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Ann Thorac Surg 1997;64:23-28
© 1997 The Society of Thoracic Surgeons

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Original Articles: Cardiovascular

Is Vertical Vein Ligation Necessary in Repair of Total Anomalous Pulmonary Venous Connection?

Division of Thoracic and Cardiovascular Surgery, Department of Surgery, University of Virginia Health Sciences Center, Charlottesville, Virginia

	Abstract

Top Footnotes Abstract Introduction Patients and Methods Diagnostic Procedures Operative Technique Statistical Analyses Results Preoperative Hemodynamic Data Time on Extracorporeal... Operative Mortality Late Mortality Follow-up Comment References

 
Background. In the repair of total anomalous venous connection, vertical vein ligation is recommended to eliminate left-to-right shunting. However, the small left heart chambers may not always tolerate the immediate increase in blood flow after combined repair and vein ligation.

Methods. A retrospective review of 23 infants and children undergoing correction of total anomalous pulmonary venous connection was undertaken to determine whether vertical vein ligation is a necessary component of successful surgical repair. In 14 patients this vein was ligated, whereas in 9 it was left patent. Six patients who underwent ligation and 5 who did not had pulmonary venous obstruction before operation.

Results. The operative mortality rate was 36% (5 of 14 patients) for the ligated group compared with 0% (0 of 9 patients) for the nonligated group (p = 0.06). All deaths occurred in patients with preoperative obstruction and a low mean left atrial pressure, and four of the deaths were directly attributable to left heart failure. Follow-up echocardiography in patients in whom the vertical vein was not ligated revealed adequate cardiac function and no residual left-to-right flow through the previously patent venous conduit.

Conclusion. Vertical vein ligation during the repair of total anomalous pulmonary venous connection is not routinely necessary and actually may be undesirable in patients with preoperative obstruction, in whom the left heart chambers are particularly small.

	Introduction

Top Footnotes Abstract Introduction Patients and Methods Diagnostic Procedures Operative Technique Statistical Analyses Results Preoperative Hemodynamic Data Time on Extracorporeal... Operative Mortality Late Mortality Follow-up Comment References

 
See also page 29.

The surgical technique for the correction of total anomalous pulmonary venous connection (TAPVC) has undergone several modifications since the initial repair of this lesion by Muller in 1951 [1]. In the current era, most surgeons prefer to ligate an anomalous ascending or descending vertical vein (VV) during TAPVC repair to prevent the potential adverse sequela of a residual left-to-right shunt. However, the issue of whether this venous channel should be interrupted routinely at the time of TAPVC repair remains controversial. Hepatic ischemia has been described after ligation of a descending VV draining to the portal system [2, 3], convincing some surgeons to leave the VV open selectively during operations for infradiaphragmatic TAPVC [3, 4]. It has been postulated that the high resistance of the liver parenchyma eventually leads to complete cessation of flow through this low-pressure venous shunt [5].

Additional concerns regarding VV ligation during the correction of TAPVC focus on the abnormal left heart chambers. Before operation and early after operation, the left atrium in TAPVC is notoriously atrophic and noncompliant, and serves as a poor reservoir [6–9]. Furthermore, both the size and the function of the left ventricle in patients with this lesion are reduced in comparison with the hearts of normal individuals [6–10]. These left heart abnormalities, which are believed to be especially striking in the presence of preoperative pulmonary venous obstruction [9, 10], may be a factor underlying the low cardiac output syndrome that occurs after some cases of TAPVC repair [8]. In some patients, the left heart may not be able to accommodate the immediate increase in pulmonary venous blood flow after it is surgically rerouted to the left atrium. Therefore, a patent VV may function as a temporary reservoir for pulmonary venous blood after TAPVC repair, volume-unloading the small, noncompliant left heart chambers until they are able to grow and adapt to the increased flow demands. In view of these considerations, in 1986 we initiated the practice of not ligating the VV in selective patients undergoing TAPVC repair at our institution. In the current study, we endeavored to ascertain whether the practice of VV ligation during the correction of TAPVC is necessary in all cases, and to describe the long-term fate of this unligated venous structure. In addition, we compared the outcomes of patients undergoing interruption of the VV at the time of repair to the outcomes of those in whom this anomalous venous conduit was left unligated.

