Theophylline, a drug frequently used to treat apnea of prematurity, has a prolonged half-life of 30 h in neonates. Severe overdoses of theophylline have an associated 10% mortality and significant morbidity. We describe a 1,220-g neonate who developed status epilepticus due to a theophylline overdose. Hemodialysis was instituted to increase elimination of theophylline. The patient tolerated the procedure without complication. The half-life of theophylline was 0.7 h during dialysis. No reported therapies used in neonates have achieved this magnitude of clearance. In fact, the clearance of theophylline in this neonate approached that obtained with hemoperfusion, the standard therapy for theophylline overdose in adult patients. This case demonstrates that hemodialysis is a safe and effective means of enhancing theophylline elimination for neonatal theophylline overdose.