Despite the frequency with which theophylline is administered to premature infants, there are few reported cases of neurotoxicity. We describe a theophylline- treated premature infant with chronic bronchopulmonary dysplasia and hepatitis who displayed a focal seizure and irritability followed by other systemic signs of theophylline intoxication, at a postnatal age of 5 months. An unrecognized accidental drug overdose combined with a prolonged elimination half-life were responsible for clinical intoxication. The potential mechanisms of theophylline's neurotoxicity are discussed briefly.