We report two subjects with tracheobronchopathia osteochondroplastica (TO), including the clinical manifestations, histological findings, and clinical treatments, which were analyzed retrospectively. One patient with TO was a 60-year-old woman, and the other was a 47-year-old man. The main clinical manifestations were cough, chest pain, and dyspnea. Computed tomography (CT) images showed that TO mainly occurred in the trachea and main bronchus. Histological analysis showed inflammatory exudation, squamous metaplasia, submucosal cartilaginous, and ossification. We present the two cases to increase physician and patient awareness of this benign disease and to improve their understanding of the disease manifestations and potential complications.