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Ann Thorac Surg 1999;68:1361-1367
© 1999 The Society of Thoracic Surgeons

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Original Articles: Cardiovascular

Hemi-Fontan procedure for hypoplastic left heart syndrome: outcome and suitability for Fontan

^a Division of Pediatric Cardiovascular Surgery, Department of Surgery, University of Michigan School of Medicine, Ann Arbor, Michigan, USA
^b Division of Pediatric Cardiology, Department of Pediatrics, University of Michigan School of Medicine, Ann Arbor, Michigan, USA

Address reprint requests to Dr Bove, Division of Pediatric Cardiovascular Surgery, F7830 Mott Hospital, University of Michigan Medical Center, 1500 E Medical Center Dr, Ann Arbor, MI 48109
e-mail: elbove{at}umich.edu

Presented at the Thirty-fifth Annual Meeting of The Soceity of Thoracic Surgeons, San Antonio, TX, Jan 25–27, 1999.

	Abstract

Top Abstract Introduction Patients and methods Results Comment References

 
Background. Following the Norwood procedure for hypoplastic left heart syndrome (HLHS), pulmonary artery distortion and hypoplasia are common and may negatively impact late outcome. The hemi-Fontan procedure (HFP) augments the central pulmonary arteries and establishes a connection between the right atrial/superior vena cava junction and the pulmonary arteries, while excluding the inferior vena cava.

Methods. The hospital records of all 114 patients undergoing a HFP for HLHS between August 1993 and April 1998 were reviewed to assess patient, procedural, and morphologic determinations of outcome. The results of cardiac catheterization, Doppler/echocardiography, 12 lead electrocardiograms, hospital and subsequent course, as well as suitability and outcome for the Fontan procedure were analyzed.

Results. Mean age was 5.4 months (range 1.5 to 15 months). Right ventricular function was normal in 95 patients, moderately depressed in 14, and severely depressed in five. Tricuspid regurgitation was absent or mild in 91 patients, moderate in 13, and severe in 10. Concomitant procedures included left superior vena cava to pulmonary artery anastomosis (12), tricuspid valve repair (10), pulmonary artery stent placement (3), coarctation repair (2), and aortic pseudoaneurysm repair (1). Hospital survival was 112/114, 98% (95% confidence interval [CI]: 95% to 100%). There were two late deaths, one noncardiac. Sinus rhythm is present in 105 patients (92%, 95% CI: 87% to 97%). To date, 79 of these patients have undergone the Fontan procedure with 74 survivors (94%, 95% CI: 89% to 99%).

Conclusions. The HFP may be performed with excellent results for HLHS. It effectively augments the central pulmonary arteries while preserving sinus rhythm in the majority. In addition, the HFP facilitates the subsequent Fontan procedure and has significantly improved the overall outcome.

	Introduction

Top Abstract Introduction Patients and methods Results Comment References

 
The construction of an anastomosis between the superior vena cava (SVC) and the pulmonary artery (PA) is now a well established part of staged surgical reconstruction for hypoplastic left heart syndrome [1–8]. The SVC to PA connection removes the obligatory volume load on the ventricle imposed by a systemic to pulmonary artery shunt which allows time for resolution of excessive ventricular hypertrophy prior to the Fontan procedure [9–11]. There are two choices of second-stage procedures, the bidirectional Glenn procedure (BDG) and the hemi-Fontan procedure (HFP). In the BDG procedure, a connection between the SVC and the undivided pulmonary arteries is made, closing the cardiac end of the SVC. This procedure is generally straightforward and usually does not include augmentation of the central pulmonary arteries. Furthermore, it does not provide an established connection for the inferior vena cava at the time of the Fontan operation. The HFP includes augmentation of the central pulmonary arteries without dividing the SVC, while excluding inferior vena caval blood from the pulmonary arteries by means of a temporary intraatrial patch [12]. The construction of a large connection between the SVC/right atrial junction and the pulmonary artery simplifies a subsequent lateral tunnel Fontan operation by eliminating the need for the additional dissection that would otherwise be required to reconnect the right atrium to the pulmonary arteries. However, the HFP is technically more complex and may result in injury to the arterial supply to the sinoatrial (SA) node, the long-term effect of which is incompletely understood [13]. In order to examine these issues in a large series of patients undergoing operation for a uniform cardiac condition, we describe our experience with the HFP for hypoplastic left heart syndrome. The early and late mortality as well as patient, procedural, and morphologic risk factors for outcome were assessed. Additionally, suitability for the Fontan procedure and cardiac rhythm status were evaluated.

