Purpose: Liposarcoma is the most common soft tissue sarcoma, and usually occurs on the thigh or in the retroperitoneal space, but rarely in the oral region. This report presents a case of liposarcoma of the cheek and includes a review of the literatures. Methods: A 21-year-old woman was admitted with a palpable mass in her cheek of about two years duration, which increased in size gradually initially, but had increased rapidly over the three months. There was no particular pain or tenderness. MRI showed a well-enhanced, well-defined mass, which suspected to be hemangioma. Results: The spherical, well-encapsulated mass was surgically excised. Biopsy results revealed myxoid liposarcoma. FDG PET-CT on the seventh postoperative day, revealed a minimal to mild FDG-uptake soft tissue lesion around the mass defect area without evidence of distant metastasis. The patient is being observed and undergoing radiation therapy. Conclusion: Liposarcoma in the head and neck region is a rare disease, and can be overlooked as a benign tumor without a pathologic diagnosis. Therefore, proper treatment and follow-up are required based on an understanding of this disease.