Pleomorphic adenoma of the cheek: a case presentation
The Egyptian Journal of Otolaryngology volume 38, Article number: 165 (2022)
Pleomorphic adenoma, also known as a benign mixed tumor, is the most common salivary gland tumor. The parotid gland accounts for 90% of the total, with the minor salivary glands accounting for 10%. Buccal minor salivary glands pleomorphic adenoma is extremely rare. It manifests as a painless, firm, slow-growing mass in most cases.
A pleomorphic adenoma in the buccal minor salivary glands was discovered in an adult 43-year-old female patient treated with wide surgical resection. A literature review of the PA of the cheek is stated.
In the differential diagnosis of cheek masses, pleomorphic adenoma should be scrutinized. The remedy of choice is wide local excision with at least a 5-year follow-up.
Salivary gland tumors are a broad group of tumors with a variety of clinical, histological, and immunohistochemical features . These tumors are extremely rare, accounting for 3% to 5% of all neoplastic processes in the jaws . They are benign neoplasms in 64.9% to 67.5% of cases [3, 4]. Minor salivary gland neoplasms, including those of the cheek mucosa, lip, and tongue, are exceptionally rare [3,4,5].
Pleomorphic adenoma (PA) is the most common benign salivary gland tumor, accounting for 33.2% to 68.4% of all cases [4, 5]. The parotid glands are the most affected, while the cheek, lip, and tongue mucosa are rarely affected [2, 4,5,6]. Females between the ages of 40 and 50 are more prone to get PA [4,5,6,7].
PA is a clinically well-defined, slow-growing, asymptomatic lesion with hard consistency and varied diameters [8, 9]. The majority of intraoral PA is solid or rubbery and is found in the submucosa. Although ulcerations are seen in certain cases, the mucosal lining stays intact [8, 10].
PA can clinically mirror other salivary gland reactive diseases and nonneoplastic proliferative processes in these intraoral locations sensitive to stress, such as the cheek mucosa, lip, and tongue.
A rare case of PA in the cheek is presented. PA’s etiology, clinical and morphological characteristics, ad differential diagnoses are introduced in this context.
A 43-year-old female patient presented to Otorhinolaryngology Department, Kafrelsheikh University Hospital, complaining of a painless, slowly growing tumor in his right cheek 12 years ago.
A swelling of 6 × 2 cm in the buccal mucosa on the right side of the face was detected; it was mildly uncomfortable and freely mobile over the underlying structures and firm in consistency (Fig. 1). We could easily palpate the mass under the skin. In addition, it was mobile, nonreducible, and unattached to the underlying structures.
The color and texture of the cheek mucosa were both normal on inspection. The cranial nerves examination was revealed to be normal.
The patient had a normal mouth opening. The lesion could easily be felt between the cheek mucosa and the skin with bimanual palpation. It was firm and slippery, with a slightly irregular surface. It was not fixed to the tissue around it. The lymph nodes examination was normal, with no enlargement.
A CT scan of the face revealed a well-defined mass in the right buccal space, not invading the neighboring structures (Fig. 1). The clinical assessment of the swelling has straightforwardly directed us to a provisional diagnosis of “fibrolipoma.”
After the patient approved appropriate consent for the surgery, we planned it with the knowledge that the lesion was mostly benign and quite superficial (Fig. 2). Ethical Committee approval of the institute was waived.
The surgical operation was under general anesthesia under aseptic conditions for wide local excision of the mass and then a sub-labial approach closure of neighboring mucosa.
The tumor was simply removed. The mass instantly burst out with no attachment to neighboring structures once the mucosa and muscle layer were dissected. The removed mass measured 6 × 2.3 × 1.5 cm and was firm and rubbery in texture (Fig. 3).
The surgical wound was closed in layers with 3/0 vicryl absorbable sutures after the mass removal (Fig. 4). Histopathological examination was done for the specimen.
The histopathological report revealed the presence of glandular epithelium and mesenchymal tissue and changes like mucoid material accumulation, hyalinization foci, and an epithelial component made up of myoepithelial cells arranged in cords and nests, in accordance with pleomorphic adenoma characteristics (Fig. 5). Postoperative follow-up for 5 years revealed no recurrence.
Pleomorphic adenoma (PA) is considered the most common benign salivary gland tumor . Willis  created the name Pleomorphic adenoma. The World Health Organization (WHO) categorized PA in 1972 as “a circumscribed tumor characterized by clearly visible epithelial tissue interspersed with the tissue of mucoid, myxoid, or chondroid appearance” . It strikes during the third and sixth decades of life, with a minor proclivity for females with a female-to-male ratio of 2:1 . The term “mixed tumor” is deceptive , despite the fact that it is made up of mesenchymal and epithelial cells . The parotid gland is frequently affected, particularly the lower pole of the superficial lobe. The minor salivary glands of the mouth are involved in 8% of PAs. Sixty to 65% of the salivary glands in the mouth  and 5.5% in the minor salivary glands of the cheek  are involved.
