A Case of an Ectopic Parathyroid Adenoma With a Concomitant 99mTc-sestamibi-avid Papillary Thyroid Carcinoma

Abstract Background: The evaluation and management of parathyroid adenomas have improved over the years. Localization of parathyroid adenomas in patients with primary hyperparathyroidism was simplified with the use of 99mTc-sestamibi scintigraphy. In the advent of minimally invasive parathyroid surgery, use of radionuclide probes reduced the need for neck exploration and intraoperative frozen section leading to fewer complications, shorter operative time and hospitalization and rapid postoperative recovery. However, limitations of these techniques should be taken into consideration in certain cases. Clinical Case: A 60 year-old female diagnosed with primary hyperparathyroidism presented with recurrent nephrolithiasis and osteoporosis. Initial laboratory evaluation showed elevated serum calcium and intact PTH (1.54 mmol/L and 146 pg/mL, respectively). 99mTc-sestamibi scintigraphy showed a sestamibi-avid focus in the inferior aspect of the right lobe suggestive of a parathyroid adenoma or hyperplasia. Pre-operative neck ultrasound showed non-specific thyroid parenchymal changes and nodules on both lobes with benign sonographic features. She underwent radionuclide-guided focused right parathyroidectomy. The identified enlarged right inferior parathyroid gland registered a highest reading of 70 cps on radionuclide probe. Post-operatively, repeat intact PTH level was still elevated (171.2 pg/mL). There was an interval non-demonstration of the sestamibi-avid focus in the inferior aspect of the right thyroid lobe with an increased sestamibi uptake in the left thyroid lobe compared to the previous parathyroid scan. Histopathologic examination showed a normocellular parathyroid gland and a multifocal papillary thyroid carcinoma. She underwent total thyroidectomy with central neck dissection and 4 parathyroid gland exploration with intraoperative parathyroid hormone assay. However, serial PTH monitoring after left inferior parathyroidectomy and after bilateral partial superior parathyroidectomy still showed elevated levels. Histopathologic examination showed mildly enlarged, normocellular parathyroid gland. The bilateral carotid sheath, retropharyngeal region and superior mediastinum were explored but no ectopic parathyroid tissues were seen. Post-operatively, calcium and PTH were still elevated (1.48 mmol/L and 200.5 pg/mL, respectively). Conclusion: This case highlights the predicaments in the management of parathyroid adenomas, recognizing the possibility of false-positive sestamibi scans due to malignant thyroid nodules and the possibility of the two diseases occurring concurrently, albeit rare.

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Rare Case of Severe Hypercalcemia Secondary to Atypical Sestamibi Negative Parathyroid Cystic Adenoma

Journal of the Endocrine Society ◽

10.1210/jendso/bvab048.540 ◽

2021 ◽

Vol 5 (Supplement_1) ◽

pp. A266-A267

Author(s):

Timur Gusov ◽

John Chen Liu ◽

Sowjanya Naha ◽

F N U Marium ◽

Joseph Theressa Nehu Parimi ◽

...

Keyword(s):

