Home Print this page Email this page Small font sizeDefault font sizeIncrease font size
Users Online: 621

 

Home About us Editorial board Search Ahead of print Current issue Archives Submit article Instructions Subscribe Advertise Contacts Login 
     

  Table of Contents  
CASE REPORT
Year : 2015  |  Volume : 56  |  Issue : 1  |  Page : 77-79  

Maxillary tumour as first sign of endocrine abnormality: A report of a rare case


Department of Oral Pathology, Chadalawada Krishnamurthy Srinivasa Theja Institute of Dental Sciences and Research, Tirupathi, Andhra Pradesh, India

Date of Web Publication13-Jan-2015

Correspondence Address:
Anitha Kuppusamy
Department of Oral Pathology, CKS Theja Institute of Dental Sciences and Research, Chadalawada Nagar, Renigunta Road, Tirupathi - 517 506, Andhra Pradesh
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0300-1652.149178

Rights and Permissions
   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 2019 Dec 14];56:77-9. Available from: http://www.nigeriamedj.com/text.asp?2015/56/1/77/149178


   Introduction Top


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. [1] 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. [2] We report a case of brown tumour of maxilla occurring in a female patient with chronic renal failure.


   Case report Top


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

Click here to view
Figure 2: Intraoral photograph showing lesion involving the anterior palate

Click here to view


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

Click here to view
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

Click here to view


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.
Figure 5: Coronal CT showing mass involving the anterior maxilla

Click here to view


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

Click here to view


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 Top


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. [3] 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. [4] 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%. [5]

The commonly involved sites are facial bones, clavicle, ribs, pelvis and femur. [6] 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 Top


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.

 
   References Top

1.
Wood NK, Pehowich DJ, Bateman RC.Generalized rarefactions of the jaw bones.In,Wood NK,Goaz PW(ed).Differential diagnosis of oral and Maxillofacial lesions,5 th edition.St.Louis,Mosby,1997;393-396.Goaz PW. Differential Diagnosis of Oral and Maxillofacial Lesions. 5 th ed. St. Louis: Mosby; 1997. p. 393-6.  Back to cited text no. 1
    
2.
Ahmad R, Hammond JM. Primary, secondary, and tertiary hyperparathyroidism. Otolaryngol Clin North Am 2004;37:701-13.  Back to cited text no. 2
    
3.
Horowitz M, Wishart JM, Need AG, Morris HA, Nordin BE. Primary hyperparathyroidism. Clin Geriatr Med 1994;10:757-75.  Back to cited text no. 3
    
4.
Triantafillidou K, Zouloumis L, Karakinaris G, Kalimeras E, Lordanidis 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. 4
    
5.
Soundarya N, Sharada P, Prakash N, Pradeep G. Bilateral maxillary brown tumors in a patient with primary hyperparathyroidism: Report of a rare entity and review of literature. J Oral Maxillofac Pathol 2011;15:56-9.  Back to cited text no. 5
[PUBMED]  Medknow Journal  
6.
Okada H, Davies JE, Yamamoto H. Brown tumor of the maxilla in a patient with secondary hyperparathyroidism: A case study involving immunohistochemistry and electron microscopy. J Oral Maxiilofac Surg 2000;58:233-8.  Back to cited text no. 6
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]


This article has been cited by
1 Systematic review of oral manifestations related to hyperparathyroidism
Benjamin Palla,Egon Burian,Riham Fliefel,Sven Otto
Clinical Oral Investigations. 2017;
[Pubmed] | [DOI]



 

Top
  
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Access Statistics
    Email Alert *
    Add to My List *
* Registration required (free)  

 
  In this article
    Abstract
   Introduction
   Case report
   Discussion
   Conclusion
    References
    Article Figures

 Article Access Statistics
    Viewed1787    
    Printed24    
    Emailed0    
    PDF Downloaded67    
    Comments [Add]    
    Cited by others 1    

Recommend this journal