Abstract
Background and purpose
The WHO recently designated salivary gland lymphoepithelial carcinoma as a unique malignant tumor that most commonly occurs in the parotid gland. This is a rare cancer and there are few reports in the literature. Among 854 patients with parotid gland tumors who were admitted to our institution, we diagnosed 12 patients (1.41%) with parotid lymphoepithelial carcinoma.
Methods
Retrospective analysis of 12 patients with parotid lymphoepithelial carcinoma diagnosed by the Department of Pathology, Xiangya Hospital of Central South University.
Results
All 12 patients had unilateral parotid gland disease and 8 had cervical lymph node metastasis. Five patients received PCR testing for the Epstein–Barr virus and two were positive. All patients received surgical treatment, two received surgical resection alone, nine received surgery and postoperative radiotherapy and chemotherapy, and one received surgery and postoperative chemotherapy. The postoperative follow-up time ranged from 13 to 77 months. As of the last follow-up, eight patients were tumor-free, one patient was lost to follow-up, and three patients died. The main cause of death was local tumor recurrence and multiple metastases throughout the body.
Conclusion
Parotid lymphoepithelial carcinoma is a malignant neoplasm characterized by proliferation, invasion, and inclusion of poorly differentiated or undifferentiated carcinoma, and a high rate of metastasis to ipsilateral cervical lymph nodes. The comprehensive treatment method consists of radical resection combined with postoperative radiotherapy and chemotherapy. After this comprehensive treatment, the 1-year, 3-year, and 5-year overall survival rates of our patients were 100%, 78.8%, and 39.4%.
Similar content being viewed by others
Availability of data and materials
The datasets generated and analyzed during the present study are available from the corresponding author on reasonable request.
Change history
25 September 2021
A Correction to this paper has been published: https://doi.org/10.1007/s00405-021-07077-w
References
Wang JQ, Deng RX, Liu H, Luo Y, Yang ZC (2020) Clinicopathological characteristics and prognostic analysis of lymphoepithelial carcinoma of salivary gland: a population-based study. Gland Surg 9:1989–1997. https://doi.org/10.21037/gs-20-464
Whelan A, Al-Sayed AA, Bullock M, Taylor SM (2020) Primary parotid lymphoepithelial carcinoma: A case report and literature review of a rare pathological entity. Int J Surg Case Rep 72:610–614. https://doi.org/10.1016/j.ijscr.2020.06.035
El-Naggar AK, Grandis JR, Takata T, Slootweg PJ (2017) WHO Classification of Head and Neck Tumours 4th eds. 285
Ambrosio MR et al (2013) Lymphoepithelial-like carcinoma of the parotid gland: a case report and a brief review of the western literature. Diagn Pathol 8:115. https://doi.org/10.1186/1746-1596-8-115
Monteiro F et al (2019) Epstein-Barr Virus-Associated With Lymphoepithelial Carcinoma: A Rare Tumor of the Larynx. Clin Med Insights Ear Nose Throat. https://doi.org/10.1177/1179550619865551
Keelawat S et al (2017) Cytokeratin-Negative Undifferentiated (Lymphoepithelial) Carcinoma of the Lacrimal Sac. Ophthalm Plast Reconstr Surg 33:e16–e18. https://doi.org/10.1097/iop.0000000000000422
Whaley RD, Carlos R, Bishop JA, Rooper L, Thompson LDR (2020) Lymphoepithelial carcinoma of salivary gland EBV-association in endemic versus non-endemic patients: a report of 16 cases. Head Neck Pathol 14:1001–1012. https://doi.org/10.1007/s12105-020-01172-w
Squillaci S et al (2000) Lympho-epithelial carcinoma (malignant lympho-epithelial lesions) of the parotid gland. Description of a case with detection of EBV by in situ hybridization. Pathologica 92:189–194
Jin Lei YH, Yufan W et al (2017) Clinicopathological analysis of 15 cases of salivary gland lymphoepithelial carcinoma. Chinese J Oral Maxillofac Surg 25:1213–1216
Kim YJ, Hong HS, Jeong SH, Lee EH, Jung MJ (2017) Lymphoepithelial carcinoma of the salivary glands. Medicine 96:e6115. https://doi.org/10.1097/md.0000000000006115
Jang SJ et al (1997) Lymphoepithelial carcinoma of the submandibular gland–a case report. J Korean Med Sci 12:252–255. https://doi.org/10.3346/jkms.1997.12.3.252
Kanjanavirojkul N, Kularbkaew C, Yutanawiboonchai W (2008) Fine needle aspiration in a malignant lymphoepithelial lesion: a case report. Acta Cytol 52:369–372. https://doi.org/10.1159/000325526
Schneider M, Rizzardi C (2008) Lymphoepithelial carcinoma of the parotid glands and its relationship with benign lymphoepithelial lesions. Arch Pathol Lab Med 132:278–282. https://doi.org/10.1043/1543-2165(2008)132[278:Lcotpg]2.0.Co;2
Raab-Traub N (1992) Epstein-Barr virus infection in nasopharyngeal carcinoma. Infect Agents Dis 1:173–184
Zhou T et al (2019) Associations between environmental factors and serological Epstein-Barr virus antibodies in patients with nasopharyngeal carcinoma in South China. Cancer Med 8:4852–4866. https://doi.org/10.1002/cam4.2348
