Journal of Dental Research and Review

CASE REPORT
Year
: 2014  |  Volume : 1  |  Issue : 2  |  Page : 97--99

Mucoepidermoid carcinoma of minor salivary gland in buccal mucosa: A rare case report


Deepak G. Kulkarni, Lakshmi Shetty, Vishal Zurange 
 Department of Oral and Maxillofacial Surgery, Dr. D. Y. Patil Dental College and Hospital, Dr. D. Y. Patil Vidyapeeth, Pimpri, Pune, Maharashtra, India

Correspondence Address:
Lakshmi Shetty
Department of Oral and Maxillofacial Surgery, Dr. D. Y. Patil Dental College and Hospital, Dr. D. Y. Patil Vidyapeeth, Pimpri, Pune, Maharashtra
India

Abstract

Minor salivary gland carcinomas are rarely reported. Low-grade mucoepidermoid carcinomas (MECs) that are of minor salivary gland origin occurrence in children and adolescents have rarely been reported, little information regarding their clinical features and biologic behavior is available. This case report represents minor salivary gland MECs in an 18-year-old female patient. She presented with swelling in the lower left buccal mucosa, which was noticed 1 year back gradually increased to its present size 1.5 cm × 1.5 cm in its greatest dimension buccally. The color of the lesion was pale pink however on the posterior aspect the swelling, which had a bluish tinge, gave a diagnostic dilemma. In children and adolescents, MECs have a female predilection and occur most commonly on the hard or soft palate or both and rare in buccal mucosa. The surgical excision of the swelling was successful with no recurrence reported.



How to cite this article:
Kulkarni DG, Shetty L, Zurange V. Mucoepidermoid carcinoma of minor salivary gland in buccal mucosa: A rare case report.J Dent Res Rev 2014;1:97-99


How to cite this URL:
Kulkarni DG, Shetty L, Zurange V. Mucoepidermoid carcinoma of minor salivary gland in buccal mucosa: A rare case report. J Dent Res Rev [serial online] 2014 [cited 2020 Jun 2 ];1:97-99
Available from: http://www.jdrr.org/text.asp?2014/1/2/97/133958


Full Text

 Introduction



Epithelial salivary gland neoplasms are rare both in adults and children, accounting for <3% of all head and neck tumors. About 5% of these tumors occur in patients younger than 18-year-old with female mostly affected, while its occurrence in newborns is exceeding rarely. [1],[2],[3],[4] Mucoepidermoid carcinoma (MEC) is one of the most common salivary gland malignancies. MEC of minor salivary gland origin in children and adolescents have rarely been reported in literature less information regarding their clinical features and biologic behavior is documented. As its name implies, MEC is composed of a mixture of cells, including mucus producing, epidermoid or squamous, and intermediate types. [5] MEC appears as asymptomatic swellings in minor salivary glands, being the second most common site of occurrence after the parotid gland. Other sites of presentation have been on the palate, in retromolar area, floor of mouth, buccal mucosa, lips, and tongue. [1],[5],[6] In our case report, we are describing salivary gland tumor in an 18-year-old female. This report describes a case of a low-grade MEC affecting the buccal mucosa.

 Case Report



An 18-year-old female patient reported to the Department of Oral and Maxillofacial Surgery of Dr. D Y Patil Dental College, Pune. She reported with the chief complaint of pain in the lower left back region of the jaw since 4 days and a painless swelling in the left side of the jaw since 1 year. One year back, she noticed a small swelling on the left side of buccal mucosa which has gradually increased to its present size [Figure 1]. She was apparently asymptomatic 4 days before she reported to our department. She gave a history of cheek bite while having food following which she experienced pain in the lower left back region of jaw. The pain was dull and continuous in nature and aggravated on mastication. The clinical examination revealed a well-defined solitary round sessile swelling present in left buccal mucosa seen at the occlusal level plane extending from 26-28 to 36-37 measuring 1.5 cm × 1.5 cm in its greatest dimension buccally. The color of the lesion was pale pink however on the posterior aspect the swelling had a bluish tinge. The surrounding mucosa appeared normal. On palpation, inspectory findings were confirmed. The swelling was nontender, hard in consistency, mobile and nonfixed to underlying structures. No history of pain, pus discharge, bleeding seen from the lesion. The swelling was nonfluctuant, noncompressible and nonpulsatile. She denied any symptoms attributed to the mass. Adjacent teeth had no mobility or displacement, and electrical pulp test results were positive. In panoramic [Figure 2] and periapical radiographs the alveolar bone had no resorption with respect to the upper and lower posteriors of the left side. No other abnormalities, including palpable submandibular and cervical lymph nodes, were found. Fine needle aspiration yielded no fluid. Since the clinical presentation appeared more towards benign entity, wide local surgical excision was performed [Figure 3]. The histopathological examination revealed a low-grade MEC [Figure 4].{Figure 1}{Figure 2}{Figure 3}{Figure 4}

