Lymphoepithelial carcinoma of the submandibular gland in a Japanese woman: A case report

Lymphoepithelial carcinoma (LEC) of the submandibular gland is a very rare neoplasm that constitutes only 0.4 % of the salivary gland neoplasms, mainly involving the parotid glands, and is well known to have a racial prevalence in Southwest Asia, southern China, Africa, and the Arctic Circle. This report describes the case of a 49-year-old Japanese woman who was diagnosed with an LEC of the right submandibular gland. The patient presented with a painless, gradually enlarging mass in the right submandibular gland for 2 years. Computed tomography scanning showed a 20 × 16 mm, slightly enhanced solid mass in the right submandibular gland; magnetic resonance imaging revealed an isointense and hyperintense mass in T1- and T2-weighted images. Fine-needle aspiration cytology revealed a few small salivary gland cells and acidophilic cells against a background of lymphocytes, suggesting Warthin tumor or chronic sialadenitis. The right submandibular gland was resected, and intraoperative frozen section analysis suggested a diagnosis of LEC of the submandibular gland. Histopathological findings from the resected submandibular gland were consistent with a diagnosis of LEC, and a definitive pathologic staging of T3N0M0. Postoperative adjuvant radiation therapy was administered, and the patient was disease free at the 4-year follow-up.