	Patients and Methods

Top Footnotes Abstract Introduction Patients and Methods Diagnostic Procedures Operative Technique Statistical Analyses Results Preoperative Hemodynamic Data Time on Extracorporeal... Operative Mortality Late Mortality Follow-up Comment References

 
We carefully reviewed the hospital records, preoperative and postoperative echocardiograms, and cardiac catheterization data of 29 infants and children who underwent surgical correction of TAPVC at our institution from 1974 to 1995. The system proposed by Darling and associates [11] was used to classify these patients according to anatomic type. Six patients had anomalous pulmonary venous drainage to the right superior vena cava, coronary sinus, or right atrium, but lacked a discernible VV. All these patients survived surgical repair with uniformly satisfactory results. The remaining 23 patients, classified as having types I, III, or IV TAPVC with a discrete ascending or descending vertical venous component, are the focus of this report. Fourteen of these patients underwent VV ligation during TAPVC repair and 9 did not.

The profile of anatomic TAPVC types among the patients who did not undergo VV ligation consisted of 4 cases of supracardiac drainage (type I), 3 of infracardiac drainage (type III), and 2 of mixed drainage (type IV). Among the patients who did undergo VV ligation, there were 11 cases of type I TAPVC, 1 of type III, and 2 of type IV. Patient ages at operation ranged from 1 day to 5 years in those who did not undergo VV ligation, and from 1 to 159 days in those who did. Except for the child operated on at age 5 years, all the patients underwent TAPVC correction within the first 6 months of life. Associated lesions included a patent ductus arteriosus in 7 patients who underwent VV ligation and 6 who did not, as well as complex cardiac defects in 2 children who did not undergo VV ligation. These latter defects were characterized by a single ventricle and L-transposition of the great vessels in 1 patient, and by valvular pulmonic/proximal pulmonary artery stenosis in the other. There were no associated complex cardiac malformations in the patients who underwent VV ligation; hence, all the patients in this group were considered to have isolated TAPVC.

	Diagnostic Procedures

Top Footnotes Abstract Introduction Patients and Methods Diagnostic Procedures Operative Technique Statistical Analyses Results Preoperative Hemodynamic Data Time on Extracorporeal... Operative Mortality Late Mortality Follow-up Comment References

 
Before 1987, two-dimensional echocardiography with pulsed Doppler was used as the initial diagnostic study in 3 of the 23 patients, whereas 12 subsequent patients were evaluated with Doppler color flow mapping from 1987 to 1995. In 4 patients, echocardiography was the sole technique used to establish the anatomic diagnosis. To delineate further the pathologic anatomy and hemodynamics, cardiac catheterization and angiocardiography were performed in 12 of the patients who underwent VV ligation and 6 of those who did not.

	Operative Technique

Top Footnotes Abstract Introduction Patients and Methods Diagnostic Procedures Operative Technique Statistical Analyses Results Preoperative Hemodynamic Data Time on Extracorporeal... Operative Mortality Late Mortality Follow-up Comment References

 
The technique used for TAPVC repair underwent few changes during the 22-year study period. All repairs were carried out under deep hypothermic circulatory arrest at a nasopharyngeal temperature of 18° to 20°C. When present, the patent ductus arteriosus was dissected and ligated before the commencement of cardiopulmonary bypass. Myocardial protection was achieved with intermittent doses of cold crystalloid cardioplegia for most patients in this series, whereas the use of cold blood multidose cardioplegia was adopted in 1994 for the last 6 consecutive patients. Supracardiac and infracardiac types of TAPVC were repaired through a transverse right atriotomy with extension of this incision through the patent foramen ovale and into the posterior wall of the left atrium. After leftward retraction of the heart to facilitate exposure, the common pulmonary vein-to-left atrium anastomosis was performed. Before 1986, this anastomosis was fashioned with 5-0 or 6-0 nonabsorbable polypropylene sutures, using a continuous technique for the entire anastomosis or, in some cases, running sutures on the posterior wall and interrupted sutures on the anterior wall to facilitate anastomotic growth. In 1986, we converted to using continuous 6-0 absorbable polydioxanone sutures for all subsequent anastomoses. Pericardial patch closure of the patent foramen ovale was undertaken in most cases to avoid narrowing the anastomosis or compromising the diameter of the left atrium. Mixed types of TAPVC were corrected with a baffle technique or direct pulmonary vein-to-left atrium anastomosis, depending on the specific pathologic anatomy. Concomitant procedures for complex cardiac defects in the 2 patients who did not undergo VV ligation included pulmonary artery banding in 1 and pulmonary valvulotomy combined with longitudinal pulmonary arteriotomy in the other.

