Coexistent parapharyngeal epithelial-myoepithelial carcinoma and bilateral Warthin’s tumors
Authors:
Z. Mateášiková 1
; R. Salzman 1
; Ivo Stárek 1
; J. Michálek 2
Authors‘ workplace:
Klinika otorinolaryngologie a chirurgie hlavy a krku LF UP a FN Olomouc
1; Ústav klinické a molekulární patologie LF UP a FN Olomouc
2
Published in:
Otorinolaryngol Foniatr, 71, 2022, No. 1, pp. 33-37.
Category:
Case Reports
doi:
https://doi.org/10.48095/ccorl202233
Overview
Epithelial-myoepithelial carcinoma (EMEC) is a very rare low-grade malignancy with an incidence of less than 1% of all salivary gland tumors. In the parapharyngeal space, it can arise either from the deep lobe of the parotid gland or from minor salivary glands of the oropharynx. We present a case of a 70-year-old female patient with progressive dysphagia and a bulging of paratonsillar space. A CT scan revealed a cystic formation in the parapharyngeal space and bilateral inhomogeneous tumors in parotid glands. Preoperative FNAB (fine needle aspiration biopsy) was undiagnostic. Definitive histologic examination after surgical removal confirmed epithelial-myoepithelial carcinoma in the parapharyngeal space and Warthin’s tumors in both parotid glands. Immunohistochemical tests of the EMEC revealed BRAF, COSM476 a COSM775 and PIK3CA mutations, however, no HRAS mutation was detected. This case report shows a rare clinical and radiological presentation that could resemble benign diagnosis. Multiple salivary gland tumors are relatively rare conditions. Warthin’s tumor is the most common tumor with this type of behaviour. The coincidence of bilateral Warthin’s tumor of parotid glands with a parapharyngeal epithelial-myoepithelial carcinoma has not been documented in literature so far.
Keywords:
epithelial-myoepithelial carcinoma – parapharyngeal space cyst – salivary gland – Warthin’s tumor
Sources
1. Arora SK, Sharma N, Bhardwaj M. Epithelial myoepithelial carcinoma of head and neck region. Indian J Otolaryngol Head Neck Surg 2013; 65 (Suppl 1): 163–166. Doi: 10.1007/s12070-011-0414-4.
2. Panda NK, Patro SK, Verma RK et al. Epithelio-myoepithelial carcinoma of masseteric space: a case report. Indian J Surg Oncol 2017; 8 (2): 240–244. Doi: 10.1007/s13193-016-0537-x.
3. Sharma S, Patankar S. An unusual case of epithelial-myoepithelial carcinoma of the parotid gland radiologically simulating a benign lesion. J Oral Maxillofac Pathol 2014; 18 (1): 118–120. Doi: 10.4103/0973-029X.131935.
4. Stárek I, Salzman R, Skálová A et al. Doporučené postupy v diagnostice a léčbě nádorů příušní žlázy. Otorinolaryngol Foniatr 2017; 66 (3): 119–126.
5. Tralongo V, Daniele E. Epithelial-myoepithelial carcinoma of the salivary glands: a review of literature. Anticancer Res 1998; 18 (1B): 603–608.
6. Bussu F, Parrilla C, Rizzo D et al. Clinical approach and treatment of benign and malignant parotid masses, personal experience. Acta Otorhinolaryngol Ital 2011; 31 (3): 135–143.
7. Chulam TC, Noronha Francisco AL, Goncalves Filho J et al. Warthin‘s tumour of the parotid gland: our experience. Acta Otorhinolaryngol Ital 2013; 33 (6): 393–397.
8. Yoo GH, Eisele DW, Askin FB et al. Warthin‘s tumor: a 40-year experience at The Johns Hopkins Hospital. Laryngoscope 1994; 104 (7): 799–803. Doi: 10.1288/00005537-199407000-00 004.
9. Maiorano E, Lo Muzio L, Favia G et al. Warthin‘s tumour: a study of 78 cases with emphasis on bilaterality, multifocality and association with other malignancies. Oral Oncol 2002; 38 (1): 35–40. Doi: 10.1016/s1368-8375 (01) 00019-7.
10. Batsakis JG, el-Naggar AK, Luna MA. Epithelial-myoepithelial carcinoma of salivary glands. Ann Otol Rhinol Laryngol 1992; 101 (6): 540–542. Doi: 10.1177/000348949210100617.
11. Simpson RH, Clarke TJ, Sarsfield PT et al. Epithelial-myoepithelial carcinoma of salivary glands. J Clin Pathol 1991; 44 (5): 419–423. Doi: 10.1136/jcp.44.5.419.
