Dermatofibrosarcoma protuberans (DFSP) is a rare cutaneous sarcoma. Limited population-based epidemiological studies on DFSP have been conducted. We aimed to estimate the incidence and disease burden of DFSP in China. We conducted a cross-sectional study using data from the national databases of the Urban Basic Medical Insurance scheme. Cases were identified by ICD code and Chinese language diagnostic terms. National incidence from 2014 to 2016 was estimated by gender and age, and associated medical costs were calculated. A total of 175 patients were confirmed with DFSP from 2014 to 2016. Crude incidence varied from 0.353 per 100,000 (95%CI: 0.203-0.503) in 2014 to 0.367 per 100,000 (95%CI: 0.279-0.455) in 2016. Incidence was higher in males than in females. The first incidence peak was observed between the ages of 20 and 39 years and the highest incidence rates were in those aged over 60 years. Average medical costs of DFSP were higher than the per capita disposable income of residents. Incidence of DFSP in mainland urban China is lower than in most developed countries and has remained relatively stable from 2014 to 2016. Further research is expected to clarify the potential pathophysiological mechanisms of DFSP.