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Ann Thorac Surg 2002;74:778-785
© 2002 The Society of Thoracic Surgeons

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Original article: cardiovascular

Fontan-type procedures: residual lesions and late interventions

^a Department of Pediatric Cardiology and Pediatric Intensive Care Medicine, Hannover Medical School, Hannover, Germany
^b Department of Thoracic, Cardiac and Vascular Surgery, Tuebingen University Hospital, Tuebingen, Germany

Accepted for publication May 7, 2002.

* Address reprint requests to Dr Kaulitz, Department of Pediatric Cardiology and Pediatric Intensive Care Medicine, Tuebingen University Hospital, Hoppe-Seyler Str 3, D-72076 Tuebingen, Germany
e-mail: renate.kaulitz{at}med.uni-tuebingen.de

	Abstract

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Background. The purpose of this study was to determine the type and incidence of hemodynamic and electrophysiological abnormalities requiring surgical or catheter-based interventions in a single-center long-term experience.

Methods. Eighty-eight patients with a follow-up of at least 5 years (mean follow-up, 9.6 ± 2.6 years) after Fontan-type procedures were included. All patients had undergone cardiac catheterization either as part of the regular postoperative protocol or because of symptomatic atrial tachycardia or increasing cyanosis.

Results. Freedom from reoperation for up to 5 years was documented for 82% of patients and decreased to 76% after 8 years. Late reoperations included conversion of an atriopulmonary anastomosis to a total cavopulmonary anastomosis in 2 patients with atrial dysrhythmia and implantation of an extracardiac conduit in 1 patient with left atrial isomerism and intrapulmonary arteriovenous malformations after a Kawashima operation. Decline in sinus node function with symptomatic bradycardia required pacemaker therapy in 10 patients (11%). Transcatheter interventions included fenestration occlusion in 5 of the 11 patients with initial baffle fenestration. In 6 of 17 patients with aortopulmonary collaterals, coil occlusion was indicated to reduce future systemic ventricular volume load. Various systemic venous collaterals were documented in 11 patients and required coil occlusion in 2. One patient with symptomatic protein-losing enteropathy underwent transcatheter fenestration creation without sustained relief of symptoms. Freedom from transcatheter interventions decreased from 94% to 82% after 5 and 10 years, respectively.

Conclusions. During long-term follow-up, reoperations are rare and mainly involve Fontan conversion to either a lateral-tunnel or extracardiac-conduit procedure. Detailed angiographic evaluation on a routine basis allows identification of the vascular sites of origin of aortopulmonary collateral vessels and systemic venous collaterals potentially developing during long-term follow-up. Transcatheter interventions including fenestration occlusion and occlusion of venous collaterals and aortopulmonary collaterals were performed to maintain and improve the Fontan circulation in clinically symptomatic and asymptomatic patients. During long-term follow-up after Fontan-type operations, a regular postoperative cardiac catheterization protocol is recommended.

	Introduction

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Modifications to the Fontan procedure and staging with a bidirectional cavopulmonary shunt have extended the indications for this operation now applied to a wide range of congenital heart defects unsuitable for biventricular repair. Despite the improvements in surgical results leading to a reduction in perioperative mortality and an expected 10-year survival rate of 80%, late deterioration in functional status can be found with a longer duration of follow-up [1–3]. Late morbidity is mainly defined by atrial arrhythmia, increasing cyanosis because of systemic venous collateralization, ventricular failure, protein-losing enteropathy, or thrombus formation [4–6]. Postoperative morbidity with the need of hospitalization also includes surgical and catheter-based reinterventions, which can result from anatomic residua or sequelae of the univentricular circulation per se.

The aim of this, retrospective study was to assess the type and incidence of hemodynamic and electrophysiological abnormalities requiring surgical or interventional procedures during long-term follow-up after Fontan-type operations. This experience represents that of a single surgeon in one center. The study was approved by the scientific committee of the Children’s Hospital at Hannover Medical School. Informed consent was obtained from each subject.

	Material and methods

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Study group
Between 1987 and 1995, 101 patients with functionally univentricular hearts underwent Fontan-type procedures at our institution. Patients who had died of cardiac-related and noncardiac causes (12 patients) were excluded from this analysis of postoperative long-term morbidity (Fig 1). In addition, 1 patient who had undergone cardiac transplantation before the cross-sectional study period could not be traced. Calendar year of operation was associated with early postoperative mortality (8 patients). Cause of early death was low cardiac output syndrome alone in 6 patients; in 1 patient, it was associated with intractable atrial tachycardia, and in another, it was complicated by intrapulmonary bleeding. Causes of late death in 4 patients were respiratory failure 5 months after total cavopulmonary anastomosis, sudden death at home, sepsis, and low cardiac output syndrome after reoperation for severe atrioventricular valve regurgitation.

