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 Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 26  |  Issue : 1  |  Page : 95-98

Submandibular gland swelling: A diagnostic dilemma


Department of Oral Medicine and Radiology, Swami Devi Dyal Hospital and Dental College, Barwala, Haryana, India

Date of Submission30-May-2014
Date of Acceptance11-Sep-2014
Date of Web Publication26-Sep-2014

Correspondence Address:
Tarun Kumar
Department of Oral Medicine and Radiology, Swami Devi Dyal Hospital and Dental College, Barwala, Haryana
India
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Source of Support: Clinical Photographs and Histopathological Reports, Conflict of Interest: None


DOI: 10.4103/0972-1363.141871

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   Abstract 

Submandibular swellings pose a diagnostic challenge to oral physicians, as many disease entities are involved in the differential diagnosis. Benign salivary gland tumors are one of them. Pleomorphic adenoma is the most common benign salivary gland tumor, which most commonly involves the parotid gland. Involvement of the submandibular gland is a rarity. This case report presents a rare case of a 60-year-old female patient with a unilateral submandibular swelling, diagnosed as a pleomorphic adenoma.

Keywords: Mixed tumor, pleomorphic adenoma, submandibular swelling


How to cite this article:
Kumar T, Puri G, Konidena A, Patil D. Submandibular gland swelling: A diagnostic dilemma . J Indian Acad Oral Med Radiol 2014;26:95-8

How to cite this URL:
Kumar T, Puri G, Konidena A, Patil D. Submandibular gland swelling: A diagnostic dilemma . J Indian Acad Oral Med Radiol [serial online] 2014 [cited 2020 Jan 22];26:95-8. Available from: http://www.jiaomr.in/text.asp?2014/26/1/95/141871


   Introduction Top


Pleomorphic adenoma (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. [1] The pleomorphic adenoma is the most common neoplasm arising in the major and minor salivary glands. It accounts for 90% of the benign salivary gland tumors. Approximately 75 to 85% of all pleomorphic adenomas occur in the parotid gland, 8% occur in the submandibular gland, and 7 to 15% occur in the minor salivary glands. [2],[3] Very few cases of pleomorphic adenomas affecting the submandibular gland are reported in literature. We are presenting herewith a case of unilateral submandibular swelling which has been diagnosed as a pleomorphic adenoma.


   Case Report Top


A 60-year-old female patient reported to the Outpatient Department of the Oral Medicine and Radiology section of our dental college, with a chief complaint of swelling in the left lower back region of the face, since two years. Her 'history of present illness' dates back to 20 years, when she had a swelling in the same region of the face, which was operated by a doctor at a government hospital. The patient did not have any record of the treatment provided earlier. At present, there is a recurrence of the swelling since the last two years. It was pea-sized and gradually increased to the present size. She was asymptomatic with no signs of fever or dysphagia. The patient was hypertensive and under medication for the same since the last five years.

On general physical examination, the patient had signs of pallor. All the other vital signs were within normal limits. On extraoral examination, a diffuse, elliptical, solitary swelling was present in the left submandibular region [Figure 1]a. It measured about 4 × 2.5 cm in maximum dimension. It extended 2 cm below the inferior border of the body of the mandible, superoinferiorly, in the left submandibular region. The surface of the swelling was lobulated with a scar mark. The overlying skin was normal, without any sinus tract formation, discharge or visible pulsations [Figure 1]b. On palpation, all the inspectory findings were confirmed. The temperature of the overlying skin was normal. The swelling was nontender, firm in consistency, and lobulated. The skin over the swelling was pinchable and mobile. The swelling was non-compressible, non-pulsatile, and non-fluctuant.
Figure 1: (a) Extraoral picture of the patient (b) Extraoral picture (Swelling)

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On intraoral examination, bimanual palpation of submandibular gland was done. The swelling was nontender and firm in consistency. Dental caries was detected in teeth numbers 16, 36, and 46 [Figure 2]. Tooth number 36 was tender on percussion and attrition was present in the mandibular anteriors.

A provisional diagnosis of chronic submandibular lymphadenitis was given. Benign salivary gland tumor, submandibular tubercular lymphadenopathy, and metastasis of unknown primary tumor were considered in the differential diagnosis. Fine needle aspiration cytology (FNAC) was performed to identify the nature of the lesion. The patient was advised a panoramic radiograph, complete hemogram, and ultrasonography, with respect to the swelling. A posteroanterior view of the chest, Renal Function Test (RFT), Liver Function Tests (LFTs), mammogram, and Papanicolaou (Pap) smear were advised, to rule out any unknown primary lesions.
Figure 2: Intraoral picture

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The panoramic radiograph revealed no significant findings [Figure 3]. A complete blood hemogram showed the patient was anemic [Table 1]. LFTs and RFT were within normal limits [Table 2]. The posteroanterior view of the chest, mammogram, and Pap smear investigation revealed no significant findings. An ultrasonograph (US) showed a lobulated homogeneous echogenic mass measuring 3.3 × 1.9 cm in the left submandibular region [Figure 4].
Figure 3: Panoramic view

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Figure 4: Ultrasonograph of the left submandibular region

