Optic perineuritis is an uncommon inflammatory disorder that involves optic nerve sheath. Numerous case reports have been published on optic perineuritis in adults, the majority of whom had bilateral presentation. There are limited data on optic perineuritis occurring in pediatric patients. We report a teenager who presented with a unilateral sign that mimicked the presentation of optic neuritis. The orbit and brain magnetic resonance imaging confirmed features of unilateral optic perineuritis. She was treated with a high dose of corticosteroids for 2weeks, and her final visual outcome was satisfactory. No signs of relapse were noted during follow-up visits.