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      Primary hyperparathyroidism due to atypical vertically long cystic adenoma


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          Parathyroid cystic adenomas are often misdiagnosed as thyroid cysts and routine preoperative diagnostic tools, such as ultrasonography (US) or 99m technetium-sestamibi (99mTc-MIBI) scans, cannot clearly distinguish between these entities. We present a 67-year-old hypercalcemic woman with a cervical cystic lesion who had negative sestamibi scan results. Her laboratory data indicated primary hyperparathyroidism (serum calcium concentration 14.0 mg/dl, phosphate concentration 2.3 mg/dl, and intact parathyroid hormone (PTH) concentration 239 pg/ml). The cervical US and computed tomography scans revealed a large and vertically long cystic mass (12×11×54 mm). A mass was located from the upper end of the left thyroid lobe to the submandibular region and was not clearly distinguishable from the thyroid. For preoperative definitive diagnosis, we carried out a parathyroid fine-needle aspiration (FNA) and PTH assay (PTH–FNA) of liquid aspirated from the cyst. The intact PTH–FNA concentration was 1.28×10 6 pg/ml, and the patient was diagnosed with primary hyperparathyroidism due to a cystic mass. She underwent a left upper parathyroidectomy and her serum calcium and intact PTH concentration immediately decreased to normal levels. This report describes the usefulness of PTH–FNA for localizing and differentiating an atypical functional parathyroid lesion from nonfunctional tissue in primary hyperparathyroidism.

          Learning points

          • Cystic parathyroid lesions, even in the case of elevated PTH levels, can produce negative results in 99mTc-MIBI scans.

          • Preoperative diagnosis of parathyroid cysts detectable on US is possible by parathyroid FNA and PTH assay (PTH–FNA) of liquid aspirated from the cyst, if malignancy is not suspected.

          • PTH–FNA could be helpful in the differential diagnosis of an equivocal cervical tumor.

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          Most cited references9

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          Parathyroid carcinoma: update and guidelines for management.

          Parathyroid carcinoma is one of the rarest known malignancies that may occur sporadically or as a part of a genetic syndrome. It accounts for approximately 1% of patients with primary hyperparathyroidism. The majority (90%) of parathyroid cancer tumors are hormonally functional and hypersecrete parathyroid hormone (PTH). Thus, most patients exhibit strong symptomatology of hypercalcemia at presentation. Sometimes, it can be difficult to diagnose parathyroid cancer preoperatively due to clinical features shared with benign causes of hyperparathyroidism. Imaging techniques such as neck ultrasound and 99mTc sestamibi scan can help localize disease, but they are not useful in the assessment of malignancy potential. Fine needle aspiration (FNA) prior to initial operation is not recommended due to technical difficulty in differentiating benign and malignant disease on cytology specimens and the possible associated risk of tumor seeding from the needle track. Complete surgical resection with microscopically negative margins is the recommended treatment and offers the best chance of cure. Persistent or recurrent disease occurs in more than 50% of patients with parathyroid carcinoma. Surgical resection is also the primary mode of therapy for recurrence since it can offer significant palliation for the metabolic derangement caused by hyperparathyroidism and allows hypercalcemia to become more medically manageable. However, reoperation is rarely curative and eventual relapse is likely. Chemotherapy and external beam radiation treatments have been generally ineffective in the treatment of parathyroid carcinoma. Typically, these patients require repeated operations that predispose them to accumulated surgical risks with each intervention. In inoperable cases, few palliative treatment options exist, although treatment with calcimimetics can effectively control hypercalcemia in some patients. Most patients ultimately succumb to complications of hypercalcemia rather than from tumor burden or infiltration.
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            The anatomic basis of parathyroid surgery.

