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Moyamoya disease can be associated with autoimmune disease such as thyrotoxicosis, but there has been only one report of association with neuromyelitis optica (NMO). We report another case of this combination with the presence of anti-SSA antibody in addition to the NMO-immunoglobulin G. The patient presented limb weakness along with unsteady gait and numbness. A magnetic resonance imaging (MRI) of the spine showed extensive intramedullary hyperintense signals at C2 to T3 levels. On the otherdoi:10.1093/omcr/omu006 pmid:25988010 pmcid:PMC4369969 fatcat:6nel4s3qwjg4zdkdhu4anxjssy