This is a case report of a patient who underwent an uneventful surgery for atlanto-axial dislocation in the prone position, after which he developed painless, unilateral loss of vision in the immediate postoperative period. Based on the ophthalmologic findings a probable diagnosis of ischemic optic neuropathy (ION) was made. Although he recovered his visual acuity completely in 1 month, the visual field defects and color vision abnormalities persisted. Intraoperative anemia, hypotension, with or without vasculopathic risk factors, and prolonged surgery in the prone position have been reported as major risk factors for the development of this complication following spine surgery. However, this healthy young man had an uneventful surgery with no such intraoperative complications. ION in this patient could have been due to a combination of factors, such as a malpositioned horseshoe headrest and surgery performed in the prone position, both of which have the potential to raise the intraocular pressure and lower the perfusion pressure of the optic nerve/nerve head. Variations in the blood supply of the optic nerve due to the presence of watershed zones could be another explanation for this dreaded complication.