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Table of Contents
CASE REPORT
Year : 2021  |  Volume : 23  |  Issue : 1  |  Page : 47-49

Giant parathyroid adenoma presenting as neck swelling: A rare case report


1 Department of Otorhinolaryngology and Head and Neck Surgery, AIIMS, Bathinda, Punjab, India
2 Department of Otorhinolaryngology and Head and Neck Surgery, Government Medical College and Hospital, Chandigarh, India

Date of Submission08-Jun-2020
Date of Decision25-Aug-2020
Date of Acceptance23-Oct-2020
Date of Web Publication06-May-2021

Correspondence Address:
Dr. Amanjot Kaur
Department of Otorhinolaryngology and Head and Neck Surgery, AIIMS, Bathinda, Punjab
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/SJOH.SJOH_28_20

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  Abstract 


The normal weight of parathyroid gland lies between 50 and 70 mg. Any adenoma weighing more than 3.5 g is labeled giant parathyroid adenoma. It forms a distinct clinical entity in terms of difficulty in differentiation from parathyroid carcinoma and more aggressive postoperative management due to higher chances of postoperative hypocalcemia. We present the case report of a 35-year-old woman presenting with multiple fractures and neck swelling which was diagnosed as giant parathyroid adenoma and managed by parathyroidectomy.

Keywords: Giant parathyroid adenoma, hypocalcemia, hypoparathyroidism, parathyroidectomy


How to cite this article:
Saini V, Gupta N, Gupta V, Kaur A, Kaur G. Giant parathyroid adenoma presenting as neck swelling: A rare case report. Saudi J Otorhinolaryngol Head Neck Surg 2021;23:47-9

How to cite this URL:
Saini V, Gupta N, Gupta V, Kaur A, Kaur G. Giant parathyroid adenoma presenting as neck swelling: A rare case report. Saudi J Otorhinolaryngol Head Neck Surg [serial online] 2021 [cited 2021 Jun 25];23:47-9. Available from: https://www.sjohns.org/text.asp?2021/23/1/47/315573




  Introduction Top


The normal weight of parathyroid gland is usually between 50 and 70 mg. Parathyroid adenomas, which are the leading cause of primary hyperparathyroidism (HRPT) in 85% of cases,[1] usually weigh <1 g.[2] Giant parathyroid adenomas have rarely been reported with weight more than 3.5 g.[3] Primary HRPT is the third most common endocrine disorder[4] with a male:female ratio of 4:1 and a peak in the fifth decade of life.[5] Only 5% of adenomas were found to weigh more than 3.5 g in a series of 300 cases of primary HRPT.[3]

To the best of our knowledge, this case report is the second case report of giant parathyroid adenoma in the Middle East presenting as visible neck swelling.[6]


  Case Report Top


A 35-year-old woman presented to the orthopedic department with chief complaints of multiple fractures and generalized fatigue. On examination, visible neck swelling was noted [Figure 1]. Her serum calcium was 3.2 mmol/L. There were no features suggestive of hyper- or hypothyroidism, dysphagia, or stridor. There was no history of constipation or diarrhea. She had no significant medical or family history.
Figure 1: 4 cm × 5 cm neck swelling

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On further evaluation, laboratory reports showed an elevated parathyroid hormone (PTH) level of 1450 pg/ml, Vitamin D was 34 nmol/L, and alkaline phosphatase level was 144 IU/L. Her thyroid-stimulating hormone and renal function tests were within normal limits. Twenty-four-hour urine calcium was 4.28 mmol/L per 24 h. Her complete blood count was within normal limits.

On examination, there was a firm, painless swelling in the lower pole of her left thyroid lobe with no palpable lymphadenopathy. Ultrasound of the neck showed a complex nodule (4.5 cm × 5 cm) in the lower pole of her left thyroid lobe. Computed tomography neck was suggestive of a hyperdense nodule measuring 5 cm × 5 cm in the lower pole of the left lobe of the thyroid gland with no airway compromise [Figure 2]. The parathyroid sestamibi scan was not done as the patient was bedridden and had a poor general condition. Differential diagnoses included parathyroid adenoma, parathyroid hyperplasia, and carcinoma. After adjusting the serum calcium of the patient, targeted parathyroidectomy was done, and 5 cm × 4 cm parathyroid adenoma was taken out [Figure 3] and [Figure 4]. The intraoperative frozen section confirmed the parathyroid tissue. Intraoperative PTH was not done as the facility was not available in our institute. Histopathological examination confirmed a parathyroid adenoma. Postoperative serum PTH on day 2 was 65 pg/ml, and on day 5, it came down to 21 pg/ml.
Figure 2: Hyperdense nodule measuring 5 cm × 5 cm in the lower pole of the left lobe of the thyroid gland

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Figure 3: Intraoperative parathyroid adenoma

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Figure 4: Removed parathyroid adenoma

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The postoperative course was uneventful. She was referred back to the orthopedic department on oral calcium and Vitamin D supplements for the management of fractures. She is currently normocalcemic and asymptomatic.


  Discussion Top


The median estimated weight of parathyroid adenomas excised from patients with primary HRPT is found to be 650 mg.[3] A retrospective review of 300 cases found that patients with giant parathyroid adenomas were found to have higher mean preoperative calcium and PTH levels.[3] Calva-Cerqueira et al. concluded that if preoperative PTH is >232 ng/L, there is a 95% likelihood of finding a physical therapist assistant weighing >250 mg.[7] However, they were also associated with higher rates of postoperative hypocalcemia.

