Abstract
Optic perineuritis (OPN) involvement in demyelinating disease is rarely encountered. To our knowledge, this is the first reported case of bilateral OPN associated with neuromyelitis optica spectrum disorder (NMOSD). We present a case of a healthy young gentleman who presented with OPN, initially presumed to have a young stroke but later diagnosed to be NMOSD. Early neuroimaging is essential to help distinguish optic neuritis (ON), and prolonged treatment of systemic immunosuppression is the mainstay of treatment.