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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 8  |  Issue : 2  |  Page : 120-123

A case report of unusual presentation of mucoepidermoid carcinoma with review of literature


1 Department of Oral Medicine and Radiology, Vishnu Dental College, Bhimavaram, Andhra Pradesh, India
2 Department of Oral and Maxillofacial Surgery, Vishnu Dental College, Bhimavaram, Andhra Pradesh, India
3 Department of Oral Pathology, Vishnu Dental College, Bhimavaram, Andhra Pradesh, India

Date of Web Publication18-Dec-2018

Correspondence Address:
Gadadasu Swathi
Department of Oral Medicine and Radiology, Vishnu Dental College, Bhimavaram, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijohs.ijohs_24_18

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  Abstract 


Head-and-neck cancers constitute about 30% of cancers in India, of which salivary gland tumor such as mucoepidermoid carcinoma (MEC) was the most common malignant salivary gland tumor. The parotid gland was the most common site of origin in both benign and malignant tumors, followed by submandibular and sublingual glands. Salivary gland tumors comprise a morphologically diverse group of rare tumors. Their multifaceted clinical presentation, varied morphologic configuration, and relatively unpredictable prognosis attract significant medical interest. Here is a rare case report of an asymptomatic swelling in the cheek region which was thought to be pleomorphic adenoma at the outset which on further investigations proved as MEC of early detection.

Keywords: Fine-needle aspiration, mucoepidermoid carcinoma, salivary glands, treatment planning, ultrasonography


How to cite this article:
Swathi G, Ramesh T, Kishore M, Kumar GR. A case report of unusual presentation of mucoepidermoid carcinoma with review of literature. Int J Oral Health Sci 2018;8:120-3

How to cite this URL:
Swathi G, Ramesh T, Kishore M, Kumar GR. A case report of unusual presentation of mucoepidermoid carcinoma with review of literature. Int J Oral Health Sci [serial online] 2018 [cited 2019 Jan 16];8:120-3. Available from: http://www.ijohsjournal.org/text.asp?2018/8/2/120/247801




  Introduction Top


Mucoepidermoid carcinoma (MEC) is the most common malignant tumor and accounts for 5% of all salivary gland tumors that commonly arise within the parotid gland. In 1945, Stewart et al. described its mucous-secreting and epidermal cellular elements, established it as a distinct pathological entity.[1] MEC accounts for 5%–10% of all submandibular glands and 4% of the sublingual glands.[2] The following case report is an attempt to report the unusual presence of MEC in an older male individual.


  Case Report Top


A 74-year-old male patient came with a chief complaint of painless swelling in the right cheek region for 8 months. The patient was apparently normal till 8 months back, when he got a trauma in the right cheek region. Since then, the patient noticed a swelling which was painful initially but regressed on the use of medication prescribed by local medical practitioner. This painless swelling which was initially peanut size gradually increased to the present state. He was having a habit of smoking three chutta per day for 10 years, which he cessated 4 months back. Medical and family history were noncontributory.

On extraoral examination, right submandibular lymph nodes were palpable which were two in number, 1 cm × 1 cm in size, oval in shape, firm on palpation, not fixed to underlying structures, and nontender on palpation. On head-and-neck examination, gross facial asymmetry was detected due to diffuse swelling involving the right middle third of the face which was round in shape, 2 cm × 2 cm in size approximately, extending from 1 cm from the ala of the nose to 1 cm from the tragus of the ear anteroposteriorly and superoinferiorly extending from 1 cm away from the infraorbital rim to 0.5 cm from level of corner of the mouth. The skin over swelling was shiny than the surrounding skin. On palpation, it was found that swelling was extending to the right parotid gland and it was firm in consistency, nontender, fluctuant, noncompressible, nonreducible, freely movable, and not fixed to underlying structures, and the skin over the swelling was pinchable with no associated secondary changes. On intraoral examination, there were generalized attrition of the teeth, missing with respect to 46, and root stumps with respect to 18, 25, and 36. There was no clinical presentation of extraoral swelling in oral cavity [Figure 1].
Figure 1: Clinical pictures

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In the 74-year-old male patient reporting with the clinical presentation and traumatic history, the most common lesions like lipoma and fibroma can be considered. Other possible lesions can be pleomorphic adenoma considering clinical presentation of firm painless swelling. Warthin's tumor can also be another option as it is more common in older individuals with male predilection and mostly seen in chronic smokers all conditions matched with the present case.

