Air embolism is a rare but dreaded complication of medical or surgical procedures with sig-nificant mortality.An abnormal air-to-blood pressure gradient and communication between the vein and atmosphere can lead to the venous air embolism.Here,we presented a rare case of venous air embolism just after the removal of the central venous catheters.A 59-year-old female was admitted to Peking Uni-versity International Hospital suffering from neuromyelitis optica spectrum disorder.After 5 rounds of im-munosorbent therapy,her hemodialysis catheter was removed.Five hours later,she suddenly experienced shortness of breath,dizziness,weakness of the lower extremities,transient loss of consciousness and sei-zures.Ultrasound showed a small amount of gas in the right internal jugular vein and brain CT showed scattered air accumulation in part of venous blood ducts and bilateral cavernous sinuses,which was diag-nosed as air embolism.High-flow oxygen inhalation,compression of the right neck extubation site,head down and feet up left decubitus position,intravenous fluid therapy were used.The patient gradually im-proved and returned to normal activities after one week.To sum up,air embolism is a rare,but serious and life-threatening complication after central catheters removal.Clinicians need to be alerted and take appropriate preventive measures.