Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 8  |  Issue : 1  |  Page : 71-73

Normocalcemic hyperparathyroidism manifesting as brown tumor of mandible: A case report


Department of Oral Medicine and Radiology, College of Dental Sciences, Davangere, Karnataka, India

Date of Web Publication6-May-2016

Correspondence Address:
Dr Dhanya S Rao
Asst Professor, Department of Oral Medicine & Radiology, AJ Institute of Dental Sciences, Kuntikana, Mangalore 575004 Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0975-8844.181933

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  Abstract 

Patients with primary hyperparathyroidism (HPT) may be asymptomatic and clinical presentation of the tumor in the jaws can be the first sign of the disease. Only 2% of all primary HPT cases occur in patients below the age of 30 years. [1] Owing to the improved methods of blood analysis in the last two decades, most of the recent cases of primary HPT are diagnosed early and asymptomatically. [2] Most cases of primary HPT are identified by hypercalcemia and hypophosphatemia on routine multi-panel serum testing. [3] This makes advanced disease with brown tumor extremely rare these days, the mandible being affected only in 4% of the cases. [2] Here, we report a case of a 23-year-old female patient who presented with a swelling at body of mandible, with hypophosphatemia, increased parathyroid hormone, and normal serum calcium levels based on which a diagnosis of brown tumor was given.

Keywords: Brown tumor, hyperparathyroidism, medullary thyroid carcinoma


How to cite this article:
Rao DS, Kalappanavar AN, Annigeri RG. Normocalcemic hyperparathyroidism manifesting as brown tumor of mandible: A case report. J Orofac Sci 2016;8:71-3

How to cite this URL:
Rao DS, Kalappanavar AN, Annigeri RG. Normocalcemic hyperparathyroidism manifesting as brown tumor of mandible: A case report. J Orofac Sci [serial online] 2016 [cited 2019 Dec 10];8:71-3. Available from: http://www.jofs.in/text.asp?2016/8/1/71/181933


  Introduction Top


Brown tumors are nonneoplastic, reactive lesions resulting from excess osteoclastic activity, differentiated from other giant-cell tumors by the presence of hyperparathyroidism (HPT). [4] The incidence of brown tumor is higher in primary, compared to secondary HPT, and has been reported to be around 3%. Although mandibular involvement has been reported in 4.5% of subjects in a 220-case HPT study it is uncommon to find a brown tumor as the first manifestation of primary HPT before the onset of general manifestations. [5]


  Case Report Top


A 22-year-old female, was referred by her dentist to our department after a mandibular lesion was discovered on a panoramic radiograph. She presented with extra oral swelling along with intraoral mass rapidly growing since 2 months associated with pain since 15 days. Patient was under treatment for microcytic hypochromic anemia since 3 months.

Extra oral examination revealed a solitary diffuse swelling present of 2 cm × 3 cm, roughly oval in shape, in left lower third of face causing facial asymmetry. The color of the swelling was normal with smooth surface. No visible pulsations or other secondary changes were present. On palpation, the swelling was tender with firm to hard consistency. Borders were well defined and no loss or alteration in sensation was present. The swelling was nonfluctuant, noncompressible, nonreducible and no pulsations were felt [Figure 5].

Intraoral examination showed a well-defined swelling in the premolar molar region of the lower left alveolar mucosa extending from distal aspect of 33-38, involving the alveolar ridge, extending into both labial and lingual vestibule, thereby obliterating the vestibule. Mucosa over the swelling was stretched and surface was corrugated. No visible pulsations or other secondary changes were present. The swelling was tender with firm consistency and definitive borders with no bleeding on palpation. Mobility and displacement of teeth were noted in all teeth of involved region that is, 34-38. The nerve sensibility was not disturbed [Figure 1].
Figure 1: Intraoral appearance of the lesion


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Pulp vitality test showed delayed response in relation to (IRT) 34, 35, 36, and no response IRT 37 and 38.

