Objective: To report a case of priapism in an adolescent male following the use of propofol for sedation. Case Report: A 13-year-old male with developmental delay was admitted to intensive care secondary to pneumonia and persistent paroxysmal choreoathetosis. In attempt to stop the choreoathetosis and progressive rhabdomyolysis, he was sedated with 50 mg of intravenous propofol, which successfully resolved the choreoathetosis. Within minutes after the injection, he developed priapism, and within 1 hour it resolved without intervention. Discussion: Priapism was considered to be caused by propofol based on temporal relationship, lack of other medications administered, and complete resolution of symptoms after withdrawal. An objective causality assessment based on the Naranjo adverse drug reaction probability scale suggests propofol is a probable cause of the priapism in our patient. The mechanism of propofol-induced priapism is unknown but likely occurs due to both local and systemic effects. Conclusion: Pediatric clinicians should be aware of this rare side effect, as it requires prompt recognition and potential urologic intervention to alleviate complications.