View larger version (10K): [in this window] [in a new window]   Fig 1. Kaplan-Meier survival curve after Fontan procedure in 100 patients. Number of patients alive and in follow-up cohort at 1 year, 5 years, and 10 years is shown.

Follow-up
All patients were followed up by regular outpatient visits, which included physical examination, 12-lead electrocardiogram, Holter monitoring, and transthoracic cross-sectional and Doppler echocardiography as well as laboratory evaluation. Cardiac catheterization data (Table 2) as well as angiographic findings were available for all 88 patients for at least 5 years postoperatively. The patients underwent cardiac catheterization under mild sedation (combination of ketamine hydrochloride and midazolam hydrochloride). Procedures such as transcatheter fenestration closure or creation requiring transesophageal echocardiographic guidance were performed under general anesthesia. Seventy-one patients were electively scheduled for cardiac catheterization every 4 to 6 years postoperatively as part of the regular postoperative surveillance protocol. The remaining 17 patients (19.3%) were studied because of symptomatic atrial tachycardia (5 patients), decrease in arterial saturation (< 90% in nonfenestrated Fontan circulation) or secondary polycythemia (hemoglobin level > 17 g/dL) (7 patients), symptoms of protein-losing enteropathy (3 patients), or history of decrease in exercise tolerance (2 patients).

	Results

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Reoperations
For the study group of 88 patients, freedom from reoperation at 5 years after a Fontan procedure was documented for 81.8% (72 patients) (Fig 2). During follow-up, 20 reoperations were performed (Table 3). Indications for reoperation were as follows: systemic venous congestion resulting from residual anastomotic stenosis; atrial tachyarrhythmia associated with anastomotic stenosis because of atrial thrombosis; persisting cyanosis because of residual interatrial shunting; chronic pericardial effusion requiring pericardiotomy; and obstruction of the ventricular outflow tract with the need to enlarge a restrictive bulboventricular foramen. Subaortic resection in 1 of our initial patients at the time of Fontan operation was followed by major aortic regurgitation leading to aortic valve replacement more than 5 years after Fontan operation (ie, 69 months). Takedown of the Fontan procedure and conversion to a bidirectional Glenn anastomosis was indicated in only 1 patient 3 months postoperatively because of persisting symptomatic pleural effusion. No patient underwent a takedown during long-term follow-up.

View larger version (13K): [in this window] [in a new window]   Fig 2. Kaplan-Meier curves showing freedom from late reintervention by either reoperation or transcatheter procedure. Number of patients free from reintervention at the given time points are shown (top line = transcatheter procedure; bottom line = reoperation.)

One patient with a complex form of univentricular heart with left atrial isomerism, interrupted inferior vena cava, and increasing systemic desaturation more than 5 years after a Kawashima operation (ie, 63 months) (arterial saturation, 79%) resulting from left-sided pulmonary arteriovenous malformations underwent an extracardiac lateral-tunnel procedure to reroute the hepatic venous blood to the systemic venous circulation (Fig 3). This resulted in only a moderate increase in arterial saturation (85%). In another patient, selective injection of contrast medium into the peripheral pulmonary arteries revealed pulmonary arteriovenous malformations without major arterial desaturation at the time of investigation; surgical intervention was not indicated.

View larger version (52K): [in this window] [in a new window]   Fig 3. (A) Cavopulmonary shunt (Kawashima operation) in patient with left atrial isomerism. (B) Selective injection of contrast medium into the left pulmonary artery demonstrates the left-sided pulmonary arteriovenous malformations. (C) Postoperative angiogram of extracardiac conduit for rerouting hepatic venous blood.

Decline in sinus node function was documented in 19 patients (21.6%), 10 of whom required a pacemaker because of symptomatic bradycardia (Fig 4). All pacing leads were placed on the epicardial surface; 8 patients were paced in DDD mode. Atrial reentry tachycardias were documented in 14 patients (15.9%); in 7 patients, supraventricular tachycardia occurred for the first time more than 5 years into follow-up. Seven patients received antiarrhythmic medication, and 5 underwent, radiofrequency ablation without recurrence of atrial reentry tachycardia in 4.

View larger version (14K): [in this window] [in a new window]   Fig 4. Incidence of atrial arrhythmias and therapeutic options. (ART = atrial reentry tachycardia; pts. = patients; RFCA = radiofrequency catheter ablation; SND = sinus node dysfunction; TCPA = total cavopulmonary anastomosis.)