	Patients and methods

Top Abstract Introduction Patients and methods Results Comment References

 
Beginning in August 1993, the HFP was exclusively used as the method of second-stage reconstruction for hypoplastic left heart syndrome (HLHS) at C. S. Mott Children’s Hospital of the University of Michigan Health System. The hospital records of all patients undergoing a HFP for HLHS between then and April 1998 were retrospectively reviewed to assess patient, procedural, and morphologic determinants for outcome. The results of cardiac catheterization, Doppler/echocardiography, electrocardiograms, inpatient hospital, and subsequent course were analyzed. Suitability for, and outcome following, the Fontan procedure were evaluated. Follow-up 12-lead electrocardiographic data were reviewed on all 114 patients. The most current study was used for analysis and, whenever possible, the electrocardiogram performed just prior to the Fontan procedure was reviewed. All patients had classic HLHS as defined by the presence of a right ventricular dependent circulation in association with aortic valve atresia or hypoplasia [8]. Patients with a dominant left ventricle (even if a Norwood was performed), discordant ventriculoarterial connections, or a normal aortic arch (not requiring arch reconstruction) were excluded. All patients underwent preoperative cardiac catheterization, either at the University of Michigan or at the referring institution.

Operative technique
The procedure was performed using single right atrial venous cannulation, deep hypothermic cardiopulmonary bypass, and circulatory arrest. The pulmonary arteries were mobilized from the right to the left upper lobe branches and the azygous vein was ligated. The pulmonary arteries were opened from a point beginning just posterior to the SVC to the left upper lobe branch (Fig 1). By individual surgeon preference, an incision was either made from the tip of the right atrial appendage medially across the cavoatrial junction to the level of the right pulmonary artery (method 1) or, alternatively, the incision was stopped before the junction between the atrium and the SVC was reached in an effort to avoid the artery to the SA node (method 2, Fig 2). The atrial septal defect was enlarged by cutting back the coronary sinus. The inferior edge of the pulmonary arteriotomy was anastomosed to the opening in the cavoatrial junction (method 1) or to the outside of the SVC and the medial edge of the atriotomy (method 2) with a running absorbable suture (Fig 3). An intraatrial patch was placed to exclude inferior vena caval blood flow from the pulmonary arteries. A patch of cryopreserved pulmonary homograft was then used to augment the pulmonary arteries and create a roof for the entire connection. The patients were managed without the use of indwelling subclavian or internal jugular vein catheters to minimize the likelihood of SVC thrombosis.

View larger version (21K): [in this window] [in a new window]   Fig 1. The initial steps in the modified technique for the hemi-Fontan procedure. The shunt is divided and the pulmonary arteries are opened from the superior vena cava to the left upper lobe branch. An incision is made in the base of the right atrial appendage towards the superior vena cava but it does not cross the cavoatrial junction.

View larger version (31K): [in this window] [in a new window]   Fig 2. The openings in the right atrial appendage and the pulmonary artery are shown. A patch of cryopreserved pulmonary allograft (upper right) is fashioned to augment the central and proximal branch pulmonary arteries. The intraatrial patch used to separate the right atrium from the cavopulmonary connection is demonstrated.

