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A case of adenoid systic carcinoma (cylindroma) of the trachea is reported. The patient was treated by radiation and survived 9 years after diagnosis, dying eventually due to mediastinal involvement. The literature of the clinical and the pathologic features of this tracheal neoplasm is discussed, and the observed histologic polymorphism and the nonbenign nature of the lesion are stressed. For a better long‐term prognosis, wide tracheal resection and reconstruction is suggested as the method of treatment.

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