Ectopic Pleomorphic Adenoma of the Cervical Region: Case Report with Integrated Clinical, Radiologic, and Embryologic Insights
DOI:
https://doi.org/10.15517/xh5dce40Keywords:
Pleomorphic adenoma; Lateral cervical mass; Head and neck pathology.Abstract
Pleomorphic adenoma (PA) is the most common benign salivary gland neoplasm. Its development in ectopic salivary tissue of the lateral neck is exceptionally uncommon, clinically deceptive, and carries a low but documented potential for malignant transformation. Understanding the embryologic basis of cervical salivary heterotopia is essential for accurate diagnosis. A 54-year-old male presented with a painless, slowly enlarging, left lateral cervical mass. Imaging and preoperative fine-needle aspiration cytology suggested a benign salivary-type neoplasm. Complete surgical excision of the mass with an intact capsule was performed. Final histopathology and immunohistochemistry confirmed the diagnosis of pleomorphic adenoma arising in heterotopic salivary tissue, with no continuity to the major salivary glands. No recurrence was detected at 12- month follow-up. Ectopic PA should be considered in the differential diagnosis of left lateral cervical masses. Complete excision and long-term surveillance are recommended because of the risk of late recurrence and malignant transformation. Embryological and molecular insights into its pathogenesis are also provided.
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References
Eveson J.W., Cawson R.A. Salivary gland tumours. A review of 2410 cases with particular reference to histological types, site, age and sex distribution. J Pathol. maggio 1985; 146 (1): 51-8. doi:10.1002/path.1711460106
Spiro R.H. Salivary neoplasms: Overview of a 35-year experience with 2,807 patients. Head Neck Surg. gennaio 1986; 8 (3): 177-84. doi:10.1002/hed.2890080309
Eneroth C.M. Salivary gland tumors in the parotid gland, submandibular gland, and the palate region. Cancer. giugno 1971; 27 (6): 1415-8. doi:10.1002/1097-0142(197106)27:6<1415::AID-CNCR2820270622>3.0.CO;2-X
Pesavento G., Ferlito A. Benign mixed tumour of heterotopic salivery gland tissue in upper neck: Report of a case with a review of the literature on heterotopic salivary gland tissue. J Laryngol Otol. giugno 1976; 90 (6): 577-84. doi:10.1017/S0022215100082475
Ferlito A., Bertino G., Rinaldo A., Mannarà G.M., Devaney K.O. A review of heterotopia and associated salivary gland neoplasms of the head and neck. J Laryngol Otol. aprile 1999; 113 (4): 299-303. doi:10.1017/S0022215100143841
Caraway J., Yang A., Mckenzie S., Meltzer N., Noller M. Ectopic Pleomorphic Adenoma: A Case Study and Scoping Review of the Literature. Cureus. 1 febbraio 2026. doi:10.7759/cureus.102785
Ismı O., Vayısoğlu Y., Arpaci R.B., Eti C., Pütürgeli T., Gorur K., et al. Carcinoma ex pleomorphic adenoma originating from ectopic salivary gland in the neck region: case report. Gland Surg. dicembre 2015; 4 (6): 567-71. doi:10.3978/j.issn.2227-684X.2015.01.03 PubMed PMID: 26645012; PubMed Central PMCID: PMC4647010.
Barnes L., Weltgesundheitsorganisation, International Agency for Research on Cancer, curatori. Pathology and genetics of head and neck tumours: ... reflects the views of a working group that convened for an Editorial and Consensus Conference in Lyon, France, July 16 - 19, 2003. Reprinted. Lyon: IARC Press; 2007. 430 p. (World Health Organization Classification of Tumours; fasc. 9).
Som P.M., Curtin H.D. Head and neck imaging. 5th ed. St. Louis: Mosby Elsevier; 2011.
Zbären P., Stauffer E. Pleomorphic adenoma of the parotid gland: Histopathologic analysis of the capsular characteristics of 218 tumors. Head Neck. agosto 2007; 29 (8): 751-7. doi:10.1002/hed.20569
Kaur J., Kaur M. Heterotopic sites of pleomorphic adenoma. J Fam Med Prim Care. maggio 2023; 12 (5): 1009-11. doi:10.4103/jfmpc.jfmpc_1591_22 PubMed PMID: 37448942; PubMed Central PMCID: PMC10336950.
Vegari S., Naderpour M., Hemmati A., Baybordi H. Pleomorphic Adenoma of the Cervical Heterotopic Salivary Gland: A Case Report. Case Rep Otolaryngol. 2012; 2012: 470652. doi:10.1155/2012/470652 PubMed PMID: 22953114; PubMed Central PMCID: PMC3420560.
La Macchia R., Stefanelli S., Lenoir V., Dulguerov N., Pache J.C., Becker M. Pleomorphic Adenoma Originating from Heterotopic Salivary Tissue of the Upper Neck: A Diagnostic Pitfall. Case Rep Otolaryngol. 2017; 2017: 1-5. doi:10.1155/2017/5767396
Hallak B., Kamel M.E., Bouayed S., Yerly S. Atypical localisation of pleomorphic adenoma issue from heterotopic salivary tissue in the supraclavicular region: diagnosis and management. BMJ Case Rep. 2 novembre 2020; 13 (11): e237583. doi:10.1136/bcr-2020-237583 PubMed PMID: 33139370; PubMed Central PMCID: PMC7607572.
