Background: Neuromyelitis optica (NMO, Devic's syndrome) is an immune mediated inflammatory demyelinating disease of the central nervous system that predominantly affects the spinal cord and optic nerves. Often confused with multiple sclerosis (MS), early diagnosis and differentiation from MS is important because of different treatment regimens. Case Report: We hereby report a sero-negative mono-phasic manifestation of this rare disease in a nine year old Indian boy. NMO was diagnosed in our case despite being sero-negative because of typical clinical findings and evidence of myelitis and optic neuritis seen in MRI. Patient was treated with pulse methyl-prednisolone followed by oral prednisolone and patient improved with some residual deformity. Conclusion: High index of suspicion is required to diagnose and treat patients at an early stage to prevent co-morbidities and complications.
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