Air embolism can occur during central venous catheter removal. We encountered a patient diagnosed with cerebral air embolism associated with the removal of an internal jugular venous catheter. The patient was 65 years old and was admitted to the intensive care unit due to pneumonia. He had a catheter placed in the right internal jugular vein. Five days thereafter, the catheter was removed in semi-Fowler's position using the Valsalva maneuver. Immediately after the catheter removal, the patient became pale, felt dyspneic, lost consciousness and exhibited seizure activity. A neurological examination, revealed left upper arm (grade I) and lower leg paralysis (grade II) as well as left facial palsy. A brain computed tomogram showed that, air was trapped in the cavernous sinus. In diffusion weighted magnetic resonance imaging, signal changes were reported in the right frontal cortex, indicating acute cerebral infarction. Hyperbaric oxygen therapy and ventilatory support with synchronized intermittent mandatory ventilation mode was applied. Twenty days thereafter, consciousness and motor functions were recovered completely except for mild weakness of the upper arm (grade IV).