Expect the unexpected: A rare case of bilateral lymphoepithelial carcinoma of the parotid gland

Lymphoepithelial carcinoma (LEC) of the salivary glands is a rare and aggressive malignancy strongly associated with Epstein–Barr virus (EBV) infection and more frequently reported in endemic regions such as Greenland and Southeast Asia. Bilateral involvement is exceptionally uncommon, particularly in non-endemic populations. We report the case of a 62-year-old male with bilateral, asymptomatic parotid swellings persisting for five years. Initial clinical assessment suggested a benign etiology, with no pain, facial nerve involvement, or other red flag symptoms. However, ultrasound, fine needle aspiration, and contrast-enhanced computed tomography of the left parotid revealed atypical features suspicious for malignancy, and histopathology confirmed LEC. The patient underwent left superficial parotidectomy with clear margins, followed by right superficial parotidectomy, which also confirmed LEC. This case represents a rare presentation of bilateral LEC in a non-endemic region. The indolent course and absence of classic malignant features highlight the diagnostic challenges of this entity. Early multidisciplinary evaluation is essential for persistent or atypical parotid swellings, and further research is warranted into the role of EBV in tumorigenesis outside endemic areas.

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Publication Details

Journal
Otolaryngology Case Reports
Published
2025-12-11
DOI
https://doi.org/10.1016/j.xocr.2025.100718
Primary Topic
Viral-associated cancers and disorders
Type
article
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article

Expect the unexpected: A rare case of bilateral lymphoepithelial carcinoma of the parotid gland

Niall James McInerney, Sangeetha Kalimuthu, О.А. Tkachuk, Umair Hafeez
Otolaryngology Case Reports
Viral-associated cancers and disorders
article

Expect the unexpected: A rare case of bilateral lymphoepithelial carcinoma of the parotid gland

Niall James McInerney, Sangeetha Kalimuthu, О.А. Tkachuk, Umair Hafeez
article en

Abstract

Lymphoepithelial carcinoma (LEC) of the salivary glands is a rare and aggressive malignancy strongly associated with Epstein–Barr virus (EBV) infection and more frequently reported in endemic regions such as Greenland and Southeast Asia. Bilateral involvement is exceptionally uncommon, particularly in non-endemic populations. We report the case of a 62-year-old male with bilateral, asymptomatic parotid swellings persisting for five years. Initial clinical assessment suggested a benign etiology, with no pain, facial nerve involvement, or other red flag symptoms. However, ultrasound, fine needle aspiration, and contrast-enhanced computed tomography of the left parotid revealed atypical features suspicious for malignancy, and histopathology confirmed LEC. The patient underwent left superficial parotidectomy with clear margins, followed by right superficial parotidectomy, which also confirmed LEC. This case represents a rare presentation of bilateral LEC in a non-endemic region. The indolent course and absence of classic malignant features highlight the diagnostic challenges of this entity. Early multidisciplinary evaluation is essential for persistent or atypical parotid swellings, and further research is warranted into the role of EBV in tumorigenesis outside endemic areas.

Otolaryngology Case ReportsVol. 38
Openalex Percentile: Top 45%
Viral-associated cancers and disorders
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Expect the unexpected: A rare case of bilateral lymphoepithelial carcinoma of the parotid gland — Niall James McInerney, Sangeetha Kalimuthu, et al. · Otolaryngology Case Reports (2025) | TGRS Research Map | TGRS