Primary Lymphoepithelial-Like Carcinoma of the Parotid Gland- Case Presentation

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Athanasios Poulopoulos1, Evangelos Parcharidis1, Christina Nikolaidou2, Aikaterini Aidonopoulou3

1 Department of Oral Medicine and Oral Pathology, Dental School, Aristotle University of Thessaloniki, Thessaloniki, Greece
2 Department of Histopathology, Ippokrateio General Hospital of Thessaloniki, Thessaloniki, Greece
3 DDS private practice, Thessaloniki, Greece

Summary

Background/Aim: Primary Lymphoepithelial carcinoma (PLEC) is a rare subtype of salivary gland cancers, which comprises only 0.4% of salivary malignant neoplasms and only a few cases have been presented previously. Case report: A patient with PLEC of the parotid gland, its management and the available literature are presented. A 53-year-old woman with initial lesion of a lump in the region of the right parotid received treatment with antibiotics which did not lead to improvement. MRI was performed that recorded the presence of a tumor in the right parotid gland and the patient subsequently underwent excision biopsy. The histopathological evaluation together with additional immunohistochemical positive staining of EMA+, EGFR+, p63+, CK 5/6+, AE1/AE3+ established the diagnosis of PLEC. A PET-CT scanning has shown nor primary mucosal source neither skin lesion to account for any possible metastatic disease, consequently a course of adjuvant post-operative radiotherapy to the region of the right parotid gland was performed. Conclusions: In the differential diagnosis of a parotid gland lump should be included the possibility of a rare salivary gland neoplasm such as PLEC. Surgical excision and radiotherapy have been proposed for the treatment of early and advanced neoplasm stages. Ages of the patient, stage of the neoplasm as well as type of therapy are significant and individual variables for the prediction of the prognosis.

Keywords: Lymphoepithelial Carcinoma; Salivary Gland Neoplasms; Parotid Neoplasms; Histopathology; Immunohistochemistry; Prognosis;Treatment

Reference

 

 

1. Eveson JW, Auclair P, Gnepp DR, El-Naggar AK. Tumours of the salivary glands. In: Barnes L, Eveson JW, Reichart P, Sidransky D, editors. World Health Organization classification of tumours: pathology and genetics of head and neck tumours. Lyon: IARC Press; 2005, pp:212-251.Google Scholar

 

2. Schneider M, Rizzardi C. Lymphoepithelial carcinoma of the parotid glands and its relationship with benign lymphoepithelial lesions. Arch Pathol Lab Med, 2008;132:278-282.Google Scholar

 

3. Squillaci S, Bertalod G, Vago L, Bertoletti F, Chiodera PL, Capitanio A. Lymphoepithelioma-like carcinoma of the parotid gland. Description of a case with detection of EBV by in situ hybridization. Pathologica, 2000;92:189-194.Google Scholar

 

4. Sone M, Nakashima T, Nagasaka T, Itoh A, Yanagita N. Lymphoepithelioma-like carcinoma of the larynx associated with an Epstein-Barr viral infection. Otolaryngol Head Neck Surg, 1998;19:134-137.CrossrefGoogle Scholar

 

5. Klijanienko J, Micheau C, Azli N, Cvitkovic E, Eschwege F, Marandas P et al. Undifferentiated carcinoma of nasopharyngeal type of tonsil. Otolaryngol Head Neck Surg, 1989;115:731-734.Google Scholar

 

6. Castro CY, Ostrowski ML, Barrios R, Green LK, Popper HH, Powell S et al. Relationship between Epstei Barr virus and lymphoepithelioma-like carcinoma of the lung: a clinicopathologic study of 6 cases and review of the literature. Hum Pathol, 2001;32:863-872.CrossrefGoogle Scholar

 

7. Wick MR, Scheithauer BW, Weiland LH, Bernatz PE Primary thymic carcinomas. Am J Surg Pathol, 1982;6:613-630.CrossrefGoogle Scholar

 

8. Shousha S, Luqmani YA. Epstein-Barr virus in gastric carcinoma and adjacent normal gastric and duodenal mucosa. J Clin Pathol, 1994;47:695-698.Google Scholar