Panoramic radiograph revealed multilocular radiolucency on the left side of body of mandible of 2 cm × 3 cm, with irregular borders, extending from first premolar to the third molar region. The lesion had caused resorption of mesial and distal roots of the first molars and mesial root of second molar as well as the second premolar. Cyst in cyst appearance was seen. Discontinuity of inferior alveolar nerve was noted. Furthermore, generalized loss of lamina dura was present [Figure 2] and [Figure 4].
Figure 2: Radiographic appearance of the lesion. Note the cyst in cyst appearance of the lesion and generalized loss of lamina dura in panoramic view


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Erosion of both buccal and lingual cortical plate was present in occlusal radiograph [Figure 3].
Figure 3: Radiographic appearance of the lesion


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Figure 4: Intraoral Intraoral periapical Radiographic appearance of the lesion appearance of the lesion


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Figure 5: Extraoral appearance


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Patient was diagnosed of brown tumor and was referred to the physician where blood analysis revealed increased alkaline phosphatase levels (762 IU/l), normal calcium (8.8 mg %), decreased phosphorous levels (1.99 mg/dl), and elevated intact parathyroid hormone (PTH) (452.5 pg/ml). There was no family history of HPT or other endocrine disorder. A biopsy specimen was obtained from the large well defined osteolytic lesion in the left side of the mandible. Histopathologically the lesion was identified as giant cell lesion. In light of the high PTH levels, a diagnosis of brown tumor secondary to primary HPT was made.

Ultrasonographic examination of the neck revealed a hyperechogenic mass on the left side mass suggestive of a parathyroid tumor but on (USG) guided fine-needle aspiration cytology cytopathological features were suggestive of medullary thyroid carcinoma.

Suspecting MEN's syndrome USG of the abdomen was done which showed normal structures thereby ruling out men's. Patient is currently under treatment for the thyroid carcinoma.


  Discussion Top


Brown tumor is a reactive lesion that arises due to excessive osteoclastic activity considered a reparative cellular process rather than a true neoplasm, [3] the incidence of which is around 3% in primary HPT and 1.2-1.7% in secondary. [6]

The ribs, clavicles, pelvic girdle, and mandible are the most commonly involved bones. [3] In HPT, due to increased osteoclastic activity osteopenia and subperiosteal bone resorption which leads to reactive osteoblastic activity resulting in the ingrowth of fibrovascular tissue in the marrow spaces. Microfractures and hemorrhage causes the influx of multinucleated macrophages and this repeated hemorrhage leads to hemosiderin deposition within the osteolytic cysts giving the characteristic brown color of the lesion. [6]

Histologically, brown tumor presents as masses of soft tissue composed of giant cells in a fibrovascular stroma, cyst like spaces lined by connective tissue, and foci of hemorrhage, changes that may be related to microfractures undergoing organization with the release of hemosiderin. [2]

Perhaps because of this normality in calcium levels, the patient presented no general signs and symptoms associated with primary HPT such as gastrointestinal and neurologic disorders, polyuria, polydipsia, nephrolithiasis, nephrocalcinosis, bone pain, arterial hypertension or lethargy. [4]

Treatment involves control of HPT and for primary HPT, partial disfiguring and symptomatic lesions, excision may be indicated. [4]


  Conclusion Top


We may now be entering a 3 rd era in the history of this disease in which patients are being discovered with normal total and ionized serum calcium concentrations but with PTH levels that are consistently elevated. [7] Hence, in young female patients who present with jaw lesions it becomes mandatory to suspect HPT as one of the differential diagnosis and evaluate the PTH level. Evidently, right and early diagnosis is the key for fruitful treatment.

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.

 
  References Top

1.
Daniels JS. Primary hyperparathyroidism presenting as a palatal brown tumor. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004;98:409-13.  Back to cited text no. 1
    
2.
Keyser JS, Postma GN. Brown tumor of the mandible. Am J Otolaryngol 1996;17:407-10.  Back to cited text no. 2
    
3.
Triantafillidou K, Zouloumis L, Karakinaris G, Kalimeras E, Iordanidis F. Brown tumors of the jaws associated with primary or secondary hyperparathyroidism. A clinical study and review of the literature. Am J Otolaryngol 2006;27:281-6.  Back to cited text no. 3
    
4.
Suarez-Cunqueiro MM, Schoen R, Kersten A, Klisch J, Schmelzeisen R. Brown tumor of the mandible as first manifestation of atypical parathyroid adenoma. J Oral Maxillofac Surg 2004;62:1024-8.  Back to cited text no. 4
    
5.
Alhusban M, Baqain ZH. Mandibular brown tumor as the first manifestation of primary hyperparathyroidism: A case report. Saudi Dent J 2011;23:107-9.  Back to cited text no. 5
    
6.
Ergen FB, Ayvaz M, Yildiz AE, Aydingoz U, Gedikoglu G. Brown tumour presenting as a soft-tissue mass. Clin Radiol 2012;67:286-9.  Back to cited text no. 6
    
7.
Bilezikian JP, Silverberg SJ. Normocalcemic primary hyperparathyroidism. Arq Bras Endocrinol Metabol 2010;54:106-9.  Back to cited text no. 7
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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Abstract
Introduction
Case Report
Discussion
Conclusion
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