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CASE REPORT
Year : 2020  |  Volume : 35  |  Issue : 4  |  Page : 333-335  

Rare imaging findings of concomitant presence of multiple parathyroid adenomas and carcinoma in a chronic kidney disease patient with tertiary hyperparathyroidism detected on 99mTc-sestamibi single-photon-emission computed tomography/computed tomography scintigraphy


1 Department of Nuclear Medicine, PET/CT and Radionuclide Therapy, Kovai Medical Center and Hospital Limited, Coimbatore, Tamil Nadu, India
2 Department of Head and Neck Surgery, PET/CT and Radionuclide Therapy, Kovai Medical Center and Hospital Limited, Coimbatore, Tamil Nadu, India
3 Department of Head and Neck Surgery, Kovai Medical Center and Hospital Limited, Coimbatore, Tamil Nadu, India
4 Department of Nephrology, Kovai Medical Center and Hospital Limited, Coimbatore, Tamil Nadu, India

Date of Submission08-May-2020
Date of Decision18-May-2020
Date of Acceptance19-May-2020
Date of Web Publication21-Oct-2020

Correspondence Address:
Dr. Koramadai Karuppusamy Kamaleshwaran
Department of Nuclear Medicine, PET/CT and Radionuclide Therapy, Kovai Medical Center and Hospital Limited, Coimbatore - 641 014, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijnm.IJNM_100_20

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   Abstract 


Parathyroid carcinoma (PC) is a rare endocrine carcinoma. It is one of the rare causes for primary hyperparathyroidism. It is very uncommon in individuals with secondary or tertiary hyperparathyroidism. We report a rare case of 53-year-old male, a known case of chronic kidney disease on dialysis, who presented with fatigue and muscle cramps. Lab reports revealed elevated parathyroid hormone (PTH) and serum calcium levels. 99mTc-Sestamibi single-photon-emission computed tomography/computed tomography scintigraphy showed 4 foci of increased tracer uptake with retention, suggestive of parathyroid adenomas, including two ectopic foci in the mediastinum. At surgery, the enlarged bilateral superior, left inferior parathyroid gland, and two calcified left superior mediastinal lesions were removed. Histopathology revealed parathyroid adenomas in the right superior, left inferior, supernumerary ectopic left superior mediastinum lesion and PC in the left superior parathyroid lesion. Intraoperative serum PTH became normal, and the patient is asymptomatic now.

Keywords: 99mTc-sestamibi single-photon-emission computed tomography/computed tomography, parathyroid adenoma, parathyroid carcinoma, tertiary hyperparathyroidism


How to cite this article:
Malipedda S, Kamaleshwaran KK, Muthusamy D, Veerasamy M, Soundararajan AP, Jayaraj AV. Rare imaging findings of concomitant presence of multiple parathyroid adenomas and carcinoma in a chronic kidney disease patient with tertiary hyperparathyroidism detected on 99mTc-sestamibi single-photon-emission computed tomography/computed tomography scintigraphy. Indian J Nucl Med 2020;35:333-5

How to cite this URL:
Malipedda S, Kamaleshwaran KK, Muthusamy D, Veerasamy M, Soundararajan AP, Jayaraj AV. Rare imaging findings of concomitant presence of multiple parathyroid adenomas and carcinoma in a chronic kidney disease patient with tertiary hyperparathyroidism detected on 99mTc-sestamibi single-photon-emission computed tomography/computed tomography scintigraphy. Indian J Nucl Med [serial online] 2020 [cited 2020 Dec 5];35:333-5. Available from: https://www.ijnm.in/text.asp?2020/35/4/333/298732




   Introduction Top


Primary hyperparathyroidism (PHPT) is caused by autonomously functioning parathyroid adenoma. More than 85% of patients have single adenoma, <5% have two adenomas, <1% have four gland hyperplasia, and fewer than 1% of patients have parathyroid carcinoma (PC). Tertiary hyperparathyroidism (THPT) presents as hypercalcemia with increased parathyroid hormone (PTH) in chronic kidney disease (CKD) patients.[1] Here, one or more glands become autonomous and no longer respond to serum PTH.99m Tc-Sestamibi single-photon-emission computed tomography/computed tomography (SPECT/CT) is used for localizing hyperfunctioning parathyroid glands before surgery, making possible minimally invasive surgery.[2] It is very rare to have multiple parathyroid adenomas and PC in a patient with CKD and THPT.[3]


