To highlight severe neurologic manifestation of rotavirus-associated leukoencephalopathy and to emphasize the importance of thorough diagnostic evaluation in infants with encephalopathy and refractory seizures. 

A previously healthy, vaccinated 3-month-old male developed diarrhea, vomiting, decreased visual tracking, and progressive irritability, followed by encephalopathy and multifocal seizures requiring intubation. EEG demonstrated frequent multifocal electroclinical seizures. MRI brain with and without contrast revealed diffuse bilateral subcortical diffusion restriction with occipital predominance, suggestive of leukoencephalopathy. CSF showed mild pleocytosis with normal protein. Enteric panel was positive for rotavirus, and it was noted that rotavirus vaccination had not yet been administered. Seizures were treated with IV loading doses of levetiracetam, phenobarbital, and a midazolam infusion, followed by maintenance therapy with phenobarbital and levetiracetam. Patient received high-dose IV steroids and IVIG to treat inflammation-associated leukoencephalopathy in the setting of active rotavirus infection. Clinical status gradually improved, with recovery of visual tracking and successful weaning of antiseizure medications. 

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In infants without prior rotavirus vaccination who present with gastrointestinal symptoms and concurrent severe neurologic manifestations, early testing for rotavirus infection should be strongly considered, along with consideration of guidance regarding use of live attenuated vaccines following infection. Early recognition of characteristic clinical and radiographic features, along with exclusion of alternative etiologies, is essential to guide timely immunomodulatory treatment and optimize neurologic outcomes.