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J Thorac Cardiovasc Surg 1998;115:547-556
© 1998 Mosby, Inc.
SURGERY FOR CONGENITAL HEART DISEASE |
From the Divisions of Cardiovascular-Thoracic Surgery and Cardiology, The Children's Memorial Hospital, and the Departments of Surgery and Pediatrics, Northwestern University Medical School, Chicago, Ill.
Presented at the Second World Congress of Pediatric Cardiology and Cardiac Surgery, Honolulu, Hawaii, May 11-15, 1997.
Received for publication June 13, 1997; revisions requested July 30, 1997; revisions received Sept. 8, 1997; accepted for publication Sept. 8, 1997. Address for reprints: Constantine Mavroudis, MD, Division of Cardiovascular-Thoracic Surgery, Mail Code 22, Children's Memorial Hospital, 2300 Children's Plaza, Chicago, IL 60614.
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Abstract |
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Introduction |
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1-5 leading to the present-day practice of total cavopulmonary artery connections using lateral tunnel 3 or extracardiac 6 techniques. Interatrial fenestration is often, but not always, used depending on the number of patient risk factors 7,8 and institutional experience. 9
However, a large number of Fontan patients had variations of atriopulmonary artery connections before the present era. These, mostly older patients, are at risk for anatomic obstructions and atrial arrhythmias that usually lead to unfavorable hemodynamics and systemic complications. 10,11 Recent reports have shown improved functional class in some patients after atriopulmonary artery Fontan conversion to total cavopulmonary artery connection. 12-15 Concomitant antiarrhythmia therapy consisting of arrhythmia circuit cryoablation in these patients has not been previously reported.
The purpose of this article is to review our surgical experience with patients with atriopulmonary artery Fontan operations who had obstructive anatomic or arrhythmia indications for conversion to total cavopulmonary artery connection, arrhythmia circuit cryoablation, and placement of atrial antitachycardia pacemaker. Not included in this review are the detailed perioperative electrophysiologic (EP) analyses and relevant discussion that are reported separately. 16,17
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Materials and methods |
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Table I reviews clinical characteristics of our 14 patients, including primary diagnoses, previous Fontan connections, indications for reoperation, components of the Fontan conversion, and outcome. Indications for reoperation were multiple and included New York Heart Association (NYHA) functional class, debilitating atrial arrhythmias, anatomic obstructions, interatrial shunts, and valvular dysfunction. Nine patients were in NYHA class IV, and three of these were considered for heart transplantation because of severe functional limitation. Four were in NYHA class III and the remaining patient was in NYHA class II, but had life-threatening atrial tachycardia, resulting in multiple syncopal episodes. Apart from functional class, the primary indications for surgery were debilitating atrial arrhythmias in 11, severe obstructive lesions in 2, and bradycardia with cyanosis caused by a right-to-left shunt in 1.
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The conduct of the operation was essentially the same for all patients except for patients 1, 2, and 11, who did not have concomitant arrhythmia circuit cryoablation.
Careful median sternotomy and relevant dissection were followed by normothermic aortobicaval cardiopulmonary bypass, allowing the heart to beat. In those patients without a right-to-left residual shunt, a right atriotomy was performed for intracardiac EP mapping and cryoablation. A right atrial bipolar wire served as the reference electrode and electrical pathway for arrhythmia stimulation. EP mapping was accomplished by activation sequence mapping during atrial reentry tachycardia (Fig. 1). An endocardial grid was used to sequentially map at least four corridors of atrial tissue, moving superiorly to inferiorly, at four to five sites for each corridor. These results were compared with the preoperative mapping findings. In some cases the areas of interest (e.g., the coronary sinus and tricuspid valve) were in the pulmonary atrium and could not be safely mapped without the risk of air embolism. On the basis of EP arrhythmia circuit identification, multiple cryoablation lesions were placed (–60° C for 90 seconds each [Frigitronics, Shelton, Conn.]) in a linear fashion between the coronary sinus os and the inferior vena cava, between the tricuspid or relevant atrioventricular valve anulus and the inferior vena cava (Fig. 2, Area 1), along the full length of the crista terminalis (Fig. 2, Area 2), along the length of the superior septal limbus, and the os of the atrial appendage (Fig. 2, Area 3). In general, Area 1 required 8 to 10 cryoablation lesions, Area 2 required 4 to 6 lesions, and Area 3 required 3 to 5 lesions. Most of the cryoablation lesions were placed during warm cardiopulmonary bypass in a beating heart. When access to the compartmentalized pulmonary atrium was required, further cryoablation was delayed until systemic cooling and cardioplegic arrest were established to avoid air embolism.
