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Persistent sciatic artery is an unusual anatomical anomaly first noted in 1832. Approximately 60 to 70 cases have been documented in the literature, but none described symptomatic persistent sciatic artery presenting in the neonate. We report a case of a newborn infant who presented after birth with an atrophic right lower extremity and ischemia. Ultrasound with Doppler and magnetic resonance angiography revealed a right persistent sciatic artery with hypoplastic external iliac artery. The common femoral artery was reconstituted above the bifurcation into the superficial femoral and profunda femoral artery via collaterals from the internal iliac and the persistent sciatic artery. The infant's blood flow to the right extremity gradually improved for the next 4 days without treatment and continues to have adequate blood flow.