Purpose: To describe the outcome of a patient with visual loss and optic nerve edema that resulted from osteopetrosis who underwent an optic nerve sheath fenestration (ONSF). Design: Interventional case report. Methods: A 33-year-old man with osteopetrosis had bilateral visual field loss and optic nerve edema. Computed tomography and magnetic resonance imaging demonstrated open optic canals. Although a lumbar puncture showed a normal opening pressure, there were other findings that were suggestive of increased intracranial pressure. The patient elected to undergo a unilateral optic nerve sheath fenestration. Results: After ONSF, the patient experienced markedly improved visual acuity, visual field, and optic nerve appearance. Conclusion: Individuals with visual loss and optic disk edema that is associated with osteopetrosis may benefit from ONSF if the optic canals appear to be open and the optic nerve edema is thought to be related to increased intracranial pressure.
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