Midazolam, one of the most common benzodiazepine derivatives, is widely used in intravenous sedation for dental treatment without severe complications. However, paradoxical reactions to midazolam, including patient's unanticipated restlessness, agitation, hostility, and rage, have been frequently reported since the introduction of benzodiazepine. During outpatient intravenous sedation using midazolam for dental treatment, we experienced a paradoxical reaction to midazolam in a 28-year-old female patient. With a thorough review of the literature, the management of this complication and its various etiologies were discussed.