Complex multiple arteriovenous fistula (AVF) of the scalp is an extremely rare disease and the traditional treatment is either endovascular embolization or surgical resection with preoperative endovascular embolization. Here, a 34-year-old male with pulsating mass and multiple tortuous vessels in front of the left tragus and scalp who was diagnosed as complex multiple arteriovenous fistula of the scalp is presented. Nearly total surgical resection of the lesion without preoperative intervention was performed. The deformity appearance and the buzzing sound in the patientÂ’s left ear were eradicated after operation. Therefore, surgical resection without preoperative embolization is still a promising method to treat complex multiple scalp AVF.