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Acute necrotizing encephalopathy of childhood (ANEC) is an extremely rare disease exclusively recorded in previously healthy infants and children. This disease has a broad clinical, radiological, and biochemical spectrum as well as high morbidity and mortality rates. Symmetric lesions in the thalami are the diagnostic hallmarks. We have reported here a previously healthy girl (age: 4 years and 8 months) who presented with intermittent fever and vomiting. She, very soon, developed generalized tonic seizures and rapid loss of consciousness. Cultures of her nasal swab specimen revealed swine influenza H1 (swH1) virus. Her brain imaging demonstrated the typical findings of ANEC. A combination of oseltamivir, immunoglo