A 48-year-old Chinese male patient was admitted to our hospital with headache and bilateral lower limb weakness for 1 month. He had no previous history of specific diseases, no family history of diseases, and no history of radiation exposure. On admission, CT scan of the head revealed uneven high-density mass at bilateral frontal parietal lobe. The T1—and T2-weighted images of the head MRI showed mixed and uneven mass signals with insignificant surrounding edema. MRI enhancement showed uneven and obvious enhancement of the lesion, involving bilateral parasagittal dura and sagittal sinus, and local brain tissue compression. Subsequently, we performed bilateral tumor resection for the patient. During the operation, we found that the tumor originated from the dura mater. The tumor was found to be soft and reddish-brown, and the frontal and parietal cortex adjacent to the tumor was brownish yellow, which may be related to hemosiderin deposition. The tumor contained a large amount of brownish yellow fluid, and local skull and brain tissue were found to be invaded. Because the lesion was located in front of the motor center of the brain, the affected brain tissue could not be completely removed in order to preserve the motor function of the limbs. After surgery, the excised mass and the affected dural tissue were sent for pathological examination, and the final pathological result was angiosarcoma. Hematoxylin and eosin staining of tissue samples showed a large number of new abnormal blood vessels, marked cell atypia, and marked nuclear staining. The tumour cells were positive for the vascular markers such as CD31, CD34, and ERG, and negative for S-100, pancytokeratin, and epithelial membrane antigen. Ki-67 showed a hight index of positivity within the tumour cells. The patient underwent further chemotherapy and radiotherapy postoperatively. Nine months after operation, no tumor recurrence was found on enhanced MRI. The patient had no obvious headache, his limb motor function was normal, and there was no seizure after operation.