Bilateral pleomorphic adenoma of the anterior tongue: a case report.

Abstract

We report a case of bilateral pleomorphic adenoma of the anterior two-thirds of the tongue in a 38-year-old man. Both tumors were excised via a transoral route. To the best of our knowledge, this is the first reported case of a bilateral pleomorphic adenoma of the anterior tongue.

Introduction

Benign pleomorphic adenomas of the tongue are not common; they have been reported to account for 0 to 2.5% of all tumors of the minor salivary glands. (1-3) The tongue ranks fourth behind the palate, lips, and buccal mucosa with respect to the incidence of benign minor salivary gland neoplasms. (3) Epker and Henny reviewed 90 cases of intraoral minor salivary gland tumors; they described 20 pleomorphic adenomas, none of which involved the tongue. (4) In a report by Eveson and Cawson, only 1 of 143 pleomorphic adenomas involved the tongue. (3)

In this article, we report our experience with a case of bilateral pleomorphic adenoma of the tongue. To the best of our knowledge, a case of bilateral pleomorphic adenoma of the anterior tongue has not been previously reported in the literature.

Case report

A 38-year-old man presented to the outpatient ENT clinic with an 11-month history of bilateral swelling of the tongue and muffled phonation. The lesions had been slowly progressive until the mass on the left began to grow rapidly during the preceding 3 weeks. The patient reported no pain, dysphagia, or paresthesia, and he had no history of trauma to the tongue.

Local examination revealed a firm, submucosal, 3 x 2cm mass in the left anterior two-thirds of the tongue and a similar 2 x 1-cm mass on the right (figure 1). Findings on the remainder of the ENT examination and a systemic examination were unremarkable. No palpable lymphadenopathy was present. The results of routine hematologic and biochemical tests were within normal ranges. Fine-needle aspiration cytology specimens were obtained from both areas of swelling; analysis of the aspirate revealed features of pleomorphic adenoma. The patient could not afford computed tomography (CT), and it was not performed.

[FIGURE 1 OMITTED]

A transoral surgical excision under general anesthesia was planned. A wide local excision of the mass on the left side was followed by a wide excision on the right. Bleeding was minimal. Because the airway was not compromised, tracheotomy was not necessary. No postoperative complications occurred.

Naked-eye examination of the excised lesions showed them to be irregularly shaped, gray-brown, soft-tissue masses. Cut sections were gray-brown. Histopathologic examination revealed that the tumors were made up of trabeculae, sheets, and cords of monomorphic epithelial cells with moderately eosinophilic cytoplasm (figure 2). These cells did not exhibit any features of atypia. The background was characterized by a scant myxoid matrix. These findings confirmed a diagnosis of pleomorphic adenoma.

[FIGURE 2 OMITTED]

At the 3-month follow-up, the patient was free of recurrence and was leading a normal life.

Discussion

Most salivary gland tumors of the adult tongue are malignant. Goldblatt and Ellis studied 55 salivary gland tumors of the tongue and found that 50 were malignant and 5 were benign. (5) All 5 of these benign tumors were pleomorphic adenomas.

Patients with tumors of the tongue can present with dysphagia, pain, muffled phonation, and/or airway obstruction, depending on the size and location of the mass.

The incidence of pleomorphic adenomas peaks during the third decade of life; such tumors have been reported in patients as young as 5 years and as old as 84 years. (1)

Diagnosis is relatively simple because it is easy to inspect the oral cavity. Diagnosis can be confirmed by fine-needle aspiration cytology and histopathology. Imaging studies--particularly CT and magnetic resonance imaging (MRI)--provide useful information on the nature of the lesion, its extent, and any involvement of surrounding structures. MRI is preferred because of its better resolution of soft tissues.

Surgery under general anesthesia (via nasal intubation) is the treatment of choice. The surgical approach depends on the size, site, and extent of the tumor. Surgical options include transoral, midline transhyoid, and lateral pharyngotomy approaches. Care must be taken to preserve the hypoglossal and lingual nerves. Use of the C[O.sub.2] laser is becoming more widespread for resection.

In elderly patients, malignant transformation of benign tumors of the minor salivary glands must always be considered.

References

(1.) Chaudhry AP, Vickers RA, Gorlin RJ. Intraoral minor salivary gland tumors: An analysis of 1,414 cases. Oral Surg Oral Med Oral Pathol 1961;14:1194-1226.

(2.) Chaudhry AP, Labay GR, Yamane GM, et al. Clinico-pathologic and histogenetic study of 189 intraoral minor salivary gland tumors. J Oral Med 1984;39:58-78.

(3.) Eveson JW, Cawson RA. Tumours of the minor (oropharyngeal) salivary glands: A demographic study of 336 cases. J Oral Pathol 1985;14:500-9.

(4.) Epker BN, Henny FA. Clinical, histopathologic, and surgical aspects of intraoral minor salivary gland tumors: Review of 90 cases. J Oral Surg 1969;27:792-804.

(5.) Goldblatt LI, Ellis GL. Salivary gland tumors of the tongue. Analysis of 55 new cases and review of the literature. Cancer 1987;60: 74-81.

Ramandeep S. Virk, MS; Harbir Hundal, MS

From the Department of Otolaryngology--Head and Neck Surgery, Virk Indus Advanced ENT Centre, Mohali (Dr. Virk), and the Department of Otolaryngology-Head and Neck Surgery, Sohana Multispecialty Hospital, Sohana, Punjab, India (Dr. Hundal).

Reprint requests: Dr. Ramandeep S. Virk, House No. 2168, Sector 15C, Chandigarh 160015, India. Phone: [phone]; fax: [phone]; e-mail: [e-mail]