Lymphoepithelial carcinoma--review of the treatment modalities and report of a rare case in the sublingual gland.

Controversies exist in the current literature regarding the treatment of lymphoepithelial carcinoma (LEC). The presented study summaries the literature controversies in the treatment of LEC and present a case of a rare LEC diagnosed in the sublingual gland of an otherwise healthy 21-year-old Arab woman. The patient was admitted for swelling in the sublingual area. A biopsy indicated a diagnosis of LEC of the sublingual gland. Treatment included a wide resection with safe margins, marginal mandibulectomy, functional neck dissection, and a radial forearm free flap for reconstruction. The patient received concomitant radiotherapy and has remained disease free since her last follow-up. The recommended primary treatment, based on the literature review, is primary surgical excision with neck dissection and optional adjuvant radiotherapy to the gland area. Chemotherapy was not found to be beneficial according to the literature.

Epstein-Barr virus (EBV) infection in salivary gland tumors: lytic EBV infection in nonmalignant epithelial cells surrounded by EBV-positive T-lymphoma cells.

To elucidate the association of Epstein-Barr virus (EBV) and salivary gland tumors, 114 cases of tumors of major salivary glands were investigated. EBV DNA was detected in all 6 cases of undifferentiated carcinoma and all 3 cases of T-cell lymphoma, but not in other tumor tissues. In situ hybridization studies for EBV DNA and EBV-encoded small RNA 1 (EBER1) showed specific localization of the EBV sequences to the undifferentiated carcinoma cells and T-lymphoma cells. Moreover, intense DNA signals were detected on nonneoplastic epithelial cells of T-lymphoma tissues. These epithelial cells were negative for EBER1 and expressed BZLF1, BALF2, and gp350/220 proteins associated with virus production. In contrast, nonmalignant epithelial cells surrounded by undifferentiated carcinoma cells showed no evidence of EBV infection or virus replication. These results indicate that there is an unusual association of salivary gland T-cell lymphomas with lytic EBV replication of nonmalignant epithelial cells.

Expression of Epstein-Barr virus in carcinomas of major salivary glands: a strong association with lymphoepithelioma-like carcinoma.

To elucidate the role of Epstein-Barr virus (EBV) in salivary gland carcinomas, 56 cases of carcinomas of major salivary glands were investigated. These included 14 mucoepidermoid carcinomas, 13 adenoid cystic carcinomas, seven malignant mixed tumors, four adenocarcinomas, four salivary duct carcinomas, two acinic cell carcinomas, two undifferentiated carcinomas without lymphoid stroma, seven lymphoepithelioma-like carcinomas (LELCs), two squamous cell carcinomas, and one small cell carcinoma. EBV transcripts were examined by in situ hybridization using digoxigenin-labeled oligonucleotide antisense probe for EBV-encoded RNA 1 (EBER1) on formalin-fixed paraffin-embedded tissue sections. EBER1 was detected in the malignant epithelial cells in all seven cases of LELC, but not in any of the other carcinomas and the neighboring normal salivary gland tissue. Because all the EBV-negative cases showed satisfactory labeling with the poly d(T) probe, the negative reaction with EBER1 was unlikely to be caused by poor RNA preservation in the tissues. The seven cases of LELC, which were histologically indistinguishable from undifferentiated nasopharyngeal carcinoma (NPC), had a disease-free 4-year survival rate of 85.7%. The results suggest that LELC of the salivary gland in Taiwanese Chinese may share similar EBV-related pathogenesis with that of NPC.