Salivary gland cystadenocarcinoma of the mobile tongue, low-grade papillary adenocarcinoma variant: a case report.

Abstract

Cystadenocarcinoma is a rare malignant neoplasm of the salivary gland. Treatment involves local excision; neck dissection and postoperative radiation therapy are used for clinically positive neck metastases. The prognosis varies according to the clinical stage and grade of the tumor.

Introduction

While squamous cell carcinoma is certainly the most common malignant oral cavity tumor, one must always be cognizant of a wide variety of less common pathologies, including minor salivary gland tumors. We describe the case of a patient with a rare variant of a minor salivary gland tumor of the mobile tongue.

Case report

A 63-year-old woman presented with a left tongue nodule. She had first noticed the nodule several months earlier when she accidentally bit her tongue. Since that time, she had not noticed any change in the size or consistency of the nodule. She denied pain, weight loss, fevers, chills, and night sweats, and she had no history of tobacco or alcohol intake. The results of the head and neck examination were unremarkable except for a submucosal lesion in the left lateral oral tongue. The 5 x 3-mm lesion was mobile, it appeared to be free of the underlying tongue musculature, and there was no alteration of the overlying mucosa.

The patient underwent an excisional biopsy of the lesion. Analysis of the pathology sections revealed that the lesion had arisen from salivary ductal epithelium; intraductal papillary formations were observed (figure). No evidence of lymphatic, perineural, or vascular invasion was seen. These findings were consistent with a salivary gland cystadenocarcinoma of the mobile tongue, low-grade papillary adenocarcinoma variant.

[FIGURE OMITTED]

Discussion

Cystadenocarcinoma is a rare malignant neoplasm of the salivary glands. In the minor salivary glands, the most common location is the lip; other areas of involvement are the buccal mucosa, palate, tongue, retromolar area, and floor of the mouth. (1) Most patients present with a slowly growing mass. (2) Depending on the grade, 5 to 17% of patients have regional lymph node involvement at the time of diagnosis. (1,3) Approximately 5% of tumors recur locally after excision? These tumors are usually unencapsulated and may be markedly invasive.

As the name implies, the architecture of cystadenocarcinomas is dominated by large cystic structures. The cell linings vary from columnar to cuboidal to simple squamous. Approximately 75% have papillary features. (1) Many of these tumors are of low to moderate grade, but tumors of high grade have been reported. (4) Malignancy is confirmed by the presence of nuclear pleomorphism, mitosis, and an infiltrative growth pattern? The prognosis varies according to the clinical stage and grade of the tumor.

Treatment involves local excision; neck dissection and postoperative radiation therapy are used for clinically positive neck metastases. (5) Postoperatively, patients should be followed closely because these tumors have a propensity for local recurrence; also, a solitary nodal metastasis may arise years after the initial resection.

References

(1.) Foss RD, Ellis GL, Auclair PL. Salivary gland cystadenocarcinomas. A clinicopathologic study of 57 cases. Am J Surg Pathol 1996;20:1440-7.

(2.) Seifert G, Sobin LH. The World Health Organization's Histological Classification of Salivary Gland Tumors. A commentary on the second edition. Cancer 1992;70:379-85.

(3.) Mostofi R, Wood RS, Christison W, Talerman A. Low-grade papillary adenocarcinoma of minor salivary glands. Case report and literature review. Oral Surg Oral Med Oral Pathol 1992;73:591-5.

(4.) Pollett A, Perez-Ordonez B, Jordan RC, Davidson MJ. High-grade papillary cystadenocarcinoma of the tongue. Histopathology 1997;31:185-8.

(5.) Nakagawa T, Hattori K, Iwata N, Tsujimura T. Papillary cystadenocarcinoma arising from minor salivary glands in the anterior portion of the tongue: A case report. Auris Nasus Larynx 2002;29:87-90.

From the Department of Otolaryngology-Head and Neck Surgery, New York

Presbyterian Hospital, Cornell Campus, New York City.

Reprint requests: Ashutosh Kacker, MD, 445 E. 77th St., Apt. 3B, New York, NY 10021. Phone: [phone]; fax: [phone]; e-mail: [e-mail]

Eli Grunstein, MD; Ashutosh Kacker, MD