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Clinical Summary At 38 weeks’ gestation, a 2334-g boy was delivered by cesarean section. Oxygen saturation of arterial blood (SaO2) was 80% with room air, and the cardiothoracic ratio was 0.92. Echocardiography demonstrated a dysplastic tricuspid valve, which showed nodular and echodense valvular tips and a tethered septal leaflet, no forward flow across the pulmonary valve without regurgitant signals, and patent ductus arteriosus. Alprostadil administration was started. The Great Ormond Street Ebstein echocardiogram score was 0.58, grade 2. On postnatal day 1, the unwell neonate required endotracheal intubation. On postnatal day 3, an emergency right hemicolectomy was done to treat meconium peritonitis. Instability of the systemic circulation and progression of the cardiomegaly indicated surgical intervention for the congenital heart defects on postnatal day 14. A mildly hypothermic cardiopulmonary bypass was instituted. After patent ductus arteriosus ligation, the heart was arrested by infusion of blood cardioplegic solution. The main pulmonary artery (PA) was transected at just above the sinotubular junction, and the atretic membrane was removed. As a result, a dilator of 9 mm was permitted to pass into the right ventricular (RV) outflow. A femoral vein allograft containing a bicuspid competent valve with an external diameter of 8 mm (supplied by the University of Tokyo Tissue Bank, Tokyo, Japan) was thawed. A segment of the allograft with the valve was interposed between the proximal and distal PA stumps (Figure 1), and the pump was terminated. The myocardial ischemic time was 22 minutes. SaO2 was 67% with 100% inspired oxygen and 10 ppm nitric oxide gas inhalation. Central venous pressure was 16 mm Hg. Systemic arterial blood pressure was 53/28 mm Hg with 10 dobutamine and dopamine and 0.2 epinephrine. A peritoneal dialysis catheter was inserted. Forward flow through the allograft was confirmed by transesophageal echocardiography. Regurgitant signals through the valve were trivial. During the night of the same day, SaO2 dropped despite 20 ppm inhaled nitric oxide. Echocardiography demonstrated dynamic RV outflow tract obstruction. The patient underwent right modified Blalock-Taussig shunt (BTS) with a 3.5-mm polytetrafluoroethylene graft the next day. Back distribution of peritoneal dialysis fluid forced us to introduce continuous venovenous hemofiltration. On the second postoperative day, cardiothoracic ratio, SaO2, and central venous pressure were improved (75%, 82%, and 8 mm Hg, respectively) with the same respirator conditions and without nitric oxide inhalation. Thereafter, however, the patient developed sepsis and disseminated intravascular coagulopathy, resulting in hemodynamic deterioration. The patient died on the 15th postoperative day. Postmortem examination demonstrated widely patent RV outflow with modified BTS. Histologic examination of the lung revealed severe pulmonary vascular disease, which showed thickening of the intima but not the media (Figure 2).

journal of thoracic and cardiovascular surgery

In situ implantation of a valved femoral vein allograft in a neonate with pulmonary atresia with intact ventricular septum and severe tricuspid regurgitation