• Yi Luo, Li Min, Yong Zhou, Fan Tang, Minxun Lu, Hongmei Xie, Yitian Wang, Hong Duan, Wenli Zhang, and Chongqi Tu.
• Department of Orthopedics, West China Hospital, Sichuan University, Chengdu, Sichuan, P. R. China.
• Medicine (Baltimore). 2021 Mar 26; 100 (12): e25262e25262.

IntroductionMyxofibrosarcoma (MFS) is a locally aggressive tumor and has the potential to be fatal because of distant metastasis. Immunotherapy targeting either programmed cell death protein 1 (PD-1) or programmed death ligand 1 (PD-L1) has recently shown a curative effect on multiple cancers including melanoma, non-small cell lung cancer, and renal cell carcinoma. Although the immunotherapy has been applied in sarcoma, there is little information about the efficiency to treat metastatic MFS.Patient ConcernsA 42-year-old male presented to the clinic with a mass in the left thigh. Mass resection and ligament replacement surgery were performed.DiagnosesThe patient was diagnosed as high-grade MFS (federation nationale des centres de lutte contre le cancer, Grade 3) with pulmonary metastasis.InterventionsIn the past few years, he was treated with surgery, chemoradiotherapy, and Anlotinib (an angiogenesis inhibitor), but the metastatic lesion continued to progress. About 40% to 50% of tumor cells in his pulmonary tissues were showed positive PD-L1 expression and his tumor mutational burden was 215Muts. Thus, he received Camrelizumab (PD-1 inhibitor).OutcomesSix months after the initiating immunotherapy of Camrelizumab, the size of pulmonary lesions showed marked shrinkage, indicating a partial response. After a follow-up of 18 months, the patient remained in good condition without progressive disease.ConclusionThis case described here demonstrated that immunotherapy of PD-1 inhibitor is a promising treatment option for refractory MFS with PD-L1 positive or tumor mutational burden -high, which could contribute to effective tumor response.Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.

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