Lymphoepithelial carcinoma of the salivary glands

Rationale: Lymphoepithelial carcinoma (LEC) is a rare malignancy with the histopathological feature of undifferentiated carcinoma and an intermixed reactive lymphoplasmacytic infiltration. Although clinically significant because of its malignant nature, it is difficult to make a differential diagnosis by preoperative imaging. Here, we report 3 cases of primary LEC arising in the major salivary glands, which showed unusual imaging features unlike other malignant tumors. Patient concerns: Our first case is a 44-year-old man with LEC in the right parotid gland, the second case is a 71-year-old woman with LEC in the right submandibular gland, and the third case is a 35-year-old woman with LEC in the right parotid gland. All of the patients presented with a palpable mass of variable duration. Diagnoses: Computed tomography (CT) scans revealed a relatively well-defined, slightly hyperattenuated exophytic solid mass that had homogeneous well-enhanced regions. Ultrasonography (US) in the first 2 cases showed well-defined, hypoechoic solid masses with posterior enhancement. The CT findings seem to be benign tumors, but US features are compatible with highly cellular and hypervascular tumors. Interventions: The resection of the involved salivary gland with postoperative radiation therapy was performed. Outcomes: There was no evidence of recurrence or metastasis after 5 years in all 3 patients. Lessons: Understanding these unusual imaging findings may be helpful in detecting LEC, and may also help clinicians provide adequate management to patients, such as surgery with adjuvant radiotherapy, because of its malignant entity.