            C. Wang (1976)
            A study of 645 normal adult parathyroid glands in 160 cadavers revealed that there is a definite pattern of anatomic distribution on the basis of the embryologic development of the parathyroid, thyroid, and thymic glands. The sites of predilection of the upper gland (Parathyroid IV) are, in order of frequency, the cricothyroid junction; the dorsum of the upper pole of the thyroid; and the retropharyngeal space. Those of the lower gland (Parathyroid III) are at the lower pole of the thyroid and the thymic tongue; rarely in the upper, the lateral neck, or the mediastinum. An understanding of the developmental relationship of the parathyroid glands to the thyroid and the thymus is fundamental in the delineation of the embryologic origin of the parathyroid glands. The parathyroid gland, located within the surgical capsule of the thyroid (subcapsular), when diseased, remains in place locally. A gland outside of the capsule (extracapsular) is often displaced into the posterior or anterior mediastinum. A collective assessment of the size, weight, color, shape, and consistency of the parathyroid gland is mandatory in the determination of its normalcy. Frozen section examination for stromal and intracellular fatty content is an added assurance of normalcy. That parathyroid glands sink in saline solution, and fat globules float, may aid in differentiating the two types of tissue. Supernumerary, fused, and intrathyroidal parathyroids, albeit rare, are of surgical importance.
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              Factors contributing to negative parathyroid localization: an analysis of 1000 patients.

              Localizing studies are the key for determining the optimal surgical strategy in patients with primary hyperparathyroidism (HP). Most of the data in the literature are retrospective in nature and from analysis on a per patient basis. This is a prospective study looking at the characteristics of the patient and the gland that determine the likelihood of an abnormal parathyroid to be detected by ultrasonography (US) and sestamibi scan (MIBI). This is a prospective analysis of 1000 consecutive patients with HP who underwent parathyroidectomy at a tertiary care center. The study group included HP with single gland disease (63%), double adenoma (15%), as well as hyperplasia (15%), familial HP (2%), and secondary/tertiary HP (6%). All patients underwent surgeon-performed neck US followed by MIBI scan. Univariate logistic regression and multivariate analyses were performed on pre- and intraoperative variables. A total of 1845 abnormal glands were analyzed. Overall, US was superior to MIBI for the detection of abnormal glands in all subgroups. On multivariate analysis, body mass index (BMI), gland size, and gland volume were the statistically significant independent factors predicting detection by both US and MIBI in primary HP. The sensitivity of US was better for single gland disease than for multigland disease in primary HP, but the sensitivity of MIBI was similar in both groups. For a given size, hyperplastic glands in primary HP imaged less well with US and MIBI than in familial or secondary/tertiary HP. This prospective study demonstrates that BMI and gland size independently predict accurate detection of abnormal parathyroid glands by US and MIBI in sporadic primary HP. Understanding the factors that affect the accuracy of parathyroid localization tests will allow the surgeon to develop a successful surgical strategy in a given patient.

                Author and article information

                Endocrinol Diabetes Metab Case Rep
                Endocrinol Diabetes Metab Case Rep
                EDM Case Reports
                Endocrinology, Diabetes & Metabolism Case Reports
                Bioscientifica Ltd (Bristol )
                1 October 2014
                : 2014
                : 140086
                [1 ]Department of Endocrinology and Metabolism , National Hospital Organization Kyoto Medical Center , Kyoto, Japan
                [2 ]Department of Diabetes , Endocrinology and Metabolism, Tango Central Hospital , Kyoto, Japan
                [3 ]Clinical Research Institute , National Hospital Organization Kyoto Medical Center , 1-1 Mukaihata-cho, Fukakusa, Fushimi-ku, Kyoto, 612-8555, Japan
                [4 ]Department of Head and Neck Surgery , National Hospital Organization Kyoto Medical Center , 1-1 Mukaihata-cho, Fukakusa, Fushimi-ku, Kyoto, 612-8555, Japan
                [5 ]Osaka Saiseikai Ibaraki Hospital , Osaka, Japan
                Author notes
                Correspondence should be addressed to T Usui Email: tusui@ 123456kuhp.kyoto-u.ac.jp
                © 2014 The authors

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivs 3.0 Unported License.

                : 23 September 2014
                : 2 October 2014
                Unique/Unexpected Symptoms or Presentations of a Disease


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