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Perioperative management of a neonate with congenital diaphragmatic hernia (CDH) is challenging because of pulmonary hypoplasia, pulmonary hypertension, and respiratory insufficiency. In this report, we present our intra-operative experience in a 4-days old and 3070 grams CDH neonate. He was admitted to neonatal intensive care unit and intubated due to severe respiratory insufficiency. He showed signs of severe pulmonary hypoplasia and his echocardiography revealed a cardiac dextroversion. The patient was relatively stabilized after four days under combined high-frequency oscillatory ventilation (HFOV) and inhaled nitric oxide (iNO). A corrective surgical intervention was sustained with dopamine, dobutamine, fentanyl and midazolam infusions. Ventilator settings were: 9 cmH2O MAP; 15-Hz frequency; 30 cmH2O amplitude and 55% FiO2. Venous-blood gas analysis indicated pH:7.38 pO2:36.2, pCO2:39.2 with SpO2:98%. We believe that HFOV and iNO combination is an effective alternative for the anesthetic management of CDH cases as it provides better gas exchange and less volutrauma.