   Case Report Top


We report a case of 53-year-old male patient, a known case of CKD on hemodialysis for the last 5 years, who presented with the complaints of fatigability, bone pain, and muscular cramps. Laboratory report showed elevated serum calcium of 12.5 mg/dl (normal value 8.5–10.5 md/dl) and PTH of 3360 ng/L (normal value 15–65 ng/L), suggestive of HPT. Ultrasound (USG) neck done was suggestive of two hypoechoic nodules in the left lobe of thyroid measuring 1.6 cm × 1.1 cm and 1.3 cm × 1.1 cm with peripheral vascularity and right lobe of thyroid was not visualized due to dialysis line and dressing. No evidence of cervical lymph nodes noted. The patient was referred for 99m Tc-sestamibi Scintigraphy for localization of parathyroid adenoma. Dual-phase 99m Tc-sestamibi planar images showed multiple foci of tracer retention in relation to superior and inferior lobes of the thyroid gland and ectopic focus in mediastinum [Figure 1]a, early image, and b, delayed image]. SPECT/CT of the neck and mediastinum [Figure 2]a, [Figure 2]b, [Figure 2]c, [Figure 2]d, [Figure 2]e, [Figure 2]f, [Figure 2]g, [Figure 2]h localized hypodense nodular lesions with increased tracer retention in the following locations, superior part of the right (a) and left lobes of thyroid gland (b), left inferior part,(c) and ectopic calcified lesions in the left upper paratracheal mediastinal location in thoracic inlet (d). CT component of the SPECT/CT did not show invasion of parathyroid nodules into adjacent structures.
Figure 1: Dual-phase (early at 10 min [a], delayed at 1 h [b]) 99mTc-sestamibi planar images showed multiple foci of tracer retention in relation to the right superior, left superior, and left inferior lobes of the thyroid gland and ectopic focus in mediastinum

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Figure 2: Single-photon-emission computed tomography/computed tomography fused and computed tomography images of the neck and mediastinum (a-h) localized hypodense nodular lesions with increased tracer retention in the following locations: right superior (a and b, adenoma) and left superior (c and d, adenoma), left inferior (e and f, carcinoma) and calcified lesion in the left upper paratracheal location in the thoracic inlet (g and h, adenoma)

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The patient underwent neck and mediastinal exploration with parathyroidectomy. At surgery, the enlarged bilateral superior and left inferior parathyroid glands were removed. Calcified adenoma like lesion from left superior mediastinum was removed along with thyrothymic tissue. The right inferior parathyroid was located in the thymus and was confirmed as hyperplastic parathyroid on frozen section (which was not visualized on sestamibi) and was autotransplanted in left brachioradialis muscle. Intraoperative calcium done showed 7.5 mg/dl and PTH was 5.27 ng/dl.

Pathological examination of the right superior, left inferior, and ectopic left superior mediastinum lesions was suggestive of parathyroid adenomas. Left superior lesion showed parathyroid with cells showing focal mild anisonucleosis, rare mitosis, broad trabeculae, foci with capsular invasion, and extracapsular vascular invasion, suggestive of PC. A final diagnosis of three parathyroid adenomas with one in ectopic location and left superior PC was made. The patient is on follow-up and asymptomatic now for HPT.


   Discussion Top


Parathyroids are small lentiform glands that are normally located adjacent to the thyroid gland. Most people have two superior and two inferior parathyroid glands, however, there may be only three, or one or more supernumerary glands may be present. Variations in parathyroid gland locations are not only due to the glands' variable embryologic descent but also to the tendency of enlarged superior parathyroid glands to migrate posteriorly and inferiorly through fibroareolar tissue. Thus, localization of a parathyroid adenoma in three dimensions and differentiation of a superior parathyroid gland at the level of the inferior thyroid from an inferior parathyroid gland is important.[4]

PC is a rare malignancy, and it accounts for <1% of all cases of PHPT. The diagnosis becomes particularly challenging when there is no palpable neck mass. Palpable neck mass was seen in only 30%–76% of patients of PC, while <5% of patients with adenomas had a palpable mass in the neck.[5] Our patient did not have a palpable neck mass. The diagnosis of malignancy was based on a strong clinical suspicion, with elevated serum calcium (>14 mg/dl) level and highly elevated levels of serum PTH (>5 times the normal levels). PC may be associated with familial HPT, in patients of HPT jaw tumor syndrome or in patients with CKD. Our patient had high level of PTH, serum calcium and CKD, suggestive of THPT.[6]