 Discussion



The patient presented with swelling seen in buccal mucosa since 1 year had bluish discoloration and localized fluctuant nodule like appearance. The clinical and radiographic differential diagnosis of a buccal mucosal mass includes mucocele, which is frequently seen in fluctuant reactive lesion of salivary glands, with transparent blue swelling including mucin. Deep mucoceles, often surrounded by a fibrous tissue wall, do not fluctuate, and if located at sites other than the lower lip cannot clinically and reliably be differentiated from salivary gland tumors. Our case was not similar to a mucocele and hence the diagnosis was ruled out.

Malignancy seen in salivary gland tumors is 50% in children and 15-25% in adults. [7] As a typical intraoral presentation this malignancy has a painless and persistent enlargement, which lasts for about a year. Intraoral lesions with paresthesia, pain, and difficulty with swallowing are noted frequently when major salivary glands and tongue are involved. High-grade lesions may be quite firm and accompanied with ulceration, resorption of bone, and numbness of adjacent teeth [6] these clinical findings were not presented in our case. High-grade MEC epithelial cells are predominantly squamous, whereas in low-grade, mucous cells predominate. [8],[9] There was no significant radiographic finding found in our case where as malignancy have poorly defined margins, local infiltrations, and solid appearances on radiographs. [10] Neurofibroma and schwannoma are occasionally encountered as compressible or firm asymptomatic nodules and pink in color unless they are secondarily traumatized. [10] The presentation of the case reported was more toward benign nature.

The tumor should be excised to obtain adequate tumor-free margins. [3] However, if there is any evidence of bony involvement, removal of a portion of the jaw is necessary in our case since there was no bony involvement so only wide local excision was advocated. Chemotherapy was not used as an adjuvant therapy in our patient and does not currently have a role in the standard treatment of MEC patients. [11]

Low-grade tumors are soft and compressible. This finding correlated with our histopathological report which showed mucous cells predominantly.

Overall survival rate has been linked to histocytologic grade with 95-100% in low-grade and 25-43% in high-grade tumors. [9] Furthermore, it should be considered that micro marsupialization, cryosurgery, and laser therapy are contraindicated in management of an intraoral submucosal mass or nodules. These kinds of treatments may result in local spread of the tumor, and more aggressive surgery may be needed. [6] Based on low recurrence and mortality rates in surgical treatment of low-grade MEC, it seems that the treatment of our patient is adequate. Radiation therapy should be used judiciously in pediatric patients with high-grade histology, positive margins, and lymph node involvement, due to its long-term consequences as facial deformity, trismus, xerostomia, osteoradionecrosis, and risk of secondary malignancy. Follow-up of the patient was done for 1 year [Figure 5] and the patient has not reported with any sign of recurrence or any other related complaints. Recent reports concerning children with second malignancies have described the development of MEC in those who have received chemotherapy and cranial irradiation for a primary malignancy or aggressive disease, including acute lymphocytic leukemia and langerhans cell histocytosis. [12] In our case although patient was in her adolescent age she did not give any history of previous malignancy. Moreover, survivors of the adolescent cancer must be followed closely throughout their lifetime for the risk of developing a secondary malignancy following the treatment of adolescent cancer. [13] MECs of the oral minor salivary glands generally have a good prognosis. [14] Moreover the central MEC and the one arising from minor salivary glands are usually of low-grade variety. [15] Rarely, MEC can arise as primary jaw or heterotopic intranodal tumours, or as laryngeal, lacrimal, nasal, paranasal, tracheal or pulmonary tumours. [16] The patient is presently also on regular follow-up.{Figure 5}

