Myoepithelioma is a benign tumor composed of sheets and islands of various proportion of spindle, plasmacytoid, epitheloid, and clear cells. We are reporting of a 38-year-old woman with an extremely rare neoplasm of the trachea, myoepithelioma. The patient had an right neck mass and diagnosed presumptively as the thyroid tumor with tracheal invasion. Resection and anastomosis of the trachea with partial thyroidectomy was done. The tumor was a well circumscribed mass with solid growth pattern and composed of spindle and epitheloid cells, which were positive for S-100 protein and smooth muscle actin. In electron microscopy, a large amount of microfilaments in the cytoplasm and layers of basement membrane-like materials in the intercellular spaces were observed, which are characteristics of myoepithelioma. Patient has been well for 8 months postoperatively.