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Hisashi Tanaka,1 Takeshi Morimoto,1 Kageaki Taima,1 Yoshihito Tanaka,1 Kunihiko Nakamura,1 Akihito Hayashi,3 Akira Kurose,2 Ken Okumura,1 Shingo Takanashi41Department of Cardiology, Respiratory Medicine and Nephrology, Hirosaki University Graduate School of Medicine, Zaifu-cho, Hirosaki, Japan; 2Department of Anatomic Pathology, Hirosaki University Graduate School of Medicine, Zaifu-cho, Hirosaki, Japan; 3Respiratory Medicine, Hachinohe City Hospital, Tamukaibishamontai, Hachinohe, Japan; 4Hirosaki University, Health Administration Center, Bunkyo, Hirosaki, JapanBackground: Thymic carcinoma is a rare neoplasm of the thymus. Systemic chemotherapy is an important therapeutic modality for thymic carcinoma. However, no standard chemotherapy for this carcinoma has yet been established. The usefulness of second-line or later-line chemotherapy has remained unclear. A case of relapsed thymic carcinoma that was successfully treated by S-1 as second-line chemotherapy is reported herein.Case presentation: A 73-year-old man diagnosed as having thymic carcinoma was treated with three cycles of first-line chemotherapy with ADOC (cisplatin, doxorubicin, vincristine, and cyclophosphamide) and additional radiotherapy (50 Gy). Since his serum cytokeratin 19 fragment level increased suddenly after 3 months of stable disease, he was considered to have progressive disease, and was given S-1 as chemotherapy. Two months later, he had partial response, and the S-1 treatment has been continued since July 2009. Progression-free survival of greater than 4 years was obtained with S-1.Conclusion: A case of relapsed thymic carcinoma that was treated with S-1, and continues to show a long progression-free survival with good quality of life on treatment is described. S-1 might be an active agent against relapsed thymic carcinoma.Keywords: thymic carcinoma, S-1, thymidylate synthase