|Year : 2015 | Volume
| Issue : 1 | Page : 77-79
Maxillary tumour as first sign of endocrine abnormality: A report of a rare case
Prasanth Thankappan, Naga Sirisha Chundru, Rajesh Amudala, Anitha Kuppusamy
Department of Oral Pathology, Chadalawada Krishnamurthy Srinivasa Theja Institute of Dental Sciences and Research, Tirupathi, Andhra Pradesh, India
|Date of Web Publication||13-Jan-2015|
Department of Oral Pathology, CKS Theja Institute of Dental Sciences and Research, Chadalawada Nagar, Renigunta Road, Tirupathi - 517 506, Andhra Pradesh
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Brown tumour is a non-neoplastic giant cell lesion resulting as a complication of hyperparathyroidism (HPT). HPT usually results from increased secretion of parathyroid hormone (PTH). We present a rare case of brown tumour of anterior maxilla presenting as a first sign of previously undiagnosed secondary HPT, to emphasise that giant cell lesions of jaw bones should routinely be screened for PTH levels to rule out underlying endocrine abnormalities and oral manifestation is the earliest manifestation of many systemic diseases and careful evaluation of oral cavity by physician gives information to underlying systemic pathology.
Keywords: Brown tumour, maxillary tumour, parathyroid hormone, secondary hyperparathyroidism
|How to cite this article:|
Thankappan P, Chundru NS, Amudala R, Kuppusamy A. Maxillary tumour as first sign of endocrine abnormality: A report of a rare case. Niger Med J 2015;56:77-9
|How to cite this URL:|
Thankappan P, Chundru NS, Amudala R, Kuppusamy A. Maxillary tumour as first sign of endocrine abnormality: A report of a rare case. Niger Med J [serial online] 2015 [cited 2020 Jul 9];56:77-9. Available from: http://www.nigeriamedj.com/text.asp?2015/56/1/77/149178
| Introduction|| |
Brown tumour is a non-neoplastic giant cell lesion caused by increased circulating levels of parathyroid hormone (PTH). Hyperparathyroidism (HPT) is a condition characterised by increased secretion of PTH. It may be primary, secondary or tertiary forms.  Primary HPT results from increased PTH secretion resulting from adenoma or hyperplasia or carcinoma of gland. Secondary HPT is characterised by increased PTH secretion in response to low serum calcium levels. Secondary HPT may be caused by low vitamin D levels acting as a direct stimulus for increased PTH secretion or chronic renal failure. Tertiary HPT is associated with renal failure and autonomous functioning of glands in long-standing secondary HPT cases.  We report a case of brown tumour of maxilla occurring in a female patient with chronic renal failure.
| Case report|| |
A 42-year-old female patient reported to us with a chief complaint of swelling in upper jaw of 2-months duration. Extraoral examination revealed gross facial asymmetry with a well-defined swelling involving the middle third of the face on the right side. There was obliteration of nasolabial fold on involved side of face [Figure 1]. Intra-oral examination revealed a well-defined solitary swelling measuring about 4 × 4 cm on the right side of hard palate not crossing midline. Posteriorly the lesion was seen extending upto the edentulous region of 15. Mucosa over the swelling was normal with no secondary changes [Figure 2]. The swelling was nontender to palpation and was hard in consistency. No lymphadenopathy was seen.
|Figure 1: Extraoral photograph showing solitary well-defined swelling on middle third of the face|
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|Figure 2: Intraoral photograph showing lesion involving the anterior palate|
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Occlusal radiograph revealed well-defined solitary radiolucent lesion involving the right side of anterior hard palate. Slight root resorption of 11 and 12 was seen. Loss of lamina dura in relation to 21, 22 and 23 region was seen [Figure 3]. Panoramic radiograph (OPG) showed findings consistent with occlusal radiograph although the lesion is superimposed with ghost image of spine. OPG also revealed generalised loss of lamina dura and thinning/loss of cortices of inferior border of mandible, walls of maxillary antrum and inferior alveolar canal suggestive of generalised rarefaction of jaw bones [Figure 4].
