Dermatofibrosarcoma protuberans (DFSP) is one of the most common dermal sarcomas, but facial DFSP is rare. The clinicopathological characteristics of 34 facial DFSPs were reviewed. Additional immunostaining (CD34) and PDGFB/COL1A1-PDGFB fluorescence in situ hybridization (FISH) detection were performed. Patients were aged from 24 to 64 years (mean 42.9 years), with a male-to-female ratio of 4.7: 1. Morphologically, classic DFSP (25/34, 73.5%), pigmented DFSP (2/34, 5.9%), DFSP with myoid differentiation (1/34, 2.9%) and fibrosarcomatous DFSP (FS-DFSP) (6/34, 17.6%) were found. Moreover, myxoid degeneration was observed in three FS-DFSP cases (3/6, 50.0%). All 29 cases that underwent CD34 immunohistochemistry exhibited positive staining (100%). Genetically, PDGFB rearrangement/COL1A1-PDGFB fusion was detected in 94.1% (16/17) of patients. Regarding prognosis, the recurrence (83.3% vs. 59.1 %) and metastasis (33.3% vs. 0%) rates were higher in FS-DFSPs than that in ordinary DFSPs. All available surgical margins were positive before DFSPs recurrence. All patients with negative excision or re-excision margins were alive without evidence of disease (mean, 81.8months; median, 81months). Facial DFSP occurs predominantly in males, while FS-DFSPs are more likely to exhibit myxoid degeneration and a worse prognosis. Notably, negative surgical margin status determined a satisfactory prognosis.