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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 28  |  Issue : 2  |  Page : 191-194

An unusual presentation of pleomorphic adenoma: A case report


1 Department of Oral Medicine Diagnosis and Radiology, M.N.D.A.V. Dental College and Hospital, Solan, Himachal Pradesh, India
2 Department of Orthodontics, Rayat Bahra Dental College, Mohali, Punjab, India

Date of Submission22-May-2015
Date of Acceptance21-Nov-2016
Date of Web Publication02-Dec-2016

Correspondence Address:
MunBhawni Bagga
Department of Oral Medicine Diagnosis and Radiology, M.N.D.A.V. Dental College and Hospital, Tatul, Solan, Himachal Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.195146

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   Abstract 

Pleomorphic adenoma of the submandibular gland is rare and can present difficulty in diagnosis. The heterogeneous histology, a possible malignant transformation, and an incomplete capsule that can determine a recurrence are the most important characteristics of pleomorphic adenoma. This paper illustrates one such atypical case resembling lymphadenopathy in the submandibular gland.

Keywords: Lymphadenopathy, malignancy, pleomorphic


How to cite this article:
Bagga M, Bhatnagar D, Bhatnagar D. An unusual presentation of pleomorphic adenoma: A case report. J Indian Acad Oral Med Radiol 2016;28:191-4

How to cite this URL:
Bagga M, Bhatnagar D, Bhatnagar D. An unusual presentation of pleomorphic adenoma: A case report. J Indian Acad Oral Med Radiol [serial online] 2016 [cited 2021 Apr 13];28:191-4. Available from: https://www.jiaomr.in/text.asp?2016/28/2/191/195146


   Introduction Top


Tumors of the salivary gland constitute a major area of concern in the field of oral and maxillofacial medicine. Salivary gland tumors constitute 2-6.5% of all head and neck neoplasms. [1] Approximately 90% of benign neoplasms in the salivary gland are associated with the parotid gland. Pleomorphic adenoma (PA) comprises 80-90% of these benign parotid neoplasms. PA of submandibular and sublingual gland is quite uncommon and comprises the rest (8-10%) of the group. [2] Its etiology is unclear with a female sex predilection affecting patients in the 4 th to 6 th decades. [1] PA is characterized by considerable histological diversity, and myoepithelial cells are considered to be responsible for the production of extracellular matrix. [3] This case report presents a case of histologically proven PA involving the submandibular gland mimicking lymphadenopathy.


   Case Report Top


A 50-year-old male patient presented in the Department of Oral Medicine and Radiology with the chief complaint of swelling below the right lower jaw region since 3 months. The swelling developed spontaneously which gradually increased in size and attained the present size. It was not associated with pain, fever, loss of weight, loss of appetite, or related to meals. Past medical history revealed that the patient suffered from tuberculosis 9 years back, following which he took medication for the same. Past dental history revealed that the patient underwent uneventful extraction of multiple teeth 1 year back due to loosening of teeth. Personal history revealed that he was a chronic smoker and used to smoke one bundle of beedi every day since 20 years. Family history was not significant. General physical examination revealed that the patient was moderately built and nourished, was calm, conscious and cooperative. No signs of anemia, cyanosis and jaundice were observed on examination.

On extraoral examination, a solitary ovoid swelling was present on the right submandibular region extending superiorly from 0.3 cm below the right border of the mandible to the upper border of sternocleidomastoid muscle inferiorly; medially, it extended 3 cm away from the chin to laterally 0.5 cm ahead of the angle of mandible. Swelling was lobulated same as the color of the facial skin measuring approximately 1.5 × 1 cm in size mediolaterally to superioinferiorly respectively [Figure 1]. On palpation, swelling was firm, lobulated, nontender with well-defined margins and was not attached to underlying structures. Skin over the swelling was pinchable with no evident secondary changes. Swelling was nonfluctuant and noncompressible with no signs of inflammation. On lymph node examination, the cervical lymph nodes were not palpable in the neck. The swelling was appreciated on superficial, i.e., submandibular lymph node examination [Figure 2]. On intraoral examination, there was no evidence of any swelling in the floor of the mouth with normal opening of Wharton's duct. On bimanual palpation, swelling was not appreciated in submandibular gland region.
Figure 1: Facial profile showing swelling in submandibular region

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Figure 2: Local examination of swelling in submandibular region

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On the basis of history and clinical examination, a provisional diagnosis of right side submandibular lymphadenopathy was proposed. Other findings noted were chronic generalized periodontitis, leukoedema bilaterally involving the buccal mucosa, smoker's palate, and partially edentulous maxillary and mandibular arches [Figure 3]. The differential diagnosis given was tuberculous lymphadenopathy (Scrofula), PA, and metastatic lymph node. Orthopantomography revealed no significant finding except that there was generalized bone loss and attrition in all his teeth [Figure 4]. Chest posterioanterior was normal. Other lab findings such as complete hemogram, Mantoux test, sputum for acid-fast bacilli, and malignant cells were negative. Fine-needle aspiration cytology revealed necrotizing inflammation, few red cells, with no cellular atypia. Ultrasonography of the case showed a hypoechoic lesion of size 27.6 × 11.7 × 23.8 mm with cystic changes in it arising from the submandibular gland with well-defined lobular margin [Figure 5]. With a standard submandibular incision, the submandibular gland and the mass was excised and sent for histopathological examination. The histopathological report revealed myxoid and cellular components were present in equal proportion. The epithelial component form ducts that contain an eosinophillic coagulum. Small sheets of spindle-shaped cells resembling myoepithelial cells were also seen. The stroma was delicately collagenous with myxoid areas. There was presence of mucoid material between tumor cells, resulting in myxomatous background confirming the diagnosis of PA of the right submandibular gland [Figure 6].
Figure 3: Intra oral picture