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Later, the patient was operated by complete surgical excision under general anaesthesia. The excised mass [Figure 5] was sent for histopathological examination. The histopathological investigation revealed a well-circumscribed, capsulated, highly cellular stroma, with strands of hyperchromatic epithelial cells [Figure 6]. The connective tissue showed chondroid, mucoid, and myxoid areas, without nuclear pleomorphism or mitotic figures. The above-mentioned features were suggestive of pleomorphic adenoma of the left submandibular gland. The patient is currently under follow up, since one year, and no recurrence has been observed [Figure 7].
Table 1: Results of blood investigations

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Table 2: Results of liver and renal function tests

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Figure 5: Excised mass

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Figure 6: Histopathological picture

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Figure 7: Postoperative extraoral picture of the patient

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   Discussion Top


Swellings in submandibular region pose a diagnostic challenge to the oral physician. Although the rate of occurrence of submandibular swelling is common, a myriad of disease entities should be considered before arriving at a final diagnosis. The various lesions include lymphadenitis, sialadenitis, and benign and malignant neoplasms, to name a few. The pleomorphic adenoma is the most common benign salivary gland tumor, but the same in the submandibular gland is a rare occasion and very few cases have been documented in literature.

Pleomorphic adenomas are derived from a mixture of ductal and myoepithelial elements. The annual incidence of PA is approximately 2-3.5 cases per 100,000 population. [4] There is a slight female (1.43: 1) predilection than males for PA. [5] It is most commonly diagnosed in the fourth to sixth decades of life. [5] The tumor is asymptomatic and the size ranges from 0.8 to 5.0 cm (average, 2.6 cm). [5] Our patient was a 60-year-old female patient with a nontender, unilateral swelling on the left submandibular region.

Various modalities like fine needle aspiration cytology, incisional biopsy, ultrasonography, computed tomography (CT), and magnetic resonance imaging (MRI) are employed for appropriate diagnosis. [1] FNAC is a widely accepted method to achieve the preoperative diagnosis of salivary gland tumors, as they are readily accessible. The reliability of FNAC in diagnosing PAs has been reported to be 89.5 to 96.2%. [6],[7] Ultrasound examination is another widely accepted method, which is widely available, relatively cheap, safe for the patient, and easily replicable. The typical ultrasound features of PAs are considered to be a well-defined contour, a regular, sometimes lobulated shape, homogeneous echogenic structure, and poor vascularization. [5] Computed tomographic scans are useful in determining the size of the lesions as well as verifying any bony involvement. An MRI helps to determine the size of the lesion, as well as gives the approximation of the lesion to vital structures like the nerves and vessels. Histopathologically, it may show epithelial and myoepithelial elements intermingled with mucoid, myxoid or chondroid tissue in the connective tissue stroma. [2],[3]

The recommended surgical approach is with a direct submandibular incision, which provides easy access. Submandibular triangle dissection is the standard treatment for submandibular gland PAs. [8] Incomplete removal of the glandular tissue paves the way for a definitive recurrence. The recurrence rate of submandibular PAs is rare when compared to parotid tumors. [9] PAs are benign tumors with a well-documented transformation to malignancy (carcinoma ex pleomorphic adenoma). It is estimated that up to 25% of the untreated PAs undergo malignant transformation. [10]


   Conclusion Top


Oral physicians play an important role in the diagnosis of such myriad lesions. By using appropriate diagnostic modalities like FNAC and ultrasound, as has been done in the present case, early diagnosis and definitive treatment is achieved, thereby decreasing the risk of recurrence and malignant transformation.

 
   References Top

1.Rai S, Sodhi SP, Sandhu SV. Pleomorphic adenoma of submandibular gland: An uncommon occurrence. Natl J Maxillofac Surg 2011;2:66-8.  Back to cited text no. 1
[PUBMED]  Medknow Journal  
2.Sadeghi EM, Darling JE. Cystic pleomorphic adenoma of the mandible. Int J Oral Maxillofac Surg 1994;23:93-5.  Back to cited text no. 2
    
3.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. 3
    
4.Jaiswal AA, Garg AK, Membally R. Pleomorphic adenoma of parotid gland with extensive bone formation - A rare case report. Egyptian J Ear Nose Throat Allied Sci 2014;15:139-42.  Back to cited text no. 4
    
5.Fu H, Wang J, Wang L, Zhang Z, He Y. Pleomorphic adenoma of the salivary glands in children and adolescents. J Pediatr Surg 2012;47:715-9.  Back to cited text no. 5
    
6.Handa U, Dhingra N, Chopra R, Mohan H. Pleomorphic adenoma: Cytologic variations and potential diagnostic pitfalls. Diagn Cytopathol 2009;37:11-5.  Back to cited text no. 6
    
7.Verma K, Kapila K. Role of fine needle aspiration cytology in diagnosis of pleomorphic adenomas. Cytopathology 2002;13:121-7.  Back to cited text no. 7
    
8.Molina EJ, Mayer K, Khurana J, Grewal H. Pleomorphic adenoma of the submandibular gland. J Pediatr Surg 2008;43:1224-6.  Back to cited text no. 8
    
9.Munir N, Bradley PJ. Pleomorphic adenoma of the submandibular gland: An evolving change in practice following review of a personal case series. Eur Arch Otorhinolaryngol 2007;264:1447-52.  Back to cited text no. 9
    
10.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. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]
 
 
    Tables

  [Table 1], [Table 2]



 

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