Ischemic perinatal stroke is a serious potential complication of delivery. In utero infection may be responsible for an underestimated proportion of perinatal stroke. Limited literature identifies objective evidence of ischemic perinatal stroke as a consequence of uterine infection. The authors report a neonate with ischemic stroke and documented findings of severe chorioamnionitis with umbilical vein thrombosis. A term neonate, after uneventful pregnancy and delivery, presented on the third day of life with seizures. Investigations for metabolic, electrolyte, infectious, and hypercoaguability derangements were normal. Extensive acute infarction in the left middle cerebral artery territory was diagnosed by magnetic resonance imaging (MRI). Placental histopathology confirmed the presence of chorioamnionitis. On follow-up assessments, mild residual neurologic deficits have persisted. Chorioamnionitis has been correlated with ischemic perinatal stroke. In addition to the recognized inflammatory cascade of in utero infection, umbilical vein thrombosis with subsequent ''paradoxical'' embolization may represent one mechanism responsible for this association.