Pleomorphic adenoma involving minor salivary

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Pleomorphic adenoma involving minor salivary glands of upper lip: A rare phenomenon ABSTRACT Pleomorphic adenoma is the most common benign tumor of salivary glands, usually occurs in major salivary glands (mostly in parotid gland). It also affects the minor salivary glands present in the oral cavity. Most of the time it occurs in postero‑lateral part of palate, but the involvement of upper lip is rare. The present report describes a case of 55-year-old male with asymptomatic firm nodular swelling of upper lip which was later diagnosed as pleomorphic adenoma. KEY WORDS: Minor salivary glands, mixed tumor, pleomorphic adenoma, upper lip

INTRODUCTION Pleomorphic adenoma or benign mixed tumor is the most common salivary gland tumor and accounts for about 60% of all salivary neoplasms.[1] About 80% of pleomorphic adenomas arise in the parotid, 10% in the submandibular gland and 10% in the minor salivary glands of the oral cavity, nasal cavity and paranasal sinuses and the upper respiratory and alimentary tracts. Hard palate is the most common site among minor glands accounting for approximately 50–60%, followed by upper lip (15–20%) and buccal mucosa (8–10%). The affected patients are between 30–50 years of age. There is a slight female predilection.[2] The clinical presentation is as an asymptomatic, slowly growing firm mass. Small tumors typically form smooth, mobile, firm lumps but larger tumors tend to become bossellated and may attenuate the overlying skin or mucosa. Palatal tumors almost always are found on the postero‑lateral aspect at the junction of hard and soft palate, presenting as smooth‑surfaced, dome‑shaped masses. We present a rare case of pleomorphic adenoma of minor salivary glands present in the upper lip. CASE REPORT A 55-year-old male reported to us with a painless swelling in the central portion of the upper lip for last 1 year [Figure 1]. He had a history of injury over the upper lip due to fall 7 years ago. His past medical history was not suggestive. On clinical examination there was 2 × 1.5 cm firm, 1096

non‑tender, nodular, circumscribed lesion present in the philtrum area, obliterating its depression just adjacent to the columella‑philtral junction of the upper lip. Both the overlying skin and the underlying mucosa were normal and intact. There was no regional lymphadenopathy. A differential diagnosis of benign tumor of either minor salivary gland or mesenchymal origin was considered. The biochemical investigations were within normal limit. Under local anesthesia via intraoral approach the tumor was separated from the adjacent structures and excised [Figure 2]. The mass was well‑encapsulated and present in between the fibers of orbicularis oris muscle and was not‑invading the adjacent structures. On histopathological examination, the section revealed proliferation of polygonal and spindle shaped myoepithelial cells in a variable background stroma containing mucoid and myxoid areas at places. Epithelial elements arranged in duct‑like structures, sheets, clumps and/or interlacing strands and consist of polygonal, spindle or stellate shaped cells. The tumor is surrounded by a fibrous pseudocapsule of varying thickness and it extends through normal glandular parenchyma in the form of finger like pseudopodia [Figures 3 and 4]. This confirmed its diagnosis as pleomorphic adenoma. After 6 months of regular follow‑up no locoregional recurrence was observed.

Akhilesh Kumar Singh1,2, Naresh Kumar1,2, Parikshit Sharma3, Shreya Singh2 Department of Oral and Maxillofacial Surgery, 2Faculty of Dental Sciences, Institute of Medical Sciences, Banaras Hindu University, Varanasi, 3 Department of Oral and Maxillofacial Pathology, Faculty of Dental Sciences, King George’s Medical University, Lucknow, Uttar Pradesh, India 1

For correspondence: Dr. Akhilesh Kumar Singh, Department of Oral and Maxillofacial Surgery, Faculty of Dental Sciences, Institute of Medical Sciences, Banaras Hindu University, Varanasi ‑ 221 005, Uttar Pradesh, India. E‑mail: georgianaks@ gmail.com

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DISCUSSION Lips are the integral structure of lower third face that acts as a muscular sphincter guarding the Journal of Cancer Research and Therapeutics - October-December 2015 - Volume 11 - Issue 4


Singh, et al.: Pleomorphic adenoma of upper lip

Figure 1: Frontal view of the patient showing localized swelling present in the middle portion, obliterating the phitral depression of upper lip

Figure 2: The nodular mass present in the muscular bed (orbicularis oris) of the upper lip

Figure 3: H and E stained photomicrograph at ×10 showing thick fibrous–capsule surrounding the tumor tissue parenchyma