The clinical features of the lesion are consistent with those described in the literature, including an asymptomatic, slow-growing nodular lesion of firm consistency with normal intact mucosa [8,9,10]. Based on the characteristics of the mass as being superficial and easily accessible by excisional biopsy, we did not recommend Fine Needle Aspiration Cytology (FNAC) of the lesion preoperatively. However, FNAC is still a precise and helpful tool for distinguishing benign from malignant tumors and establishing the ideal treatment strategy.
The histopathological report of the case described the interaction of epithelial components clustered in duct-like structures with myoepithelial cells in the stroma with various patterns such as mucoid, myxoid, cartilaginous, or hyaline. These findings coincide with PA's microscopic features [8, 15].
The tumor under inquiry has a wide range of histological features that reflect the polymorphic pattern seen in salivary gland neoplasms. The histology of these malignancies, which is linked to the type of cell, handles a wide range of histomorphology [16, 17]. PA’s histogenesis is linked to the intercalated duct’s reserve cells. The ability of reserve cells to develop into multiple subtypes points out the morphological variability seen in salivary gland neoplasms, which can even differ within the same tumor .
Soft-tissue neoplasms, like neurilemoma [8, 10] neurofibroma, lipoma [8, 14] neoplasms of minor salivary glands, may also be considered for the differential diagnosis , in addition to inflammatory and reactive lesions [8,9,10].
Finally, while developing diagnostic hypotheses is critical, the significance of making definitive scrutiny is emphasized. Whereas the comprehensive look at a lesion can imply a variety of theories, the anatomopathological investigation is the only way to figure out the definitive findings. The recommended treatment for PA is surgical excision with preservation of the capsule and a perimeter of normal tissue around it [8,9,10, 14, 15, 19,20,21]. PA should be diagnosed as soon as feasible because, despite its rarity [19, 22, 23], later malignant change to carcinoma ex pleomorphic adenoma might occur [7, 11, 21,22,23]. Consequently, in our study, we followed up the case for 5 years with no recurrence.
Pleomorphic adenoma of the minor salivary gland, particularly when involving the cheek, is a rare lesion that can be difficult to detect, even for the most experienced surgeons. In the differential diagnosis of cheek swellings, it should be considered.
The recommended treatment is the surgical removal of the tumor with adequate safety margins. As a result, early detection and treatment, as well as regular follow-up for at least 5 years, are critical.
Availability of data and materials
Cavalcante RB, Nonaka CFW, Rabenhorst SHB, Miguel MCC, Pinto LP, de BatistaSouza L (2017) Pleomorphic adenoma and adenoid cystic carcinoma of salivary glands: E-cadherin immunoexpression and analysis of the CDH1-160C/A polymorphism. Arch Oral Biol 73:48–54. https://doi.org/10.1016/j.archoralbio.2016.09.005
Khandekar VS, Dive A, Munde P, Wankhede ND (2015) Pleomorphic adenoma of the buccal salivary gland. J Oral Maxillofac Pathol 19:111–116. https://doi.org/10.4103/0973-029X.157222
Ito FA, Ito K, Vargas PA, Almeida OP, Lopes MA (2005) Salivary gland tumors in a Brazilian population: a retrospective study of 496 cases. Int J Oral Maxillofac Surg 34:533–536. https://doi.org/10.1016/j.ijom.2005.02.005
Jones AV, Craig GT, Speight PM, Franklin CD (2008) The range and demographics of salivary gland tumors diagnosed in a UK population. Oral Oncol 44:407–417. https://doi.org/10.1016/j.oraloncology.2007.05.010
Pires FR, Pringle GA, Paes de Almeida O, Chen S (2007) Intra-oral minor salivary gland tumors: a clinicopathological study of 546 cases. Oral Oncol 43:463–470. https://doi.org/10.1016/j.oraloncology.2006.04.008
