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Acute necrotizing encephalopathy is characterized by multiple, symmetrical lesions involving the thalamus, brainstem, cerebellum, and white matter and develops secondarily to viral infections. Influenza viruses are the most common etiological agents. Here, we present the first case of acute necrotizing encephalopathy to develop secondarily to human bocavirus.A 3-year-old girl presented with fever and altered mental status. She had had a fever, cough, and rhinorrhea for five days. The patient was admitted to the intensive care unit with an initial diagnosis of encephalitis when vomiting, convulsions, and loss of consciousness developed. Signs of meningeal irritation were detected upon physical examination. There was a mild increase in proteins, but no cells, in the cerebrospinal fluid (CSF). Brain magnetic resonance imaging showed symmetrical, heterogeneous hyperintensities bilaterally in the caudate nuclei and putamen. Ammonium, lactate, tandem mass spectroscopy, and urine organic acid were normal. No bacteria were detected in the CSF cultures. Human bocavirus was detected in a nasopharyngeal aspirate using real-time PCR, while no influenza was detected. Oseltamivir, acyclovir, 3% hypertonic saline solution, and supportive care were used to treat the patient, who was discharged after two weeks. She began to walk and talk after one month of physical therapy and complete recovery was observed after six months.Human bocavirus is a recently identified virus that is mainly reported as a causative agent in respiratory tract infections. Here, we present a case of influenza-like acute necrotizing encephalopathy secondary to human bocavirus infection. [ABSTRACT FROM AUTHOR]