In a total of 14 repairs, 8 of which occurred before 1986, the ascending or descending VV was dissected and ligated during the systemic cooling phase of cardiopulmonary bypass. However, beginning in 1986, we adopted the practice of neither dissecting nor ligating this vein in selected cases. Patients in whom VV patency was maintained intentionally during TAPVC repair included those with type III drainage, severe pulmonary hypertension, or severe pulmonary venous obstruction. To date, a total of 9 infants and children have undergone TAPVC repair at our institution without concomitant VV ligation.

	Statistical Analyses

Top Footnotes Abstract Introduction Patients and Methods Diagnostic Procedures Operative Technique Statistical Analyses Results Preoperative Hemodynamic Data Time on Extracorporeal... Operative Mortality Late Mortality Follow-up Comment References

 
Numerical data, such as hemodynamic parameters, arterial blood gas values, and time on extracorporeal circulation, are reported as the mean plus or minus the standard error of the mean. These values were analyzed for significant between-group differences by Fisher's exact t test. All other data, such as mortality statistics or the number of patients with preoperative pulmonary venous obstruction, are reported as the fraction, percentage, or both, of the total number of patients in each group. These data were analyzed by the ² test. For all analyses, a p value of less than 0.05 was considered significant.

	Results

Top Footnotes Abstract Introduction Patients and Methods Diagnostic Procedures Operative Technique Statistical Analyses Results Preoperative Hemodynamic Data Time on Extracorporeal... Operative Mortality Late Mortality Follow-up Comment References

 
Preoperative Patient Features
A comparison of preoperative demographic and clinical characteristics for the two groups in whom the VV was either ligated or left patent during TAPVC repair is presented in Table 1. As demonstrated, the two groups were similar with regard to age and weight at operation, percentage of patients with pulmonary venous obstruction, and arterial pH and partial pressure of oxygen on hospital admission. However, there was a substantially higher percentage of patients who did not undergo VV ligation who required both preoperative intubation and inotropic support compared with patients who did undergo VV ligation, with the former parameter exhibiting a trend toward statistical significance (p = 0.15).

	Preoperative Hemodynamic Data

Top Footnotes Abstract Introduction Patients and Methods Diagnostic Procedures Operative Technique Statistical Analyses Results Preoperative Hemodynamic Data Time on Extracorporeal... Operative Mortality Late Mortality Follow-up Comment References

	Time on Extracorporeal Circulation

Top Footnotes Abstract Introduction Patients and Methods Diagnostic Procedures Operative Technique Statistical Analyses Results Preoperative Hemodynamic Data Time on Extracorporeal... Operative Mortality Late Mortality Follow-up Comment References

	Operative Mortality

Top Footnotes Abstract Introduction Patients and Methods Diagnostic Procedures Operative Technique Statistical Analyses Results Preoperative Hemodynamic Data Time on Extracorporeal... Operative Mortality Late Mortality Follow-up Comment References

 
There were 5 early (less than 30 postoperative days) deaths in the series, all of which occurred before 1985 and in patients who had undergone VV ligation. The operative mortality rate was 36% (5 of 14) for patients who underwent VV ligation compared with 0% (0 of 9) for those who did not, and this difference was nearly statistically significant (p = 0.06). All early mortality occurred in patients with preoperative pulmonary venous obstruction. Three of these deaths occurred during operation and were characterized by inability to wean from cardiopulmonary bypass; 2 were attributed to primary cardiac failure and the other to severe, refractory pulmonary hypertension. The remaining 2 patients died within 1 hour after TAPVC repair, both of primary cardiac failure. Postmortem examinations were performed in 4 of these patients and confirmed that the TAPVC repair was a technical success, but that primary left heart failure was the underlying cause of death in all cases. In three of these autopsy reports, it was specifically mentioned that the left ventricle was markedly smaller than the right ventricle.

