Pentamidine isethionate is a diamidine compound used in the treatment of a number of parasitic diseases, notably Pneumocystis carinii pneumonia. Although cases of sudden death have been reported during the administration of pentamidine, there have been no reported cases in the literature of pentamidine-associated arrhythmias. Reported in this study are two cases of torsade de pointes occurring during the prolonged administration of pentamidine. In addition, electrocardiographic changes of marked QT interval prolongation, pronounced precordial T wave abnormalities, and ST segment changes were shown in both patients. Mild hypomagnesemia coexisted in both cases, but torsade de pointes persisted in one patient and electrocardiographic changes remained in both cases despite magnesium replacement. QT interval prolongation and electrocardiographic abnormalities resolved slowly over several days to weeks, paralleling the known elimination kinetics of pentamidine. These data suggest a proarrhythmic effect of pentamidine isethionate.