Valstar M.H., Mast H., ten Hove I., Moonen L.R., Balm A.J., Smeele L.E., et al. Malignant transformation of salivary gland pleomorphic adenoma: proof of principle. J Pathol Clin Res. 7 maggio 2021; 7 (5): 432-7. doi:10.1002/cjp2.216 PubMed PMID: 34390320; PubMed Central PMCID: PMC8363925.
Skandalakis J.E., Gray S.W., curatori. Embryology for surgeons: the embryological basis for the treatment of congenital anomalies. 2nd ed. Baltimore: Williams & Wilkins; 1994. 1101 p.
Sadler T.W. Langman’s medical embryology. Fourteenth edition. Philadelphia: Wolters Kluwer; 2019. 432 p.
Hallak B., Kamel M.E., Bouayed S., Yerly S. Atypical localisation of pleomorphic adenoma issue from heterotopic salivary tissue in the supraclavicular region: diagnosis and management. BMJ Case Rep. novembre 2020; 13 (11): e237583. doi:10.1136/bcr-2020-237583
Howlett D.C. High resolution ultrasound assessment of the parotid gland. Br J Radiol. aprile 2003; 76 (904): 271-7. doi:10.1259/bjr/33081866
Christe A., Waldherr C., Hallett R., Zbaeren P., Thoeny H. MR Imaging of Parotid Tumors: Typical Lesion Characteristics in MR Imaging Improve Discrimination between Benign and Malignant Disease. Am J Neuroradiol. agosto 2011; 32 (7): 1202-7. doi:10.3174/ajnr.A2520
Schmidt R.L., Hall B.J., Wilson A.R., Layfield L.J. A Systematic Review and Meta-Analysis of the Diagnostic Accuracy of Fine-Needle Aspiration Cytology for Parotid Gland Lesions. Am J Clin Pathol. luglio 2011; 136 (1): 45-59. doi:10.1309/AJCPOIE0CZNAT6SQ
Tryggvason G., Gailey M.P., Hulstein S.L., Karnell L.H., Hoffman H.T., Funk G.F., et al. Accuracy of fine-needle aspiration and imaging in the preoperative workup of salivary gland mass lesions treated surgically. The Laryngoscope. gennaio 2013; 123 (1): 158-63. doi:10.1002/lary.23613
Marcos Ordóñez M., García Lagarto E., Santos Pérez J., Moráis Pérez D. [Carcinoma on pleomorphic adenoma in cervical salivary heteropia. Handling of one case and literature review]. Acta Otorrinolaringol Esp. ottobre 2007; 58 (8): 371-4. PubMed PMID: 17949666.
Skálová A., Hyrcza M.D., Leivo I. Update from the 5th Edition of the World Health Organization Classification of Head and Neck Tumors: Salivary Glands. Head Neck Pathol. 21 marzo 2022; 16 (1): 40-53. doi:10.1007/s12105-022-01420-1 PubMed PMID: 35312980; PubMed Central PMCID: PMC9018948.
Katabi N., Gomez D., Klimstra D.S., Carlson D.L., Lee N., Ghossein R. Prognostic factors of recurrence in salivary carcinoma ex pleomorphic adenoma, with emphasis on the carcinoma histologic subtype: a clinicopathologic study of 43 cases. Hum Pathol. 1 luglio 2010; 41 (7): 927-34. doi:10.1016/j.humpath.2009.12.011
Bahrami A., Dalton J.D., Shivakumar B., Krane J.F. PLAG1 Alteration in Carcinoma Ex Pleomorphic Adenoma: Immunohistochemical and Fluorescence In Situ Hybridization Studies of 22 Cases. Head Neck Pathol. 8 aprile 2012; 6 (3): 328-35. doi:10.1007/s12105-012-0353-8.PubMed PMID: 22485045; PubMed Central PMCID: PMC3422590.
Wolk R.A., Cipriani N.A. Update of newly-recognized salivary gland neoplasms: molecular and immunohistochemical findings and clinical importance. Histopathology. gennaio 2025; 86 (2): 183-98. doi:10.1111/his.15289 PubMed PMID: 39108216; PubMed Central PMCID: PMC11649518.
Wittekindt C., Streubel K., Arnold G., Stennert E., Guntinas-Lichius O. Recurrent pleomorphic adenoma of the parotid gland: Analysis of 108 consecutive patients. Head Neck. settembre 2007; 29 (9): 822-8. doi:10.1002/hed.20613
Bartuli F.N., Luciani F., Cardoni G., Muzzi F., Caddeo F., Ottria L., Arcuri C. “Oral Cavity rare lesion: 15 year histories” Oral&Implantology – 2011 (4); 3-4: 18-22. ISSN: 1974-5648.
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