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Introduction: Neuromyelitis optica (NMO) is an inflammatory demyelinating disease with predilection for thespinal cord and optic nerves. In relation to Multiple Sclerosis, has lower prevalence,however, the number and severity of relapses is higher, with a worst responseto acute phase treatment.Methods: Report of two cases Case description: Patient1: JKS, female, presented her first outbreak at seven yearsold with tetraparesia. MRI: extensive transverse myelitis. Nowadays she is14years old and presented five other outbreaks. MRI showed inflammation ofoptical nerves, what may represent a subclinical outbreak. Asymptomatic usingazathioprine and oral prednisone - EDSS 1,0.Patient 2: MAS, female, fist symptoms at 11 years old with a seriesof self-limited episodes of crural paresis. At 14, presented tetraplegia.  She did not search medical attention for thissevere episode wich resolved spontaneously. At 15, presented blurred vision thatprogressed to bilateral amaurosis. After three days of pulsotherapy withsteroids she partially recovered vision. MRI: longitudinally extensive hyperssinal.Admitted at our hospital with irregular usage of oral prendisone, changed tomonthly pulsotherapy for non-adherence. Nevertheless presented another threeoutbreaks. Present medication is azathioprine plus monthly pulsotherapy,without clinical relapses, but with great sequelae (EDSS 5.5).Both patients were positive for anti-aquaporin 4 antibody.Discussion:We present two distinct cases and consider differences in treatment and outcomeaccording to the time of diagnosis and treatment, considering early recognitionand  adherence to treatment as importantvariables for reducing future outbrakes and sequelae.