 

9. Dadmanesh F, Peterse JL, Sapino A, Fonelli A, Eusebi V. Lymphoepithelioma-like carcinoma of the breast: lack of evidence of Epstein-Barr virus infection. Histopathol, 2001;38:54-61.Google Scholar

 

10. Amin MB, Ro JY, Lee KM, Ordóñez NG, Dinney CP, Gulley ML et al. Lymphoepithelioma-like carcinoma of the urinary bladder. Am J Surg Pathol, 1994;18:466-473.Web of ScienceGoogle Scholar

 

11. Mills SE, Austin MB, Randall ME. Lymphoepitheliomalike carcinoma of the uterine cervix. A distinctive, undifferentiated carcinoma with inflammatory stroma. Am J Surg Pathol, 1985;9:883-889.CrossrefGoogle Scholar

 

12. Ambrosio MR, Rocca BJ, Mourmouras V, Onorati M, Mastrogiulio MG, Di Mari N et al. Lymphoepitheliomalike carcinoma of the endometrium. Pathologica, 2010;102:57-61.Google Scholar

 

13. Ambrosio MR, Rocca BJ, Onorati M, Mourmouras V, Mastrogiulio MG, Crispino S et al. Lymphoepithelioma- like carcinoma of the ovary. Int Surg Pathol, 2011;19:514-517.Google Scholar

 

14. Bosmuller H, Haitchi-Petnehazy S, Gruber C, Roithmeier F, Stummvoll W, Webersinke G. Lymphoepithelioma-like carcinoma of the vulva, an underrecognized entity? Case report with a single inguinal micrometastasis detected by sentinel node technique. Diagn Pathol, 2011, 6:1-4.Web of ScienceGoogle Scholar

 

15. Ambrosio MR, Mastrogiulio MG, Barone A, Rocca BJ, Gallo C, Lazzi S et al. Lymphoepithelial-like carcinoma of the parotid gland: a case report and a brief review of the western literature. Diagn Pathol, 2013;8:115-120.Web of ScienceGoogle Scholar

 

16. Abdulla AK, Mian MY. Lymphoepithelial carcinoma of salivary glands. Head Neck, 1996;18:577-581.Google Scholar

 

17. Hsu YC, Lu HF, Huang CC, Hsu RF, Su CY. Malignant lymphoepithelial lesions of the salivary gland. Otolaryngol Head Neck Surg, 2006;134:661-666.Google Scholar

 

18. Jang SJ, Paik SS, Lee WM, Park YW, Jang KJ, Tae K et al. Lymphoepithelial carcinoma of the submandibular gland -a case report. J Korean Med Sci, 1997;12:252-255.CrossrefGoogle Scholar

 

19. Roy L, Moubayed SP, Ayad T. Lymphoepithelial carcinoma of the sublingual gland: case report and review of the literature. J Oral Maxillofac Surg, 2015;73:1878 e1-5.Google Scholar

 

20. Tian Z, Li L, Wang L, Hu Y, Li J. Salivary gland neoplasms in oral and maxillofacial regions: a 23-year retrospective study of 6982 cases in an eastern Chinese population. Int J Oral Maxillofac Surg, 2010;39:235-242.Web of ScienceGoogle Scholar

 

21. Wu DL, Shemen L, Brady T, Saw D. Malignant lymphoepithelial lesion of the parotid gland: a case report and review of the literature. Ear Nose Throat J, 2001;80:803-806.Google Scholar

 

22. Bialas M, Sinczak A, Choinska-Stefanska A, Zygulska A. EBV-positive lymphoepithelial carcinoma of salivary gland in a woman of a non-endemic area – a case report. Pol J Pathol, 2002;53:235-238.Google Scholar

 

23. Kott ET, Goepfert H, Ayala AG, Ordonez NG. Lymphoepithelial carcinoma (malignant lymphoepithelial lesion) of the salivary glands. Arch Otolaryngol, 1984;110:50-53.Google Scholar

 

