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

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 ◽  
...  

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.

2021 ◽  
Vol 64 (2) ◽  
pp. 38-45
Author(s):  
Isaías Adrián Barrientos López ◽  
◽  
Bertha Beatriz Castillo Ventura ◽  

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.


2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Craig Jenkins ◽  
Matthew Kemm ◽  
Lydia Jenkins

Abstract Background: More than 95% of patients with primary hyperparathyroidism will be cured with the initial operation by an experienced surgeon. However, localization of hyperparathyroid recurrences, especially after extensive surgery becomes challenging. For patients with transplanted parathyroid glands into the forearm, there may be utility in bilateral arm serum PTH testing to help with localization. Clinical Case: A 65-year-old woman presented to the clinic with primary hyperparathyroidism in 2008. After a localization study, she had a partial parathyroidectomy but continued to have persistent biochemical hyperparathyroidism despite negative localization studies. She was then referred to another institution for further studies. Follow up Sestamibi scans were negative but 4D-CT scans assisted in localizing the presence of a superior parathyroid gland adenoma which was later removed in 2011. During this time, the left inferior parathyroid gland was auto-transplanted into the left forearm. Again, her calcium and PTH levels rose despite negative Sestamibi scans showing no abnormalities in post-operative beds or in the forearm. Review of previous labs revealed elevated PTH levels in the ranges of 80-110 pg/mL since 2012-2019. The patient’s most recent PTH was 2408 pg/mL. At that point, the decision was made to repeat the labs on the left and right forearms simultaneously and labs showed PTH levels of 1283 pg/mL and 118 pg/mL, respectively. Repeat Sestamibi scan following these labs demonstrated evidence of increased radiotracer uptake in the region of the prior transplanted parathyroid tissue with no neck uptake concerning for hyperparathyroidism due to auto-transplanted hyperplastic tissue. Conclusion: This case demonstrates the utility of bilateral arm serum PTH testing in the evaluation of recurrent hyperparathyroidism in patient’s status-post parathyroid auto-transplantation.


2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A170-A171
Author(s):  
Maria Nikki Castillo Cruz ◽  
Celeste Ong Ramos

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.


2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Nitish Singh Nandu ◽  
Janice L Gilden

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.


2007 ◽  
Vol 92 (5) ◽  
pp. 1785-1790 ◽  
Author(s):  
Hiroya Takeuchi ◽  
Nancy C. Greep ◽  
Dave S. B. Hoon ◽  
Armando E. Giuliano ◽  
Nora M. Hansen ◽  
...  

Abstract Context: Retention of technetium-99m-sestamibi (99mTc-sestamibi) by parathyroid adenomas appears to be due to the loss of at least one membrane transporter, multidrug resistance 1 (MDR1), and possibly another, multidrug resistance-associated protein 1 (MRP1). Objective: The objective was to determine whether hypermethylation of either gene plays a role in their expression and 99mTc-sestamibi retention. Design: This was a retrospective study on a convenience sample of paraffin-embedded parathyroid glands. Setting: The study was performed at the John Wayne Cancer Institute at Saint John’s Health Center (Santa Monica, CA). Patients: Forty-eight patients with primary hyperparathyroidism and five patients without parathyroid disease undergoing thyroid surgery provided 27 adenomatous, 10 hyperplastic, and 16 normal parathyroid glands. Intervention: We performed immunohistochemistry, real-time quantitative RT-PCR, and methylation-specific PCR for MDR1 and MRP1 on archival parathyroid tissue and correlated these results with the patient’s 99mTc-sestamibi scan. Main Outcome Measure: The main outcome measure was to determine whether hypermethylation of the genes for either transporter is associated with loss of their expression and with a positive 99mTc-sestamibi scan. Results: The MDR1 gene was methylated in none of 12 normal glands, 19 of 27 adenomas, and three of 10 hyperplastic glands. Methylation of the MRP1 gene was uncommon (five of 48 tested glands). Methylation of the gene affected the transcript level only for MDR1. Among all glands, hypermethylation for MDR1 was more likely in 99mTc-sestamibi-positive scans (P < 0.001). Conclusion: In parathyroid tissue, hypermethylation of the MDR1 gene decreases its expression and is associated with increased detection of parathyroid adenomas by 99mTc-sestamibi parathyroid scans.


2011 ◽  
Vol 3 (3) ◽  
pp. 128-130
Author(s):  
Ana Karena Neukirch ◽  
Christian Heckmann ◽  
Norbert Weyerbrock ◽  
Silke Günther ◽  
Cornelia Dotzenrath

ABSTRACT A 56-year-old female patient with the phenotype of Albright's hereditary osteodystrophy (AHO) and pseudohypoparathyroidism 1a (PHP) diagnosed in 1987 was shown to have a heterozygote inactivating mutation on the GNAS1 gene. The patient has been treated with oral calcium and vitamin D since diagnosis of PHP 1a and developed primary hyperparathyroidism (pHPT) in 2009. Ultrasound as well as 99Tcsestamibi could demonstrate a lesion in the right lower position. Intraoperatively, a solitary parathyroid adenoma was found and resected leading to a ‘normalization’ of PTH and calcium. This case highlight is the rare coincidence of PHP, AHO and pHPT.


