Tracheopathia osteoplastica is a rare disease of unknown cause and characterized by cartilaginous or bony projection into the tracheobronchial lumen, usually not involved posterior membranous portion of tracheobronchial tree. In the past, most of the cases were diagnosed incidentally at autopsy. But after the introduction of bronchoscopy and computed tomography, antemortem diagnosis was reported. Because of initial presenting symptoms were indolent and non-specific, misdiagnosis was reported frequently and correct diagnosis was delayed usually. We report two cases of tracheopathia osteoplastica diagnosed by fiberoptic bronchoscopic biopsy.