Invasive myoepithelial carcinoma ex pleomorphic adenoma of the major salivary gland: two case reports


Background:
Myoepithelial carcinoma (MEC) is a rare salivary gland tumor. Its long-term prognosis remains unknown because of the paucity of reported cases with long-term follow-up. Although some case series exist, the clinical features of MEC vary considerably depending on the site of origin. Therefore, accumulation of these rare cases is important.

Case presentation:
Case 1: An 89-year-old man presented with a 10-year history of a mass originating from the right parotid gland and involving the neck. The mass grew rapidly for 3 months, reaching approximately 8 cm. There was no facial paralysis. MEC ex pleomorphic adenoma (PA) was suspected. Superficial parotid gland resection was performed in 2013; the tumor grade was pT3N0M0, and the resection margins were free of carcinoma. Because of several high-risk factors for metastasis (i.e., invasive carcinoma ex PA, high MIB1 index, and mutant p53 protein positivity), radiotherapy and chemotherapy were recommended as adjuvant therapy. Although the patient refused adjuvant therapy, he was recurrence-free at 36 months after surgery.

Case 2:
A 54-year-old woman presented with a >10-year history of a right submandibular mass, which grew rapidly for 1 year, reaching approximately 6 cm. Preoperative diagnosis was PA of the right submandibular gland. Submandibular gland resection was performed in 2013. Pathological analysis revealed invasive MEC ex PA, pT3N0M0; in addition, the carcinoma portion had an extra capsule and had invaded the platysma muscle close to the margin. An MIB1 index of 40 % and mutant p53 protein positivity indicated a high risk for metastasis. Additional resection and right neck dissection revealed no residual carcinoma. The patient refused adjuvant chemotherapy. One year after surgery, metastasis to the right pulmonary hilar node and both lungs were detected. Chemotherapy prevented recurrent growth of the lesion and extended survival. The patient was alive with cancer 30 months after the first surgery.

Conclusions:
High expression of the Ki67 labeling index might reflect prognosis of these cases. Chemotherapy for distant metastasis was effective, as expected. Further accumulation of cases and long follow-up data are needed to elucidate the pathophysiology and prognosis of MEC ex PA.

Keywords:
Case report, Myoepithelial carcinoma, Malignant myoepithelioma, Ex pleomorphic adenoma, Major salivary gland, Ki67 labeling index


Autoři: Takahiro Wakasaki 1,3*;  Marie Kubota 1;  Yutaka Nakashima 2;  Eri Tomonobe 1;  Takenao Mihara 1;  Junichi Fukushima 1
Působiště autorů: Department of Otorhinolaryngology, Japanese Red Cross Fukuoka Hospital, -1-1 Okusu, Miniami-ku, Fukuoka 815-8555, Japan. 1;  Department of Pathology, Japanese Red Cross Fukuoka Hospital, -1-1 Okusu, Miniami-ku, Fukuoka 815-8555, Japan. 2;  Department of Head and Neck Surgery, National Hospital Organization, Kyushu Cancer Center, 3-1-1 Notame, Miniami-ku, Fukuoka 811-1395, Japan. 3
Vyšlo v časopise: BMC Cancer 2016, 16:827
Kategorie: Case report
prolekare.web.journal.doi_sk: 10.1186/s12885-016-2871-3

© 2016 The Author(s).

Open access
This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
The electronic version of this article is the complete one and can be found online at: http://bmccancer.biomedcentral.com/articles/10.1186/s12885-016-2871-3

Souhrn

Background:
Myoepithelial carcinoma (MEC) is a rare salivary gland tumor. Its long-term prognosis remains unknown because of the paucity of reported cases with long-term follow-up. Although some case series exist, the clinical features of MEC vary considerably depending on the site of origin. Therefore, accumulation of these rare cases is important.

