Myxoid liposarcoma occurs predominantly in the deep soft tissues of the extremities, and tends to metastasize to a wide range of soft tissue or bone locations. We report a case of myxoid liposarcoma arising in the ankle of a 19-year-old man. A diagnosis of myxoid liposarcoma was made by open biopsy, and a wide resection requiring plastic surgical reconstruction was performed. Cytogenetic analysis of the resected tumor exhibited a reciprocal translocation t(12;16)(q13;p11) as the sole anomaly, which is found in more than 90% of myxoid liposarcoma cases. The patient had no evidence of local recurrence or metastasis within 7 years of follow-up. Although myxoid liposarcoma is rare in the ankle, it should be considered in the differential diagnosis of a painless soft tissue mass in this region.