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Ann Thorac Surg 2006;82:1316-1321
© 2006 The Society of Thoracic Surgeons

Original Articles: Cardiovascular

Total Transatrial Correction of Tetralogy of Fallot: No Outflow Patch Technique

Balram Airan, MCh*, Shiv Kumar Choudhary, MCh, Honnakere Venkataiya Jayanth Kumar, MS, Sachin Talwar, MCh, Jayesh Dhareshwar, MCh, Rajnish Juneja, DM, Shyam Sunder Kothari, DM, Anita Saxena, DM, Panangipalli Venugopal, MCh

Cardiothoracic Centre, All India Institute of Medical Sciences, New Delhi, India

Accepted for publication March 31, 2006.

* Address correspondence to Dr Airan, Department of Cardiothoracic and Vascular Surgery, All India Institute of Medical Sciences, New Delhi, 110029 India (Email: iactscon_2004{at}yahoo.co.in).

Presented at the Forty-second Annual Meeting of The Society of Thoracic Surgeons, Chicago, IL, Jan 30–Feb 1, 2006.


   Abstract
Top
 Abstract
Introduction
 Patients and Methods
 Results
 Comment
 Discussion
 References
 
BACKGROUND: The aim of this study was to analyze the feasibility and early results of transatrial total correction of tetralogy of Fallot (TOF).

METHODS: Of the 860 patients undergoing total correction for TOF between January 2000 and July 2005, 334 patients were considered morphologically suitable for transatrial total correction. The ventricular septal defect (VSD) closure, infundibular resection, and pulmonary valvotomy were performed through the right atrium without a right ventriculotomy. Age ranged from 6 months to 40 years (median, 2.8 years), and weight ranged from 5.5 to 70 kg (median, 14 kg).

RESULTS: Peroperatively, 34 patients required right ventriculotomy and transannular patch; hence, they were excluded from the study. In addition, pulmonary arteriotomy was required in 71 patients (22.9%). There were 4 hospital deaths. There were 4 early reoperations (residual/additional VSD in 3 and tricuspid regurgitation in 1). Two patients had complete heart block requiring permanent pacemaker. Echocardiography at discharge showed a peak right ventricular outflow tract gradient of 20 ± 5.2 mm Hg. Mean follow-up was 26.8 ± 4.2 months (range, 1 to 52 months). The right ventricular outflow tract gradients reduced to 13 ± 4.2 mm Hg after a mean interval of 18.8 ± 5.2 months. Follow-up New York Heart Association class was I in 240 cases (82%), II in 49 (16%), and III in 7 (2%). There were no late deaths or reoperations.

CONCLUSIONS: Transatrial total correction of TOF can be accomplished in selected patients with good early results. In 300 cases (90%), the feasibility of transatrial total correction could be predicted accurately.


   Introduction
Top
 Abstract
 Introduction
Patients and Methods
 Results
 Comment
 Discussion
 References
 
Since the first description by Hudspeth and colleagues [1], others [2] have also shown the feasibility of transatrial approach for total correction of tetrology of Fallot (TOF) so as to avoid the risk of sudden death from ventricular arrhythmias and also to prevent progressive right ventricular dysfunction [3]. Here, we discuss our experience with transatrial total correction for TOF.


   Patients and Methods
Top
 Abstract
 Introduction
 Patients and Methods
Results
 Comment
 Discussion
 References
 
Between January 2000 and July 2005, 860 patients underwent total correction for TOF at the All India Institute of Medical Sciences, New Delhi. Of these, 334 patients were considered suitable for transatrial total correction preoperatively. Thirty-four of these patients eventually required right ventriculotomy and transannular patching at operation; hence, they were excluded from the study. Informed consent was obtained from all the patients. All these patients were individually provided detailed information about the nature of the surgical procedure and the need for detailed regular follow-up studies without individually identifying them. Because this was a retrospective analysis of a clinical series involving a well-established surgical procedure, the chairman of the Ethics Committee waived the need for a formal approval from the committee as long as the identity of individual patients was not disclosed.

Of all patients, 192 were male; median age was 2.8 years (range, 6 months to 40 years). Median weight was 14 kg (range, 5.5 to 70 kg). Age and weight distribution is shown in Figures 1 and 2, Go respectively. Twenty-six patients (8.6%) had a prior modified Blalock -Taussig shunt. Fourteen patients required coil embolisation of the aortopulmonary collaterals. Associated cardiac anomalies were present in 74 patients (24.6%; Table1). Twenty patients (6.7%) had an anomalous left anterior descending coronary artery crossing the right ventricular outflow tract (RVOT). One patient had absent left pulmonary artery. The ventricular septal defect (VSD) closure, infundibular resection, and pulmonary valvotomy were performed through the right atrium. There was no ventriculotomy.