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Figure 4: Panoramic view

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Figure 5: Ultrasonograph of right submandibular region

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Figure 6: Histopathological picture

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   Discussion Top


Submandibular swellings pose a diagnostic challenge to oral physicians because many disease entities such as lymphadenitis, sialadenitis, lymphoepithelial cyst, and benign and malignant neoplasms are involved in the differential diagnosis. [2] PA is an epithelial tumor of complex morphology, possessing epithelial and myoepithelial elements intermingled with mucoid, myxoid, or chondroid tissue arranged in a variety of patterns and embedded in a mucopolysaccharide stroma. [4]

PA is commonly seen in the parotid gland, i.e., 85% and 8% occur in submandibular gland followed by 6.5% of cases in minor salivary glands and 0.5% in sublingual gland. [5] The annual incidence of PA was approximately 2-3.5% cases per 100,000 population. It is most commonly diagnosed in the 4 th to 6 th decade of life. There is slight female predilection (1.43:1). Mostly the tumor is asymptomatic and the size ranges from 0.8 cm to 5.0 cm. Our patient also was a 50-year-old male patient with a small painless, quiescent nodule which slowly increased in size and attained the present size.

Here, the case report illustrates the characteristic clinical rare case of PA mimicking scrofula of submandibular lymph nodes because the lymph nodes are localized in the gland parenchyma with a positive history of past tuberculosis. However, metastatic submandibular lymph node from other primary tumor is also a possibility because the patient was old aged and salivary glands had intense drainage with lymph vessels but rare in frequency, i.e., 1.5% in lung cancer and 1% in renal cancer. [6]

Various modalities such as fine-needle aspiration cytology, ultrasonography, computed tomography and incisional biopsy are employed for appropriate diagnosis. Standard radiological tools help in determining the size and extent of lesion whereas adjunctive procedures such as fine needle aspiration helps in preoperative diagnosis to distinguish benign from malignant neoplasms. [7],[8] PA are benign tumors with a well-documented transformation to malignancy (carcinoma ex pleomorphic adenoma). It is estimated that up to 25% of untreated PAs undergo malignant transformation. [9] Therefore, early definitive treatment is strongly recommended.


   Conclusion Top


Early diagnosis of PA at a rare site in the submandibular region is a diagnostic challenge. A key to diagnose PA emphasizes on the prior clinical knowledge of the clinical features and investigations. Submandibular swellings pose a diagnostic challenge as many disease entities are involved in differential diagnosis. We as oral physician play an important role in early diagnosis and timely management of the case to prevent the risk of malignant changes associated with the pathology.

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.
Shetty MM, Rithesh KB, Shetty A, Banerjee S. Pleomorphic adenoma of the upper lip in adolescent. Indian Dent Res Rev 2015;9:22-4.  Back to cited text no. 1
    
2.
SomPM, Brandwein MS. Salivary glands: Anatomy and pathology. In: P.M. Som, D.C. Curtin, editors. Head and neck imaging. 4 th ed. Mosby, St Louis, MO; 2003:2067-76.  Back to cited text no. 2
    
3.
Auclair PR, Ellis GL, Gnepp DR, Wenig BM, Janney GG. Salivary gland neoplasms general considerations. In: Ellis GL, Auclair PR, Gnepp DR. Surgical pathology of the salivary glands. Philadelphia: WB Saunders Co.; 1991. p. 135-64.  Back to cited text no. 3
    
4.
Van der Wall I. Salivary gland neoplasm. In: Prabhu SR, Wilson DF, Daftary DK, Johnson NW, editors. Oral Disease in the tropics. New York: Oxford University Press; 1992. p. 478-86.  Back to cited text no. 4
    
5.
Spiro RH. Salivary neoplasm: Overview of a 35 year experience with 2807 patients. Head Neck Surg 1986;8:177-84.  Back to cited text no. 5
    
6.
Seifert G, Hennings K, Caselitz J. Metastatic tumours of parotid and submandibular gland - Analysis and differential diagnosis of 108 cases. Pathol Res Pract 1986;181:684-92.  Back to cited text no. 6
    
7.
LiVolsi VA, Perzin KH. Malignant mixed tumors arising in salivary glands. I. Carcinomas arising in benign mixed tumors: A clinicopathological study. Cancer 1977;39:2209-30.  Back to cited text no. 7
    
8.
Sadeghi EM, Darling JE. Cystic pleomorphic adenoma of the mandible. Int J Oral Maxillofac Surg 1994;23:93-5.  Back to cited text no. 8
    
9.
Pinkston JA, Cole P. Incidence rates of salivary gland tumors: Results from a population-based study. Otolaryngol Head Neck Surg 1999;120:834-40.  Back to cited text no. 9
    


    Figures

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



 

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