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17 ventricular vent placement, atrial wall reduction (performed in most patients), and atriorrhaphy. The atrial pacemaker lead is placed in the "red" atrium by means of the transmural technique reported by Hoyer and associates. 18 This is followed by de-airing, aortic crossclamp removal, rewarming, vent removal, and extracardiac inferior vena cava connection when elected (Figs. 3, 4
). Separation from cardiopulmonary bypass is followed by transesophageal echocardiographic assessment. Patients were usually extubated the following day and underwent EP study and attempted arrhythmia induction before hospital discharge.
Follow-up.
Clinical status was assessed on the basis of the functional classification of the NYHA. Arrhythmia assessment included review of clinical history, electrocardiograms, 24-hour continuous Holter monitoring, and pacemaker interrogation at regular 3-month intervals. All patients were placed on lifelong warfarin sodium (Coumadin) therapy (international normalized ratio maintained in the 1.5 to 2.0 range). Specific antiplatelet therapy was not used.
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Results |
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Resternotomy was accomplished safely and without significant cavitary entry in 12 of 14 patients. Two patients had unwanted cavitary entry during the resternotomy. One of these (second-time redo Fontan, discussed above) was managed by expeditious femoral-femoral partial cardiopulmonary bypass with intracavitary cardiotomy sucker assistance until completion sternotomy and aortobicaval cardiopulmonary bypass could be established. Another patient was quickly managed with femoral-femoral partial cardiopulmonary bypass but without intracavitary cardiotomy sucker assistance. This prevented air from entering the right atrium and allowed completion sternotomy with aortobicaval conversion without a period of instability. The patient had no complications after a successful Fontan conversion and arrhythmia circuit cryoablation. Subsequently, one patient with a gigantic right anterior atrium was placed on femoro-femoral bypass for atrial decompression before sternotomy. This was then converted to aortobicaval cannulation for the conduct of the operation.
One patient required reoperation (during the same admission) for retrieval of a Lock clamshell device (USCI/C.R. Bard, Inc., Bellerica, Mass.) after a failed attempt at postoperative atrial fenestration closure. We have since abandoned atrial fenes-tration for these patients, speculating that postoperative improved hemodynamics and maintenance of systemic oxygen saturation will benefit the patient more than the enhanced cardiac output and obligatory systemic desaturation that accompanies a fenestration. 19
Transmural steroid-eluting atrial leads and atrial antitachycardia pacemakers were placed in 10 patients who had arrhythmia circuit cryoablation. The rationale for this therapy is that the atrial wire is more easily placed at the revision operation than at a future time of need when obligatory thoracotomy would be necessary, and that the atrial antitachycardia pacemaker may be needed in the event of recurrent atrial tachycardia or the development of a new atrial arrhythmia that might arise from existing atrial suture lines. One patient with sinus bradycardia had an atrial rate-responsive (AAIR) pacemaker and another patient with single ventricle and l-transposition had a dual-chamber (DDD) pacemaker placed because of atrioventricular dissociation after arrhythmia circuit cryoablation. Postoperatively, the patient regained normal atrioventricular node function without inducible atrial tachycardia, which allowed pacemaker conversion to an AAI mode. Should atrial tachycardia recur, this pacemaker may be manually programmed for arrhythmia conversion. The remaining two patients had total cavopulmonary artery conversion before our arrhythmia protocol was initiated.
The average postoperative length of stay in the remaining 13 survivors was 10 ± 3 days. Chest tube output was minimal (removal of last chest tube, postoperative day 7 ± 3). Postoperative atrial tachycardia developed in one patient (No. 9) while receiving inotropic support, requiring procainamide infusion. Another patient (No. 14) had transient junctional ectopic tachycardia develop responding to intravenous amiodarone, which was subsequently withdrawn with no evidence of inducible supraventricular tachycardia before discharge.
No long-term deaths occurred. The mean duration of follow-up in the 13 survivors is 1.7 years (standard deviation 1.6 years, range 5 months to 5 years). All patients are currently considered to be in NYHA class I or II, with resumption of normal activities. Among the 11 patients whose primary indication for reoperation was medically refractory atrial tachycardia, only 2 patients are receiving antiarrhythmic medications other than digoxin. One patient (No. 9, see above) had a single clinical recurrence of adenosine-sensitive atrial tachycardia 4 weeks postoperatively, and no recurrences in the subsequent 14 months while taking procainamide; her preoperative arrhythmia was not adenosine-sensitive. Another patient (No. 4) with incessant, hemodynamically unstable rapid atrial tachycardia preoperatively, had a slow, asymptomatic atrial tachycardia develop 21 months postoperatively, responding to ß-blocker therapy. In the 10 patients with atrial antitachycardia pacemakers, tachycardia has been successfully detected and terminated by the pacemaker in three of 10 patients (Table I), all occurring in the first 3 months postoperatively, with no subsequent recurrences. None of these patients have been aware of clinical episodes of tachycardia.