Figure 1
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  		Fig 1. Age distribution of the patients.

 

Figure 2
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  		Fig 2. Weight distribution of the patients.

 

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  		Table 1. Associated Anomalies in Patients Undergoing Total Transatrial Repair for Tetralogy of Fallot (N = 300)
 
Selection Criteria
Preoperative echocardiography, cardiac catheterization, and cineangiography were performed in all patients as a matter of institutional policy. Patients with favorable anatomy were planned for a right atrial approach for total correction. Favorable cardiac anatomy was defined as discrete infundibular obstruction with adequate pulmonary annulus and good-sized branch pulmonary arteries. All Z-values upto –3 were considered suitable for transatrial approach. A long segment infundibular hypertrophy was not an absolute contraindication. The pulmonary valve morphology did not influence the decision making.

Absolute contraindication for the trans–right atrium correction was the need for the enlargement of the pulmonary annulus or pulmonary arterioplasty as judged by echocardiography and cineangiography.

The degree of aortic override did not alter the approach for correction. However, as a matter of institutional policy, total correction was planned in patients older than 3 months or weighing 5 kg or more.

We performed transatrial repair with the above criteria even in patients with severe infundibular hypertrophy with a arterial saturation as low as 60% and also in situations requiring emergency surgery.

Surgical Technique
The operation was performed under standard cardiopulmonary bypass with bicaval cannulation and moderate hypothermia upto 28°C. Sodium nitroprusside infusion (0.5 µg · kg–1 · min–1) was started at the time of aortic cannulation. The operative procedure is schematically depicted in the Figure 3. Interrupted horizontal mattress sutures around the posterosuperior margin of the VSD helped in retracting the septal leaflet of the tricuspid valve for improved exposure. An intracardiac sucker was placed in the infundibular ostium, and resection was started with sharp dissecting scissors all around its edges. Particular care was taken not to resect too much of the parietal band and along the anterior margin of the VSD. Also, it was borne in mind that the ostium does not lie in a single plane. All the fibrous tissue was excised. That was followed by a wedge resection of the RVOT anteriorly and longitudinally, slitting the infundibulam to the pulmonary annulus.


Figure 3
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  		Fig 3. (a) Exposure of the ventricular septal defect and the infundibulam through the tricuspid valve. (b) Infundibular resection is complete; the pulmonary valve is now seen. (c) Pulmonary valvotomy is done. (d) Ventricular septal defect closure completed.

 
After adequate resection, the pulmonary valve was visualized, and its cusps were pulled down and everted, and valvotomy was performed. If pulmonary valve exposure was unsatisfactory, a small longitudinal arteriotomy was made in the main pulmonary artery (n = 71). The pulmonary valve was inspected through the arteriotomy, and commissurotomy was performed, if required. That was mainly in the early part of our experience, particularly if the infundibular resection was considered inadequate. However, with increasing experience, that is rarely required now. To avoid undersizing the annulus in a flaccid arrested heart, it was ensured that a Hegar's dilator at least two sizes more than that predicted by the Rowlatt chart [4] for that particular patient could be passed easily. The patch closure of VSD was then performed. A small atrial septal defect or patent foramen ovale was left open in borderline cases or in children less than 10 kg body weight.

Low-dose dopamine infusion was started at the initiation of rewarming. Right ventricular pressures were not routinely measured intraoperatively if the patient was uneventfully weaned off bypass with good hemodynamics.

Follow-Up
The patients were usually discharged on the 5th postoperative day after a baseline chest radiograph, an electrocardiogram, and postoperative echocardiography. The patients were then followed up at 1 week, and at 1, 3, 6, and 12 months, and then on a yearly basis. At each visit, the patients were assessed clinically and underwent electrocardiography and chest radiography. Follow-up echocardiography assessment was done for residual VSD, presence of tricuspid regurgitation, RVOT gradient, pulmonary regurgitation, aortic regurgitation, and biventricular function. Postoperative echocardiography was performed after 3 months' follow-up and then yearly. Between January 2005 and July 2005, 290 of the 300 patients (96%) underwent a detailed assessment as described above, and this interval was considered the closing interval for the study.

Statistical Methods
The accumulated data were analyzed using SPSS for Windows 10.0 Software package (SPSS, Chicago, Illinois). Data are expressed as numbers and simple percentages. Mean, median, and standard deviation are calculated for continuous variables.