2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Maria Alexandra Carranceja Villapol ◽  
Maria Princess L Kanapi

Abstract Introduction: This is the case of a pregnant woman on her 3rd trimester who was diagnosed with primary hyperparathyroidism. Since there are two patients involved, the potential complications that can be brought about by the diagnostic tests and the treatment had to be weighed against the benefits. Case: The patient is SA, a 34-year old female on her 29th week of pregnancy, admitted due to a month history of abdominal pain described as intermittent, crampy, generalized, non-radiating, and mild-to-moderately severe in intensity. She was advised to do tests but did not comply. In the interim, there was recurrence of symptoms but with resolution. However, the crampy abdominal pain recurred, now localized at the right upper quadrant and epigastric areas, radiating to the right upper back, moderate in intensity, and with associated nausea and vomiting, leading to admission. She was first managed under OB-Gynecology, given hydration, pain management and Betamethasone. She was also referred to Cardiology for blood pressure control, and Surgery for evaluation of the abdominal pain. Due to an increasing trend of her blood glucose, she was referred to Endocrinology and started on insulin. Mild bilateral nephrocalcinoses seen in an abdominal ultrasound prompted work-up showing an elevated serum ionized calcium at 1.88 meq/L (n 1.12-1.32 meq/L), elevated intact PTH at 451.13 pg/ml (n <67.9 pg/ml), and low Vitamin D at 10.96 ng/ml (n >30ng/ml). Parathyroid ultrasound showed nonthyroidal tissue measuring 0.4 x 0.6 cm at the right inferior area. Saline hydration and diuresis with Furosemide were started to manage the hypercalcemia. A multi-disciplinary meeting was held to discuss the options for management and risks involved. The goal was to deliver the baby in stable condition possibly to term, while keeping maternal calcium levels and blood pressure normal. However on her 30th week of gestation, she had persistent elevated blood pressure and underwent emergency caesarian section. After delivery, the patient was advised against breastfeeding for adequate management of her hypercalcemia. She was started on Cinacalcet, Calcitonin, and Ibandronic Acid. A Parathyroid Sestamibi Scan done showed a parathyroid adenoma on the right inferior lobe, and she underwent right inferior parathyroidectomy, with left thyroidectomy and isthmusectomy. Findings showed a right inferior parathyroid adenoma and a benign follicular nodule on the left thyroid. She was started on Calcium Carbonate and Calcitriol, and discharged stable. Conclusions: This case shows that when two lives are at stake every step of the management, whether diagnostic or therapeutic, must be communicated well to the patient and to the other members of the team. It is ultimately a choice made by the expectant mother but through the proper guidance and updated knowledge of the team, combined with a good clinical eye especially in the treatment of pregnant women.


2018 ◽  
Vol 2018 ◽  
pp. 1-4
Author(s):  
Fernando Mendoza-Moreno ◽  
Ángel Rodriguez-Pascual ◽  
María Rocío Díez-Gago ◽  
Marina Pérez-González ◽  
Laura Jiménez‐Alvárez ◽  
...  

Introduction. The variability of the location of the parathyroid glands is directly related to the events that occur during embryonic development. The impact that an individual submits more than four parathyroid glands is close to 13%. However the presentation of a parathyroid adenoma in a supernumerary gland is an uncommon event. Case report. A 30-year-old man diagnosed with primary hyperparathyroidism with matching findings on ultrasonography and scintigraphy for parathyroid adenoma localization lower left regarding the thyroid gland. A cervicotomy explorer showed four orthotopic parathyroid glands. The biopsy of the inferior left gland was normal. No signs of adenoma were seen in the biopsy. Following mobilization of the ipsilateral thyroid lobe, fifth parathyroid gland was found increased significantly in size than proceeded to remove, confirming the diagnosis of adenoma. After the excision, the levels of serum calcium and parathyroid hormone were normalized. Conclusions. The presentation of a parathyroid adenoma in a supernumerary gland is a challenge for the surgeon. The high sensitivity having different imaging techniques has been a key to locate preoperatively the pathological parathyroid gland. Analytical or clinical persistence of primary hyperparathyroidism after parathyroid surgery can occur if the location of the adenoma is a supernumerary or ectopic gland location.


2010 ◽  
Vol 2 (3) ◽  
pp. 131-133
Author(s):  
Geoffrey B Thompson ◽  
Benzon M Dy ◽  
Bianca Vazquez ◽  
Peter J Tebben ◽  
Seema Kumar

ABSTRACT Introduction Severe hypercalcemia due to primary hyperparathyroidism (PHPT) is rare in the setting of MEN 2A. Materials and methods Two patients with MEN 2A and severe hypercalcemia were identified recently. Their clinical presentation, evaluation, surgical management and outcomes are reviewed. Results Two patients with MEN 2A were identified with severe hypercalcemia secondary to a parathyroid adenoma. Calcium levels were elevated to 12.7 mg/dL and 15.1 mg/dL, respectively (normal range = 8.9-10.1 mg/dL). In each case, a single parathyroid adenoma was identified and surgically excised with normalization of parathyroid and calcium levels postoperatively. Clinical manifestations at the time of diagnosis included constipation, polyuria, hypercalciuria, and decreased bone mineral density. Conclusion Severe elevation of serum calcium is a rare presentation of PHPT in MEN2A. The differential diagnosis should include parathyroid adenoma, hyperplasia and parathyroid carcinoma. Early surgical management is essential in the treatment of hyperparathyroidism with severe hypercalcemia to prevent further complications.


2011 ◽  
Vol 135 (12) ◽  
pp. 1521-1521

An abstract published in the September 2011 issue of the Archives (Murugan P et al. Tumor-to-Tumor Metastasis: A Rare Case of Cutaneous Melanoma Metastatic to a Parathyroid Adenoma [CAP abstract 109, session 100]. Arch Pathol Lab Med. 2011;135[9]:1132) contains incorrect data in line 10 when referring to the right inferior parathyroid gland that was removed. The weight of the gland should have been shown as “…1200-mg (normal, 30–70 mg)…”


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