A rare case of tracheopathia osteochondroplastica complicated with malignancy is reported. Further examination of hemosputum following thyroid cancer operation revealed an abnormal finding of the trachea, suggesting the recurrence of thyroid cancer. However, the serum level of thyroglobulin was within the normal range. In contrast, the findings of bronchofiberscopy and the histological analysis confirmed the presence of tracheopathia osteochondroplastica. Therefore, measurement of serum thyroglobulin level and the bronchofiberscopy procedure were valuable for the differential diagnosis of metastasis of thyroid cancer in this case. Additionally, case reports of tracheopathia osteochondroplastica in Japan are summarized.