Lam WKJ et al (2019) Methylation analysis of plasma DNA informs etiologies of Epstein-Barr virus-associated diseases. Nat Commun 10:3256. https://doi.org/10.1038/s41467-019-11226-5
Tsao SW, Tsang CM, Lo KW (2017) Epstein-Barr virus infection and nasopharyngeal carcinoma. Philos Trans Roy Soc London Series B Biol Sci. https://doi.org/10.1098/rstb.2016.0270
Young LS, Dawson CW (2014) Epstein-Barr virus and nasopharyngeal carcinoma. Chin J Cancer 33:581–590. https://doi.org/10.5732/cjc.014.10197
Lu SY, Huang CC, Hsiung CY, Eng HL, Huang HY (2006) Primary lymphoepithelioma-like carcinoma of minor salivary gland: a case report with immunohistochemical and in situ hybridization studies. Head Neck 28:182–186. https://doi.org/10.1002/hed.20312
Nagao T et al (1996) Epstein-Barr virus-associated undifferentiated carcinoma with lymphoid stroma of the salivary gland in Japanese patients comparison with benign lymphoepithelial lesion. Cancer 78:695–703. https://doi.org/10.1002/(sici)1097-0142(19960815)78:4%3c695::Aid-cncr1%3e3.0.Co;2-e
Leung SY et al (1995) Lymphoepithelial carcinoma of the salivary gland: in situ detection of Epstein-Barr virus. J Clin Pathol 48:1022–1027. https://doi.org/10.1136/jcp.48.11.1022
Schiødt M et al (1992) Natural history of HIV-associated salivary gland disease. Oral Surg Oral Med Oral Pathol 74:326–331. https://doi.org/10.1016/0030-4220(92)90069-3
Terry JH, Loree TR, Thomas MD, Marti JR (1991) Major salivary gland lymphoepithelial lesions and the acquired immunodeficiency syndrome. Am J Surg 162:324–329. https://doi.org/10.1016/0002-9610(91)90141-y
Abdelkrim SB et al (2009) Primary lymphoepithelial carcinoma of the parotid gland in a North African woman. Rare tumors 1:e16. https://doi.org/10.4081/rt.2009.e16
Zhan KY, Nicolli EA, Khaja SF, Day TA (2016) Lymphoepithelial carcinoma of the major salivary glands: Predictors of survival in a non-endemic region. Oral Oncol 52:24–29. https://doi.org/10.1016/j.oraloncology.2015.10.019
Yin X, Xu G, Zhang L, Chen Z, Liu X, Zhang J (2013) Lymphoepithelial carcinoma in salivary glands: clinical and pathological analysis of 17 cases. J Clin Otorhinolaryngol Head Neck Surg 27(21):1171–1174
Zhu GC, Sun XF, Xiao DJ (2017) Analysis of the lymphoepithelial carcinoma of the salivary gland: 12 cases report. Lin chuang er bi yan hou tou jing wai ke za zhi J Clin Otorhinolaryngol Head Neck Surg 31:1093–1096 https://doi.org/10.13201/j.issn.1001-1781.2017.14.010
Wang P, Yang J, Yu Q (2017) Lymphoepithelial carcinoma of salivary glands: CT and MR imaging findings. Dentomaxillofac Radiol 46:20170053. https://doi.org/10.1259/dmfr.20170053
Yin L, Huang X, Liu X, Zhang Y, Wang X (2017) Distribution of lymph node metastasis from lymphoepithelial-like carcinoma of the parotid. Oncotarget 8:84841–84846. https://doi.org/10.18632/oncotarget.11002
Dubey P et al (1998) Nonnasopharyngeal lymphoepithelioma of the head and neck. Cancer 82:1556–1562. https://doi.org/10.1002/(sici)1097-0142(19980415)82:8%3c1556::aid-cncr18%3e3.0.co;2-3
Abdulla AK, Mian MY (1996) Lymphoepithelial carcinoma of salivary glands. Head Neck 18:577–581. https://doi.org/10.1002/(sici)1097-0347(199611/12)18:6%3c577::Aid-hed13%3e3.0.Co;2-5
Myers JN, Peel RL, Myers EN (1995) Pathologic quiz case 1. Malignant lymphoepithelial lesion of the parotid gland. Arch Otolaryngol Head Neck Surg 121:479–482
Zhang Q, Qing J, Wei MW, Guo ZM (2005) Clinical analysis of sixteen cases of lymphoepithelial carcinoma of salivary gland. Ai zheng Aizheng Chinese J Cancer 24:1384–1387
Acknowledgements
This work was supported by the Natural Science Foundation of Hunan Province for Young Scholars (No. 2020JJ5957).
Funding
Natural Science Foundation of Hunan Province for Young Scholars (No. 2020JJ5957).
Author information
Authors and Affiliations
Contributions
Analysis planning: lS, DD, ZY Conceptualization: lS, DD, ZY Data curation: lS, DD, ZY Draft manuscript: lS, DD, ZY Investigation: IS, Manuscript editing: lS, DD. Methodology: lS, DD, ZY.
Corresponding author
Ethics declarations
Competing interests
The authors declare that they have no competing interests, and all authors attest to the accuracy of the results.
Ethics approval
This study was approved by the Ethics Committee of the Xiangya Hospital of Central South University prior to commencement.
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
The original version of this article was revised as affiliations were incorrectly tagged to the authors Zhuo-miao Ye, Zhe Xu and affiliations are assigned correctly in this version.
Rights and permissions
About this article
Cite this article
Deng, Df., Zhou, Q., Ye, Zm. et al. Clinical analysis of 12 patients with primary lymphoepithelial carcinoma of the parotid gland. Eur Arch Otorhinolaryngol 279, 2003–2008 (2022). https://doi.org/10.1007/s00405-021-06947-7
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00405-021-06947-7