   Results
Top
 Abstract
 Introduction
 Patients and Methods
 Results
Comment
 Discussion
 References
 
Among the 334 patients selected preoperatively for the transatrial repair, 34 patients required a ventriculotomy and transannular patch. In 29 patients (8.5%), after the infundibular resection was performed, the pulmonary annulus was considered to be inadequate as judged by the Hegar sizing. In 5 patients, the resection and the pulmonary annulus was considered to be adequate; however, after the termination of the bypass, the hemodynamics were not satisfactory, and pressure measurements demonstrated significant residual RVOT obstruction. In these 5 patients, cardiopulmonary bypass was reinstituted, and a transannular patch was used. These 34 patients were considered as failures and were excluded from the study.

The mean cardiopulmonary bypass time was 70 ± 21.8 minutes (range, 43 to 162), and the mean aortic cross clamp time was 41 ± 12.8 minutes (range, 28 to 106). Postoperative outcome and complications have been outlined in Table 2. Five patients had low cardiac output syndrome; in all 5, a residual surgical problem was identified. There were 4 hospital deaths. The first was an 11-year-old girl with a borderline left ventricular dimensions. Although surgery was uneventful, features of low output and congestive heart failure developed 6 hours after surgery. Echocardiography revealed a small additional muscular VSD, which was repaired on cardiopulmonary bypass. However, the patient did not improve thereafter. Two patients died of ventricular arrhythmias on the third and fourth postoperative days. In 1 of the patients, an aggressive attempt at resection (in the early part of the series) resulted in injury to the left anterior descending coronary artery. In this patient, a homograft saphenous vein was used for the bypass grafting to the left anterior descending coronary artery, but she died of intractable ventricular arrhythmias. In the other patient, the cause of the ventricular arrhythmia was not identifiable. One 6-month-old female patient developed cardiac arrest after aspiration on the fourth postoperative day; she was resuscitated but died of aspiration pneumonitis on the 16th postoperative day. The mean hospital stay was 5.7 ± 0.8 days (range, 5 to 26).


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  		Table 2. Postoperative Complications
 
Postoperative Echocardiographic Results
Early postoperative echocardiography revealed mean peak gradients of 20 ± 5.2 mm Hg (range, 0 to 46 mm Hg). Of these, 16 patients had peak RVOT gradient of more than 30 mm Hg (Table 3). Residual insignificant VSD flow was present in 6 patients. Mild aortic regurgitation was present in 4 patients, pulmonary regurgitation was present in 44 (25 mild, 13 moderate, and 6 severe), and mild tricuspid regurgitation was present in 10 patients.


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  		Table 3. Profile of 16 Patients With Right Ventricular Outflow Tract (RVOT) Gradient Greater Than 30 mm Hg
 
Follow-Up
During the closing interval, all 296 survivors were seen in the out patient clinic; however, echocardiographic data were available for 290 patients. The follow-up ranged from 1 to 52 months (mean, 26.8 ± 4.2) and totaled 710.4 patient-years. There was no late death or reintervention. The mean interval of echocardiographic follow-up was 18.8 ± 4.2 months. Follow-up echocardiograms showed mean peak RVOT gradients of 13 ± 4.2 mm Hg (0 to 40 mm Hg), with 6 patients having peak gradients more than 30 mm Hg. Thirty-eight patients had pulmonary regurgitation (32 mild, 6 severe). Ten patients had mild tricuspid regurgitation and 1 had moderate aortic regurgitation. On follow-up, the residual VSD closed spontaneously in 4 patients; 2 patients continue to have insignificant residual VSD.

At last follow-up, New York Heart Association (NYHA) class was class I in 240 cases (81.1%), class II in 49 (16.5%), and class III in 7 (2.4%). All patients in NYHA class I are without any medication, those in class II are well controlled on medication, and those in class III have significant pulmonary regurgitation (n = 6) and moderate aortic regurgitation (n = 1) and are being closely followed up.


   Comment
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
Discussion
 References
 
Over the years, with better understanding of the pathophysiology of TOF, modern surgical techniques, and improved myocardial preservation and postoperative care, mortality for total correction has steadily decreased, and most of the centers report mortality rates of 1% to 5% in all age groups [2, 5]. With the decline in early mortality, the focus has now shifted to the late outcome.

The concerns of right ventriculotomy (classical right ventricular approach) are low cardiac output in the early postoperative period, a higher incidence of arrhythmias, and a risk of late sudden death [3, 6, 7]. The transannular patch results in pulmonary regurgitation, further depressing the right ventricular performance [3, 6, 8].

