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CASE REPORT
Year : 2020  |  Volume : 19  |  Issue : 2  |  Page : 149-151

An unusual case of hyperparathyroidism, presenting as multiple skeletal lesions


1 Department of Nuclear Medicine and PET/CT, Mahajan Imaging Centre, Sir Ganga Ram Hospital, New Delhi, India
2 Department of Nuclear Medicine and PET/CT, Manipal Hospital, New Delhi, India
3 Department of Pathology, Sir Ganga Ram Hospital, New Delhi, India

Date of Submission26-Aug-2019
Date of Acceptance15-Oct-2019
Date of Web Publication14-Jan-2020

Correspondence Address:
Dr. Nitin Gupta
Department of Nuclear Medicine and PET/CT, Mahajan Imaging Centre, Sir Ganga Ram Hospital, New Delhi - 110 060
India
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DOI: 10.4103/wjnm.WJNM_69_19

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   Abstract 


We report the case of a 43-year-old male patient, who presented with severe osteoporosis and pain at multiple sites and a chest X-ray suggestive of few cystic lesions in bilateral ribs. Subsequently, the patient was referred for bone scan, in view of the suspicion of polyostotic fibrous dysplasia or neoplastic lesions. Bone scan showed homogenously increased tracer uptake in the axial and appendicular skeletal system, suggestive of metabolic superscan. Computed tomography neck localized right inferior parathyroid adenoma and expansile lytic skeletal lesions suggestive of brown tumors were also seen. This case highlights the importance of bone scan, helping in the differential diagnosis of multiple skeletal lesions.

Keywords: Brown tumors, hyperparathyroidism, Methylene diphosphonate (MDP) bone scan, skeletal lesions


How to cite this article:
Gupta N, Pruthi A, Verma R, Manocha A. An unusual case of hyperparathyroidism, presenting as multiple skeletal lesions. World J Nucl Med 2020;19:149-51

How to cite this URL:
Gupta N, Pruthi A, Verma R, Manocha A. An unusual case of hyperparathyroidism, presenting as multiple skeletal lesions. World J Nucl Med [serial online] 2020 [cited 2020 Jul 4];19:149-51. Available from: http://www.wjnm.org/text.asp?2020/19/2/149/275849




   Introduction Top


Majority of the hyperparathyroidism cases are diagnosed when a routine assay shows hypercalcemia. Symptoms include recurrent renal stones, asthenia, neuromuscular or psychiatric disorders, gastrointestinal manifestations, or osteoporosis. Brown tumors are rare nonneoplastic lesions that arise in the setting of hyperparathyroidism. They can arise as solitary or multiple lesions of any bone, more common in the extremities, clavicle, ribs, and pelvis. We report an unusual case in which a patient presented with multiple skeletal lesions and bone scan and further investigations helped in the diagnosis.


   Case Report Top


A 43-year-old male patient who presented with pain at multiple sites was referred for whole-body bone scan with suspicion of polyostotic fibrous dysplasia, other differentials being multiple myeloma or metastatic lesions. Whole-body bone scan, anterior and posterior views [Figure 1]a and [Figure 1]b, was suggestive of homogenously increased tracer uptake in the axial and appendicular skeleton system with faint visualization of the kidneys, likely suggestive of metabolic superscan. Since chest X-ray revealed cystic lesions, and relevant biochemical parameters were raised serum calcium (12.8 mg/dl, range: 8.6–10.2 mg/dl), raised alkaline phosphatase (592 IU/L, range: 20–140 IU/L), and low inorganic phosphorus (1.8 mg/dl, range: 2.5–4.5 mg/dl), suspicion of parathyroid adenoma was raised. Noncontrast computed tomography neck, transaxial and coronal sections [Figure 1]c and [Figure 1]e localized large hypodense nodular lesion in relation to the inferior pole of the right lobe of the thyroid gland, and bone window transaxial and coronal sections [Figure 1]d and [Figure 1]f revealed expansile lytic lesions involving the ribs and scapula. On further blood test, serum parathyroid hormone was ~1700 pg/ml (range: 10–65 pg/ml), correlating with other findings and confirming diagnosis of primary hyperparathyroidism with multiple skeletal lesions. Subsequently, the lesion was excised, and histopathology report [Figure 2] confirmed parathyroid adenoma.
Figure 1: Whole-body bone scan, anterior and posterior views (a and b), suggestive of homogenously increased tracer uptake in the axial and appendicular skeleton system with faint visualization of the kidneys. Noncontrast computed tomography neck, transaxial and coronal sections (c and e), localized large hypodense nodular lesion in relation to the inferior pole of the right lobe of the thyroid gland, and bone window transaxial and coronal sections (d and f) revealed expansile lytic lesions involving the ribs and scapula

