Abstract
Purpose
We report the diagnosis, treatment outcomes and prognosis of the patients with soft tissue malignant myoepithelioma in the extremities.
Materials and Methods
We retrospectively reviewed 6 patients with soft tissue malignant myoepithelioma in the extremities who were treated at our institution between 2008 and 2014. Two patients received unplanned excision at another hospital and remaining 4 patients underwent the biopsy procedures and received wide excision at our hospital.
Results
There were 3 men and 3 women with mean age of 41 (33−54) years. The average follow up was 28 (9−45) months. Among the 6 patients, only 4 patients underwent biopsy procedures under the impression of malignant soft tissue sarcoma. Surgical margins for these 4 patients were negative. Two patients who had unplanned excision received another re-excision and one of them showed no residual tumor in the resected specimen. Local recurrences were developed in all patients and distant metastasis in 4 patients. All 4 patients who developed distant metastasis died due to disease progression. Among the 2 patients who developed local recurrence only, one patient has another local recurrence after reoperation and remaining one patient is no evidence of disease for 2 years after resection of locally recurred mass.
References
1. Sheldon W. So-called mixed tumors of the salivary glands. Arch Pathol. 1943; 35:1–20.
2. Bigotti G, Di Giorgio CG. Myoepithelioma of the breast: histologic, immunologic, and electromicroscopic appearance. J Surg Oncol. 1986; 32:58–64.
3. Tuncel U, Ergul G, Ozlugedik S, Unal A. Myoepithelial carcinoma in the nasopharynx: an unusual localization. Yonsei Med J. 2004; 45:161–5.
4. Martínez-Madrigal F, Santiago Payán H, Meneses A, Domínguez Malagón H, Rojas ME. Plasmacytoid myoepithelioma of the laryngeal region: a case report. Hum Pathol. 1995; 26:802–4.
5. Hornick JL, Fletcher CD. Cutaneous myoepithelioma: a clinicopathologic and immunohistochemical study of 14 cases. Hum Pathol. 2004; 35:14–24.
6. Veeramachaneni R, Gulick J, Halldorsson AO, Van TT, Zhang PL, Herrera GA. Benign myoepithelioma of the lung: a case report and review of the literature. Arch Pathol Lab Med. 2001; 125:1494–6.
7. Alberghini M, Pasquinelli G, Zanella L, et al. Primary malignant myoepithelioma of the distal femur. APMIS. 2007; 115:376–80.
8. Park JS, Ryu KN, Han CS, Park YK. Malignant myoepithelioma of the humerus with a satellite lesion: a case report and literature review. Br J Radiol. 2010; 83:e161–4.
9. Burke T, Sahin A, Johnson DE, Ordóñez NG, Mackay B. Myoepithelioma of the retroperitoneum. Ultrastruct Pathol. 1995; 19:269–74.
10. Lee JR, Georgi DE, Wang BY. Malignant myoepithelial tumor of soft tissue: a report of two cases of the lower extremity and a review of the literature. Ann Diagn Pathol. 2007; 11:190–8.
11. Hornick JL, Fletcher CD. Myoepithelial tumors of soft tissue: a clinicopathologic and immunohistochemical study of 101 cases with evaluation of prognostic parameters. Am J Surg Pathol. 2003; 27:1183–96.
12. Gleason BC, Fletcher CD. Myoepithelial carcinoma of soft tissue in children: an aggressive neoplasm analyzed in a series of 29 cases. Am J Surg Pathol. 2007; 31:1813–24.
13. Mahdi Y, Zouaidia F, Zouhair A, et al. Combined myoepithelial carcinoma and myoepithelioma in soft tissue: a case report and review of the literature. J Med Case Rep. 2014; 8:317.
14. Harada O, Ota H, Nakayama J. Malignant myoepithelioma (myoepithelial carcinoma) of soft tissue. Pathol Int. 2005; 55:510–3.
15. Combalía A, Marco V, Seijas R, Domínguez R. Rare presentation of a soft-tissue myoepithelial carcinoma. J Orthop Sci. 2013. [Epub ahead of print].
16. Neto AG, Pineda-Daboin K, Luna MA. Myoepithelioma of the soft tissue of the head and neck: a case report and review of the literature. Head Neck. 2004; 26:470–3.
17. Rastrelli M, Passuello N, Cecchin D, Basso U, Tosi AL, Rossi CR. Metastatic malignant soft tissue myoepithelioma: a case report showing complete response after locoregional and systemic therapy. J Surg Case Rep. 2013; 2013:pii: rjt109.
18. Kilpatrick SE, Hitchcock MG, Kraus MD, Calonje E, Fletcher CD. Mixed tumors and myoepitheliomas of soft tissue: a clinicopathologic study of 19 cases with a unifying concept. Am J Surg Pathol. 1997; 21:13–22.
19. Nagao T, Sugano I, Ishida Y, et al. Salivary gland malignant myoepithelioma: a clinicopathologic and immunohistochemical study of ten cases. Cancer. 1998; 83:1292–9.
20. Bombí JA, Alós L, Rey MJ, et al. Myoepithelial carcinoma arising in a benign myoepithelioma: immunohistochemical, ultrastructural, and flow-cytometrical study. Ultrastruct Pathol. 1996; 20:145–54.