Although anaphylactic shock during the perioperative period is rare, it can be lethal due to severe cardiovascular and respiratory collapse. Midazolam is generally used as premedication for relieving anxiety about the operation, and the danger of anaphylactic shock after intramuscular injection is not widely recognized. We report the first case of anaphylactic shock occurring during the perioperative period after intramuscular injection of midazolam. Since anaphylactic shock after intramuscular injection can be of slow onset, the operation should be delayed if an anaphylactic reaction is suspected, even if the symptoms are limited. In addition, anesthesiologists should be prepared for the occurrence of anaphylaxis at any time in the perioperative period.