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Figure 2: Histopathology images of the specimen. Section (a) shows an encapsulated tumor composed of cells arranged in nests separated by thin-walled vasculature (H and E, ×4). Section (b) shows an encapsulated tumor composed of cells arranged in nests separated by thin-walled vasculature (H and E, ×10). The adenoma cells show (c) monomorphic nuclei with stippled chromatin, inconspicuous nucleoli, and moderate amount of clear-to-pale eosinophilic cytoplasm (H and E, ×20)

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   Discussion Top


Primary hyperparathyroidism is a disease characterized by excessive secretion of parathormone. Multiple skeletal lesions (brown tumors) represent a rare clinical manifestation of prolonged hyperparathyroidism and reported in approximately 3% of patients with primary hyperparathyroidism.[1] Differential diagnosis of multiple bony lesions include metastatic lesions, bone cysts, osteosarcoma, and giant-cell tumor. There are various reports of brown tumors imitating skeletal metastases.[2] Although homogeneously increased tracer uptake in bone scan, with greater uptake in the appendicular skeleton compared to axial skeleton and faint visualization of kidneys, raises suspicion of metabolic superscan and in presence of raised calcium, primary hyperparathyroidism appears more likely with less chances of malignancy, but few cases of secondary hyperparathyroidism have been reported in prostate cancer patients with bone metastases.[3] However, there was no known history of carcinoma prostate in our patient, and serum prostate-specific antigen levels were also normal (0.5 ng/ml, range: 0.2–4.0 ng/ml); later, histopathology report of parathyroid lesion confirmed parathyroid adenoma in the patient. Few cases have been reported in literature, where multiple skeletal lesions were later diagnosed due to hyperparathyroidism.[2],[4],[5],[6],[7] The classical radiographic features of skeletal lesions (brown tumors) in hyperparathyroidism are osteopenia and diffuse subperiosteal bone resorption.[8] Thus, in case of hypercalcemia and multiple lytic lesions, primary hyperparathyroidism should always be kept in differential diagnosis, and further biochemical evaluation and bone scan can help in management.

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.
Azria A, Beaudreuil J, Juquel JP, Quillard A, Bardin T. Brown tumor of the spine revealing secondary hyperparathyroidism. Report of a case. Joint Bone Spine 2000;67:230-3.  Back to cited text no. 1
    
2.
Ullah E, Ahmad M, Ali SA, Redhu N. Primary hyperparathyroidism having multiple brown tumors mimicking malignancy. Indian J Endocrinol Metab 2012;16:1040-2.  Back to cited text no. 2
    
3.
Berruti A, Cook R, Saad F, Buttigliero C, Lipton A, Tampellini M, et al. Prognostic role of serum parathyroid hormone levels in advanced prostate cancer patients undergoing zoledronic acid administration. Oncologist 2012;17:645-52.  Back to cited text no. 3
    
4.
Yang Q, Sun P, Li J, Yang Z, Li X, Li Z, et al. Skeletal lesions in primary hyperparathyroidism. Am J Med Sci 2015;349:321-7.  Back to cited text no. 4
    
5.
Sager S, Aliyev A, Halac M, Oztürk T. Positron emission tomography/computed tomography imaging of Brown tumors mimicking multiple skeletal metastases in patient with primary hyperparathyroidism. Indian J Endocrinol Metab 2012;16:850-2.  Back to cited text no. 5
    
6.
Misiorowski W, Czajka-Oraniec I, Kochman M, Zgliczyński W, Bilezikian JP. Osteitis fibrosa cystica-a forgotten radiological feature of primary hyperparathyroidism. Endocrine 2017;58:380-5.  Back to cited text no. 6
    
7.
Jouan A, Zabraniecki L, Vincent V, Poix E, Fournié B. An unusual presentation of primary hyperparathyroidism: Severe hypercalcemia and multiple brown tumors. Joint Bone Spine 2008;75:209-11.  Back to cited text no. 7
    
8.
Silverberg SJ, Bilezikian JP. Primary hyperparathyroidism: Still evolving? J Bone Miner Res 1997;12:856-62.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2]



 

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