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Background and objective Adenoid cystic carcinoma is primary bronchopulmonary carcinoma with low malignancy, and 43 patients treated in the past 50 years in our hospital were retrospectively studied. The aim of this study is to discuss the clinical symptoms, pathologic characteristic and therapeutic method of primary tracheal or bronchuotracheal adenoid cystic carcinoma. Methods This study summarized total 43 patients of primary tracheal or bronchus adenoid cystic carcinoma treated in our hospital from Jan. 1958 to Dec. 2007. Among them, 40 patients were treated by surgical resection, and 3 patients were treated by fiberoptic bronchoscope’s interventional treatment. Results The 1-yr, 3-yr, 5-yr survival rates of the 43 patients above were 100% (41/41), 89.5% (34/38), 87.1% (27/31), respectively. Conclusion Primary tracheal or bronchus adenoid cystic carcinoma are rare and low malignancy carcinoma. The clinical symptoms of them are not typical. The best treatment is early detection and taking measures of operation plus radiotherapy. The other palliative treatment is fiberoptic bronchoscope’s interventional treatment.