Pediatric autoimmune neuropsychiatric disorders associated with streptococcal infections (PANDAS) are rare neurobehavioral disorders in children. Affected children may have a diverse array of perioperative manifestations including compulsive behavior, agitation, and abnormal movements. Perioperative management might be more challenging for anesthesiologists. Series of therapeutic plasmapheresis in these children may complicate their anesthesia management. We describe the first case of a patient with PANDAS and its anesthesia implications.