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Case Reports
. 2023 Sep;17(3):871-876.
doi: 10.1007/s12105-023-01546-w. Epub 2023 Apr 6.

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

Jahg Wong  1 , Olga Gologan  1 , Khouloud Ahmad  2 , Raja R Seethala  3 , Jérémie Berdugo  4   5
Affiliations
Case Reports

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

Jahg Wong et al. Head Neck Pathol. 2023 Sep.

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.

Keywords: Epstein–Barr virus; Lymphadenoma; Lymphoepithelial carcinoma; Parotid tumor; Salivary gland tumor.

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Conflict of interest statement

The authors declare that they have no conflict of interest.

Figures

Fig. 1
Fig. 1
Computed tomography scan shows a solid and well-demarcated lesion of the left superficial parotid lobe measuring 1.3 cm in diameter (arrow)
Fig. 2
Fig. 2
a The tumor is well demarcated and is composed of epithelial cells within a lymphoid stroma (H&E 20X), b Monotonous abluminal cells surround the luminal carcinoma component (H&E 100X), c Sebaceous lymphadenoma in relation to malignant component (H&E 100X), d Squamous differentiation (H&E 100X)
Fig. 3
Fig. 3
a Tumor (H&E, 400X), b Epstein–Barr Virus in situ hybridization shows positivity in the luminal carcinoma and abluminal lymphadenoma components, c The abluminal component shows positivity for p40, d SOX10 is expressed in abluminal cells
See this image and copyright information in PMC

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