Shengfang Bao, Hua Huang, Yingying Jin, Fei Ding, Zhen Yang, Xuemei Xu, Chenxi Liu, Xiqiong Han, Liping Wang, Jingyi Lu, Jiani Ma, Yanliang Jin Department of Rheumatology & Immunology, Shanghai Children’s Medical Center, School of Medicine, Shanghai Jiao Tong University, Shanghai, 200127, People’s Republic of ChinaCorrespondence: Yanliang Jin, Email jinyanliang@scmc.com.cnAbstract: Kikuchi-Fujimoto disease (KFD), also known as histiocytic necrotizing lymphadenitis, is a rare, benign, and self-limiting condition characterized by lymph node inflammation. While KFD is rarely associated with ocular manifestations, our case report highlights bilateral optic neuritis in a 13-year-old male patient with KFD. We also provide a comprehensive review of similar cases in the literature.Keywords: Kikuchi-Fujimoto disease, optic neuritis, neuromyelitis optica spectrum disorder, vision deficiency