The efficacy of the right atrial approach has been demonstrated by several investigators [2, 9, 10]. In addition to preserving the right ventricular function, the resultant pulmonary regurgitation after limited transannular patching is less severe than that which occurs after transventricular repair [3]. The incidence of ventricular arrhythmias is less in all series, and the risk of atrial arrhythmias is not much, either [3, 11].

An important advantage of the transatrial approach is in the cases of an anomalous coronary artery crossing the RVOT, which renders the transventricular approach impossible. The use of transatrial approach in the setting of an anomalous coronary pattern allows adequate correction without the use of a conduit for reconstruction of the RVOT. This was feasible in 20 patients (6.7%) in our study.

Only 5 of our patients had low output syndrome. In all 5, a residual surgical problem could be identified, and there was no evidence of primary right ventricular dysfunction. The majority of the patients could be weaned off ionotropes within 48 hours. We had an overall mortality rate of 1.3%, which is comparable to most other series [2, 5].

Residual RVOT obstruction is an important factor that affects the outcome of corrective surgery for TOF. To avoid this, we have ensured that, in the flaccid arrested heart, a Hegar dilator at least two sizes bigger than that predicted by the Rowlatt chart for that particular patient could be passed easily [4]. We had a mean residual gradient of 20 ± 5.2 mm Hg on early postoperative echocardiography (mean interval of 5.7 days) that dropped to 13 ± 4.2 mm Hg at the follow-up echocardiography (mean interval of 18.8 ± 5.2 months). It has been shown that the RVOT gradient usually decreases progressively after surgery [12].

Pulmonary regurgitation caused by the use of transannular right ventricular outflow patch is associated with increased hospital mortality, although its effect is not as obvious as that which occurs with residual pulmonary stenosis [13]. By avoiding the use of an outflow patch, we have had very low incidence of severe pulmonary regurgitation (6 of 296; 2%).

The cause of right bundle branch block after correction of TOF is due to both the right ventriculotomy and the closure of the VSD [14]. It is natural that the incidence of right bundle branch block would be less with a transatrial approach. We had an incidence of 35% of right bundle branch block in the present series. There were no clinically significant arrhythmias on late follow-up electrocardiograms, although no patient underwent 24-hour Holter monitoring. However, as the mean follow-up period is only 26 months, it may not be possible to comment on the full implication of transatrial approach in consideration of arrhythmias.

Initial investigators had concern about the use of transatrial approach in younger patients. Edmunds and colleagues [15] used the transatrial approach in children above the age of 2 years only. They found it technically difficult in small infants and thought it desirable to use a large outflow patch in the smaller age group. The feasibility of transatrial repair is best judged preoperatively by calculating Z-scores on angiocardiography. According to traditional criteria, patients with Z-scores less than –2 are likely to require a transannular patch [16]. There has been a recent report of transatrial repair being successfully performed in patients with Z-scores of as low as – 4 [17]. In our own experience, patients with a Z-score of –3 or less can be considered for the transatrial repair. This is a matter of continuing debate, and recommendations will become clearer once more widespread experience is gained and long-term follow-up of this cohort of patients is available.

In our present series, 30 patients (10%) less than 1 year of age and 82 patients (27.7%) weighing less than 10 kg underwent successful correction (Figs 1 and 2). In our surgical practice, we do not perform correction below 3 months of age or in patients weighing less than 5 kg. Ours being a tertiary level center, we get patients from all over the country, many of whom have undergone a palliative systemic to pulmonary artery shunt outside. We believe that, with timely referral, patients aged 3 months or weighing more than 5 kg are likely to be suitable for this approach. We believe that the patients presenting in the neonatal period have severe forms of infundibular and annular hypoplasia and may not be suitable for the transatrial repair.

In our experience, the extent and length of infundibular obstruction, the degree of aortic override, and the size, position, and number of VSDs did not alter the decision to use this approach. This approach was feasible in patients with anomalous coronaries. The approach can be used in patients with a single branch pulmonary artery, avoiding the use of a conduit, as the native pulmonary valve is preserved. The absolute contraindications for this approach in our setup were inadequate pulmonary annulus and cases for which pulmonary arterioplasty was required.

Study Limitations
There was no randomization of the patients, and there is no comparative group; however, it is not unreasonable for us to compare our results with the available literature. We also did not routinely measure the ratio of the right and left ventricular pressures after the correction in the operating room. As demonstrated by others [12], we also believe that if the patient has stable hemodynamics after the termination of the bypass, then the intraoperative pressure studies are not essential. Finally, an angiographic follow-up would have been ideal.