12. Jin XL, Ding CN, Chu Q. Epithelial-myoepithelial carcinoma arising in the nasal cavity: a case report and review of literature. Pathology 1999; 31 (2): 148–151. Doi: 10.1080/003130299105 340.
13. Pelosi G, Fraggetta F, Maffini F et al. Pulmonary epithelial-myoepithelial tumor of unproven malignant potential: report of a case and review of the literature. Mod Pathol 2001; 14 (5): 521–526. Doi: 10.1038/modpathol.3880 341.
14. Guan M, Cao X, Wang W et al. Epithelial-myoepithelial carcinoma of the hypopharynx: A rare case. Oncol Lett 2014; 7 (6): 1978–1980. Doi: 10.3892/ol.2014.2024.
15. Donath K, Seifert G, Schmitz R. Diagnosis and ultrastructure of the tubular carcinoma of salivary gland ducts. Epithelial-myoepithelial carcinoma of the intercalated ducts. Virchows Arch A Pathol Pathol Anat 1972; 356 (1): 16–31.
16. Seifert G. Histological classification of salivary gland tumours. In: Histological Typing of Salivary Gland Tumours. World Health Organization. Springer, Berlin, Heidelberg: 1991. Doi: 10.1007/978-3-642-84506-2_2.
17. Politi M, Robiony M, Avellini C et al. Epithelial-myoepithelial carcinoma of the parotid gland: Clinicopathological aspect, diagnosis and surgical consideration. Ann Maxillofac Surg 2014; 4 (1): 99–102. Doi: 10.4103/2231-0746.133 085.
18. Corio RL, Sciubba JJ, Brannon RB et al. Epithelial-myoepithelial carcinoma of intercalated duct origin. A clinicopathologic and ultrastructural assessment of sixteen cases. Oral Surg Oral Med Oral Pathol 1982; 53 (3): 280–287. Doi: 10.1016/0030-4220 (82) 90304-8
19. Bakshi J, Budhiraja G, Gupta K et al. Epithelial-myoepithelial carcinoma of parotid gland – a surprising clinical entity. Surgical Science 2012; 3 (5): 5. Doi: 10.4236/ss.2012.35055.
20. Chiosea SI, Miller M, Seethala RR. HRAS mutations in epithelial-myoepithelial carcinoma. Head Neck Pathol 2014; 8 (2): 146–150. Doi: 10.1007/s12105-013-0506-4.
21. Urano M, Nakaguro M, Yamamoto Y et al. Diagnostic significance of HRAS mutations in epithelial-myoepithelial carcinomas exhibiting a broad histopathologic spectrum. Am J Surg Pathol 2019; 43 (7): 984–994. Doi: 10.1097/PAS.0000000000001258.
22. Maurer MH, Gartenschlager S, Dietrich C. Giant epithelial-myoepithelial carcinoma of the parotid gland. Clin Imaging 2009; 33 (3): 237–239. Doi: 10.1016/j.clinimag.2008.11. 013.
23. Ochal-Choinska A, Bruzgielewicz A, Osuch-Wojcikiewicz E. Synchronous multiple unilateral parotid gland tumors of benign and malignant histological types: case report and literature review. Braz J Otorhinolaryngol 2019; 85 (3): 388–392. Doi: 10.1016/j.bjorl.2016.03.002.
24. Hattori K, Murai N, Kusano J et al. Bilateral epithelial-myoepithelial carcinoma of the parotid glands. Head Neck 2016; 38 (3): E73–75. Doi: 10.1002/hed.24179.
25. Stárek I. Multiplicitní nádory slinných žláz. Otorinolaryngol Foniatr 1993; 42 (1): 25–27.
26. Stárek I, Černý L, Komínek P. Nádory hlubokého laloku příušní žlázy. Chor hlavy a krku 1993; 2 (3/4): 48–51.
27. Wemmert S, Willnecker V, Sauter B et al. Genomic alterations in Warthin tumors of the parotid gland. Oncol Rep 2014; 31 (4): 1899– –1904. Doi: 10.3892/or.2014.3028.
28. Chaudhari P, Kaur J, Nalwa A et al. Epithelial myoepithelial carcinoma of parotid gland. Indian J Otolaryngol Head Neck Surg 2019; 71 (Suppl 1): 62–65. Doi: 10.1007/s12070-016-1025-x.
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Audiology Paediatric ENT ENT (Otorhinolaryngology)Article was published in
Otorhinolaryngology and Phoniatrics
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