|Figure 3: Occlusal radiograph revealed hypodense solitary radiolucent lesion involving the right side of anterior hard palate|
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|Figure 4: OPG revealed generalised loss of lamina dura and thinning of cortices of inferior border of mandible, walls of maxillary antrum and inferior alveolar canal suggestive of generalised rarefaction of jaw bones|
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Coronal CT showed a solitary hypodense mass in the maxillary anterior region measuring approximately 3 × 4 cm, spherical shaped and encroaching the nasal fossa/cavity [Figure 5]. The radiological differential diagnosis included giant cell lesion associated with HPT. The patient was then subjected to incisional biopsy and gross examination revealed multiple friable soft tissue bits that were brown in colour.
Histopathological examination revealed numerous multinucleated giant cells set in a background of fibro-cellular connective tissue stroma showing numerous blood vessels, blood-filled spaces, suggestive of central giant cell lesion [Figure 6].
|Figure 6: H & E Section (10×) showing numerous multi-nucleated giant cells. Inset showing giant cells|
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In view of the radiological and histological evidence of giant cell lesion, the patient was further subjected to blood examination which included estimation of PTH, serum calcium, alkaline phosphatase and renal functional test. Blood examination revealed abnormally elevated PTH levels of 1810.9 pg/ml, increased Alkaline phosphatase 431 U/l, increased serum creatinine 2.5 mg/dl, elevated blood urea nitrogen[BUN] 35 gm/dl, increased serum phosphatase levels 7.5 mg/dl, decreased serum calcium 7.6 mg/dl, decreased haemoglobin 9 gm%. Based on clinical, radiographical, histopathological and lab findings the patient was diagnosed as Brown tumour resulting from secondary HPT due to underlying chronic renal failure. The patient was then referred to endocrinologist for further management.
| Discussion|| |
Brown tumours are non-neoplastic lesions resulting from abnormal bone metabolism in HPT. Bone involvement is a late manifestation of HPT and includes classic skeletal lesions such as bone resorption, bone cysts, brown tumours and generalised osteopenia.  Brown tumour is a kind of giant cell lesion and is so named because of its friable red-brown appearance resulting from vascularity, haemorrhage and haemosiderin deposits.  They usually present as painless enlargement with slow growth rate occurring with the frequency of 4.5% in primary HPT and 1.5% to 1.7% in case of secondary HPT with overall incidence of 0.1%. 
The commonly involved sites are facial bones, clavicle, ribs, pelvis and femur.  They are more common in mandible compared to maxilla and are three times more frequent in women aged over 50 years. The present case occurred in maxilla in 42-year-old female and was asymptomatic. Secondary HPT represents a frequent complication in patients with chronic renal failure. Hypocalcemia, hyperphosphatemia may be significant in chronic renal failure and may stimulate increased PTH secretion, which in turn alters intra and extracellular calcium ratio thereby causing increased bone resorption, decreased bone density as seen in the present case.
The histopathological features of Brown tumour are suggestive but not sufficient enough to establish the diagnosis or differentiation from other giant cell lesion such as central giant cell granuloma, giant cell tumour, etc.
In the present case the histopathological features resembled central giant cell granuloma but the diagnosis of Brown tumour associated with secondary HPT was established in view of the clinical, radiographical and laboratory reports.
They are often asymptomatic as in the present case and sometimes may be, the first impending sign of an underlying previously undiagnosed endocrine disorder.
Treatment should commence with treatment of underlying HPT which in turn results in complete resolution. Controlling HPT is mandatory and may be done by carrying out total parathyroidectomy.
| Conclusion|| |
Although rare, Brown tumours of jaws should be considered in the differential diagnosis of giant cell lesions of bone and the dental surgeon should be aware of oral manifestations associated with underlying systemic disease and need for careful clinical, radiological, serological examination cannot be over-ruled.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]