Primary Hyperparathyroidism ◽

Parathyroid Adenoma ◽

Parathyroid Gland ◽

Severe Hypercalcemia ◽

Parathyroid Adenomas ◽

Sestamibi Scan ◽

Serum Pth ◽

The Right ◽

99Mtc Sestamibi ◽

Cystic Adenoma

Abstract Primary hyperparathyroidism (PHPT) is defined as excessive secretion of parathyroid hormone (PTH) originating from the parathyroid gland. The most common cause is a single parathyroid adenoma which is typically solid. Cystic parathyroid adenomas (CPA) are the cause of about 1–2% of cases of primary hyperparathyroidism. It is known that cystic parathyroid adenomas are a result of degeneration of an existing parathyroid adenoma. SestaMIBI is an imaging study based on uptake of radioactive technetium99 and used to localize parathyroid adenomas. We describe an unusual case of severe hypercalcemia secondary to 99mTc sestaMIBI negative atypical parathyroid cystic adenoma. A 56-year-old male presented to our facility with nausea and vomiting. His past medical history included hypertension and hepatitis C with no history of fractures or kidney disease. Physical examination was normal. Upon admission the patient was afebrile with blood pressure of 170/120 mmHg and heart rate of 62 bpm. Chemistry showed Calcium of 14.5 mg/dL (8.6–10.2mg/dL), phosphorus 2.2 (2.7–4.5) mh/dL, magnesium 1.8 (1.7–2.6)mg/dL, intact PTH of 375 (15–65) pg/mL, PTH-related peptide <2.0 pmol/L(<2 pmol/L), 25-OH vitamin D of 19 ng/ml (30–80), Creatinine 1.22 (0.7–1.2)mg/dL, alkaline phosphatase 95 (40–129) units/L. He was started on aggressive hydration, calcitonin 4 units/kg, 4 mg of IV Zolendroninc acid. Neck sonogram revealed a large, complex, predominantly anechoic lesion with solid vascular components and thick internal septations in the inferior and medial aspect of the right thyroid lobe measuring 3 x 2 x 5.5 cm. Findings were confirmed with CT of the neck. Since Sestamibi scan (planar and SPECT/CT) did not show uptake in parathyroid glands, the cyst was thought to be of thyroid origin. Fine needle aspiration was not able to detect cellular material, but PTH was >100 pg/ml on the FNA sample. Otolaryngology service was consulted for parathyroidectomy. During the surgical treatment, the right upper parathyroid gland was removed with no changes in serum PTH. Next, the cystic lesion was removed with normalization of serum PTH (from 218 pg/ml to 35.2 pg/ml respectively). Intraoperative frozen section analysis was read as a cystic parathyroid adenoma. The final pathology report revealed cystic parathyroid tissue favoring parathyroid adenoma with focal atypia. Hypercalcemia resolved. Conclusions: Atypical cystic parathyroid adenomas are a rare cause of PHPT. 90% of parathyroid cysts are nonfunctional. Above mention is a case of a patient presenting with hypercalcemic crisis secondary to cystic parathyroid adenoma, which posed a diagnostic challenge as both neck ultrasound and 99mTc sestaMIBI scan were inconclusive. These findings should trigger suspicion for functional parathyroid lesions. Cystic components should be evaluated for PTH levels and if significantly elevated should be treated as a parathyroid adenoma.

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SUN-473 Primary Hyperparathyroidism and Papillary Thyroid Carcinoma, Association or Coincidence?

Journal of the Endocrine Society ◽

10.1210/jendso/bvaa046.1478 ◽

2020 ◽

Vol 4 (Supplement_1) ◽

Author(s):

Nitish Singh Nandu ◽

Janice L Gilden

Keyword(s):

Papillary Thyroid Carcinoma ◽

Thyroid Gland ◽

Primary Hyperparathyroidism ◽

Thyroid Carcinoma ◽

Parathyroid Adenoma ◽

Parathyroid Gland ◽

Total Serum ◽

Papillary Thyroid ◽

Thyroid Lobe ◽

The Right

Abstract Background: The Parathyroid glands originate from the Pharyngeal pouches, Inferior from the third and superior from the fourth. Rarely these glands migrate to the thyroid gland, isthmus or thymus and become ectopic. Primary Hyperparathyroidism (PHPT) is a common cause of hypercalcemia in ambulatory patients. It is also more frequent in women and increases with age. Its treatment is often surgical removal of the affected parathyroid gland. We present a case of an ectopic parathyroid adenoma hidden within the thyroid lobe, treated by thyroid lobectomy ultimately leading to the diagnosis and management of Papillary thyroid carcinoma. Case report: A 73-Year-old female with DM, HTN, hyperlipidemia, osteoporosis was referred to the Endocrine clinic for a history of fractures to the right upper and lower extremities after trivial falls, She was subsequently evaluated for metabolic bone disease, noted to have a PTH 78 (n=14-64 pg/ml) with a total serum calcium 9.7 (n=8.6-10.4 mg/dl), 25-OH Vit-D 14 (n=30-100 ng/ml), urinary calcium to creatinine ratio 20 (n=10-320 mg/g). The parathyroid scan showed persistent activity in the area of the inferior margin right thyroid lobe, suggesting a parathyroid adenoma. She had a parathyroidectomy and during the procedure, the parathyroid gland was unable to be visualized. Hence the Right inferior thyroid lobe was removed. The pathology also showed papillary thyroid carcinoma and the patient had a total thyroidectomy. Discussion: The relationship between PHPT and Papillary thyroid carcinoma still remains unclear. Our patient demonstrated a rare circumstance, wherein the presence of a parathyroid adenoma within the thyroid gland has led to early diagnosis and timely treatment of papillary thyroid carcinoma. Few authors reported thyroid malignancy as the most prevalent cancer among patients with PHPT as the primary disorder. While others report concurrence as a coincidental pathology. Nevertheless, we emphasize the importance of surveillance for thyroid pathology in patients with PHPT that can provide better overall patient outcomes. References: 1. Vargas-Ortega, G., et al. (2018). “Symptomatic Primary Hyperparathyroidism as a Risk Factor for Differentiated Thyroid Cancer %J Journal of Thyroid Research.” 2018: 6. 2. Miccoli, P., et al. (2006). “Incidental thyroid carcinoma in a large series of consecutive patients operated on for benign thyroid disease.” ANZ J Surg 76(3): 123-126. 3. Bentrem, D. J., et al. (2002). “Is preoperative investigation of the thyroid justified in patients undergoing parathyroidectomy for hyperparathyroidism?” Thyroid 12(12): 1109-1112.

