Objective To improve the recognization of long term interval between optic neuropathy and spinal cord injury of neuromyelitis optica. Methods One 51-year old male patient with 37 years'interval between optic neuropathy and spinal cord injury of neuromyelitis optica underwent the examination of plasma and cerebrospinal fluid and head and spinal MRI examinations, who was also followed up. His clinical data were analyzed and related literature was reviewed. Results The myelin basic protein and IgG index in his cerebrospinal fluid was high, his oligoclonal band of cerebrospinal fluid was positive, and abnormal finding in visual evoked potential. Abnormal intramedullary long T2 signals was showed in spinal cord MRI at T1-7 segment. When dignosed as neuromyelitis optica, the clinical symptom and signs was improved with corticosteroid and gamma globulin therapy. The patient was in stable condition at present.Conclusions There could be a long term interval between optic neuritis and myelitis. One should pay attention to clinical features and imaging examination of subclinical lesion in spinal cord and brain and conoide the possibility of developing neuromyelitis optica or multiple sclerosis.