Fine-needle aspiration cytology (FNAC) was done intraorally 1 cm above Stenson's duct revealed straw-colored fluid [Figure 2]. Extraoral FNAC was performed as intraoral FNAC did not reveal anything. Extraoral FNAC revealed blood-tinged fluid [Figure 3]. Radiographic investigations were performed by keeping radiographic film intraorally onto buccal mucosa corresponding to extraoral swelling which revealed negative for calcifications. Then, the patient was advised for ultrasonographic examination which described the swelling as a well-defined mass with solid and cystic areas in the right cheek region with altered texture of associated parotid gland, supposing it to be a case of pleomorphic adenoma involving right parotid gland.
Figure 2: Straw-colored fluid from intraoral fine-needle aspiration cytology

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Figure 3: Blood-tinged fluid from extraoral fine-needle aspiration cytology

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Hence, to confirm, we headed for an incisional biopsy of lesion [Figure 4] followed by subsequent histopathological examination of the biopsy specimen, under magnification of 10× revealed cystic areas with ductal spaces, and under magnification of 40× [Figure 5] those ductal spaces filled with mucin, characteristic sign of MEC, are seen. There is also evidence of clear cells and pleomorphic cells with hyperchromatism and hyperplasia with different degrees of atypia highly suggestive of peripheral type of intermediate-grade MEC. The patient was counseled regarding the severity of the condition and was referred to the cancer center for further treatment.
Figure 4: Incisional biopsy specimen

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Figure 5: Histopathological presentation under ×40 showing ductal cells filled with mucin cells

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  Discussion Top


Painless extraoral swelling turned out to be salivary gland neoplasm in spite of the absence of intraoral presentation. Clinical presentation diversified from common nature of lesion. MEC with such clinical presentation is very rare to be found in the literature. Hence, all investigations have to be carried on in arriving proper diagnosis of lesion. Lesions have different presentations which cannot be assessed.

The MEC was first described by Stewart et al. in 1945 as a mucoepidermoid tumor categorized into “favorable” (benign) and “unfavorable” (malignant) variants. It later, however, became apparent that all variants of the neoplasm are malignant and capable of locoregional and distant metastasis, hence the adoption and endorsement of the term “MEC” by the second 2005 World Health Organization (WHO) classification of salivary gland neoplasms.[3],[4]

The WHO classified MEC as a malignant neoplasm of epithelial origin that represents 3%–15% of all salivary gland tumors.[5] The palate is the most common site of involvement for minor salivary gland neoplasms, with more than 50% presenting at the junction between the hard and soft palate where minor salivary glands are most numerous.[6] Other sites that may be involved include buccal mucosa, upper and lower lips, and the retromolar region.[7]

MEC has a slight female preponderance and is more common in adults in their fourth to sixth decades, with the highest prevalence during the fifth decade of life. The most common presenting symptom is a slowly enlarging painless mass of several years duration clinically, mimicking a pleomorphic adenoma or other benign neoplasm. Pain or facial nerve palsy may develop, usually in association with high-grade tumors.[8]

Histologically, MEC is characterized by three main cell types as follows: epithelial cells, mucin-producing cells, and intermediate cells. The histopathological grades are based on the degree of cytological atypia, amount of cyst formation, and relative numbers of mucous, epidermoid, and intermediate cells. MECs are classified into three grades: low, intermediate, and high grade. Low-grade tumors have a higher ratio of mucous cells and are less aggressive lesions, while high-grade tumors have a smaller proportion of mucous cells and are considered to be a more malignant tumor with poorer prognosis.[9] Spiro et al. classified MEC as low, intermediate, or high grade, with 5-year survival rates of 92%, 83%, and 24%, respectively.[10]

MECs are capable of metastasizing, and metastases most often occur in high-grade lesions. In addition to lymph nodes, the usual sites of metastases are the lungs and bone. Radiological features are seen in central MEC as appears as a unilocular or multilocular radiolucent lesion with sclerotic and well-defined margins.[11]