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Persistent primary hyperparathyroidism: an uncommon location for an ectopic gland - Case report and review

Arquivos Brasileiros de Endocrinologia & Metabologia ◽

10.1590/s0004-27302012000600009 ◽

2012 ◽

Vol 56 (6) ◽

pp. 393-403 ◽

Cited By ~ 8

Author(s):

Sofia Gouveia ◽

Dírcea Rodrigues ◽

Luísa Barros ◽

Cristina Ribeiro ◽

Anabela Albuquerque ◽

...

Keyword(s):

Primary Hyperparathyroidism ◽

Parathyroid Gland ◽

Median Sternotomy ◽

Parathyroid Glands ◽

Aortopulmonary Window ◽

Genetic Syndromes ◽

Subtotal Parathyroidectomy ◽

Common Causes ◽

Ectopic Parathyroid ◽

99Mtc Sestamibi

Primary hyperparathyroidism (PHPT) is a common endocrine disorder that mainly affects middle-aged women. Patients are usually asymptomatic. The disease might be ascribable to hyperplasia, carcinoma, and single or multiple adenomas. PHPT may be sporadic or familial, the latter comprising multiple endocrine neoplasia type 1 or 2A, familial benign hypocalciuria hypercalcemia, and hyperparathyroidism-jaw tumor syndrome. The most common causes for persistent PHPT are multiglandular disease, and missed abnormal ectopic or orthotopic parathyroid glands. Imaging localization studies should precede a new surgical intervention. Ectopic parathyroid glands are rarely located at the aortopulmonary window. For diagnosis confirmation, 99mTc-sestamibi SPECT/CT seems to be an advantageous test. Another possibility is to perform 99mTc-sestamibi followed by thoracic CT or MRI. Parathyroidectomy may be performed by means of median sternotomy, thoracotomy, or video-assisted thoracoscopy. We describe a case of persistent primary hyperparathyroidism due to the presence of an ectopic parathyroid gland found at the aortopulmonary window. As the investigation necessary to clarify the etiology of recurrent nephrolithiasis proceeded, the diagnosis of PHPT was determined. The patient underwent subtotal parathyroidectomy; nevertheless, PHPT persisted. Genetic syndromes that could account for this condition were excluded. Imaging studies available at that time were not able to locate abnormal glands; moreover, the patient refused to undergo surgical exploration. Later, the patient underwent 99mTc-sestamibi SPECT/CT, which revealed a parathyroid gland at the aortopulmonary window.

