SUCCESSFUL PRENATAL DIGOXIN THERAPY FOR EBSTEINS-ANOMALY WITH HYDROPS-FETALIS - A CASE-REPORT

BACKGROUND: Ebstein anomaly is a rare tricuspid valve anomaly. Some fe tuses with Ebstein's anomaly have concurrent severe cardiac function i mpairment, which results in hydrops fetalis. Most of these fetuses are inevitably terminally ill. No reports have demonstrated the potential prenatal therapy for fetuses under such conditions. CASE: Ebstein's a nomaly and hydrops fetalis were detected at 28 weeks' gestation. Tricu spid regurgitation with congestive heart failure was observed. From 28 to 34 weeks' gestation, intrauterine therapy with digoxin, 0.75 mg/d, was administered. The fetal hydrops status improved gradually, while the tricuspid valve regurgitation persisted. At 36 weeks' gestation th e fetus was delivered normally. During the neonatal phase, digoxin was continued adn gradually tapered off. The tricuspid valve regurgitatio n and cardiomegaly gradually improved. CONCLUSIONS: The favorable outc ome in this case supports the positive effect of prenatal digoxin ther apy for Ebstein's anomaly with hydrops fetalis. In such conditions, up on the appearance of hydrops and congestive cardiac failure, immediate digoxin therapy may be useful. This successful trial encouraged us to manage such fetuses more aggressively.