MECs are best treated by surgery, which depends on the location, size, and histopathological grading. Local resection is the treatment option for less aggressive low-grade tumors, while high-grade tumors require wide resection with the involvement of adjacent structures.[11] Radiotherapy should be used only in selected cases because of long-term adverse effects. Prognosis is dependent on clinical stage, site, grading, and adequacy of surgery.[12]

In our present case, lesion was presented with cystic consistency in unusual site (middle one-third of the face), with palpable submandibular lymph nodes deflecting the diagnosis. However, sequential and analytical diagnostic procedures helped for a definitive diagnosis of MEC.


  Conclusion Top


Although MEC arising from the parotid gland is a common finding, its clinical presentation of the above case is entirely diverse and not found in the literature. As a result, thorough monitoring of different types of lesions with their typical presentations is utmost important for all medical professionals. Careful examination and evaluation of lesion with a multidirectional approach in an analytical manner can only lead to the proper diagnosis so that treatment can be initiated at the earliest for better prognosis.

Declaration of patient consent

We, being authors of this case report certify that we 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.
Banerjee P, Jain A, Rathi S. Central mucoepidermoid carcinoma of the mandible: A rare case report with diagnostic dilemma and review of the literature. Egypt J Otolaryngol 2016;32:110.  Back to cited text no. 1
  [Full text]  
2.
Verma RK, Sunku SK, Bal A, Panda NK. Giant cystic primary mucoepidermoid carcinoma of mandible: A rare case and literature review. Otolaryngol Pol 2014;68:328-32.  Back to cited text no. 2
    
3.
Ellis GL, Auclair PL; Armed Forces Institute of Pathology (U.S.), Universities Associated for Research and Education in Pathology. Tumors of the Salivary Glands. Washington DC: Armed Forces Institute of Pathology, under the auspices of Universities Associated for Research and Education in Pathology; 1996. p. 468.  Back to cited text no. 3
    
4.
Stewart FW, Foote FW, Becker WF. Muco-epidermoid tumors of salivary glands. Ann Surg 1945;122:820-44.  Back to cited text no. 4
    
5.
Eveson J, Nagao T. Diseases of the salivary glands. In: Barnes L, editor. Surgical Pathology of the Head and Neck. 3rd ed., Vol. 1. New York: Informa Healthcare; 2008. p. 546-52.  Back to cited text no. 5
    
6.
Abrahão AC, de Santos Netto JN, Pires FR, Santos TC, Cabral MG. Clinicopathological characteristics of tumours of the intraoral minor salivary glands in 170 Brazilian patients. Br J Oral Maxillofac Surg 2016;54:30-4.  Back to cited text no. 6
    
7.
Mishra S, Mishra YC. Minor salivary gland tumors in the Indian population: A series of cases over a ten year period. J Oral Biol Craniofac Res 2014;4:174-80.  Back to cited text no. 7
    
8.
Rajendran R. Tumors of the salivary glands. Shafer's Text book of Oral Pathology. 6th ed. India: Elsevier; 2009. p. 232-4.  Back to cited text no. 8
    
9.
Kumar AN, Nair PP, Thomas S, Raman PS, Bhambal A. Mucoepidermoid carcinoma of sublingual gland: A malignant neoplasm in an uncommon region. BMJ Case Rep 2011;2011. pii: bcr0220113864.  Back to cited text no. 9
    
10.
Spiro RH, Huvos AG, Berk R, Strong EW. Mucoepidermoid carcinoma of salivary gland origin. A clinicopathologic study of 367 cases. Am J Surg 1978;136:461-8.  Back to cited text no. 10
    
11.
Ali SA, Memon AS, Shaik NA, Soomro AG. Mucoepidermoid carcinoma of parotid presenting as unilocular cyst. J Ayub Med Coll Abbottabad 2008;20:141-2.   Back to cited text no. 11
    
12.
Neville WB, Damm DD, Allen CM, Bouquot J. Salivary gland pathology. Oral And Maxillofacial Pathology. 2nd ed. Penslyvania: Saunders; 2002. p. 420-3.  Back to cited text no. 12
    


    Figures

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



 

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