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Introduction: Influenza is an acute febrile infectious disease with respiratory manifestations, in a variable degree of severity, and a seasonal epidemic distribution. In healthy children, it usually presents as mild and self-limiting, but it can evolve into severe forms with significant morbidity and mortality. These include neurological manifestations such as influenza-associated encephalopathy and acute necrotizing encephalopathy. Objective: To report case of a previously healthy child who developed severe pneumonia due to influenza B and subsequent blindness associated with ANE. Case Report: previously healthy, five years old male, not vaccinated for seasonal influenza, was admitted with four days of fever and respiratory symptoms. Initial evaluation reveled respiratory impair and he was admitted to the critical care unit for respiratory distress syndrome. Influenza B antigen was detected in upper respiratory swab by immunochromatography. Initial laboratory: White blood count; 20300/L, C- protein reaction: 300mg/L, AST 176 U/L, LDH 2549 U/L. He required 11 days of invasive respiratory support due to severe hypoxemia. When support was withdrawn, severe bilateral visual impairment was observed without other neurological manifestations. Magnetic Resonance Image revels bilateral temporo-occipital cortico-subcortical lesions and in both cerebellar hemispheres, with a lacunar image in the left thalamus; no involvement of the optic nerve was observed. Close watching was workup without pharmacologic intervention. He starts recovering vision 5 month later. Discussion: influenza-associated encephalopathy and acute necrotizing encephalopathy are recognized as rare inflammatory complications associated with influenza and other virus infections. Cases of blindness associated with necrotizing encephalopathy are reported. Treatment with corticosteroids, intravenous immunoglobulin, hypothermia, and plasmapheresis are propose; roll of oseltamivir remain unknown. Tocilizumab represents a potentially rational approach to ANE management. Conclusion: Influenza infection could be severe, including neurological complication. Vaccination is the only primary prevention tool.