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A Case of Hyperparathyroidism due to a Large Intrathyroid Parathyroid Adenoma with Recurrent Episodes of Acute Pancreatitis.

ABSTRACT: We report a case of a 66-year-old woman who developed hyperparathyroidism due to a large intrathyroid parathyroid adenoma with episodes of acute pancreatitis. She had previously been treated for acute pancreatitis twice. Serum calcium was 12.4 mg/dL, and intact parathyroid hormone was 253 pg/dL. Ultrasonography and computed tomography of the neck with contrast enhancement revealed a soft tissue mass (28 mm transverse diameter) within the left lobe of the thyroid. 99mTc-MIBI scintigraphy demonstrated focal accumulation due to increased radiotracer uptake in the left thyroid lobe. Left hemithyroidectomy was performed. Histopathology showed no signs of invasion, and this is consistent with parathyroid adenoma. Immunostaining was positive for expression of chromogranin A and parathyroid hormone. The patient had no episode of pancreatitis after the operation. In a patient with recurrent episodes of pancreatitis, the possibility of complication with hyperparathyroidism should be considered.

SUBMITTER: Kageyama K

PROVIDER: S-EPMC5549494 | biostudies-other | 2017

REPOSITORIES: biostudies-other

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Project description:Autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED) is a rare monogenic disorder, associated with endocrine deficiencies and non-endocrine involvement. Gastrointestinal (GI) manifestations appear in approximately 25% of patients and are the presenting symptom in about 10% of them. Limited awareness among pediatricians of autoimmune enteropathy (AIE) caused by destruction of the gut endocrine cells in APECED patients delays diagnosis and appropriate therapy. We describe an 18-year-old female presenting at the age of 6.10 years with hypoparathyroidism, oral candidiasis and vitiligo. The clinical diagnosis of APECED was confirmed by sequencing the autoimmune regulator-encoding (AIRE) gene. Several characteristics of the disease-Hashimoto's thyroiditis, Addison's disease, diabetes mellitus type 1 and primary ovarian insufficiency-developed over the years. She had recurrent episodes of severe intractable hypocalcemia. Extensive GI investigations for possible malabsorption, including laboratory analyses, imaging and endoscopy with biopsies were unremarkable. Revision of the biopsies and chromogranin A (CgA) immunostaining demonstrated complete loss of enteroendocrine cells in the duodenum and small intestine, confirming the diagnosis of AIE. Management of hypocalcemia was challenging. Only intravenous calcitriol maintained calcium in the normal range. Between hypocalcemic episodes, the proband maintained normal calcium levels, suggesting a fluctuating disease course. Repeated intestinal biopsy revealed positive intestinal CgA immunostaining. The attribution of severe hypocalcemic episodes to AIE emphasizes the need for increased awareness of this unique presentation of APECED. The fluctuating disease course and repeated intestinal biopsy showing positive CgA immunostaining support a reversible effect of GI involvement. CgA immunostaining is indicated in patients with APECED for whom all other investigations have failed to reveal an explanation for the malabsorption.

Project description:Background and aimsEctopic parathyroid adenoma is a rare entity. Its clinical management is challenging due to varying locations in the mediastinum. The aim of our study is to report our experience about the preoperative localization of the ectopic parathyroid adenoma and to emphasize the major role of the intraoperative parathyroid hormone assay in such circumstances.MethodsIt is a monocentric, retrospective study about patients diagnosed with EPA (ectopic parathyroid adenoma) from January 2015 to December 2016. Clinical aspects, preoperative management as well as the surgical procedures have been analyzed.ResultsThere were 7 women, with an average age of 59.14 years. Six patients presented with biological disorders of the phosphocalcic metabolism such as spontaneous bone fracture and recurrent renal lithiasis. In one case, EPA was discovered in the setting of malignant hypercalcemia. The topographic preoperative assessment with a cervicothoracic CT (computed tomography) showed spontaneously hyperdense tissular masses of variable localizations in the mediastinum. A Tc-99 m (99mTc - MIBI) scintigraphy was performed in 5 patients and showed uptake in all cases. We performed cervicotomy in 1 case, manubriotomy in 2 patients, neck manubriotomy in 2 cases, total vertical sternotomy in 1 case, and posterolateral thoracotomy in 1 patient. The lesion was localized in the mediastinum in 1 patient in the perithymic fat in 1 case; EPA was laterotracheal in 1 case, retro tracheal in 1 case, intra-thymic in 2 cases, inter-jugulo-carotidian with contact with the left subclavial artery in 1 case, and anterior mediastinal in 1 patient. The 1-h after-parathormonemia following removal of the surgical specimens showed a decrease value of 45 and 80% of the baseline value. No surgical morbidity was noted after an average follow-up of 7.9 months (range of 5-18 months).ConclusionThe preoperative topographic diagnosis of ectopic parathyroid adenoma is challenging for the surgeon despite progress in the morphological assessment. The intraoperative parathyroid hormone assay is a valuable tool for an appropriate surgical management.

Project description:Persistent or recurrent renal hyperparathyroidism may occur after total parathyroidectomy and transcervical thymectomy with forearm autograft under continuous stimulation due to uremia. Parathyroid hormone (PTH) levels may reflect persistent or recurrent renal hyperparathyroidism because of the enlarged autografted parathyroid glands in the forearm or remnant parathyroid glands in the neck or mediastinum. Detailed imaging requires predictive localization of causative parathyroid glands. Casanova and simplified Casanova tests may be convenient. However, these methods require avascularization of the autografted forearm for >10 min with a tourniquet or Esmarch. The heavy pressure during avascularization can be incredibly painful and result in nerve damage. An easier method that minimizes the burden on patients in addition to predicting the localization of causative parathyroid glands was developed in this study. Ninety patients who underwent successful re-parathyroidectomy for persistent or recurrent renal hyperparathyroidism after parathyroidectomy between January 2000 and July 2019 were classified according to the localization of causative parathyroid glands (63 and 27 patients in the autografted forearm and the neck or mediastinum groups, respectively). Preoperatively, intact PTH levels were measured from bilateral forearm blood samples following a 5-min avascularization of the autografted forearm. Cutoff values of the intact PTH ratio (intact PTH level obtained from the non-autografted forearm before re-parathyroidectomy/intact PTH level obtained from the autografted forearm before re-parathyroidectomy) were investigated with receiver operating characteristic curves to localize the causative parathyroid glands. Intact PTH ratios of <0.310 with an area under the curve (AUC) of 0.913 (95% confidence interval [CI]: 0.856-0.970; P < 0.001) and >0.859 with an AUC 0.744 (95% CI: 0.587-0.901; P = 0.013) could predict causative parathyroid glands in the autografted forearm and the neck or mediastinum with diagnostic accuracies of 81.1% and 83.3%, respectively. Therefore, we propose that the intact PTH ratio is useful for predicting the localization of causative parathyroid glands for re-parathyroidectomy.

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A Case of Hyperparathyroidism due to a Large Intrathyroid Parathyroid Adenoma with Recurrent Episodes of Acute Pancreatitis.

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