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Inflammatory myofibroblastoma tumor (IMT) is rare neoplasm that has high recurrence rate and low likelihood of metastasis. Here, we report the efficacy and safety of microwave ablation (MWA) combined with transarterial embolization in a patient with unresectable retroperitoneal IMT measuring 89 mm in maximum diameter. Multiple courses of MWA, the last of which was performed under magnetic resonance imaging guidance, and one course of transarterial embolization resulted in complete response without complications. After more than 1.5 years of the last MWA treatment, follow-up showed sustained complete response. This case suggested that the combination of MWA and transarterial embolization is a new option for retroperitoneal IMT. A multicenter and large-sample clinical trial is required to confirm the efficacy and safety of MWA combined with transarterial embolization for retroperitoneal IMT.