Congenital cystic adenomatoid malformation (CCAM) is a lung lesion that is now commonly diagnosed in utero with fetal ultrasonography. The described treatment of this lesion includes observation with treatment delivery, a single aspiration, thoraco-amniotic shunts, and fetal resection. This patient had an in utero diagnosis of a Stocker type I CCAM associated with hydrops. Fetal resection was not an option because of patient refusal. The fetus was treated with multiple serial aspirations. There was marked improvement of the anasarca, and subsequently the baby was born without respiratory distress. On the second day of life the CCAM began to expand, and the right lower lobe was resected. The baby's postoperative course was uneventful. CCAM with hydrops is associated with a high mortality rate. Current recommended therapy for these lesions is fetal resection or thoracoamniotic shunt. The authors' patient was treated with serial fetal thoracocenteses, with an excellent outcome. This therapy may be an alternative to fetal surgery or an adjunct to fetal surgery in selected cases.