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J Thorac Cardiovasc Surg 2003;125:421-423
© 2003 The American Association for Thoracic Surgery
Brief Communications |
From the Department of Pediatric Cardiology and Cardiac Surgery, The Chaim Sheba Medical Center, Tel Hashomer, Israel, affiliated to the Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel.
Received for publication June 10, 2002. Accepted for publication July 1, 2002. Address for reprints: David Mishaly, MD, Department of Congenital and Pediatric Cardiac Surgery, Haim Sheba Medical Center, Tel Hashomer 52621, Israel (E-mail:
Ligation of the vertical vein (VV) during the correction of total anomalous pulmonary venous drainage (TAPVD) is controversial.
1-3 Most surgeons usually ligate or divide the VV, but others support a potential benefit in leaving it patent to serve as a possible vent for relatively hypoplastic, noncompliant left heart chambers. 1,2 The fate of an unligated VV late after correction of a TAPVD is unclear; both no flow 1,4 and patent veins with considerable left-to-right shunting 3,5 have been reported. We report a rare case of persistent cyanosis after a Fontan procedure in a patient with a VV left open after correction of infracardiac TAPVD and emphasize the importance of VV ligation in this special setting of single ventricle and TAPVD.
Clinical summary
A 3.5-year-old girl was seen with persistent cyanosis after completion of the Fontan operation. She had a diagnosis from birth of infracardiac TAPVD, a double-outlet right ventricle, transposition of the great arteries, mitral atresia, and heterotaxy with right isomerism and asplenia. Correction of total anomalous venous connection without ligation of the infracardiac VV and pulmonary artery banding was performed. When she was 1 year old, she underwent a bidirectional Glenn shunt and division of the pulmonary artery and had saturation levels in the 80% range after that operation. At the age of 3.5 years, she underwent completion of the nonfenestrated Fontan operation with an 18-mm polytetrafluoroethylene extracardiac conduit.* She was more profoundly cyanotic after that operation than she had been before it. There was no respiratory explanation for the low saturation levels. In a search for a residual systemic vein draining into the atrium, she underwent catheterization; however, injection into the inferior vena cava and both arms demonstrated no residual right-to-left shunt causing the persistent cyanosis.
Spiral contrast computed tomography revealed a patent infracardiac VV (Figure 1). This was confirmed by ultrasonography, which corroborated the diagnosis and showed reversal of flow from the inferior vena cava through the hepatic veins to the hepatic sinusoids and portal veins through the patent VV into the common atria, creating a significant right-to-left shunt.
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VV ligation during TAPVD repair is controversial, as is the fate of the unligated VV. Failing to demonstrate flow in an unligated vein in long-term follow-up, some authors have concluded that cessation of flow occurs spontaneously. 1,4 Others have found patent veins with persistent left-to-right shunting. 3,6 Most surgeons ligate the ascending or descending VV to prevent potential adverse sequelae of left-to-right shunting 1; others leave the VV unligated to serve as a "pop off valve" to the left heart chambers, which are believed to be hypoplastic and noncompliant 7 and to cause low cardiac output after the operation. Hepatic ischemia after infradiaphragmatic VV ligation draining to the portal vein has convinced several surgeons to selectively leave the VV open. 6 To determine whether VV ligation is essential to successful surgical repair, Cope and colleagues 1 reviewed their experience with 23 infants who underwent correction of TAPVD and concluded that VV ligation is not routinely necessary and may even be undesirable in patients with pulmonary vein obstruction. Furthermore, Caspi and associates 2 reviewed 34 infants after operation and concluded that maintaining the VV patent in selected patients with infracardiac TAPVD contributes to a favorable postoperative outcome. Others, however, have described significant left-to-right shunts as being serious consequences of an unligated VV. 3,5
Operative mortality is high in patients with single ventricle and TAPVD, and long-term outcome is poor. 8 Leaving the VV patent in these high-risk patients at the time of TAPVD repair is logical, because it probably contributes to a smoother postoperative course. Furthermore, left-to-right shunting has no meaning in this setting, because the pulmonary veins and the inferior vena cava drain into the same atrial chamber during the first two stages of single-ventricle repair.
In our case the flow reversed through the hepatic veins, hepatic parenchyma, and the portal vein (without disruption of liver functions) through the VV into the common atrium and caused a significant right-to-left shunting, as in a large Fontan fenestration, because systemic venous pressure is higher than pulmonary venous pressure at the time of the completion of the Fontan operation. Ligation of the infradiaphragmatic VV resulted in immediate improvement. In light of the our described experience, we support the advocates of ligation of the VV, at least when the Fontan operation is completed, to prevent late cyanosis and shunting.
Acknowledgments
We thank Esther Eshkol for editorial assistance.
Footnotes
* Gore-Tex conduit; registered trade name of W. L. Gore & Associates, Inc, Flagstaff, Ariz. 
References
adR shunt: a serious consequence of TAPVC repair without ligation of vertical vein. Ann Thorac Surg. 2000;70:971-3.
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