 Conclusion



Low-grade MEC and other tumors are rare in buccal mucosal region. The minor salivary gland location in the buccal mucosa is also rare and hence the patient has been under close follow-up to elicit any recurrence. Thus patients with these kinds of swelling must be cautiously evaluated and proper treatment can lead to successful treatment.

References

1Hicks J, Flaitz C. Mucoepidermoid carcinoma of salivary glands in children and adolescents: Assessment of proliferation markers. Oral Oncol 2000;36:454-60.
2Luna MA, Batsakis JG, el-Naggar AK. Salivary gland tumors in children. Ann Otol Rhinol Laryngol 1991;100:869-71.
3Auclair PL, Ellis GL. Mucoepidermoid carcinoma. In: Ellis GL, Auclair PL, Gnepp DR, editors. Surgical Pathology of the Salivary Glands. 2 nd ed. Philadelphia: WB Saunders; 1991. p. 269-98.
4Ashok Kumar S, Ramakrishnan M. Mucocele in lower lip as a result of improper use of feeding bottle: A case report. Case Rep Dent 2013;2013:520425.
5da Cruz Perez DE, Pires FR, Alves FA, Almeida OP, Kowalski LP. Salivary gland tumors in children and adolescents: A clinicopathologic and immunohistochemical study of fifty-three cases. Int J Pediatr Otorhinolaryngol 2004;68:895-902.
6Flaitz CM. Mucoepidermoid carcinoma of the palate in a child. Pediatr Dent 2000;22:292-3.
7Baker SR, Malone B. Salivary gland malignancies in children. Cancer 1985;55:1730-6.
8Bower CM, Dyleski RA. Diseases of the salivary glands. In: Bluestone CD, editor. Pediatric Otolaryngology. 5 th ed. Philadelphia: Saunders; 2003. p. 1251-67.
9Khadaroo RG, Walton JM, Ramsay JA, Hicks MJ, Archibald SD. Mucoepidermoid carcinoma of the parotid gland: A rare presentation in a young child. J Pediatr Surg 1998;33:893-5.
10Som PM, Brandwein MS. Anatomy and pathology of the salivary glands. In: Som PM, Curtin HD, editors. Head and Neck Imaging. 2 nd ed. St. Louis: Mosby Elsevier; 2011. p. 2448-609.
11Kupferman ME, de la Garza GO, Santillan AA, Williams MD, Varghese BT, Huh W, et al. Outcomes of pediatric patients with malignancies of the major salivary glands. Ann Surg Oncol 2010;17:3301-7.
12Prasannan L, Pu A, Hoff P, Weatherly R, Castle V. Parotid carcinoma as a second malignancy after treatment of childhood acute lymphoblastic leukemia. J Pediatr Hematol Oncol 1999;21:535-8.
13Savelli SL, Klopfenstein KJ, Termuhlen AM. Mucoepidermoid carcinoma of the parotid gland as a second malignant neoplasm. Pediatr Blood Cancer 2005;45:997-1000.
14Neville BW. Salivary gland pathology. In: Neville BW, Damm DD, Allen CM, Bouquot JE, editors. Oral and Maxillofacial Pathology. 2 nd ed. Edinburgh: Saunders; 2002. p. 420-2.
15Brookstone MS, Huvos AG. Central salivary gland tumors of the maxilla and mandible: A clinicopathologic study of 11 cases with an analysis of the literature. J Oral Maxillofac Surg 1992;50:229-36.
16Wedell B, Burian P, Dahlenfors R, Stenman G, Mark J. Cytogenetical observations in a mucoepidermoid carcinoma arising from heterotopic intranodal salivary gland tissue. Oncol Rep 1997;4:515-6.