Primary lymphoepithelioma-like carcinoma of minor salivary gland: A case report with immunohistochemical and in situ hybridization studies

Department of Pathology, Chang Gung Memorial Hospital, Kaohsiung Medical Center, No.123, Ta-Pei Rd., Niao-song Township, Kaohsiung County 833, Taiwan.
Head & Neck (Impact Factor: 2.64). 02/2006; 28(2):182-6. DOI: 10.1002/hed.20312
Source: PubMed


Lymphoepithelioma-like carcinomas (LEC) of salivary glands represent rare epithelial malignancies, with most cases affecting the parotid gland. To our knowledge, there was only one LEC arising from the minor salivary gland described in the English-language literature.
We report the second LEC of the minor salivary gland in the buccal area of a 50-year-old Taiwanese woman, who underwent surgical resection and adjuvant radiotherapy and remained alive and well after 120 months of follow-up. Histologically, irregular tumor nests of undifferentiated epithelial cells with syncytial cell boundaries were found embedded within rich lymphoplasmacytic stroma. The tumor cells also showed strong c-KIT expression and evidence of Epstein-Barr virus (EBV) infection.
Our case suggests potential pathogenic implications of both c-KIT and EBV in LEC of the minor salivary gland that can be cured by the combination of surgery and radiotherapy and has a very favorable long-term prognosis.

Download full-text


Available from: Hock-Liew Eng
  • Source
    • "Among these, LMP-1 has a driving role. Expression of LMP-1 prevents apoptosis, induces abnormal cell proliferation, deregulates epithelial growths and inhibits differentiation, with the epithelial cells showing the features of transformed cells [34]. However, the expression of this protein was observed only in few EBV-linked salivary LELC from Asian patients and our case is the first one in which LMP-1 positivity was detected in a Western patient. "
    [Show abstract] [Hide abstract]
    ABSTRACT: Primary lymphoepithelial-like carcinoma of the parotid gland is a rare tumour with an increased incidence among Eskimos and Orientals. In these populations, it is usually associated with Epstein-Barr virus. In Western countries, salivary gland lymphoepithelial-like carcinoma are uncommon and only 14 cases have been described so far; among these, only three cases showed Epstein-Barr virus positivity.Case report: A 45-year-old woman was admitted to Siena Hospital for evaluation of a pre-existent (2 years) painless and tender submandibular mass, rapidly enlarging since two months. On physical examination, a 2.5-cm mass was found in the right parotid. It was firm, mobile and non-tender. Laboratory data were within reference range. Nuclear magnetic resonance detected a 2,5x1,5x1-cm well-circumscribed mass in the deep lobe of the right parotid. A right total paroditectomy with dissection of a satellite lymph node was performed. On the basis of morphological, immunohistochemical and molecular biology findings, a diagnosis of stage II (according to TNM7) Epstein Barr-virus positive, undifferentiated lymphoepithelial-like carcinoma of the parotid gland was made. Twenty months after surgery she was free of disease. Further studies seem to be necessary to completely elucidate the oncogenic role of Epstein Barr-virus in these tumors, which have identical morphology but different prognosis and variable presence of the virus.
    Full-text · Article · Jul 2013 · Diagnostic Pathology
  • [Show abstract] [Hide abstract]
    ABSTRACT: Lymphoepithelial carcinoma (LEC) is a rare category of malignant neoplasms that share morphologic features with undifferentiated nasopharyngeal carcinoma, which can be regarded as the prototype Epstein-Barr virus (EBV)-positive epithelial neoplasm. A similar association with EBV is emerging for LEC of the oral cavity, which appears to be strongly influenced by the ethnic origin of the patient. A rare case of primary LEC of the lower lip in a 73-year-old Caucasian man is described. The tumor showed a striking microscopic resemblance to undifferentiated nasopharyngeal carcinoma (lymphoepithelioma) with features of a syncytial growth pattern, large undifferentiated tumor cells with vesicular nuclei, prominent eosinophilic nucleoli, and a dense intratumoral lymphocytic infiltrate. There was, however, no evidence of EBV infection of the tumor cells by immunohistochemistry and polymerase chain reaction, suggesting that EBV probably does not play a role in the pathogenesis of LEC of the lip in Caucasian patients.
    No preview · Article · Jun 2008 · Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology
  • Source
    [Show abstract] [Hide abstract]
    ABSTRACT: Individuals with acquired immunodeficiency syndrome (AIDS) manifest an increased risk of cancer, particularly cancers caused by oncogenic viruses. Because some salivary gland and nasopharyngeal cancers are associated with Epstein Barr virus, the impact of AIDS on these cancers needs further evaluation. We used linked U.S. AIDS and cancer registry data (N = 519,934 people with AIDS) to derive standardized incidence ratios (SIRs) comparing risk of salivary gland and nasopharyngeal cancers to the general population. For salivary gland cancers (N = 43 cases), individuals with AIDS had strongly elevated risks for lymphoepithelial carcinoma (SIR 39, 95% CI 16-81) and squamous cell carcinoma (SIR 4.9, 95% CI 2.5-8.6). Among nasopharyngeal cancers (N = 39 cases), risks were elevated for both keratinizing and nonkeratinizing carcinomas (SIR 2.4, 95% CI 1.5-3.7 and SIR 2.4, 95% CI 1.2-4.4, respectively). The elevated risks of salivary gland and nasopharyngeal cancers among people with AIDS suggest that immunosuppression and oncogenic viral infections are etiologically important.
    Full-text · Article · Jan 2009 · International Journal of Cancer
Show more