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Discussion |
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3-11,19 Long-term follow-up will determine the efficacy of these techniques with regard to improved hemodynamics and arrhythmia control. 3,4,6,8,19-23 However, increasing concern exists that atriopulmonary Fontan patients, who underwent repair before the present era, are at risk for anastomotic obstructions and atrial arrhythmias. 12-15,24-29 In addition, pulmonary arteriovenous fistulas are frequently present in those patients with discontinuous pulmonary arteries caused by an established right Glenn procedure and atrial–left pulmonary artery connections. 30 Other patients will have left-sided valvular or obstructive lesions, acquired right-to-left shunts, and protein-losing enteropathy develop. 10,11,31
Reoperation in patients with atriopulmonary Fontan procedures to correct obstructive lesions and convert them to total cavopulmonary artery connections has been successfully performed by several groups. 12-15 Hemodynamic improvement and increase in functional class were achieved in most patients with acceptable mortality and morbidity. Our experience compares favorably with these reports, demonstrating a low mortality (7%) and a uniform increase in functional class. The significance of these cumulative results is profound considering that the only alternative for many of these patients is cardiac transplantation, which was subsequently performed once in McElhinney's series 13 and once in Kreutzer's series. 14
The rationale for arrhythmia therapy in these previously reported cases 12-15 was based on reduction atrioplasty and elimination of the high-pressure atrium 25-27 by lateral tunnel or extracardiac techniques. Except for perioperative atrial antitachycardia pacemaker placement in selected patients, no specific attempt at arrhythmia circuit interruption was made. As a consequence, atrial arrhythmias persisted in a significant number of their patients and developed anew in others who had extensive atrial reduction. 13 These results are not surprising based on experimental animal studies by Gandhi and associates 32 that implicated preexisting Fontan intraatrial suture lines as the substrate for reentry atrial tachycardia. In a concurrent comparative clinical report, 16 we found that atriopulmonary Fontan conversion to total cavopulmonary artery connection without arrhythmia circuit cryoablation results in uniform recurrence of atrial arrhythmias, supporting in human beings what was found in Gandhi's animal studies. 32 We conclude from these studies that preexisting Fontan atrial suture lines are critical to the pathogenesis of reentry atrial tachycardia and that relief of atrial hypertension by means of lateral tunnel or extracardiac techniques, although important for optimal hemodynamic results, will not effectively control atrial arrhythmias unless accompanied by arrhythmia circuit cryoablation.
Reoperation in Fontan patients with established atriopulmonary artery connection (with or without discontinuous pulmonary arteries) involves three important components to achieve improved hemodynamics and effective arrhythmia control. The first is safe resternotomy and effective total cavopulmonary artery reconstruction; the second is EP mapping and comprehensive arrhythmia circuit cryoablation; and the third, in our practice, is the prophylactic placement of an atrial antitachycardia pacemaker.
Resternotomy in Fontan patients with large, thin-walled, and oftentimes anterior right atria requires careful consideration of anatomic and pathophysiologic conditions that can easily be destabilized in the event of unwanted and uncontrolled cavitary entry. Indeed, one of our patients eventually died because of a presumed cerebral air embolism caused by cavitary entry during resternotomy despite expeditious femoral cannulation and effective total cavopulmonary artery conversion. In selected patients with the aforementioned risk factors (one in this series), we recommend femoral artery–femoral vein partial cardiopulmonary bypass to decompress the right atrium for more effective and safer resternotomy. 13 After dissection, conversion to aortobicaval cannulation and femoral vessel repair is performed. Conversion from atriopulmonary to total cavopulmonary artery connection is relatively straightforward. In most cases, the pulmonary arteries must be completely dissected because a significant number of connections are anteroaortic and should be converted to a retroaortic position. All pulmonary artery stenoses should be addressed by arterioplasty (we prefer PTFE), which can be followed by intraatrial lateral tunnel or extracardiac inferior vena cava–pulmonary artery connection. The retrospective reconstructive trends in this small series of patients are less use of atrial fenestration and greater use of aortic homografts and PTFE tubes for the extracardiac connection. 6,20,21 Our unsubstantiated bias is to avoid atrial fenestration and systemic desaturation despite the increased cardiac output 19 in favor of the resultant improved hemodynamics in a patient already adapted to the increased systemic venous pressure of the "Fontan physiology." The extracardiac connection is attractive because it is generally easier to perform and has the added benefit of fewer atrial suture lines that are one of the substrates for atrial tachycardia. 33
The exact sites for arrhythmia circuit cryoablation in our patients were based on perioperative EP studies 16 and previous reports 34-36 that defined the predominant anatomic critical isthmus for slow conduction in the area between the coronary sinus os, tricuspid anulus, and the inferior vena cava os. We identified three major anatomic sites thought to be critical to the tachycardia circuit (Fig. 2). These were Area 1, the area between the coronary sinus os and the inferior vena cava os, as well as the area between the atrioventricular valve anulus and the inferior vena cava os; Area 2, the lateral atriotomy, which corresponded to the length of the crista terminalis; and Area 3, the region at the superior limbus corresponding to the rim of the prior atrial septal defect patch. All patients had cryoablation lesions placed in Area 1. In those selected patients requiring further therapy in Areas 2 and 3, cryoablation lesions were linearly placed to connect these areas with an anatomic orifice (i.e., vertical lesions along the crista terminalis from the superior vena cava to the inferior vena cava or lesions to connect the superior limbus to the superior vena cava). With this strategy, all patients had acutely successful atrial arrhythmia ablation, although two patients had new atrial arrhythmias develop that were easily controlled with single-drug therapy.
