Cardiac arrest (CA) in the operating room is rare. Among known perioperative CA causes, Takotsubo syndrome (TTS) is a well-recognized one. Perioperative TTS is more common than existing reported cases therefore anesthesiologists should be aware of its diagnosis and management. TTS is an acquired and selflimited stress cardiomyopathy usually triggered by a precipitating stress factor and should be considered in any hospitalized patient presenting with symptoms such as acute coronary syndrome, cardiac arrhythmias or CA. A 67-year-old woman presented for lumbar discectomy in knee-chest position suffered CA two hours after the beginning of surgery. After a thorough examination, TTS was determined as the cause. This case report highlights the importance of TTS as a differential diagnosis of CA in the intraoperative setting as well as the usefulness of multimodal monitoring with cerebral monitoring to assist the prompt pulseless electric activity diagnosis. To our knowledge, this is the first case report in such circumstances.