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Rare concurrence of ectopic intrathyroidal parathyroid gland and papillary thyroid carcinoma within a thyroid lobe

Medicine ◽

10.1097/md.0000000000016893 ◽

2019 ◽

Vol 98 (34) ◽

pp. e16893 ◽

Cited By ~ 1

Author(s):

Jianguo Chen ◽

Yingying Wan ◽

Shuguang Chen

Keyword(s):

Papillary Thyroid Carcinoma ◽

Thyroid Carcinoma ◽

Parathyroid Gland ◽

Papillary Thyroid ◽

Thyroid Lobe

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Escisión transcervical de un adenoma paratiroideo mediastínico gigante

Studies in the Economic History of Southern Africa ◽

10.22201/fm.24484865e.2021.64.2.06 ◽

2021 ◽

Vol 64 (2) ◽

pp. 38-45

Author(s):

Isaías Adrián Barrientos López ◽

◽

Bertha Beatriz Castillo Ventura ◽

Keyword(s):

Primary Hyperparathyroidism ◽

Parathyroid Adenoma ◽

Thyroid Lobe ◽

Selective Treatment ◽

Case Presentation ◽

Parathyroid Adenomas ◽

History Of ◽

Thyroid Profile ◽

The Right ◽

Similar Location

Primary hyperparathyroidism (HPTP) is the third most common neuroendocrine disorder, its main cause is parathyroid adenoma. Within the classification, those adenomas that weigh more than 3.5 g are classified as giant parathyroid adenomas (APG), and are associated with a worse evolution and malignancy. The purpose of this article is to present our diagnostic and therapeutic approach of a patient with HPTP ssociated with a mediastinal APG. A search in the international literature of the last 10 years revealed that only 8 other centers ha ve reported the extraction of an APG of similardimensions to ours and in a similar location. Case presentation: A 66-year-old woman with a history of multinodular goiter, thyroid profile and parathormone (PTH) measurement were requested, with a report of 350.60 pg/mL. A thyroid/parathyroid scintigraphy was performed with 99mTc sestaMIBI which reported persistence of a focal area of the concentration located in the topography of the lower pole of the right thyroid lobe. This study was complemented with SPECT-CT, which reported an image of hyperfunctioning right inferior parathyroid compatible with adenoma and the patient was diagnosed with PTH. Finally, resection was performed, obtaining a tumor measuring 7.0 × 4.5 × 2.0 cm, with a weight of 24.5 g. Conclusions: Our case highlights the use of imaging studies to facilitate localization and achieve diagnosis together with clinical presentation and biochemical profile. Selective treatment was achieved safely through a minimally invasive transcervical technique, combined with the measurement of PTH. Keywords: Parathyroid adenoma; neoplasia; hypercalcemia; primary hyperparathyroidism; case report.

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Ectopic Parathyroid Adenoma in Piriform Sinus: Case Report and Review of the Literature

Ear Nose & Throat Journal ◽

10.1177/0145561318822933 ◽

2019 ◽

Vol 98 (1) ◽

pp. 14-17

Author(s):

Michael J. Connolly ◽

Dorothy Lazinski ◽

Katherine A. Aoki ◽

Laurie McLean ◽

Carlos Torres ◽

...

Keyword(s):

Computed Tomography ◽

Parathyroid Adenoma ◽

Emission Computed Tomography ◽

Piriform Sinus ◽

Ectopic Parathyroid Adenoma ◽

Sestamibi Scintigraphy ◽

Parathyroid Adenomas ◽

Ectopic Parathyroid ◽

The Right ◽

Increased Activity

During routine blood work, a 53-year-old female patient was noted to have asymptomatic hypercalcemia and subsequently found to have hyperparathyroidism. Localization studies for a suspected parathyroid adenoma included 99mTc Sestamibi scintigraphy, Single Photon Emission Computed Tomography (SPECT)/computed tomography (CT) study, and ultrasound of the neck, which were initially read as negative for parathyroid adenoma. A contrast-enhanced CT scan of the neck was performed to locate the suspected parathyroid adenoma and demonstrated a soft tissue lesion within the right piriform sinus. Flexible fiber optic nasolaryngoscopy revealed a submucosal lesion in the right piriform sinus. Following these findings, the initial 99mTc Sestamibi scintigraphy and SPECT/CT were reviewed with confirmation of a focal area of increased activity superior to the right thyroid lobe, corresponding to a nodule in the right piriform sinus that demonstrated increased activity on SPECT/CT. The patient was brought to the operating room for surgical management where a laryngoscope and operating microscope were utilized. The encapsulated lesion was dissected and excised in total. The parathyroid hormone and ionized calcium levels normalized postoperatively. Pathology confirmed a parathyroid adenoma. Parathyroid adenomas are the most common cause of primary hyperparathyroidism. Sixteen percent of parathyroid adenomas can be situated in an ectopic location. Ectopic parathyroid adenomas in the piriform sinus are rare with only a few previously documented cases. We document a rare case of ectopic parathyroid adenoma in the piriform sinus overlooked on initial imaging studies. These lesions can be challenging to localize, however, an understanding of embryology, close scrutiny of possible ectopic locations, and the application of complementary imaging techniques may prove useful for surgeons and clinicians.