	Late Mortality

Top Footnotes Abstract Introduction Patients and Methods Diagnostic Procedures Operative Technique Statistical Analyses Results Preoperative Hemodynamic Data Time on Extracorporeal... Operative Mortality Late Mortality Follow-up Comment References

 
There were three late deaths. One patient died with fixed pulmonary hypertension while being considered for lung transplantation. Echocardiography and cardiac catheterization in this patient confirmed adequate repair. A second child with single ventricle who had undergone concomitant pulmonary artery banding died 4 months after operation of progressive respiratory failure unrelated to any cardiac disease. A third patient in this group died 33 days after initial repair of mixed-type TAPVC while undergoing reoperation for stricture at the common pulmonary vein-to-left atrium anastomosis. In this child, severe pulmonary hypertension precluded his weaning from cardiopulmonary bypass. Although the total late mortality rate for the patients who did not undergo ligation was 33% (3 of 9), none of these deaths were attributed to left ventricular dysfunction or right ventricular failure from persistent shunting through a patent anomalous VV. To date, there has been no late mortality among the patients who underwent VV ligation.

	Follow-up

Top Footnotes Abstract Introduction Patients and Methods Diagnostic Procedures Operative Technique Statistical Analyses Results Preoperative Hemodynamic Data Time on Extracorporeal... Operative Mortality Late Mortality Follow-up Comment References

 
All early and late survivors in the series are doing well and have minimal or no restriction of their physical activity at a mean follow-up of 55 months (range, 1 to 139 months). Pulmonary venous stenosis has developed in 2 children. One of these patients, in whom the VV was not ligated, sustained a stricture at the anastomosis and died during reoperation for this complication. The other patient, in whom the VV was ligated, had isolated, complete obstruction of the left pulmonary veins 25 months after operation and has had a satisfactory response to medical therapy alone.

Of the 6 long-term survivors among the patients who did not undergo VV ligation, including 2 with supracardiac drainage, 3 with infracardiac drainage, and 1 with mixed drainage, all have undergone postoperative evaluation with two-dimensional echocardiography. At a range of 1 day to 106 months after operation, echocardiography failed to reveal flow in any of the unligated ascending or descending VVs. Furthermore, none of the patients in whom the VV was left patent during repair demonstrated any stigmata of a chronic left-to-right shunt, such as marked right ventricular hypertrophy or dilatation. Only 1 long-term survivor who did not undergo VV ligation was evaluated with cardiac catheterization after operation, which was performed 43 months after repair of a type III TAPVC. During levophase, contrast medium filled the upper portion of the previously patent unligated descending VV, but the remainder of the lumen was obliterated. Normal right-sided cardiac chamber pressures, including right ventricular systolic/diastolic pressures of 28/3 mm Hg and a mean right atrial pressure of 3 mm Hg, corroborated the angiographic findings of the absence of residual shunt flow through the VV. Interestingly, cardiac catheterization performed 32 days after surgical correction in the patient with mixed TAPVC in whom anastomotic stricture developed and death occurred demonstrated suprasystemic pulmonary artery pressures, a markedly elevated pulmonary capillary wedge pressure, and a widely patent descending VV. Figures 1 and 2 compare preoperative and postoperative angiographic images of these 2 patients.

View larger version (71K): [in this window] [in a new window]   Fig 1. . Angiographic images obtained during levophase after pulmonary artery injection in a child with type III total anomalous pulmonary venous return, in whom the descending vertical vein was not ligated at the time of repair. (A) Preoperative angiogram demonstrating an anomalous descending vertical vein to the portal system. The arrow indicates the site of obstruction at the level of the diaphragm. (B) Angiographic image obtained 43 months after the operation revealing normal filling of the left atrium (open arrow) and only the uppermost portion of the unligated vertical vein (solid arrow). The remaining portion of the vein has undergone spontaneous closure.

View larger version (253K): [in this window] [in a new window]   Fig 2. . Angiographic studies performed in a patient with mixed total anomalous pulmonary venous return and a descending vertical venous component, in whom the latter was left open during the operation. (A) Preoperative anteroposterior view of the descending vein (arrow). (B) Anteroposterior image obtained 32 days after operation, disclosing persistent patency of the unligated vein (arrow). (C) Lateral projection revealing a stricture at the common pulmonary vein-to-left atrium anastomosis (solid arrow), accounting for failure of the vertical vein (open arrow) to undergo spontaneous closure.