Case presentation:
Case 1: An 89-year-old man presented with a 10-year history of a mass originating from the right parotid gland and involving the neck. The mass grew rapidly for 3 months, reaching approximately 8 cm. There was no facial paralysis. MEC ex pleomorphic adenoma (PA) was suspected. Superficial parotid gland resection was performed in 2013; the tumor grade was pT3N0M0, and the resection margins were free of carcinoma. Because of several high-risk factors for metastasis (i.e., invasive carcinoma ex PA, high MIB1 index, and mutant p53 protein positivity), radiotherapy and chemotherapy were recommended as adjuvant therapy. Although the patient refused adjuvant therapy, he was recurrence-free at 36 months after surgery.

Case 2:
A 54-year-old woman presented with a >10-year history of a right submandibular mass, which grew rapidly for 1 year, reaching approximately 6 cm. Preoperative diagnosis was PA of the right submandibular gland. Submandibular gland resection was performed in 2013. Pathological analysis revealed invasive MEC ex PA, pT3N0M0; in addition, the carcinoma portion had an extra capsule and had invaded the platysma muscle close to the margin. An MIB1 index of 40 % and mutant p53 protein positivity indicated a high risk for metastasis. Additional resection and right neck dissection revealed no residual carcinoma. The patient refused adjuvant chemotherapy. One year after surgery, metastasis to the right pulmonary hilar node and both lungs were detected. Chemotherapy prevented recurrent growth of the lesion and extended survival. The patient was alive with cancer 30 months after the first surgery.

Conclusions:
High expression of the Ki67 labeling index might reflect prognosis of these cases. Chemotherapy for distant metastasis was effective, as expected. Further accumulation of cases and long follow-up data are needed to elucidate the pathophysiology and prognosis of MEC ex PA.

Keywords:
Case report, Myoepithelial carcinoma, Malignant myoepithelioma, Ex pleomorphic adenoma, Major salivary gland, Ki67 labeling index


Zdroje

1. Nagao T, Sugano I, Ishida Y, Tajima Y, Matsuzaki O, Konno A, Kondo Y, Nagao K. Salivary gland malignant myoepithelioma: a clinicopathologic and immunohistochemical study of ten cases. Cancer. 1998;83(7):1292–9.

2. Vilar-Gonzalez S, Bradley K, Rico-Perez J, Vogiatzis P, Golka D, Nigam A, Sivaramalingam M, Kazmi S. Salivary gland myoepithelial carcinoma. Clin Trans. 2015;17(11):847–55.

3. Kane SV, Bagwan IN. Myoepithelial carcinoma of the salivary glands: a clinicopathologic study of 51 cases in a tertiary cancer center. Arch Otolaryngol Head Neck Surg. 2010;136(7):702–12.

4. Mejia-Hernandez IJ, Valdez AMC, De L-TD, Luna-Ortiz K. Malignant myoepithelioma of the soft palate. Auris Nasus Larynx. 2013;40(2):231–4.

5. Jiang Y-H, Cheng B, Ge M-H, Zhang G. The prognostic significance of p63 and Ki-67 expression in myoepithelial carcinoma. Head Neck Oncol. 2012;4(1):9.

6. Briasoulis A, Salem N, Siddiqui F, Rashed A, Othman M. Myoepithelial carcinoma in the mediastinum involving the left atrium. Cardiovas. 2016; 25(2):158–60.

7. Yu G, Ma D, Sun K, Li T, Zhang Y. Myoepithelial carcinoma of the salivary glands: behavior and management. Chin Med J. 2003;116(2):163–5.

8. Di Palma S, Guzzo M. Malignant myoepithelioma of salivary glands: clinicopathological features of ten cases. Vichows Archiv A Pathol Anat. 1993;423(5):389–96.

9. ZhengZheng L, Lv X, Shi Y, Zhang Y, Yu G, Zhang J. (125)I interstitial brachytherapy for the treatment of myoepithelial carcinoma of the oral and maxillofacialregion. 2016;15(2):240–5.

10. Andry G, Hamoir M, Locati LD, Licitra L, Langendijk JA. Management of salivary gland tumors. Expert Rev Anticancer Ther. 2012;12(9):1161–8.

11. Savera AT, Sloman A, Huvos AG, Klimstra DS. Myoepithelial carcinoma of the salivary glands: a clinicopathologic study of 25 patients. Am J Surg Pathol. 2000;24(6):761–74.