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Incidental diagnosis of the tall-cell variant of the papillary microcarcinoma of the thyroid gland requires completion lymphadenectomy: case report

Polish Journal of Surgery ◽

10.5604/01.3001.0010.1023 ◽

2017 ◽

Vol 89 (3) ◽

pp. 40-43

Author(s):

I. Bartella ◽

F. Meyer ◽

K. Frauenschläger ◽

K. Reschke ◽

Th. Wallbaum ◽

...

Keyword(s):

Thyroid Gland ◽

Rare Tumor ◽

Capsular Invasion ◽

Papillary Thyroid ◽

Thyroid Lobe ◽

Tall Cell Variant ◽

Tall Cell ◽

Cell Variant ◽

The Right

Papillary thyroid carcinoma is the most common neoplasm of the thyroid gland which is usually associated with a very good prognosis. The aim of this case report is to present the disease course of a rare tumor of the thyroid gland, which is worthwhile due to its extraordinary appearance and specific management. A 46-year-old patient presented with a pronounced right-sided, but bilateral, multinodular goiter, with a volume of approximately 80 mL, as assessed on ultrasonography. Surgical removal was indicated as scintigraphy showed a 4-cm cold nodule that almost completely took up the right thyroid lobe. Because of the micronodular texture of the left thyroid lobe, complete thyroidectomy was performed according to well-established guidelines. Histopathological investigation of the specimen revealed a follicular adenoma without any malignancy in the right thyroid lobe and the tall-cell variant of the papillary thyroid microcarcinoma in the left lobe, with a capsular invasion and diameter of 0.6 cm. Because this rare tumor subtype is known for its aggressive behavior, and there was capsular invasion, low-grade differentiation, and an increased risk for lymphatic metastases, completion lymphadenectomy of the central compartments was performed after an interdisciplinary board decision. On histopathology, there were 30 tumor-free lymph nodes; final TNM classification was as follows: pT3 pN0 [0/30] L0 V0 Pn0 R0). The postoperative course was uneventful, and surgery was followed by radioiodine therapy. Six months after the surgery, clinical follow-up did revealed any sign of recurrence. The tall-cell variant is a rare and aggressive subtype of the papillary thyroid carcinoma, and it is characterized by poor 5-year survival and high recurrence rate. According to our understanding and based on current literature, this disease requires an aggressive surgical treatment and a close follow-up, as recommended by the current guidelines.

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Giant parathyroid adenoma and challenges with preoperative differentiation from malignancy

BMJ Case Reports ◽

10.1136/bcr-2021-241554 ◽

2021 ◽

Vol 14 (4) ◽

pp. e241554

Author(s):

Grace Wong ◽

Andrew Ghabbour ◽

Farzad Borumandi

Keyword(s):

Differential Diagnosis ◽

Parathyroid Adenoma ◽

Parathyroid Gland ◽

Parathyroid Carcinoma ◽

Thyroid Lobe ◽

En Bloc ◽

Parathyroid Adenomas ◽

Preoperative Diagnostic ◽

Parathyroid Tumour ◽

Benign Tumours

Giant parathyroid adenomas are rare and underreported benign tumours of parathyroid gland. Preoperative differentiation between giant parathyroid adenoma (GPA) and parathyroid carcinoma can be challenging, as they both may present as a large parathyroid tumour with hyperparathyroidism. Very few GPAs had been described in the literature, with only 60 cases including our present case. We report a case of GPA and the preoperative diagnostic challenges we faced in differential diagnosis of a potential parathyroid malignancy. As a parathyroid malignancy could not be excluded preoperatively the parathyroid tumour was excised en bloc with surrounding thyroid lobe. The histology was contributory to exclude parathyroid malignancy confirming a benign GPA.

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