Figure 4: H and E stained photomicrograph at ×20 showing proliferation of duct like epithelial cells, polygonal shaped myoepithelial cells along with areas of myxoid, mucoid and eosinophilic substance mimicking osseous changes

opening of the oral cavity. It is made up of both epithelial and mesenchymal connective tissue components that is skin, adipose tissue, orbicularis oris muscle, minor salivary glands, blood vessels and nerves. Among both the lips, upper lip is more prone to developmental anomalies such as cleft lip whereas lower lip is more affected by pathological conditions like mucocele and other neoplastic conditions. Daley reviewed the clinical differential diagnosis of a swelling of the upper lip, listing mucocele, fibroma, lipoma, mucus retention cyst, sialolith, phlebolith and salivary gland neoplasm as possibilities. Extravasation mucoceles are usually the result of traumatic damage to a duct and obstruction to the drainage of a minor salivary gland. It is more common on lower lip. Fibromas vary in consistency from soft to very firm. Lipomas are soft and fluctuant, consisting of mature adipose tissue, are uncommon in the oral cavity, but can occur on lips. Sialoliths and phleboliths are also uncommon and usually appear opaque in the radiograph.[3]

Nasolabial cyst, which derive from epithelial remnants of the nasolacrimal duct, is another rare condition that may involve upper lip and nasolabial fold region.[4] Epidermoid and dermoid cysts of the head and neck constitute only about 7% of all such cysts. Some (25%) of these head and neck cysts develop in the floor of the mouth, with the remainder generally found in the tongue, lips, palate and jaws.[5] Few cases of dermoid and epidermoid cyst of upper lip have also been reported in literature.[6,7] In younger age, swellings of upper lip may be suspected as hemangiomas or lymphangiomas. Usually blue in colour, these blanch under digital pressure, which distinguishes them from pigmented lesions such as nevi, mucoceles, hematomas and melanomas.[8] Deshmukh et al., reported a rare occurrence of oral cysticercosis present as an aymptomatic nodule over the upper lip.[9] Among benign salivary gland pathologies, Canalicular adenoma almost always occurs in the upper lip, most often at the midline. Its histologic appearance is dominated by double rows

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of columnar epithelial cells, which branch and interconnect; peak incidence is in the seventh decade of life.[10] Pleomorphic adenoma is the second most common benign tumor of the upper lip.[11] Acinic cell adenocarcinoma is the most common malignant salivary gland neoplasm of the upper lip whereas mucoepidermoid carcinoma is more common in lower lip.[12] Palpation of submucosal swellings is necessary to differentiate between cyst and tumors. Usually cystic lesions such as mucocele exhibit fluctuance whereas solid masses are indicative of benign tumors. As pleomorphic adenoma is rare in this location, clinical examination and histopathology study is required for definitive diagnosis. The tumor is usually encapsulated, so surgical excision with adequate surrounding tissue margins is treatment of choice. Recurrence is rare in minor salivary gland tumors. REFERENCES 1. Spiro RH. Salivary neoplasms: Overview of a 35‑year experience with 2,807 patients. Head Neck Surg 1986;8:177‑184. 2. Eveson JW, Cawson RA. Salivary gland tumours. A review of 2410 cases with particular reference to histological types, site, age and sex distribution. J Pathol 1985;146:51‑8. 3. Daley TD. The canalicular adenoma: Considerations on differential diagnosis and treatment. J Oral Maxillofac Surg 1984;42:728‑30.

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4. Langlais RP, Miller CS. Nodules of the lip. Color atlas of common oral diseases. 1st ed. Philadelphia: Lippincott Williams and Wilkins; 1992. p. 32‑3. 5. Rontal E, Adams GL, Boies LR Jr. Cysts in the floor of the mouth. Minn Med 1971;54:829‑31. 6. Herlin C, Béziat JL, Koppe M, Nimeskern N, Gleizal A. Bilateral dermoid cysts of the upper lip. J Craniofac Surg 2011;22:2414‑5. 7. Kuroyanagi K, Kawabata T, Tooi M. Epidermoid cyst of the upper lip: Report of a case. Bull Tokyo Dent Coll 1973;14:95‑8. 8. Eversole LR. Red and pigmented lesions. Clinical outline of oral pathology: Diagnosis and treatment. 3rd ed. Philadelphia: Lea and Febiger; 1992. p. 31‑61. 9. Deshmukh A, Avadhani A, Tupkari J, Sardar M. Cysticercosis of the upper lip. J Oral Maxillofac Pathol 2011;15:219‑22. 10. Ellis GL, Auclair PL. Benign epithelial neoplasms. In: Atlas of tumor pathology, 3rd series fascicle 17: Tumors of the salivary glands. Washington: Armed Forces Institute of Pathology; 1995. p. 39‑153. 11. Ellis GL, Auclair PL, Gnepp DR. Mixed tumor (pleomorphic adenoma) and myoepithelioma. In: Surgical pathology of the salivary glands. Volume 25 of Major Problems in pathology. Philadelphia: W. B. Saunders Co.; 1991. p. 165‑86. 12. Ellis GL, Auclair PL, Gnepp DR. Salivary gland neoplasms: General considerations. In: Surgical pathology of the salivary glands. Volume 25 of Major Problems in pathology Philadelphia: W. B. Saunders Co.; 1991. p. 135‑64. Cite this article as: Singh AK, Kumar N, Sharma P, Singh S. Pleomorphic

adenoma involving minor salivary glands of upper lip: A rare phenomenon. J Can Res Ther 2015;11:1025. Source of Support: Nil, Conflict of Interest: None declared.

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