24. Manganaris A, Patakiouta F, Xirou P, Manganaris T. Lymphoepithelial carcinoma of the parotid gland: is an association with Epstein-Barr virus possible in non-endemic areas? Int J Oral Maxillofac Surg, 2007;36:556-559.Google Scholar

 

25. Kountakis SE, SooHoo W, Maillard A. Lymphoepithelial carcinoma of the parotid gland. Head Neck, 1995;17:445-450.Google Scholar

 

26. Li F, Zhu G, Wang Y, Wang Y, Chen T, Ji Q. A clinical analysis of 37 cases with lymphoepithelial carcinoma of the major salivary gland treated by surgical resection and postoperative radiotherapy: a single institution study. Med Oncol, 2014;31:957-965.CrossrefWeb of ScienceGoogle Scholar

 

27. Wenig BM. Lymphoepithelial-like carcinomas of the head and neck. Semin Diagn Pathol, 2015;32:74-86.Google Scholar

 

28. Zhan KY, Nicolli EA, Khaja SF, Day TA. Lymphoepithelial carcinoma of the major salivary glands: Predictors of survival in a non-endemic region. Oral Oncology, 2016;52:24-29.Web of ScienceCrossrefGoogle Scholar

 

29. Ma H, Lin Y, Wang L, Rao H, Xu G, He Y et al. Primary lymphoepithelioma-like carcinoma of salivary gland: sixty-nine cases with long-term follow-up. Head Neck, 2014;36:1305-1312.Google Scholar

 

30. Wang CP, Chang YL, Ko JY, Lou PJ, Yeh CF, Sheen TS. Lymphoepithelial carcinoma versus large cell undifferentiated carcinoma of the major salivary glands. Cancer, 2004;101:2020-2027.Google Scholar

 

31. Collela G, Cannavale R, Flamminio F, Foschini MP et al. Fine needle aspiration cytology of salivary gland lesions: a systematic review. J Oral Maxillofac Surg, 2010;68:2146-2153.Google Scholar

 

32. Ban X, Wu J, Mo Y, Yang Q, Liu X, Xie C et al. Lymphoepithelial carcinoma of the salivary gland: morphologic patterns and imaging features on CT and MRI. Am J Neuroradiol, 2014;35:1813-1819.Google Scholar

 

33. Zao W, Deng N, Gao X, Chen TB, Xie J, Li Q et al. Primary lymphoepithelial-like carcinoma of salivary glands: a clinicopathologic study of 21 cases. Int J Exp Pathol, 2014;7:7951-7956.Google Scholar

 

34. Tang CG, Schmidtknecht TM, Tang GY. Lymphoepithelial carcinoma: A case of a rare parotid gland tumor. Perm J, 2012;16:60-62.Google Scholar

 

35. Jen KY, Cheng J, Li J, Wu L, Li Y, Yu S et al. Mutational events in LMP1 gene of Epstein- Barr virus in salivary gland lymphoepithelial carcinomas. Int J Cancer, 2003;105:654-660.Google Scholar

 

36. Lu SY, Huang CC, Hsiung CY, Eng HL, Huang HY. Primary lymphoepithelioma-like carcinoma of minor salivary gland: a case report with immunohistochemical and in situ hybridization studies. Head Neck, 2006;28:182-186.Google Scholar

 

37. Hjalgrim H, Friborg J, Melbye M. The epidemiology of EBV and its association with malignant disease. In: Arvin A, Campadelli-Fiume G, Mocarski E, Moore PS, Roizman B, Whitley R, et al. editors. Human herpesviruses: biology, therapy, and immunoprophylaxis. Cambridge: Cambridge University Press; 2007 chapter 53.Google Scholar

 

38. Hsu JL, Glaser SL. Epstein-barr virus-associated malignancies: epidemiologic patterns and etiologic implications. Crit Rev Oncol Hematol, 2000;34:27-53.CrossrefGoogle Scholar

 

Balkan Journal of Dental Medicine, Volume 22, Issue 1, Pages 43–48, ISSN (Online) 2335-0245,DOI: https://doi.org/10.2478/bjdm-2018-0008.

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