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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 13  |  Issue : 1  |  Page : 63-65

Pleomorphic adenoma of the cheek: Report of a rarity


Department of Oral and Maxillofacial Surgery, Sharad Pawar Dental College, Datta Meghe Institute of Medical Sciences, Wardha, Maharashtra, India

Date of Web Publication10-Sep-2018

Correspondence Address:
Dr. Akash P Kasatwar
Department of Oral and Maxillofacial Surgery Sharad Pawar Dental College, Datta Meghe Institute of Medical Sciences, Sawangi (Meghe), Wardha - 422 004, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jdmimsu.jdmimsu_108_17

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  Abstract 


Pleomorphic adenoma (PA) is the most common tumor of the salivary glands. Approximately, 90% of these tumors occur in the parotid gland and 10% in the minor salivary glands. The buccal mucosa or cheek is a rarely affected site by PAs of the minor salivary glands. Herein, we report a case of PA of the cheek, presented with intraoral swelling, and conclude that complete surgical excision can be a curative treatment for this benign tumor with 5-year follow-up.

Keywords: Benign tumor, cheek, minor salivary glands, pleomorphic adenoma


How to cite this article:
Fidvi MZ, Jadhav AA, Kasatwar AP, Borle RM. Pleomorphic adenoma of the cheek: Report of a rarity. J Datta Meghe Inst Med Sci Univ 2018;13:63-5

How to cite this URL:
Fidvi MZ, Jadhav AA, Kasatwar AP, Borle RM. Pleomorphic adenoma of the cheek: Report of a rarity. J Datta Meghe Inst Med Sci Univ [serial online] 2018 [cited 2019 Jun 26];13:63-5. Available from: http://www.journaldmims.com/text.asp?2018/13/1/63/240888




  Introduction Top


Pleomorphic adenoma (PA) is a mixed tumor of benign nature which presents as slow-growing, painless, firm swelling of glandular origin that does not cause ulceration.[1] The high variability in morphology of this neoplasm is the result of the interplay between these elements. Recently, it is widely accepted that both epithelial and mesenchymal (myxoid, hyaline, chondroid, and osseous) elements often arise from same cell clone, which may be a myoepithelial or ductal reserve cell.[2] Approximately, 8% of PA involves the minor salivary glands. Out of which, palate is the most common site (60%–65%).[3] The mucosa of the cheek is an uncommon site of occurrence with 5.5% incidence. The present report describes a case of PA arising in the buccal space, which underwent therapeutic wide local excision with wide margins. Although PA arising from the minor salivary glands rarely affects the buccal space, the condition should still be considered in the differential diagnosis of buccal mucosa masses.


  Case Report Top


A 40-year-old female patient reported to the Department of Oral Surgery with a complaint of painless swelling over the left cheek associated with difficulty in mastication for 6 months. On clinical examination, extraorally, single swelling of size approximately 3 cm × 3 cm was observed over the cheek region. A single left submandibular lymph node was palpable of size 1 cm × 1 cm approximately, roughly oval, firm, mobile, and nontender in nature. Intraorally, a domed-shaped swelling was present on the left buccal mucosa extending anterio-posteriorly from the left corner of mouth to second molar region and superio-inferiorly from 1 cm above the occlusal plane to the depth of lower vestibule which was roughly round, smooth, color was same as that of the adjacent mucosa, with well-defined margins. Indentation of opposing teeth was seen over swelling [Figure 1].
Figure 1: Intraoral swelling over the left cheek mucosa

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The differential diagnosis included buccal abscess, dermoid cyst, sebaceous cyst, neurofibroma, lipoma, mucoepidermoid carcinoma, and polymorphous low-grade adenocarcinoma.[4]

All the necessary blood investigations were done; on ultrasonography, there is well-defined, round-to-oval shape, mixed echogenic mass lesion seen in the left cheek, possibly in the submucosal region, size 27 mm × 20 mm F/S/O benign neoplastic etiology versus malignant etiology lesion. Fine-needle aspiration cytomorphology was performed which was S/O chronically infected submucosal cyst, nonmalignant cells. Since the lesion was small in size, excision of the lesion was planned. A horizontal incision was given over the prominence of the swelling parallel to the Stenson's duct, submucosal blunt dissection was carried out, and tumor mass was excised [Figure 2] and sent for histopathological evaluation. On histopathological examination, specimen showed glandular structures lined by oval-to-round cells having hyperchromatic nuclei, pink cytoplasm with no mitotic figures, or necrosis suggestive of benign PA of minor salivary glands of left buccal mucosa [Figure 3] and [Figure 4].
Figure 2: Intraoperative exposure of the tumor mass