View larger version (34K): [in this window] [in a new window]   Fig 3. The lower edge of the pulmonary artery incision is anastomosed to the opening in the right atrium, using the outside of the superior vena cava until the atrial incision is reached. Very superficial bites are taken to avoid injuring the sinoatrial nodal artery. The pulmonary allograft patch is sutured beginning over the left pulmonary artery and is used to form a roof for the cavopulmonary connection.

Data collection and statistical analysis
In addition to describing demographic, preoperative clinical characteristics, and clinical outcomes of this cohort, we aimed to determine clinical and demographic features associated with early and midcourse outcomes. Because the mortality rate was low in this cohort, we formulated a composite outcome which included both mortality and unsuitability for a Fontan procedure. Thus, patients who died at the time of the hemi-Fontan, died after the hemi-Fontan but before the Fontan, or are determined to be unsuitable for the Fontan operation are included in the adverse outcome group for this composite variable. All other patients were considered to have a favorable outcome for the purposes of these analyses. A second group of analyses were performed to determine demographic, hemodynamic, and morphologic features associated with the presence of sinus rhythm as determined by standard 12 lead electrocardiograms. Bivariate analyses to assess conditions associated with each of these outcomes utilized Student t test for continuous variables and Fischer’s exact and Wilcoxon rank sum tests for non-parametric variables. Multivariable models were tested with logistic regression. Data was stored in Excel and data analysis was performed using SAS statistical software (SAS Institute, Cary, NC).

Patient data
There were 114 patients included in the study. There were 71 males. Mean age at the time of the HFP was 5.4 months (range, 1.5 to 15 months) and weight 6.1 kg (range, 4.1 to 10.0 kg). Only 7 patients were older than 7 months of age. Preoperative Doppler/echocardiography revealed normal systolic right ventricular function in 95 patients, moderately depressed function in 14 patients, and poor function in 5 patients. Right ventricular function was determined semiquantitatively using standard imaging techniques. Tricuspid regurgitation was absent or mild in 91 patients, moderate in 13 patients, and severe in 10 patients. Preoperative angiography (Figs 4, 5) frequently demonstrated central pulmonary artery distortion and hypoplasia, most commonly at the origin of the left pulmonary artery (ductal insertion site). Additional sites of pulmonary artery distortion or narrowing were often identified at the right pulmonary artery origin or at the shunt anastomosis itself.

View larger version (168K): [in this window] [in a new window]   Fig 4. Angiogram performed just prior to the hemi-Fontan procedure. The modified Blalock–Taussig shunt is demonstrated with opacification of the branch pulmonary arteries. Both pulmonary arteries are small and, although it is not well seen in this view, there is a stenosis at the origin of the left pulmonary artery.

View larger version (156K): [in this window] [in a new window]   Fig 5. Follow-up angiogram performed just prior to the Fontan procedure in the same patient as shown in Figure 1. Significant enlargement of the pulmonary arteries is demonstrated with contrast filling all branches. The intraatrial patch is shown.

	Results

Top Abstract Introduction Patients and methods Results Comment References

Morbidity
Major complications included mediastinitis (6), phrenic nerve paralysis requiring diaphragm plication (5), pulmonary artery thrombosis (5), need for extracorporeal membrane oxygenation (2), heart block (2), and necrotizing enterocolitis (1).

One patient developed complete heart block after the Norwood procedure, and 2 other patients developed heart block after the HFP; one of these patients had atrial flutter prior to operation. All 3 of these patients required a permanent pacemaker. Of the remaining patients, 6 have standard 12 lead electrocardiographic evidence of sinus node dysfunction (low atrial rhythm in 4, junctional rhythm in 1, and ectopic atrial rhythm in 1) and 105 are in normal sinus rhythm (105/114, 92%, 95% CI: 87% to 97%). There was no difference between the two operative techniques with regards to the cardiac rhythm. However, in a separate analysis involving a subgroup of these patients, those in whom the cavoatrial junction was not incised had a significantly higher incidence of normal sinus rhythm at the first postoperative electrocardiogram performed in the intensive care unit, whereas patients in whom the cavoatrial junction was incised were more likely to have junctional rhythm early and regain sinus rhythm within the first few postoperative days (data not shown).

