Pleomorphic adenoma of cheek: A rare case report ...

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CASE REPORT Pleomorphic adenoma of cheek: A rare case report and review of literature Pradhuman Verma, Suresh K Sachdeva, Kanika Gupta Verma1, Kompal Sachdeva2 Departments of Oral Medicine and Radiology, 1Pedodontics and Preventive Dentistry, 2 Conservative and Endodontics, Surendera Dental College and Research Institute, Sri Ganganagar, Rajasthan, India Received : 18-02-13 Review completed : 12-04-13 Accepted : 21-09-13

ABSTRACT Pleomorphic adenoma (PA) is the most common tumor of the salivary glands. About 90% of these tumors occur in the parotid gland and 10% in the minor salivary glands. The most common sites of PA of the minor salivary glands are the palate, followed by lips and cheeks. Throat, floor of the mouth, tongue, tonsil, pharynx, retromolar area and nasal cavity are rarely involved. Here, we report a case of PA of the cheek in a 42-year-old female. The mass was excised and the patient was followed for 3 years post operatively with no recurrence. Key words: Cheek, minor salivary gland, myoepithelial cells, pleomorphic adenoma

Pleomorphic adenoma (PA) is defined by World Health Organization in 1972 as a circumscribed tumor characterized by its pleomorphic or mixed appearance clearly recognizable epithelial tissue being intermingled with tissue of mucoid, myxoid and chondroid appearance. Among all salivary gland tumors, PA is the most frequently encountered lesion (50%).[1] It account for 53-77% of parotid tumors, 44-68% of submandibular tumors, 6.4% occur in minor salivary glands. Among minor salivary glands, palate is considered as the most common intraoral site, followed by upper lip and cheek.[2] They are more common in adult females from the 3rd to 5th decades.[3] PA is of glandular origin, usually presenting as a slowly growing, painless, firm swelling that does not cause ulceration of the overlying mucosa.[4] Histologically, PA consists of cells with epithelial and mesenchymal differentiation. The treatment of choice for PA is surgical removal with safety margins, to prevent the recurrence. Recurrence rate of 5-30% has been found for PA, so a periodic follow-up is must, due to the important relapse potential and aggressivity of these lesions.[5] The purpose of this article is to report a rare case of PA of cheek in 42-year-old female patient with characteristic clinical, imaging, histological features and treatment. Address for correspondence: Dr. Suresh K Sachdeva E-mail: [email protected] Access this article online Quick Response Code:

Website: www.ijdr.in PMID: *** DOI: 10.4103/0970-9290.131166

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CASE REPORT A 42-year-old female patient visited the Department of Oral Medicine and Radiology with the chief complaint of small painless swelling over the left side of cheek since 4 years causing unesthetic appearance. History of presenting illness revealed that the swelling was gradual in onset, appeared first intraorally, which has grown slowly to attain the present size. There was no history of fever, bleeding, pain, sensory changes and disturbance of salivation or trauma. The past dental history and medical history was unremarkable and no other abnormalities were found. Personnel history revealed that patient was married with two kids. The family history was non-significant. General physical examination showed that patient was of moderate built, height and well-oriented to time and place and all the vitals were in normal limits. Hard tissue examination showed incipient dental caries in 26 and 37. The gingiva was mildly inflamed with both stains and calculus was present. The salivary flow was normal with thin and watery consistency. Extra oral examination showed a mild facial asymmetry due to diffuse solitary swelling on the left side of cheek extending from the ala-tragus line superiorly up to 2 cm above the lower border of mandible of the same side inferiorly. Anteriorly, it extends from 1 cm posterior to oral commissure up to line drawn perpendicular from the lateral canthus of the eye to the body of mandible posteriorly [Figure 1]. The skin over the swelling appeared normal and was pinchable with no localized increase in temperature. Intraorally, the mass was round oval in shape, measuring 3.5 × 2.5 cm in maximum diameter, firm in consistency, non-fluctuant and non-reducible, non-pulsatile and freely movable. Overlying mucosa was of normal in color [Figure 2]. Indian Journal of Dental Research, 25(1), 2014


Verma, et al.:

Pleomorphic adenoma of cheek

Based upon the history and clinical examination, the provisional diagnosis of benign buccal sub mucosal nodule was made and the differential diagnosis of neurofibroma, benign minor salivary gland tumor, lipoma, dermoid cyst, intraoral sebaceous cyst fibrosed mucocele and hemangioma were considered.

INVESTIGATIONS Patient was subjected to hematological investigations, which showed white blood cells = 6.7 × 109/μL, red blood cells = 3.59 × 106/μL, hemoglobin = 10.4 g/dL, bleeding time = 2.30 s, clotting time = 4.40 s and erythrocyte sedimentation rate of 10 mm/1st h. Radiological examination showed no abnormality in the panoramic radiograph. The ultrasound examination revealed oval hypoechoic well-marginated mass (2.0 × 1.1 × 1.4 cm) inferio-lateral to mouth on the left side without any calcification and increased vascularity [Figure 3]. The biopsy was planned and the mass was excised with safety margins under local anesthesia. Grossly, the lesion was ovoid in shape, well demarcated partially encapsulated, grey-white rubbery mass measuring 2.2 × 1.2 × 1.6 cm with solid cut surface [Figure 4]. On histopathological examinaion, specimen

showed biphasic population of epithelial and myoepithelial cells. The former was composed of glandular structures lined by oval round cells having hyperchromatic nuclei, pink cytoplasm with no mitotic figures or necrosis suggestive of benign PA of minor salivary glands of cheek. Based upon history, clinical examination, laboratory investigations, ultrasound examination and histopathological findings the final diagnosis of benign PA of minor salivary glands of left cheek was made. The post-operative period was uneventful. Patient was followed-up over a period of 3 years and no recurrence was observed.

