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Complete situs inversus is a congenital condition which involves complete transposition of all thoracic and abdominal organs. This condition poses a technically challenging issue for the procedure of transcatheter pulmonary vein isolation to treat atrial fibrillation. We have recently experienced one case of situs inversus complicated with cardiac tamponade after successful ablation. The patient is a 67-year-old woman with a history of situs inversus totalis. She presented to our emergency department with repeated episodes of drug-resistant paroxysmal atrial fibrillation. Radiofrequency ablation was preferred. During the procedure, we performed interatrial septal puncture under intracardiac ultrasound guidance and isolation of all 4 pulmonary veins was achieved. Ten minutes after removing the sheaths from left atrium the patient developed cardiac tamponade and prompt pericardiocentesis was needed. Pericardial effusion resolved on the next day and she was discharged 3 days later. No recurrence of atrial fibrillation has been documented after the procedure. Judging from clinical progress, pericardial effusion is speculated to occur after removal of the sheath.