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Xiaoyan Li, Hongjun Gao, Chuanhao Tang, Xiaoqing Liu Department of Lung Cancer, Affiliated Hospital of Academy of Military Medical Sciences, Beijing, People’s Republic of China Abstract: Sunitinib, an oral tyrosine kinase inhibitor, has been approved by the US Food and Drug Administration for the treatment of metastatic renal cell carcinoma and imatinib-refractory gastrointestinal stromal tumor. In non-gastrointestinal stromal tumor soft tissue sarcomas, the activity of this small-molecule drug has been rarely reported. Herein, we report a patient with lung metastases from renal leiomyosarcoma who responded favorably to sunitinib after the failure of conventional chemotherapy. Adverse effects of sunitinib, which include fatigue, hand-foot syndrome, and stomatitis were observed following its administration. Withdrawal of sunitinib led to progression of disease, and resuming use of sunitinib was still effective for multiple lung metastases. Sunitinib might be an effective treatment for renal leiomyosarcoma, especially when conventional chemotherapy fails. Keywords: sunitinib, gastrointestinal stromal tumor, GIST, renal leiomyosarcoma