Congenital hypoplasia of bilateral internal carotid arteries (ICAs) is an extremely rare anomaly with less than 25 reported cases in literature till date. We present a case of a 30-year-old primigravida, who developed seizures and subsequently loss of consciousness just few minutes after the delivery of a healthy male child. To the best of our knowledge, this is the first case with bilateral ICA hypoplasia presenting in postpartum female who developed infarct in bilateral frontal region and subarachnoid hemorrhage (SAH).
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