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Cerebral air embolism is the most serious form of air embolism potentially caused by invasive procedures. An 81-year-old man abruptly developed left hemiplegia and conjugate eye deviation to the right after removing a central venous catheter in his left internal jugular vein. Brain CT demonstrated entrapped air bubbles in right frontal cortex and bilateral cavernous sinus. Brain MRI performed at the next day confirmed acute right frontal lesion relevant to clinical manifestations, which was thought to be attributed to arterial air embolism. However, serial diffusion-weighted images and apparent diffusion coefficient maps showed atypical features which were not compatible with those of usual thromboembolic arterial ischemia. We discussed the pathophysiologic mechanism of neuronal injury in cerebral air embolism.