Background: Tracheobronchopathia osteochondroplastica (TO) is a rare benign disease. We report a case of TO. Methods: Chest CT scan and bronchoscope with biopsy was performed for diagnosis and blood tests explored for the latent etiology. Results: Chest CT scan and bronchoscopic images showed multiple nodular protrusions in the trachea and main bronchi. Histopathology demonstrated sub-mucosal ossification and inflammatory cell infiltration. Laboratory inspection showed adenovirus and coxsackie B virus IgM antibodies were positive. Conclusions: The patient recently had a virus infection and inflammation was observed in histopathology, which indicated adenovirus and coxsackie B virus may play a role in the occurrence or exacerbation of TO.