Optic neuritis is an inflammatory demyelinating disorder of the central nervous system. Anti-NMDA receptor (NMDAR) antibodies encephalitis is also mediated by the inappropriate immunological response. We report a girl who was readmitted because of unilateral eye pain and vision impairment in the remission period of anti-NMDAR encephalitis. Based on the limited available literature on the treatment of optic neuritis associated with anti-NMDA receptor antibody, she was treated with a combination of corticosteroids and intravenous immunoglobulin with clinical improvement. Optic neuritis in the remission phase of anti-N-methyl-D-aspartate receptor encephalitis remains relatively uncommon. It is important to document and follow these patients to understand the pathogenesis of optic neuritis in the setting of anti-NMDAR encephalitis and formulate effective therapeutic strategies.