A 67-year-old woman who was followed as a patient with bronchial asthma for 1.5 years visited our hospital with progressive dyspnea. Although the chest radiography findings were normal, a chest computed tomography scan revealed a mass obliterating the intrathoracic tracheal lumen. The patient's symptoms disappeared immediately after tumor excision, and no recurrence was observed during a 1.5-year follow-up period. Microscopically, the tumor was composed of densely packed polygonal-, oval- and spindle-shaped cells that were positive for pan-cytokeratin, α-smooth muscle actin and p63. These pathological findings confirmed the diagnosis of benign myoepithelioma. Chest physicians should recognize that benign myoepithelioma can develop in the trachea, although it is very rare.