| Summary: | We report an atypical manifestation of Antiphospholipid syndrome (APS) in a 40-year-old male with nonartertic anterior ischemic optic neuropathy (NAION) presenting with 12-day history of a headache and left eye pain not associated with visual changes. On his initial evaluation, our differential diagnoses included optic neuritis, ischemic optic neuropathy (ION), and intracranial hemorrhage. Onset and course of the disease, lack of vision changes as well as the presence of retinal hemorrhages on fundoscopic examination excluded optic neuritis. The diagnosis was made after exclusion of all other causes including infectious and noninfectious causes of optic neuritis, ION, and intracranial hemorrhage. This case demonstrates the importance of including APS as a differential diagnosis of NAION in middle-aged individuals.
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