| Summary: | 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). On a postpartum three-dimensional (3D) computed tomography (CT) angiography, bilateral ICA hypoplasia was confirmed and the manifestations of infarcts were probably the consequence of altered hemodynamics of pregnancy. In conclusion, a patient in her late pregnancy and postpartum period, having nonspecific cerebral symptoms or having suffered a cerebrovascular accident, should not only be evaluated for pregnancy or puerperium-related complications but also whenever possible a baseline screening with Doppler study of neck vessels and a noncontrast magnetic resonance imaging (MRI) angiography of neck and cerebral vessels should be performed to rule out congenital anomalies.
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