Pleomorphic adenomas are benign salivary gland tumors that represent about 3-10% of the neoplasm of the head and neck region. PA is the most common benign neoplasm of the salivary glands. The palate is considered as the most common intraoral site (42.8-68.8%), followed by the upper lip (10.1%) and cheek (5.5%). Other rare sites include the throat (2.5%), retromolar region (0.7%), ?oor of the mouth and the alveolar mucosa [1-2]. PA usually presents as a mobile slowly growing, painless ?rm swelling that does not cause ulceration of the overlying mucosa that does not cause ulceration of the overlying mucosa [3]. The mucosa of cheek is an uncommon site of occurrence for intraoral pleomorphic adenoma and most of these cases have been reported in adults [4]. Histopathological elements of the PA include: capsule, epithelial and myoepithelial cells, and mesenchymal or stromal elements (Barnes). The aetiology of PA’s is unknown; however, some authors mention the myoepithelial cell are responsible for the development of this pathology (Ledesma-Montes). Seifert (1976) mentioned that PA’s can be divided according to the proportion of cellular and stromal elements within the tumor, and therefore be classified as myxoid, classic or cellular [5]. In children, very few cases have been reported to date. Here, we report a case of PA in a 13-year-old male.

A 13 years old male patient reported to the OPD, Private clinic with a chief complaint of swelling on upper left cheek since past 2 years. Patient also had a history of cheek biting. During the same duration patient noticed a swelling occurring in the cheek region, after which he discontinued the habit. The swelling persisted even after discontinuing the habit. There was no history of pain, bleeding, or irritation.

Figure 1: Clinical intra oral view with visible swelling over left cheek mucosa, overlying mucosa with no abnormality in colour or texture noted. 1x1cm, 0.2x0.2cm, 0.3x0.3xm approx.

The intraoral examination on palpation findings were swelling was found to be non-tender, firm in consistency, fluctuant, mobile, having a smooth surface and with the colour same as the adjacent mucosa. Swelling was not appreciable extra orally. The laboratory tests Hb, CBC, ESR, CT, BT were investigated and findings were unremarkable. The di?erential diagnosis was Mucocele and Focal fibrous hyperplasia (fibroma). Excision biopsy was advised as an investigation. The patient was scheduled for surgical excision of the lesion. Consent was taken. Surgical excision was under the effect of the infiltration with 2 % lidocaine plus epinephrine 1:100,000.

Figure 2: Excision biopsy was advised as an investigation.

Proceed and removed minor salivary gland with laser (PICASSO DENTAL LASER-AMD lasers) used laser in 3.0 Walt, Coagulate the area, Remove any remaining overhanging accessory salivary gland. Transfer the specimen to formalin container and submitted to pathology department for examination.

Figure 3: Showing the gross specimen which was submitted to Pathology department for HP examination.

Histopathological examination revealed well encapsulated mass of epithelium circumscribing connective tissue. Epithelium appears to be glandular in origin forming duct like structure with eosinophilic coagulum within. Tumor cells are present in the form of sheets, cords and nest of cells with atypical features (Figure 4). Connective tissue stroma shows myxoid areas (Figure 5) and endothelial lined blood vessels.

Figure 4: Sheets and cords of cells.

Figure 5: Myxoid areas.

Figure 6: postoperative reassessment A) 30 days, B) 60 days, C) 90 days postoperative reassessment.

Thus, diagnosis of PA of minor salivary gland was made. The patient was followed-up over a period of 3 months and no recurrence was observed.