The development of systemic venous collateralization with consecutive arterial desaturation was documented in 11 patients (12.5%). Various venous communications were observed with tortuous channels originating from the innominate vein or superior vena cava with connection to the pulmonary venous or left atrial level. Transcatheter closure of these communications was performed in 2 patients (arterial saturation, < 85%) and resulted in an increase in arterial saturation of 90% or higher. In addition, small vascular channels originating from the inferior vena cava and connecting to the inferior phrenic veins and further to the pericardial veins without clear opacification of the left atrium were identified in 5 of these patients. In 1 additional patient, a communication was found between the juxtapositioned left atrial appendage and the left atrium by way of multiple small channels. None of the patients underwent transcatheter occlusion of these unusual venous communications because of clinical irrelevance and small volume of the right-to-left shunt.

Aortopulmonary collateral vessels were documented in 65% of the patients and were the most frequent pathological angiographic findings. In only about one third (17 patients), the origin of the vessel was identified and opacity of the pulmonary arteries or veins was associated with an increase in oxygen saturation of 5% from the superior vena cava to the peripheral pulmonary arteries. Coil embolization was undertaken in 6 of the 17 patients to reduce the total and the ineffective pulmonary blood flow and the systemic ventricular volume load.

Of the 15 patients undergoing catheterization, 7 had transcatheter intervention on the basis of angiographic or hemodynamic findings or both.

Symptoms of protein-losing enteropathy developed in 4 patients (4.5%). One severely ill patient underwent transseptal puncture followed by serial dilation of the newly created defect. With an acute decrease in oxygen saturation from 90% to 84%, the mean right atrial pressure before and after intervention ranged from 15 to 17 mm Hg. However, in the presence of systemic ventricular dysfunction, this procedure did not lead to clinical improvement.

	Comment

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Late sequelae of the Fontan circulation have been described in many recent and fairly recent reports [1, 4–6]. Postoperative morbidity characterized by atrial arrhythmia, reduced exercise capacity, thromboembolic events, protein-losing enteropathy, or myocardial dysfunction can necessitate lifelong medical treatment in some patients. Quality of life during long-term follow-up can be compromised further by recurrent hospitalizations for invasive diagnosis, intervention, and eventually redo operations to maintain or to improve the Fontan circulation. In this study comprising the long-term experience of a single center, freedom from reoperation during long-term follow-up decreased continual to 81.8% and 76.2% at 5 years and 8 years, respectively. Corresponding results were reported in a larger but similar patient group in Boston [2].

None of the patients who underwent reoperation 5 to 16 years postoperatively required revision of the direct atriopulmonary or cavopulmonary anastomosis. This documents the preservation of a primary ideal anatomic result for many years. On the other hand, a recent morphometric study [7] demonstrated that despite absent gradients, the area of the Fontan anastomosis relative to the systemic venous area is significantly reduced. This might be an important finding contributing to the reasons for late functional deterioration, which is not yet completely understood in patients with a Fontan circulation.

The spectrum of reoperations, after a Fontan procedure, which include replacement of a stenosed or thrombosed atrioventricular conduit, resection of subaortic stenosis, thrombectomy from right atrial cavities, closure of a residual atrial right-to-left shunt, or takedown of the Fontan operation [2, 4, 8–10], has changed over the years corresponding to new technical modifications to and staging concepts for the Fontan procedure. Thorough preoperative patient selection and the increasing number of facilities performing transcatheter-based interventions might be contributing to the decrease in late reoperations. Thus, it can be expected that after a perfect Fontan procedure with an ideal anatomic result, reoperations will be restricted in future to conversion of an atriopulmonary anastomosis to a lateral-tunnel cavopulmonary anastomosis or implantation of an extracardiac conduit. The necessity for surgical pacemaker insertion because of major atrial arrhythmia is an underestimated cause of postoperative morbidity requiring hospitalization.

In our study, postoperative morbidity was mainly comprised atrial arrhythmia with a 22% incidence of sinus node dysfunction and a 16% incidence of atrial reentry tachycardia at follow-up of longer than 5 years. Younger age at palliation and use of an intermediate bidirectional Glenn anastomosis might have contributed to a relatively high incidence of sinus node dysfunction compared with previous reports [11, 12]. However, there was no significant difference between patients who had undergone a staged or primary Fontan procedure. Fifty-three percent of our patients with sinus node dysfunction required pacemaker implantation; antibradycardia pacing in the DDD mode was preferred, as it increases pulmonary venous return and cardiac output [13].

Atrial tachyarrhythmia refractory to medical treatment in patients with total cavopulmonary anastomosis was electrophysiologically evaluated, and radiofrequency ablation was effective in 4 of 5 patients. Complexity of electrophysiological mechanisms and variability in suture lines and scarring procedure explain the ineffectiveness of ablation procedures and recurrence of atrial reentry tachycardia in such patients [14], even with the use of modern endocardial mapping techniques [15].