DISCUSSION Tumors originating in the minor salivary glands are uncommon neoplasms of upper aero digestive tract. In 2002, Jansisyanont et al.,[6] suggested that a total of 80 minor salivary gland tumors were identified in 49 female patients and 31 male patients and the ratio range from 1.2:1 to

Figure 2: Intra oral photograph showing oval shaped submucosal nodule Figure 1: Extra oral photograph showing diffuse swelling of left cheek

Figure 3: Ultrasonography of the lesion showing hypoechoic, marginated mass Indian Journal of Dental Research, 25(1), 2014

Figure 4: Gross specimen measuring 2.2 × 1.2 × 1.6 cm with smooth, faintly lobulated surface 123


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Pleomorphic adenoma of cheek

1.9:1. A study conducted on a Mexican sample showed that in minor salivary gland tumors, 64.2% are benign neoplasms and only 35.8% were malignant.[7] PAs are benign heterogeneous tumors of salivary gland origin, which are made up of myoepithelial and epithelial components. In the registry of Armed Forces Institute of Pathology of 6880 cases, PA records only 126 cases of affecting minor salivary glands of cheek (1.8%).[8] This tumor of salivary glands is also known as benign mixed tumor. PA occurs commonly between the 3th and 5th decades with female predilection. The PA of minor salivary glands clinically present as painless, slow growing sub mucosal masses. The covering mucosa is seldom affected unless it is secondarily traumatized. The majority of intraoral mixed tumors are less than 3.0 cm in diameter. They are usually solitary and well-circumscribed. The findings of the case presented here is in agreement with those of other investigators.[3,4,9] The differential diagnosis of PA cheek includes buccal abscess, dermoid cyst, sebaceous cyst, neurofibroma, lipoma, mucoepidermoid carcinoma and polymorphous low grade adenocarcinoma.[10] The possibility of buccal space abscess was ruled out due to absence of sign of inflammation. The solid nature of lesion coupled with lack of tissue representing the three germ layers rule out the possibility of mature dermoid cyst. Absence of punctum and freely movable nature of the mass differentiate PA from sebaceous cyst. As on histological picture both epithelial and myoepithelial cells were seen, which rules out mucoepidermoid carcinoma. The negative slip test clinically and absence of lipomatous component histologically rules out lipoma. The absence of perineural invasion and mitotic figures obscure the chances of polymorphic low grade adenocarcinoma. There are three histological subtypes, myxoid (80% stroma), cellular (myoepithelial cells predominating) and mixed (classic). PA is known to produce recurrence either due to spillage, inadequate removal or enucleation at the time of operation, but is not known to produce distant metastasis. A recurrence rate of 2-44% has been reported in the literature. The ideal treatment of choice for PA is wide local excision with good safety margins and follows-up for at least 3-4 years.[9]

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CONCLUSION PA of the cheek is a rare neoplasm and therefore its diagnosis requires a high index of suspicion. PA should always be considered in the differential diagnosis of cheek masses. Complete wide surgical excision is the treatment of choice. Recurrence after many years of surgical excision as well as malignant transformation should be a concern and therefore long-term follow-up is necessary.

REFERENCES 1.

Traiger J, Rosen MB. Mixed tumor of the cheek; report of a case. Oral Surg Oral Med Oral Pathol 1965;19:711-4. 2. Wang D, Li Y, He H, Liu L, Wu L, He Z. Intraoral minor salivary gland tumors in a Chinese population: A retrospective study on 737 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;104:94-100. 3. Rao PK, Shetty SR, Hegde D. Ectopic pleomorphic adenoma. N Am J Med Sci 2012;4:190-2. 4. Dalati T, Hussein MR. Juvenile pleomorphic adenoma of the cheek: A case report and review of literature. Diagn Pathol 2009;4:32. 5. Pons Vicente O, Almendros Marqués N, Berini Aytés L, Gay Escoda C. Minor salivary gland tumors: A clinicopathological study of 18 cases. Med Oral Patol Oral Cir Bucal 2008;13:E582-8. 6. Jansisyanont P, Blanchaert RH Jr, Ord RA. Intraoral minor salivary gland neoplasm: A single institution experience of 80 cases. Int J Oral Maxillofac Surg 2002;31:257-61. 7. Ledesma-Montes C, Garces-Ortiz M. Salivary gland tumours in a Mexican sample. A retrospective study. Med Oral 2002;7:324-30. 8. Chidzonga MM, Lopez Perez VM, Portilla Alvarez AL. Pleomorphic adenoma of the salivary glands. Clinicopathologic study of 206 cases in Zimbabwe. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1995;79:747-9. 9. Aggarwal A, Singh R, Sheikh S, Pallagatti S, Singla I. Pleomorphic adenoma of minor salivary gland: A case report. RSBO 2012; 9:97-101. 10. Jorge J, Pires FR, Alves FA, Perez DE, Kowalski LP, Lopes MA, et al . Juvenile intraoral pleomorphic adenoma: Report of five cases and review of the literature. Int J Oral Maxillofac Surg 2002;31:273-5. How to cite this article: Verma P, Sachdeva SK, Verma KG, Sachdeva K. Pleomorphic adenoma of cheek: A rare case report and review of literature. Indian J Dent Res 2014;25:122-4. Source of Support: Nil, Conflict of Interest: None declared.

Indian Journal of Dental Research, 25(1), 2014