Five patients had pulmonary artery occlusion in the early postoperative period. One patient died and one ultimately underwent successful orthotopic cardiac transplantation following successful pulmonary artery thrombectomy. One additional patient underwent a successful thrombectomy and is awaiting a Fontan. The left pulmonary artery remains occluded in the remaining 2 patients.

Suitability for Fontan
To date, 79 patients have undergone the Fontan procedure with 74 survivors (94%, 95% CI: 89% to 99%) (Fig 6). Twenty-five patients are currently awaiting the Fontan procedure while 6 are considered unacceptable candidates secondary to right ventricular dysfunction and/or pulmonary artery distortion or occlusion. Of those 6 patients, 1 patient underwent intraoperative stenting of the left pulmonary artery during the HFP which failed to adequately relieve pulmonary artery stenosis, and 3 had pulmonary artery thrombosis in the postoperative period. The remaining 2 patients have poor right ventricular function. Of the 25 patients awaiting the Fontan procedure, 4 patients are considered marginal candidates secondary to residual pulmonary artery stenosis, and there is depressed right ventricular function in all 4 patients.

View larger version (16K): [in this window] [in a new window]   Fig 6. Overall survival and suitability for the Fontan procedure.

	Comment

Top Abstract Introduction Patients and methods Results Comment References

 
The application of a superior vena cava to pulmonary artery connection has been well accepted in the staged reconstruction of patients with functional single ventricle, including those with HLHS. The SVC to PA connection removes the volume overload on the right ventricle associated with a systemic to pulmonary artery shunt while increasing effective pulmonary blood flow to maintain acceptable systemic arterial oxygen saturation.

The HFP has been our preferred choice over the BDG since 1993. This decision evolved from our earlier experience with the BDG procedure which, although initially favorable, was associated with an increase in complications at the time of the Fontan due to subtle but significant pulmonary artery stenoses, particularly at the origin of the left pulmonary artery, and the need to reestablish a connection between the pulmonary arteries and the right atrium [14]. It became apparent that the majority of patients with HLHS require augmentation of the central pulmonary arteries, and that this was best accomplished at the second stage procedure rather than at the Fontan for a variety of reasons. Although longer cardiopulmonary bypass time has been identified as a risk factor for survival following the Fontan procedure [14–16], this appears to be better tolerated during the HFP. After the Fontan, an increase in extravascular lung water and myocardial diastolic dysfunction associated with long periods of cardiopulmonary bypass and aortic cross-clamping may limit cardiac output in the early postoperative period [17]. However, after a HFP, cardiac output is maintained by the inferior vena caval flow which does not go through the pulmonary vascular bed. Additionally, routine augmentation of the pulmonary arteries and the establishment of the connection between the cavoatrial junction and the pulmonary artery at the HFP reduces the dissection required at the Fontan procedure and, therefore, the potential for bleeding as well as injury to the phrenic nerve and SA node [18]. Therefore, the need to augment the pulmonary arteries and the desire to limit operative dissection, as well as bypass and cross-clamp times during the Fontan procedure, led to the use of the HFP as our procedure of choice for HLHS. Although the HFP is a more extensive operation which is performed with a period of circulatory arrest, the smooth postoperative recovery and the brief period of intubation and hospital length of stay in the majority of patients support its continued use. With increasing experience, the circulatory arrest times have been gradually reduced and neurodevelopmental testing after all three stages has been reported by our institution to be associated with excellent outcomes [19].

