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Background: Spinal dural arteriovenous fistulas (DAVF) in the cervical spine are known to cause subarachnoid hemorrhage. Vasospasm after rupture of a DAVF, however, has not previously been reported., Case Presentation: A 48-year-old woman who presented with the sudden onset of altered mental status. Imaging demonstrated extensive subarachnoid hemorrhage and spinal DAVF at C1 to C2. The patient underwent a suboccipital craniotomy for DAVF ligation. On post-operative day three, she began having acute weakness in all her extremities with proprioception and vibration preserved, whereas pain and temperature sensation was lost. An angiogram demonstrated bilateral vertebral artery vasospasm with no filling of the anterior spinal artery. Bilateral angioplasty of the vertebral arteries was performed successfully and post-angioplasty, the right vertebral artery was filling the anterior spinal artery. The patient clinically improved. She subsequently required treatment with n-butyl cyanoacrylic acid (nBCA) embolization and gamma knife radiosurgery to achieve obliteration of the lesion., Conclusions: For patients with subarachnoid hemorrhage of unknown origin, differential diagnosis should include DAVF. This patient also presented with vasospasm in the context of ruptured DAVF, a complication previously unreported in the literature. This finding suggests that close monitoring for vasospasm after rupture of DAVF is warranted., (Copyright © 2011 by the American Society of Neuroimaging.)