0000-0001-7081-2299
                Article
                10.1155/2018/3745239
                6079486
                30123260
                8c224625-8b75-4729-ad60-54c445a0ac5f
                Copyright © 2018 Ya Hu et al.

                This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 30 January 2018
                : 21 June 2018
                Funding
                Funded by: Peking Union Medical College
                Award ID: 2016
                Funded by: Chinese Academy of Medical Sciences
                Award ID: 2017-I2M-1-001
                Categories
                Research Article

                Endocrinology & Diabetes
                Endocrinology & Diabetes

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