Abrahão AC, Santos Netto JN, Pires FR, Santos TC, Cabral MG (2016) Clinicopathological characteristics of tumors of the intraoral minor salivary glands in 170 Brazilian patients. Br J Oral Maxillofac Surg 54:30–34. https://doi.org/10.1016/j.bjoms.2015.10.035
Kiciński K, Mikaszewski B, Stankiewicz C (2016) Risk factors for recurrence of pleomorphic adenoma. Otolaryngol Pol 70:1–7. https://doi.org/10.5604/00306657.1193691
Rahnama M, Orzędała-Koszel U, Czupkałło L, Lobacz M (2013) Pleomorphic adenoma of the palate: a case report and review of the literature. Contemp Oncol (Pozn) 17:103–106. https://doi.org/10.5114/wo.2013.33438
Singh AK, Kumar N, Sharma P, Singh S (2015) Pleomorphic adenoma involving minor salivary glands of the upper lip: A rare phenomenon. J Cancer Res Ther 11:1025. https://doi.org/10.4103/0973-1482.148682
Qureshi MY, Khan TA, Dhurjati VN, Gaddikeri K, Khany MZ (2016) Pleomorphic adenoma in the retromolar area: a very rare case report and review of the literature. J Clin Diagn Res 10:ZD03–ZD05. https://doi.org/10.7860/JCDR/2016/16269.7067
Ito FA, Jorge J, Vargas PA et al (2009) Histopathological findings of pleomorphic adenomas of the salivary glands. Med Oral Patol Oral Cir Bucal 14(2):57–61 PMID: 19179950
Rajendran S, Sivapathasundaram S (2009) Shafer's Textbook of Oral Pathology. 6th edition. Elsevier, India, 219–24
Verma P, Sachdeva SK, Verma KG et al (2014) Pleomorphic adenoma of cheek: a rare case report and review of the literature. Indian J Dent Res 25:122–124. https://doi.org/10.4103/0970-9290.131166
Rivera-Bastidas H, Ocanto RA, Acevedo AM (1996) Intraoral minor salivary gland  tumors a retrospective study of 62 cases in a Venezuelan population. J Oral Pathol Med 25:1–4. https://doi.org/10.1111/j.1600-0714.1996.tb01214.x
Neville BW, Damm DD, Allen CM, et al. (2009) Oral & Maxillofacial Pathology. 3 rd ed. St. Louis, Saunders Elsevier. 477–79. https://doi.org/10.7860/JCDR/2015/14035.6770
Toida M, Shimokawa K, Makita H et al (2005) Intraoral minor salivary gland tumors: a clinicopathological study of 82 cases. Int J Oral Maxillofac Surg 34:528–532. https://doi.org/10.1016/j.ijom.2004.10.010
Dwivedi N, Agarwal A, Raj V, Chandra S (2013) Histogenesis of salivary gland neoplasms. Indian J Cancer 50:361–366. https://doi.org/10.4103/0019-509X.123629
Kadeh H, Derakhshanfar G, Saravani S (2016) Comparative Study of Mast Cell Count in Oral Reactive Lesions and Its Association with Inflammation. Turk Patoloji Derg 32:22–26. https://doi.org/10.5146/tjpath.2015.01338
Goyal P, Sehgal S, Ghosh S, Agrawal D, Singh S (2016) Rare carcinoma ex pleomorphic adenoma of buccal mucosa: case report and review of the literature. Rare Tumors 8:11–13. https://doi.org/10.4081/rt.2016.6138
Laturiya R, Kasim JS, Jankar AS, Mohiuddin SA (2016) Pleomorphic adenoma of minor salivary gland arising de novo in the parapharyngeal space - a rare case report. J Clin Diagn Res 10:ZD01–ZD03. https://doi.org/10.7860/JCDR/2016/18435.7356
Misra S, Bhandari A, Misra N, Gogri P, Mahajan S (2016) Pleomorphic adenoma of a deep orbital ectopic lacrimal gland. Orbit 35:295–297. https://doi.org/10.1080/01676830.2016.1193526
Kini Y, Desai C, Mahindra U, Kalburgue J (2013) Rare carcinoma ex pleomorphic adenoma of the buccal minor salivary gland causing a therapeutic dilemma. Contemp Clin Dent 3:209–211. https://doi.org/10.4103/0976-237X.96832
Mariano FV, Egal ES, Pramio D, Fidalgo F, Sara É, Costa AF et al (2016) Evaluation of a subset of tumor suppressor gene for copy number and epigenetic changes in pleomorphic adenoma and carcinoma ex-pleomorphic adenoma carcinogenesis. Oral Surg Oral Med Oral Pathol Oral Radiol 122:322–331. https://doi.org/10.1016/j.oooo.2016.05.002
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Abdelhamid, A.S., Elzayat, S., Essa, A.A. et al. Pleomorphic adenoma of the cheek: a case presentation. Egypt J Otolaryngol 38, 165 (2022). https://doi.org/10.1186/s43163-022-00352-5