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Epstein–Barr Virus-Associated Lymphoepithelial Carcinoma Arising in a Salivary Sebaceous Lymphadenoma

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Abstract

Background

Lymphadenomas are rare benign tumors of the major salivary glands that are further classified as sebaceous and non-sebaceous. No association with viruses has been reported so far. Little is known about the mechanisms that allow lymphadenomas to undergo malignant transformation. Among these rare instances, there has never been a malignant transformation to Epstein–Barr virus (EBV)-associated lymphoepithelial carcinoma.

Methods

Clinical data of the reported case were retrieved from the patient’s electronic medical record. Hematoxylin & eosin-stained slides, immunohistochemical tests, and in situ hybridization performed for routine diagnostic purposes were reviewed.

Results

We report a salivary gland sebaceous lymphadenoma in which the luminal components were mostly replaced by malignant epithelial cells with markedly atypical nuclear features. Presence of EBV was demonstrated in all components by EBER. The morphological and immunohistochemical findings were consistent with a lymphoepithelial carcinoma arising from a sebaceous lymphadenoma.

Conclusion

We report the first case of an Epstein–Barr virus-associated lymphoepithelial carcinoma arising from a sebaceous lymphadenoma.

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Data Availability

All material included in manuscript. No supplementary data to provide.

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Funding

The authors have no relevant financial or non-financial interests to disclose. This study was not supported by any funding.

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JW performed literature review and wrote the manuscript. JB supervised the project, substantially edited the manuscript, and provided images. OG, KA, and RRS reviewed the manuscript. All authors read and approved the final manuscript.

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Correspondence to Jérémie Berdugo.

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Wong, J., Gologan, O., Ahmad, K. et al. Epstein–Barr Virus-Associated Lymphoepithelial Carcinoma Arising in a Salivary Sebaceous Lymphadenoma. Head and Neck Pathol 17, 871–876 (2023). https://doi.org/10.1007/s12105-023-01546-w

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