ABSTRACT
Background Acute necrotic encephalopathy（ANE） in children is a very rare complication of Severe Acute Respiratory Syndrome Coronavirus 2（SARS-CoV-2） infection, which has rarely been reported worldwide. Case presentation A 45-day-old girl was admitted to our hospital with fever and listlessness. A nose swab tested positive for novel coronavirus nucleic acid, and her cerebrospinal fluid was positive for SARS-CoV-2. An early head magnetic resonance imaging (MRI) scan indicated multiple abnormal signals in her bilateral cerebral hemispheres, and encephalitis was diagnosed. Twenty-three days after hospitalization, bilateral cerebral atrophy-like changes were observed by MRI, with multiple softening lesions in the bilateral cerebral hemispheres, accompanied by convulsions. She was admitted to hospital for mechanically-assisted ventilation, and her condition improved after treatment of her symptoms, including anti-epileptic medication, anti-infection drugs, glucocorticoids, and immunoglobulins. Conclusions Acute necrotic encephalopathy associated with novel coronavirus infection in children should be detected and treated as early as possible. Satisfactory short-term efficacy can be obtained, but long-term neurological sequelae often linger.