PC may be associated with hyperplasia of other parathyroid glands in TPHT. However, it was rarely associated with adenoma of the other normal and ectopic supernumary parathyroid glands. The occurrence of PC in a case of TPHT is very rare. Berland et al. reported the first case of PC developing in a patient on hemodialysis.[7] Only 17 cases of PC in THPT in English literature were reported as mentioned by Khan et al.[8] Compared to different parathyroid imaging methods,99m Tc-sestamibi scintigraphy for preoperative localization was found to be superior than other imaging modalities. A meta-analysis of the medical literature reported that the overall sensitivity of dual-phase 99m Tc-sestamibi scintigraphy in comparison with high-resolution ultrasonography was 88% versus 78% for single adenomas, 30% versus 16% for double adenomas, and 44% versus 35% for multiple-gland hyperplasia.[9]

However, the role of a sestamibi scan in diagnosing PC in secondary or tertiary HPT is as yet not clear. Sestamibi can be used as an indicator of proliferative parathyroid gland activity. PC can be assumed to have higher proliferative activity than parathyroid hyperplasia. Therefore, when a lesion of PC coexists with other lesions of parathyroid hyperplasia, an increased uptake in the sestamibi scan is suggestive of PC.[10] In our patient, there is concomitant occurrence of multiple adenomas and PC associated with TPHT, which is a very rare presentation.


   Conclusion Top


PC is a rare cancer, and occurs rarely in patients with THPT and CKD. In a patient with highly elevated serum PTH, associated PC, which is a rare presentation, should be suspected.99m Tc SPECT/CT sestamibi scintigraphy is a very useful one-stop shop investigation for parathyroid adenoma or carcinoma which will give the exact anatomical location of adenoma with its CT component and help surgeons to locate and remove all lesions. Our case represents a very rare occurrence of multiple parathyroid adenomas in normally located parathyroid and ectopic location along with PC visualized in 99m Tc-Sestamibi scintigraphy.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Lunn MR, Mendoza JM, Pasche LJ, Norton JA, Ayco AL, Chertow GM. Hyperparathyroidism with hypercalcaemia in chronic kidney disease: Primary or tertiary? NDT Plus 2010;3:366-71.  Back to cited text no. 1
    
2.
Custódio MR, Montenegro F, Costa AF, dos Reis LM, Buchpiguel CA, Oliveira SG, et al. MIBI scintigraphy, indicators of cell proliferation and histology of parathyroid glands in uraemic patients. Nephrol Dial Transplant 2005;20:1898-903.  Back to cited text no. 2
    
3.
Yang J, Li Y, Li J, Yang P, Huang L. Hyperparathyroidism due to concurrent parathyroid carcinoma and parathyroid adenoma. Clin Nucl Med 2019;44:844-7.  Back to cited text no. 3
    
4.
Heideh KE, Harvey AZ. Parathyroid scintigraphy in patients with primary hyperparathyroidism:99m Tc Sestamibi SPECT and SPECT/CT. Radio Graph 2008;28:1461-76.  Back to cited text no. 4
    
5.
Fernandes JM, Paiva C, Correia R, Polónia J, da Costa AM. Parathyroid carcinoma: From a case report to a review of the literature. Int J Surg Case Rep 2018;42:214-7.  Back to cited text no. 5
    
6.
Kulkarni PS, Parikh PM. The carcinoma of parathyroid gland. Indian J Cancer 2004;41:51-9.  Back to cited text no. 6
[PUBMED]  [Full text]  
7.
Berland Y, Olmer M, Lebreuil G, Grisoli J. Parathyroid carcinoma, adenoma and hyperplasia in a case of chronic renal insufficiency on dialysis. Clin Nephrol 1982;18:154-8.  Back to cited text no. 7
    
8.
Khan MW, Worcester EM, Straus FH 2nd, Khan S, Staszak V, Kaplan EL. Parathyroid carcinoma in secondary and tertiary hyperparathyroidism. J Am Coll Surg 2004;199:312-9.  Back to cited text no. 8
    
9.
Moghadam RN, Amlelshahbaz AP, Namiranian N, Sobhan-Ardekani M, Emami-Meybodi M, Dehghan A, et al. Comparative diagnostic performance of ultrasonography and 99m Tc-sestamibi scintigraphy for parathyroid adenoma in primary hyperparathyroidism; systematic review and meta-analysis. Asian Pac J Cancer Prev 2017;28:3195-32.  Back to cited text no. 9
    
10.
Kim BS, Ryu HS, Kang KH, Park SJ. Parathyroid carcinoma in tertiary hyperparathyroidism. Asian J Surg 2016;39:255-9.  Back to cited text no. 10
    


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  [Figure 1], [Figure 2]



 

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