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Malposition of the extracorporeal membrane oxygenation (ECMO) cannulas is not frequently reported in central cannulation technique: we aimed to present an ECMO cannula malposition in follow-up in a neonate case. The patient was followed up three days on ECMO support with optimum hemodynamics. On the third day swelling occurred in the left face area within hours. Chest radiography showed that the arterial cannula tip was identified upwardly facing, which implied malposition of the cannula in the left carotid artery. After repositioning the cannula, swelling on the left side of the face completely disappeared in 4 hours. Clinicians should be alert for ECMO cannula malposition. Chest radiography which can be accessed quickly and easily can be beneficial for the diagnosis.