Immediate visual loss following craniotomy in the supine position is a disastrous complication in neurosurgical patients. The incidence is unknown and little is known on the definite pathogenesis. Also, preventive or restorative interventions are unclear. We describe the rare case of post-craniotomy optic neuropathy and sudden visual loss after craniotomy in the supine position for an olfactory groove meningioma, discuss the possible pathophysiology and review the literature on the pathogenesis, risk factors, and outcome. Although rare, neurosurgeons, as well as neuroanesthesiologists should be aware of the possibility of this devastating complication in the high-risk group of patients.