Background: Congenital anomalies of the external iliac arteries are rare and may be associated with other congenital abnormalities, including vertebral, cardiac, renal, or limb anomalies, as well as anal atresia and tracheoesophageal fistula. Tamisier’s classification divides these anomalies into three groups: those concerning origin or course, hypoplasia or atresia compensated by a persistent sciatic artery (PSA), and isolated hypoplasia or atresia. While the first group is often identified on autopsy, the latter two may present clinically due to limb ischemia, as observed in this case.Case Presentation Summary: A male term infant was delivered via emergency cesarean section for suspected asphyxia at 40 weeks’ gestation (birth weight: 4040 g; length: 52 cm; Apgar scores: 10, 10). Maternal history was notable for gestational diabetes. At birth, the infant displayed no distress or complications.Within the first 24 hours, documented hypoglycemia was corrected with oral glucose. On day two, cyanosis of the left lower extremity was observed with delayed capillary refill (6 seconds) and oxygen saturation between 60% and 70%. Other extremities demonstrated normal saturation levels. The infant was admitted to the NICU, where elevated inflammatory markers prompted a seven-day course of antibiotics, with blood cultures remaining negative. Echocardiography ruled out cardiac defects. Due to intermittent cyanosis of the left leg, Doppler ultrasound was performed, and suggested high-grade stenosis of the external iliac artery. MRI angiography confirmed the diagnosis without evidence of PSA or other vascular anomalies. The coagulation profile was within normal limits.During hospitalization, the infant remained stable, with no further episodes of cyanosis and satisfactory oxygenation in the affected limb. He was discharged with planned pediatric surgical follow-up and remained symptom-free at subsequent evaluations.Learning Points Discussion: Arterial development in the lower extremities begins around gestation days 35-37 and is completed by the third month. Genetic and environmental factors - including hypoxemia, hyperglycemia, and hyperinsulinemia - may disrupt normal vessel formation. In utero limb growth remains unaffected due to collateral circulation; however, chronic postnatal ischemia may result in limb atrophy, weakness, and motor delay. The presence of a PSA increases the risk for aneurysm and thrombosis, potentially requiring early intervention.Notably, this appears to be only the third reported case of external iliac artery anomaly in infants, emphasizing the importance of early recognition and coordinated multidisciplinary care.
