Dermatofibrosarcoma protuberans (DFSP) of the scalp is a rare soft tissue sarcoma. A 22-year-old male with DFSP, who had undergone several surgical excisions and radiotherapy, presented with a mild left lower-limb paresis. An MRI scan demonstrated a huge mass extending throughout the frontoparietal convexity. A macroscopically complete removal of the mass was performed. Despite this, there were several recurrences; thereafter, the patient underwent surgery, stereotactic radiosurgery and chemotherapy. The histological findings demonstrated a DFSP that both abutted and occasionally invaded the adjacent brain cortex, while extending along the Virchow-Robin spaces. Due to the highly infiltrating characteristics of DFSP, another approach, such as the use of molecular-targeted agents, will be required to significantly improve the clinical outcome.