	Comment

Top Footnotes Abstract Introduction Patients and Methods Diagnostic Procedures Operative Technique Statistical Analyses Results Preoperative Hemodynamic Data Time on Extracorporeal... Operative Mortality Late Mortality Follow-up Comment References

Previous studies have documented that in TAPVC, left atrial volume is smaller than normal, left atrial pressure is low, and the left atrium lacks both normal compliance and reservoir function [6–9]. Proposed explanations for these abnormalities include failure of the common pulmonary vein to fuse with the left atrium during development [9, 12] and reduced atrial filling and chamber underloading as a consequence of the characteristically large left-to-right shunt [9]. In the presence of obstruction to pulmonary venous drainage, left atrial size may be even smaller than in nonobstructed cases [9]. Although we did not evaluate cardiac chamber size in the current study, the significantly lower mean left atrial pressure in nonsurvivors who underwent VV ligation suggests that in these obstructed patients, the left atrium was seriously underloaded before repair. Because it has been demonstrated that a diminutive, noncompliant left atrium may substantially compromise cardiac output [8, 13, 14], it is conceivable that the left heart failure and eventual demise of 4 of the 5 patients who underwent VV ligation were related in part to abnormal left atrial characteristics.

Previous investigations have concluded that the left ventricle also may be atrophic and dysfunctional in TAPVC [6–10], and that such derangements are even more pronounced when there is coexisting pulmonary venous obstruction [9, 10]. Such chamber abnormalities have been attributed to decreased left ventricular flow and perturbation of the normal left ventricular diastolic pressure-volume relation secondary to elevated right ventricular diastolic pressure or volume [6, 15, 16]. Although not quite statistically significant, the right ventricular-to-left ventricular diastolic pressure ratio was three times higher in the patients who did not undergo VV ligation compared with those who did. This implies that in the former patients, the left ventricle was remarkably small and dysfunctional. In these patients, the advantages of a temporary pulmonary venous reservoir provided by the unligated VV may have been critical to their early postoperative hemodynamic stability and survival.

In our series, postoperative echocardiography and angiocardiography failed to detect flow in the unligated VV of any long-term survivors. This finding is consistent with the results of an earlier report by Jegier and associates [5], who described the repair of an infradiaphragmatic TAPVC in an infant with a small left atrium and left ventricle in whom the descending VV to the portal vein was left patent. Angiocardiography performed 6 months after operation in the same patient demonstrated that both left heart chambers had grown to a normal size and the anomalous pulmonary venous shunt had become obliterated. The authors speculated that the high resistance of the hepatic capillary bed accounted for the eventual termination of flow through the VV. As opposed to the suggestion of Mustard and colleagues [17] that the patent foramen ovale and VV be closed during the second procedure of a two-stage repair, Jegier and associates concluded, as do we, that definitive correction of type III TAPVC is possible without initial ligation of the descending VV and without the need for reoperation to close this anomalous pathway. We propose that such an approach also is valid for the management of an ascending VV in supracardiac TAPVC.

	Footnotes

Top Footnotes Abstract Introduction Patients and Methods Diagnostic Procedures Operative Technique Statistical Analyses Results Preoperative Hemodynamic Data Time on Extracorporeal... Operative Mortality Late Mortality Follow-up Comment References

 
Presented at the Forty-third Annual Meeting of the Southern Thoracic Surgical Association, Cancun, Mexico, Nov 7–9, 1996.

Address reprint requests to Dr Kron, Department of Surgery, University of Virginia Health Sciences Center, Box 310, Charlottesville, VA 22908.

	References

Top Footnotes Abstract Introduction Patients and Methods Diagnostic Procedures Operative Technique Statistical Analyses Results Preoperative Hemodynamic Data Time on Extracorporeal... Operative Mortality Late Mortality Follow-up Comment References

 

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13. Trusler GA, Bull RC, Hoeksema T, Mustard WT. The effect on cardiac output of a reduction in atrial volume. J Thorac Cardiovasc Surg 1963;46:109–16.[Medline]
14. Suga H. Importance of atrial compliance in cardiac performance. Circ Res 1974;35:39–43.[Abstract/Free Full Text]
15. Nakazawa M, Jarmakani JM, Gyepes MT, Prochazka JV, Yabek SM, Marks RA. Pre and postoperative ventricular function in infants and children with right ventricular volume overload. Circulation 1977;55:479–84.[Abstract/Free Full Text]
16. Taylor RR, Covell JW, Sonnenblick EH, Ross J Jr. Dependence of ventricular distensibility on filling of the opposite ventricle. Am J Physiol 1967;213:711–18.[Free Full Text]
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This Article Abstract Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Stanton P. Nolan Irving L. Kron Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Cope, J. T. Articles by Kron, I. L. Search for Related Content PubMed PubMed Citation Articles by Cope, J. T. Articles by Kron, I. L. Related Collections Related Article