J Korean Neurosurg Soc 48 : [380][381][382] 2010 The authors present an extremely rare case of a pseudoaneurysm of the ulnar artery as a complication of a two-portal endoscopic carpal tunnel release (ECTR). A 70-year-old man with chronic renal failure and on maintenance hemodialysis with a left arteriovenous fistula presented with paresthesia of his right hand. A clinical diagnosis of right carpal tunnel syndrome was confirmed by ultrasonography and an electrophysiologic study. He underwent two-portal ECTR, and the paresthesia was much improved. However, he presented to us one month after operation with severe pain, a tender mass distal to the right wrist crease and more aggravation of the paresthesia in the ulnar nerve distribution. Doppler ultrasound was performed and revealed a hypoechoic lesion 20 mm in diameter in the right palm, with arterial Doppler flow inside connected to the palmar segment of the ulnar artery. An ulnar artery pseudoaneurysm was diagnosed and treated by ultrasound-guided percutaneous thrombin injection. Transverse color Doppler ultrasound image showed complete thrombosis of the pseudoaneurysm and flow cessation after a total injection of 500 units of thrombin. The symptoms were also improved. KEY WORDS :Endoscopic carpal tunnel release˙ Complication˙ Pseudoaneurysm.