12. Antony J, Gopalan V, Smith RA, Lam AK. Carcinoma ex pleomorphic adenoma: a comprehensive review of clinical, pathological and molecular data. Head Neck Pathol. 2012;6(1):1–9.

13. Lagha A, Chraiet N, Ayadi M, Krimi S, Allani B, Rifi H, Raies H, Mezlini A. Systemic therapy in the management of metastatic or advanced salivary gland cancers. Head Neck Oncol. 2012;4:19.

14. Chhieng DC, Paulino AF. Cytology of myoepithelial carcinoma of the salivary gland. Cancer. 2002;96(1):32–6.

15. Klijanienko J, El-Naggar AK, Vielh P. Fine-needle sampling findings in 26 carcinoma ex pleomorphic adenomas: diagnostic pitfalls and clinical considerations. Diagn Cytopathol. 1999;21(3):163–6.

16. Nouraei SA, Hope KL, Kelly CG, McLean NR, Soames JV. Carcinoma ex benign pleomorphic adenoma of the parotid gland. Plast Reconstr Surg. 2005;116(5):1206–13.

17. Yang S, Zeng M, Zhang J, Chen X. Clear cell myoepithelial carcinoma of minor salivary gland: a case report. Int J Oral Maxillofac Surg. 2010;39(3):297–300.

18. Cassidy M, Connolly CE. Clear cell carcinoma arising in a pleomorphic adenoma of the submandibular gland. J Laryngol Otol. 1994;108(6):529–32.

19. Pfister DG, Spencer S, Brizel DM, Burtness B, Busse PM, Caudell JJ, Cmelak AJ, Colevas AD, Dunphy F, Eisele DW, et al. Head and neck cancers, version 1.2015. J Natl Compr Canc Netw. 2015;13(7):847–55. quiz 856.

20. Michal M, Skalova A, Simpson RH, Rychterova V, Leivo I. Clear cell malignant myoepithelioma of the salivary glands. Histopathology. 1996;28(4):309–15.

21. Passador-Santos F, Gronroos M, Irish J, Gilbert R, Gullane P, Perez-Ordonez B, Makitie A, Leivo I. Clinicopathological characteristics and cell cycle proteins as potential prognostic factors in myoepithelial carcinoma of salivary glands. Virchows Arch. 2016;468(3):305–12.

Štítky
Detská onkológia Onkológia

Článok vyšiel v časopise

BMC Cancer

Číslo 827

2016 Číslo 827

Najčítanejšie v tomto čísle

Tejto téme sa ďalej venujú…


Kurzy

Zvýšte si kvalifikáciu online z pohodlia domova

nový kurz
Autori: MUDr. Petr Výborný, CSc., FEBO

Autori: MUDr. Jiří Horažďovský, Ph.D

Zánětlivá bolest zad a axiální spondylartritida – Diagnostika a referenční strategie
Autori: MUDr. Monika Gregová, Ph.D., MUDr. Kristýna Bubová

Krvácení v důsledku portální hypertenze při jaterní cirhóze – od pohledu záchranné služby až po závěrečný hepato-gastroenterologický pohled
Autori: PhDr. Petr Jaššo, MBA, MUDr. Hynek Fiala, Ph.D., prof. MUDr. Radan Brůha, CSc., MUDr. Tomáš Fejfar, Ph.D., MUDr. David Astapenko, Ph.D., prof. MUDr. Vladimír Černý, Ph.D.

Rozšíření možností lokální terapie atopické dermatitidy v ordinaci praktického lékaře či alergologa
Autori: MUDr. Nina Benáková, Ph.D.

Všetky kurzy
Prihlásenie
Zabudnuté heslo

Nemáte účet?  Registrujte sa

Zabudnuté heslo

Zadajte e-mailovú adresu, s ktorou ste vytvárali účet. Budú Vám na ňu zasielané informácie k nastaveniu nového hesla.

Prihlásenie

Nemáte účet?  Registrujte sa