BACKGROUND : Formation of a dural arteriovenous fistula (DAVF) after lumbar spine decompression in the lumbar spine has been reported as a late complication, but is extremely rare in the cervical spine. METHODS : A 57-year-old man who underwent a C5-6, C6-7 anterior fusion 5 years previously for cervical myelopathy at another hospital presented with progressive gait disturbance and thermal hypoalgesia on the right side of the body. Magnetic resonance imaging showed flow voids in the spinal cord dorsally at the C4-7 level, which had not been detected before the initial operation. Digital subtraction angiography showed a fistulous connection at the C7 level between the perimedullary vein and a segmental artery from the right vertebral artery, which was diagnosed as a DAVF. Surgical interruption of the intradural draining vein with multilevel posterior cervical decompression was performed, and his symptoms improved. CONCLUSIONS : To the best of our knowledge, this is the first report of a DAVF developing after multilevel anterior cervical discectomy and fusion. All the published cases of secondary DAVF, especially after spinal surgery, are reviewed.