Spinal dural arterio-venous fistula (SDAVF) is a common type of spinal vascular malformation. Surgical obliteration of the fistula can cure SDAVF anatomically, but the functional outcome is unsatisfactory.The aim of the study was to evaluate the effect of hyperbaric oxygen therapy (HBOT) on the functional recovery of postoperative SDAVF patients.This prospective cohort study included postoperative SDAVF patients. Patients were divided into control and HBOT groups. Patients in control group received conventional treatment, whereas those in the HBOT group received conventional treatment plus HBOT (2.0 atmospheric pressure absolute, 14 days). Follow-up was done at 1, 3, 6, 12, and 24 months after surgery for evaluation, including symptoms. To assess the effectiveness of HBOT on SDAVF patients, we compared the postoperative magnetic resonance imaging and neurological outcomes of each group with respect to modified Aminoff-Lougue scale and modified Denis Pain and Numbness Scale.From September 1, 2013 to January 31, 2014, 33 SDAVF patients (27 male) treated by microsurgery were included in this study. Sixteen patients were in the HBOT group and 17 patients were in the control group. At 24 months follow-up, the improvement of mDPNS for the HBOT group was significantly larger than those of the control group (2.25 vs 0.88; P=0.005). In the HBOT group, the average length of hypersignal in magnetic resonance imaging T2 image decrease at 3 months after surgery was 3.25 compared with 2.29 in the control group (P=0.009). No major adverse effects were reported for all 16 patients who received HBOT.The current findings suggest that HBOT is an effective and safe treatment to relieve lower body pain and numbness for postoperative SDAVF patients.