Anastomotic stenosis and atrial arrhythmia after an atriopulmonary anastomosis can lead to hemodynamic and systemic complications [14, 16, 17]. Patients in this category represented an older group in whom conversion to a total cavopulmonary anastomosis and surgical cryoablation of the arrhythmia circuit at the time of operation can be highly effective [18]. To date 2 (12.5%) of our 16 adolescent patients with an atriopulmonary anastomosis have undergone conversion to a total cavopulmonary anastomosis for atrial reentry tachycardia refractory to medical treatment. This extensive intervention was more successful than radiofrequency ablation to control intraatrial reentrant tachycardia. Future studies will be necessary to determine whether modifications to suture lines or creation of an extracardiac cavopulmonary anastomosis can reduce the incidence of late atrial reentry tachycardia [19, 20].

In recent years, an increasing number of catheter-based interventions have been performed with the aim of improving the Fontan circulation by reducing the systemic ventricular volume load and thereby avoid major surgical revisions. Although the angiographic findings are very heterogeneous and sometimes unique, transcatheter interventions focus mainly on elimination of cyanosis and volume overload of the systemic ventricle.

Residual shunting was evident in 23% of the patients on postoperative angiograms, but systemic desaturation was rare. In patients with small defects in the suture lines, the incidence of thromboembolic complications is unknown, and the potential risk for either surgical or catheter-based interventions is probably higher than the risk for paradoxical embolism [21]. Among the 11 patients with a fenestrated lateral-tunnel procedure, transcatheter occlusion was indicated in 5 because of increasing cyanosis during exercise. Fenestration closure was not routinely performed. It should not be done in patients with preoperative risk factors (age at operation and preoperative hemodynamics), residual or even mild pulmonary artery distortion or anastomotic stenosis, and signs of systemic ventricular dysfunction [21].

After total cavopulmonary anastomosis, various unusual systemic venous collateral vessels have been described [22, 23]. Collateral vessels can develop from the innominate vein to the left atrium by way of pericardial veins or the coronary sinus and also from the superior vena cava to the pulmonary veins. These channels either evolve or enlarge with reversal of flow toward the lower-pressure left atrium. Although not noted before operation, they may have been present and then develop postoperatively within hours [5, 24]. Preceding reports as well as our study have not proved any relationship to underlying cardiac malformation or postoperative hemodynamic data. These communications have been described mainly after total cavopulmonary anastomosis [24, 25]. The feasibility and the effectiveness of device occlusion were reported for patients with arterial desaturation [23]. However, as risk factors for the development of systemic venous collaterals have not been clearly defined, development of new communications and recurrence over time have to be suspected in patients with increasing or persisting cyanosis. Particular attention must be paid to these collaterals at detailed angiographic assessment to identify their most common sites.

Aortopulmonary collateral vessels, frequently found after a Fontan procedure, often are present preoperatively [26, 27]. Their prevalence and clinical significance are not known, and identification partially depends on the site of selective angiography. In addition, there is a high risk of recurrence after coil occlusion. Thus, preoperative coil embolization or intraoperative ligation of the internal mammary artery has been performed to reduce early postoperative morbidity and mortality [26]. This might explain the low rate of postoperative coil embolization of collateral vessels in our patients (6/17 patients). However, patients with aortopulmonary collateral are at risk for both chronic increased volume load on the functionally univentricular heart and increased pressure in the pulmonary vascular system. Therapeutic options for patients with a failing Fontan circulation and symptomatic protein-losing enteropathy are few and carry high risk [4, 5, 28, 29]. In one study [30], transcatheter fenestration creation was effective in select patients and led to hemodynamic improvement. In our study, this transcatheter procedure prolonged the decision period for heart transplantation. In patients with associated sinus node dysfunction, atrial pacing might be an alternative way to increase cardiac output while avoiding the risk of hypoxemia potentially associated with fenestration [31].

In contrast to the early postoperative period, the spectrum of reoperations during long-term follow-up has changed and now is limited mainly to conversion of an atriopulmonary anastomosis to a total cavopulmonary anastomosis (intracardiac or extracardiac procedure) in patients with atrial reentry tachycardia refractory to medication. However, for maintenance and improvement of the Fontan circulation, a large and increasing number of transcatheter interventions have been performed. Indications include systemic venous collateralization, residual shunting, aortopulmonary collaterals, and, extremely rarely, requirement for fenestration creation. A regular postoperative cardiac catheterization program is recommended to identify early these abnormalities in asymptomatic patients to schedule appropriate therapy.

	References

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This Article Abstract Full Text (PDF) 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): Gerhard Ziemer Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Kaulitz, R. Articles by Hausdorf, G. Search for Related Content PubMed PubMed Citation Articles by Kaulitz, R. Articles by Hausdorf, G. Related Collections Congenital - cyanotic