In the absence of invasion of adjacent structures, the differentiation between giant parathyroid adenoma and parathyroid carcinoma becomes a diagnostic challenge. However, hypercalcemia symptoms are more severe in parathyroid carcinoma, which was the case in our patients who presented with multiple bone fractures. On ultrasonography, both present as hypoechoic masses, but parathyroid carcinomas have a heterogeneous appearance, whereas granulomatosis with polyangiitis (GPA) has a homogeneous appearance with smooth borders. The depth/width ratio is more significant than 1 in 95% of cases of PCs and smaller than 1 in 94% of GPAs.[8]

The localization of GPA is imperative before proceeding with surgery. A combination of neck ultrasound and 99mTc-sestamibi scintigraphy scan is used most commonly. The limitation of neck ultrasound is that it will not show the extent of the lesion and will completely miss the lesion in case of the ectopic gland.[1] Patients with GPA have a single-gland disease in the majority of the cases.[9]

Genetically, GPAs are almost similar to the usual parathyroid adenomas with regard to MEN1 mutation and infrequently HRPT2 gene mutation. They are more akin to carcinoma in terms of parafibromin expression and gain of chromosome 5.[9]

The management of parathyroid adenomas is according to the guidelines by the National Institutes of Health US in 2014.[10] Our case met the surgical criteria and was treated with parathyroidectomy.

The hungry bone syndrome refers to a severe decline in serum calcium following parathyroidectomy due to the abrupt withdrawal of PTH in patients with severe HRPT. The incidence of hungry bone syndrome following parathyroidectomy was 13% in one case series.[11] The size of the gland is a more predictive factor of HBS than other factors such as old age, alkaline phosphatase values, and blood urea nitrogen values. Treatment involves oral or intravenous calcium and magnesium supplementation. Prophylactic use of preoperative bisphosphonates and Vitamin D supplementation is currently under study.[12]

In our case, the weight of the adenoma was 6.5 g. Although higher weight is associated with more chances of postoperative hypocalcemia, our patient did well with oral calcium and Vitamin D supplements.

Gamma probe is available in some centers to detect any other hyperfunctioning parathyroid tissue.

Intraoperative nerve monitoring reduces the risk of recurrent laryngeal nerve injury during parathyroidectomy, especially among new generation of head-and-neck surgeons.[13]


  Conclusion Top


Giant parathyroid adenoma presents with a neck mass and features of HRPT, i.e., painful bones, abdominal groans, renal stones, and psychic moans. Extremely elevated serum calcium levels are more suggestive of carcinoma or parathyroid carcinoma than parathyroid adenoma. Hungry bone syndrome is a serious postoperative complication that needs aggressive management. Accurate diagnosis and early management are imperative in case of GPA to minimize the risk of pre- and postoperative complications.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Garas G, Poulasouchidou M, Dimoulas A, Hytiroglou P, Kita M, Zacharakis E. Radiological considerations and surgical planning in the treatment of giant parathyroid adenomas. Ann R Coll Surg Engl 2015;97:e64-6.  Back to cited text no. 1
    
2.
Neagoe RM, Sala DT, Borda A, Mogoantă CA, Műhlfay G. Clinicopathologic and therapeutic aspects of giant parathyroid adenomas - three case reports and short review of the literature. Rom J Morphol Embryol 2014;55:669-74.  Back to cited text no. 2
    
3.
Spanheimer PM, Stoltze AJ, Howe JR, Sugg SL, Lal G, Weigel RJ. Do giant parathyroid adenomas represent a distinct clinical entity? Surgery 2013;154:714-8.  Back to cited text no. 3
    
4.
Madkhali T, Alhefdhi A, Chen H, Elfenbein D. Primary hyperparathyroidism. Ulus Cerrahi Derg 2016;32:58-66.  Back to cited text no. 4
    
5.
Fraser WD. Hyperparathyroidism. Lancet 2009;374:145-58.  Back to cited text no. 5
    
6.
Al-Hassan MS, Mekhaimar M, El Ansari W, Darweesh A, Abdelaal A. Giant parathyroid adenoma: A case report and review of the literature. J Med Case Rep 2019;13:332.  Back to cited text no. 6
    
7.
Calva-Cerqueira D, Smith BJ, Hostetler ML, Lal G, Menda Y, O'Dorisio TM, et al. Minimally invasive parathyroidectomy and preoperative MIBI scans: Correlation of gland weight and preoperative PTH. J Am Coll Surg 2007;205:S38-44.  Back to cited text no. 7
    
8.
Hara H, Igarashi A, Yano Y, Yashiro T, Ueno E, Aiyoshi Y, et al. Ultrasonographic features of parathyroid carcinoma. Endocr J 2001;48:213-7.  Back to cited text no. 8
    
9.
Sulaiman L, Nilsson IL, Juhlin CC, Haglund F, Höög A, Larsson C, et al. Genetic characterization of large parathyroid adenomas. Endocr Relat Cancer 2012;19:389-407.  Back to cited text no. 9
    
10.
Bilezikian JP, Brandi ML, Eastell R, Silverberg SJ, Udelsman R, Marcocci C, et al. Guidelines for the management of asymptomatic primary hyperparathyroidism: Summary statement from the Fourth International Workshop. J Clin Endocrinol Metab 2014;99:3561-9.  Back to cited text no. 10
    
11.
Brasier AR, Nussbaum SR. Hungry bone syndrome: Clinical and biochemical predictors of its occurrence after parathyroid surgery. Am J Med 1988;84:654-60.  Back to cited text no. 11
    
12.
Witteveen JE, van Thiel S, Romijn JA, Hamdy NA. Hungry bone syndrome: Still a challenge in the post-operative management of primary hyperparathyroidism: A systematic review of the literature. Eur J Endocrinol 2013;168:R45-53.  Back to cited text no. 12
    
13.
Ghani U, Assad S, Assad S. Role of intraoperative nerve monitoring during parathyroidectomy to prevent recurrent laryngeal nerve injury. Cureus 2016;8:e880.v  Back to cited text no. 13
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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