Our series demonstrates that the HFP may be performed with a low operative mortality, despite the extensive nature of the operation and the high risk cardiac diagnosis. Poor right ventricular systolic function was associated with adverse outcome, although only one patient died following the HFP. However, right ventricular function generally remained poor, and these patients were not felt to be suitable for a Fontan procedure (Fig 7). In contrast, there was no evidence that preoperative tricuspid regurgitation was associated with adverse outcome. Of the 10 patients who required tricuspid valve repair, there were no early or late deaths following the HFP indicating that tricuspid regurgitation can be successfully treated by standard operative techniques [20, 21]. Younger age, lower weight, and earlier year of operation were also associated with adverse outcome, either death or unsuitable for Fontan. It is not surprising that experience was associated with improved outcome. Of note, no particular age or weight had meaningful positive or negative predictive value. Thus, these factors would not deter us from performing the HFP on an otherwise appropriate younger age or lower weight patient.

View larger version (15K): [in this window] [in a new window]   Fig 7. Impact of poor right ventricular systolic function on outcome. (RV = right ventricular; HFP = hemi-Fontan procedure.)

Nearly 95% of long-term survivors in our series (104/110) have either undergone or remain good candidates for the Fontan procedure. When evaluating the merits of this approach, it is important to also examine the results of the subsequent Fontan procedure. A detailed report of the results of the Fontan procedure for HLHS at this institution is available in a separate report [14]. The mortality for the Fontan procedure has been reported to be between 5% and 10% in a number of recent series [7, 15]. Before 1993, when the BDG procedure was used routinely as the second stage of reconstruction, the mortality of the Fontan procedure for HLHS at the University of Michigan was 21% [14]. Since 1993, when the HFP became used routinely as the second step, the Fontan mortality has been reduced to 2%. Although other factors may have played an important role in this improvement, we believe that the type of second-stage procedure itself is a major reason. In the current series, four of the five deaths following the Fontan occurred early in our experience and there have been no deaths in the last 57 patients undergoing a Fontan for HLHS.

Because atrial dysrhythmias are an important cause of early and late morbidity following the Fontan procedure, we investigated whether the technique of the HFP as employed in this series was associated with a loss of sinus rhythm. Early in this experience, it was noted that many patients were in junctional rhythm for the first 24 to 48 hours following operation. The cause of this finding was believed to be sinus node dysfunction secondary to injury to the SA nodal artery [13]. This led one surgeon to change the technique, modifying the incision in the right atrium to avoid crossing the superior vena cava/right atrial junction (Figs 2, 3). Although the prevalence of sinus rhythm on the first electrocardiogram performed in the intensive care unit immediately following the HFP significantly improved with the modified technique, no difference between the two techniques was found on the late rhythm evaluation and only 6 patients were found to have sinus node dysfunction late after operation. However, a more detailed electrophysiologic investigation into sinus node function in these patients has demonstrated significantly longer sinus node recovery times among those patients with the original technique when compared to the modified approach [22]. The long-term impact of these findings remains unknown, but these data suggest that avoiding injury to the SA nodal artery may be associated with improved late sinus node function.

A number of recent studies have demonstrated improved hemodynamics with the use of caval offset to avoid competitive flow between the superior and inferior vena cavae [23–25]. These reports indicate that it is better to augment inferior caval flow, even at the expense of superior caval return, because superior vena caval hypertension appears to be better tolerated. An additional, although as yet unproved, benefit of the HFP is that caval offset is obtained in an anteroposterior direction. Flow from the inferior vena cava enters the pulmonary arteries in a direct, streamlined path while that from the superior vena cava turns posteriorly (Fig 8).

View larger version (138K): [in this window] [in a new window]   Fig 8. Angiographic appearance of the caval pathways after the Fontan procedure as shown in the lateral projection. Note the direct path for the inferior vena cava to the pulmonary arteries. The superior vena cava is more anterior and its venous return must turn posteriorly to enter the pulmonary arteries, favoring inferior vena caval return. The fenestration is profiled.

	Footnotes

 
This article has been selected for the open discussion forum on the STS Web site: http://www.sts.org/section/atsdiscussion/

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