Midazolam is generally effective and safe to be widely used in the management of seizures, procedural sedation, and anxiolytic. However, paradoxical reactions, though rare, present clinical challenges. Flumazenil is the preferred option for reversing the reactions. Additionally, ketamine, physostigmine, and haloperidol have been successfully used to counteract the adverse effects (AEs). Herein, we report an 11-year-old girl with epilepsy who was brought to the emergency department with a breakthrough seizure. Approximately 10 minutes after a midazolam administration, she developed increased agitation, confusion, uncontrollable crying, and excessive random movements. Despite receiving 4 doses of flumazenil, no improvement was observed. However, the AEs ceased following the administration of ketamine. This report may increase awareness of the uncommon AEs and equip clinicians to effectively handle such occurrences.