Benign Myoepithelioma in the Intrathoracic Trachea

Abstract

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.

Tracheal myoepithelioma resected by using rigid bronchoscopy: a case report and review of the literature

“…To the best of our knowledge, to date, only four cases of benign myoepitheliomas in the trachea have been reported in the literature, excluding our case [ 7 – 10 ]. Generally, primary tracheal tumors are rarely seen.…”

“…Since Strickler et al [ 17 ] presented the first case of myoepithelioma in the lung, only a few studies have been reported these neoplasms in the respiratory tract [ 16 , 18 , 19 ], with the lung, bronchus, and trachea as reported locations of the tumors in the pulmonary organs. To the best of our knowledge, excluding our case, only four cases of endotracheal myoepithelioma were reported in the literature [ 7 – 10 ]. Table 1 reviews clinical manifestations, pathology, immunohistochemistry, treatment, and follow-up of these cases.…”

“…For instance, surgical intervention is necessitated in conditions as our case, in which the location of the tumor causes near-total obstruction of the trachea that may lead to serious complications. As shown in Table 1 , all previously recognized benign myoepitheliomas in the trachea have undergone surgical resection, of which two were excised with endoscopic snare and cautery [ 7 , 8 ], and two were treated with resection and anastomosis of the trachea [ 9 , 10 ]. It is worth mentioning that, in the case reported by Pfeiffer et al [ 9 ], multiple recurrences occurred following multiple endoscopic procedures.…”

“…Tracheal tumors include less than 0.2% of all tumors in the respiratory tract [ 6 ], while extremely rare cases of endotracheal myoepitheliomas have been reported in the literature [ 7 – 10 ]. Despite their benign nature, surgical intervention is necessary in most cases due to complications.…”