Although we believe that the cryoablation technique is critical to interrupt the reentrant tachycardia circuit, the limitations of our mapping technique and the possibility of creating new arrhythmia substrates may result in postoperative tachycardia. Transvenous catheter access to the heart for radiofrequency catheter ablation or pacing will be limited postoperatively, particularly in patients with an extracardiac tunnel. Reoperation for placement of an antitachycardia pacemaker will require an additional sternotomy. The rationale for the atrial wire and antitachycardia pacemaker represents "backup therapy" in the event of cryoablation failure. Early in our experience, finding a favorable right atrial wire location for the antitachycardia pacemaker was often problematic because of the endocardial fibrosis often present in the right side of the atrium. We subsequently found that the left atrial appendage is a superior site, yielding favorable EP measurements. 17 The advantages of this technique include accurate wire placement and fixation aided by cardioplegic arrest and lack of left atrial endocardial fibrosis because reconstructive suture lines are rarely placed in this area. The disadvantage of this technique is that the atrial wire can only be tested after cavitary closure and separation from cardiopulmonary bypass, when manipulation is severely limited. Thus far, we have not experienced systemic complications from the left-sided atrial wires, perhaps owing to the improved hemodynamics and long-term warfarin administration. 37 We therefore continue to recommend prophylactic atrial antitachycardia pacemaker placement in these patients to add another measure of therapy should cryoablation fail or a new arrhythmia circuit develop.
The primary indication for surgery in most of our patients was medically refractory atrial arrhythmias associated with syncope, stroke, or severe functional incapacity. Present-day management of atrial tachycardia in Fontan patients by pharmacotherapy, 38 radiofrequency ablation, 34-36 and antitachycardia pacing 39 has met with variable success and significant morbidity. 38Our own comparative study 16 and reports from others 12-15t 28 indicate that Fontan conversion without specific arrhythmia therapy improves hemodynamics but does not alter the course of atrial arrhythmias. We have shown that intraoperative mapping and selective cryoablation can interrupt the arrhythmia circuit in most patients without pharmacotherapy or dependence on atrial antitachycardia pacemakers.
Reoperation in Fontan patients with significant debilitating atrial arrhythmias with or without obstructive lesions can be accomplished with low morbidity and mortality, resulting in oftentimes dramatic increases in patient activity and function. We recommend that patients with atriopulmonary artery Fontan connections be closely followed up for obstructive lesions, valvular dysfunction, and atrial arrhythmias with a low threshold for reoperation, even if the only indication is debilitating atrial arrhythmias. Prophylactic cryoablation in first-time Fontan reconstructions to prevent atrial arrhythmias is an intriguing issue that will require a controlled study to determine its efficacy.
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C. Mavroudis, C. L. Backer, L. M. Kohr, B. J. Deal, J. Stinios, A. J. Muster, and D. F. Wax Bidirectional Glenn shunt in association with congenital heart repairs: the 1 1/2 ventricular repair Ann. Thorac. Surg., September 1, 1999; 68(3): 976 - 981. [Abstract] [Full Text] [PDF]
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S. A. Thorne, I. Barnes, P. Cullinan, and J. Somerville Amiodarone-Associated Thyroid Dysfunction : Risk Factors in Adults With Congenital Heart Disease Circulation, July 13, 1999; 100(2): 149 - 154. [Abstract] [Full Text] [PDF]
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 E. Magosso, S. Cavalcanti, and M. Ursino Theoretical analysis of rest and exercise hemodynamics in patients with total cavopulmonary connection Am J Physiol Heart Circ Physiol, March 1, 2002; 282(3): H1018 - H1034. [Abstract] [Full Text] [PDF]
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