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Figure 3: Excised tumor mass

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Figure 4: Histomicrograph showing cuboidal to low columnar cells and hyalinized connective tissue stroma

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  Discussion Top


PAs are benign heterogeneous tumors of salivary gland origin. Salivary gland tumors represent <5% of all head-and-neck tumors, two-thirds of these tumors are PAs.[3] PA is the most common salivary gland tumor that affects both major and minor salivary glands. In head-and-neck region, they arise mainly in the major salivary glands mostly parotid (85%), submandibular gland (8%), and intraoral (7%) salivary glands.[5] Among the minor salivary glands, palatal glands are the most commonly affected with PA with a frequency ranging from 43% to 70% followed by the upper lip (10.1%) and cheek (5.5%). Other rare sites include the throat (2.5%), retromolar region (0.7%), floor of the mouth, and the alveolar mucosa.[6] In 2002, Jansisyanont et al.[7] 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 1.9:1. PAs are benign heterogeneous tumors of salivary gland origin, which are made up of myoepithelial and epithelial components. In the registry of the Armed Forces Institute of Pathology of 6880 cases, PA records only 126 cases of affecting minor salivary glands of the cheek (1.8%).[8] PA occurs commonly between the 3rd and 5th decades with female predilection. Etiology of PA is still obscure. However, PA is described to have an epithelial origin, along with clonal chromosome abnormalities with genetic aberrations involving 8q12 and 12q15 in the literature.[9] The PA of minor salivary glands clinically present as painless, slow-growing submucosal masses. The covering mucosa is seldom affected unless it is secondarily traumatized. A majority of intraoral mixed tumors are <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.[6],[10] Due to the absence of sign of inflammation, buccal space abscess was ruled out. Dermoid cyst was ruled out because of the solid nature of lesion coupled with lack of tissue representing the three germ layers. Sebaceous cyst was differentiated with PA as there was the absence of punctum and freely movable nature of the mass. As on histological picture, both epithelial and myoepithelial cells were seen, which rules out mucoepidermoid carcinoma. The negative slip test clinically and the absence of lipomatous component histologically rules out lipoma. The absence of perineural invasion and mitotic figures obscure the chances of polymorphic low-grade adenocarcinoma.

PA is known to produce recurrence either due to the spillage, inadequate removal, or enucleation, at the time of operation, but is not known to produce distant metastasis. Local recurrence has been attributed to microscopic interruptions in the pseudocapsule, incomplete excisions, or rupture of the capsule causing spillage of the tumor content during excision. 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 as PA has malignant potential, hence regular follow-up for at least 3–4 years has been recommended.


  Conclusion Top


Given the rarity of the lesion, a careful consideration is warranted in the differential diagnosis of slow-growing, painless cheek masses. Complete surgical excision is the treatment of choice. Recurrence after many years of surgical excision and malignant transformation as well should be a concern, long-term follow-up is necessary.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Dalati T, Hussein MR. Juvenile pleomorphic adenoma of the cheek: A case report and review of literature. Diagn Pathol 2009;4:32.  Back to cited text no. 1
    
2.
Lee PS, Sabbath-Solitare M, Redondo TC, Ongcapin EH. Molecular evidence that the stromal and epithelial cells in pleomorphic adenomas of salivary gland arise from the same origin: Clonal analysis using human androgen receptor gene (HUMARA) assay. Hum Pathol 2000;31:498-503.  Back to cited text no. 2
    
3.
Greenberg M, Glick M. Burket's Oral Medicine. 11th ed. Philadelphia: Lippincott; 2008. p. 172.  Back to cited text no. 3
    
4.
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.  Back to cited text no. 4
    
5.
Bakshi PS, Jindal N, Roy S, Khanna K. Pleiomorphic adenoma of buccal mucosa: A rare case. OHDM 2015;14.  Back to cited text no. 5
    
6.
Geetha NT, Deepa BV, Umashankara KV, Kithikumar R. Pleomorphic adenoma of minor salivary gland in the cheek. Int J Oral Health Sci 2015;5:117-20.  Back to cited text no. 6
  [Full text]  
7.
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.  Back to cited text no. 7
    
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.  Back to cited text no. 8
    
9.
Farina A, Pelucchi S, Grandi E, Carinci F. Histological subtypes of pleomorphic adenoma and age-frequency distribution. Br J Oral Maxillofac Surg 1999;37:154-5.  Back to cited text no. 9
    
10.
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.  Back to cited text no. 10
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