For salivary gland tumors in children, it has been suggested that they have the proclivity to be malignant than to be benign regardless of the site. Mucoepidermoid carcinoma is the most common malignant salivary gland tumor, while pleomorphic adenoma is the most common benign counterpart [9]. Pleomorphic adenomas do not usually present sexual predisposition and they can appear at any age with same clinical behavior. They are generally round, slow growing tumors that are painless and firm in consistency [11]. Salivary gland tumors are rare in children and when they do arise, they preferentially a?ect major salivary glands, but minor salivary gland tumors have also been reported. The etiology of pleomorphic adenoma is not known [8]. The mean age at initial presentation of 42 patients treated by Peter Zbaren mentioned that age of recurrence observed was 34 years and the interval between initial surgery and tumor recurrence varied between 3-15yrs [2-4]. Yamamoto et al. reported a 9-year-old Japanese girl with PA of the cheek mucosa. Cohen and Kronenberg reported two more cases of juvenile PAs of the cheek (girls, age <18 years) [5-6]. Histopathological features of pleomorphic adenoma are a variable pattern of epithelium in a loosely fibrous stroma which may be myxoid, chondroid or mucoid. The epithelium is usually arranged in sheets or strands, and ductal structures, often bilayered, are typical. The main histologic subgroups have been identified: myxoid (80% stroma) endothelial lined blood vessels [12]. The differential diagnosis of the juvenile PA of the cheek includes buccal space abscess, dermoid cyst, foreign body reaction, fibroma, lipoma, neurofibroma, rhabdomyosarcoma, mucoepidermoid carcinoma, adenoid cystic carcinoma, polymorphous low-grade adenocarcinoma and carcinoma ex-PA [7]. Renehan A has suggested adjuvant radiotherapy for Pleomorphic multi nodular recurrent case. On the contrary Glas AS 2001 have stated that only when surgery is contraindicated for medical reasons should radiotherapy be even considered. Samson MJ 1991 have suggested that radiation may be justified if surgical risk of sacrificing the adjunct nerve is relatively high [1-2]. The surgical treatment for the PA in both juvenile and adult patients is principally the same and includes complete wide surgical excision with good safety margins. Treatment for PA is excision with normal margins due to high recurrence rate and malignant potential. The reason for recurrence is rupture of tumor capsule or incomplete excision during the procedure and leaving behind live residual tumor cells. The recurrence rate of PA is 2–44%. PA has excellent prognosis with a cure rate of 95%. Close follow?up is needed, so our case is also being followed?up due to risk of recurrence [10].

To conclude, this case report alerts the clinicians about the unusual causes of intraoral swellings. As the swelling is usually asymptomatic, it may be discovered on routine oral examination. Biopsy and histopathological study are the gold standard to diagnose this pathology. PA of the cheek is rare and should be considered in the differential diagnosis of intraoral swellings of the buccal mucosa. 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.

1. Pillai A, Satpathy M, Nahar S, Moghe S. Pleomorphic Adenoma in Cheek: An Uncommon Finding. IJSS Case Reports and Reviews. 2014; 1.
2. Gupta MK, Bailoor DN, Mhaske S, Raghuvanshi V, Nahar S, Ragavendra RT. Juvenile Pleomorphic Adenoma of the Cheek - A rare case with review of literature, IOSR Journal of Dental and Medical Sciences. 2013; 6: 45-48.
3. Dalati T, Hussein MR. Juvenile pleomorphic adenoma of the cheek: a case report and review of literature. Diagnostic pathology. 2009; 4: 32.
4. Kumar JK, Anilkumar MG, Krishna Kumar HC, Maggad R. Pleomorphic adenoma of the cheek in a child: A case report. Dent Res J. 2014; 11: 522-524.
5. Contreras W, Fernandez C. Palatal Pleomorphic Adenoma in a Pediatric Patient: Case Report. Int J Odontostomat. 2017; 11: 147-150.
6. Cohen MA. Pleomorphic adenoma of the cheek. Int J oral and maxillofacial surgery. 1986; 15: 777-779.
7. Cerulli G, Renzi G, Perugini M, Becelli R. Differential diagnosis between adenoid cystic carcinoma and pleomorphic adenoma of the minor salivary glands of palate, J Craniofac Surg. 2004; 15: 1056-1060.
8. AlShehri AA, Al-Qarni SN, Hudise JY, Sumaily IA. Juvenile buccal pleomorphic adenoma: a rare case report. Int J Otorhinolaryngology and Head and Neck Surgery. 2017; 3: 431-433.
9. Dhanuthai k, Sappayatosok k, Kongin k. Pleomorphic adenoma of the palate in a child: A case report. Med Oral Patol Oral Cir Bucal. 2009; 14: 73-75.
10. Thomas DC, Nair VV, Thomas S. Pleomorphic adenoma: An unusual presentation on upper lip ? A case report. J Indian Acad Oral Med Radiol. 2017; 29: 217?219.
11. Ahuja G, Marya J, Sood P. Pleomorphic adenoma of minor salivary gland in buccal mucosa : An uncommon finding. DJAS. 2015; 3: 47-50.
12. Mukul SK, Kumar A, Mokhtar EA, Pandey S. Pleomorphic adenoma of buccal mucosa: An uncommon presentation among cheek swellings, Indian J Anatomy and Surgery of Head, Neck and Brain. 2017; 3: 48-50.