Liposarcoma as one of the most common soft-tissue sarcomas in adults rarely occurs in oral and maxillofacial region. Its rarity makes a systematic and in-depth understanding of these uncommon entities extremely difficult. Here we aimed to characterize and analyze the epidemiology, clinicopathologic characteristics, treatment and prognosis of primary oral and maxillofacial liposarcomas by presenting our experience from a Chinese tertiary referral hospital and literature review. All relevant information was manually collected from medical records from the authors' department (1993-2009) and literature retrieval (Jan.1940-Sep.2009). A total number of 150 patients were identified and included with mean age 49.1 years old and a slight male preponderance. These lesions mostly occurred in tongue (32.0%) followed by buccal region (28.7%) and were diagnosed as atypical lipmatous tumor/well-differentiated (58.45%) and myxoid liposarcomas (27.5%) by pathologic re-evaluation and re-categorization based on 2002 WHO classification scheme. Complete surgical excision with negative margins was the primary treatment modality, while therapeutic utilities of adjuvant radiotherapy/chemotherapy remained controversial. Our data reinforced histopathologic subtype as one of key prognostic factors irrespective of gender, age and primary sites for this malignancy. More importantly, our analysis further revealed that tumor size (especially when larger than 3.6 cm) served as another important prognostic factor suggesting higher rates of disease-related death. Taken together, these findings might for the first time provide comprehensive information regarding the epidemiology, clinicopathologic features, treatment and prognosis of oral and maxillofacial liposarcoma.