Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) is a clinico-radiological syndrome in children secondary to viral or bacterial infections. The causes include viral (influenza, human herpes virus-6, adenovirus, rota) as well as bacterial infections. However, AESD with dengue infection has not been reported earlier. Here, we present an infant with dengue infection and AESD which recovered completely following treatment with intravenous human immunoglobulin therapy. A 9-month-old girl presented with seizures following fever and loose stools. Seizures recurred after 2 days of seizure-free interval. Cerebrospinal fluid analysis was not contributory. Dengue infection was confirmed by lab tests. Magnetic resonance imaging brain after the second seizure revealed diffusion restriction involving the bilateral frontal and parietal white matter, both hemispheres with a typical central perisylvian sparing lesion suggestive of AESD. This case report expands the reported spectrum of neurological manifestations of dengue infection.