| Summary: | Abstract High‐grade myofibroblastic sarcoma is a rare mesenchymal tumor with a high recurrence and metastatic rate. Few cases of high‐grade myofibroblastic sarcomas have been reported. Herein, we report a rare case of undifferentiated, high‐grade myofibroblastic sarcoma with an unclear primary site, initially presenting with oral symptoms. High‐grade myofibroblastic sarcoma was diagnosed following an excisional biopsy of a gingival tumor. After this excisional biopsy, systemic imaging revealed multiple metastases in the tonsil, lung, liver, kidney, and eye. The patient underwent two cycles of chemotherapy (doxorubicin). During follow‐up, the tumor progressed rapidly and metastasized to the skin of the head and neck. The patient expired three months after the initial examination.
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