In conclusion, the transatrial approach for total correction for TOF provides adequate relief of right ventricular outflow tract obstruction. This approach is simple and reproducible and can be used for significant subgroup of patients. Preoperatively, in the majority, the feasibility of transatrial total correction could be predicted accurately.


   Discussion
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 Discussion
References
 
DR RICHARD A. JONAS (Washington, DC): Doctor Levitsky, Dr Wood, members and guests. This year, 2006, is the 50th anniversary of the introduction of the transannular patch by Lillehei just 1 year after John Kirklin had pioneered the repair of tetralogy using cardiopulmonary bypass. Congenital cardiac surgeons have been arguing ever since as to how best to manage tetralogy. This anomaly has provided program committees an endless source of topics for debate controversies, including the relative merits of transventricular versus transatrial repair and the risks versus benefits of early primary repair in infancy.

Professor Airan, you and your colleagues are to be congratulated on these excellent results. You have used a trans–right atrial approach in 35% of a total population of 860 patients with tetralogy, with an early mortality of 1%. You have emphasized the importance of relieving outflow tract obstruction by muscular excision rather than a patch plasty enlargement of the hypoplastic infundibulum.

Right ventricular outflow tract obstruction in tetralogy can occur at multiple levels. If the infundibulum itself is underdeveloped, then the surgeon has a choice of either enlarging the lumen by extensive muscle excision or enlarging the circumference of the infundibulum by placing an infundibular patch. A similar choice applies with left ventricular outflow tract obstruction where tunnel-like subaortic stenosis can be managed by extensive resection and septal myectomy or by a modified Konno procedure with a patch placed in the ventricular septum. Some of us believe that the patch plasty approach is less damaging to the ventricle than widespread endocardial resection, which may lead to more extensive endocardial scarring. However, there are critically important technical factors in the patch plasty approach, including the length of the ventriculotomy, the preservation of small coronary artery branches, and limitation of the width of the patch.

The success of the patch plasty approach has been well supported by an analysis that we undertook of 57 of Aldo Castaneda's early primary repair patients in whom reparative surgery was undertaken in infancy between 1972 and 1977. Median follow-up was more than 23 years. Actuarial survival was 90% at 20 and 25 years. Interestingly, use of a transannular patch was associated with a lower probability of reintervention relative to those who did not have a transannular patch.

Professor Airan, your series presented this morning had a median age of 2.8 years and fewer than 10% of your patients had a shunt, suggesting that these patients had a mild form of tetralogy. Is it correct for us to infer that you limited the total transatrial approach to patients with a mild form of tetralogy? I am also interested to know what your group's philosophy is regarding the optimal age of repair in the current era for the asymptomatic child with tetralogy, and your approach to the child who presents in the first 6 to 12 months who is symptomatic with cyanosis. Thank you for the opportunity to comment on this outstanding series.

DR AIRAN: Thank you, Dr Jonas. We really congratulate the Boston Children's Hospital for their extensive work on tetralogy of Fallot in infancy, and I am aware of the article with the long-term follow-up with excellent results. But as I said earlier, most of our patients are referred late, and we don't have the choice to operate on them in infancy, but if they do come in infancy, we still consider them for transatrial repair. The only contraindications, as I said, are if the patient needs a pulmonary root enlargement or a pulmonary arterioplasty. If the patient is shunted also, even if they have malformed tetrology of Fallot, we will take these patients for correction of tetralogy of Fallot through the transatrial route, and it has been possible in our patients. Thank you.


   References
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 Discussion
 References
 

  1. Hudspeth AS, Cordell AR, Johnston FR. Transatrial approach to total correction of Tetralogy of Fallot Circulation 1963;27:796-800.[Abstract/Free Full Text]
  2. Karl TR, Sano S, Pornviliwan S, Mee RBB. Tetralogy of Fallot: favorable outcome of nonneonatal transatrial transpulmonary repair Ann Thorac Surg 1992;54:903-907.[Abstract]
  3. Dietl CA, Cazzaniga ME, Dubner JS, et al. Life threatening arrhythmias and RV dysfunction after surgical repair of tetrology of Fallot Circulation 1994;5:7-11.
  4. Rowlatt UF, Rimoldi HJA, Lev M. The quantitative anatomy of the normal child's heart Pediatr Clin North Am 1963;10:499-508.
  5. Van Arsdell GS, Maharaj GS, Tom J, et al. What is the optimal age for repair of tetrology of Fallot? Circulation 2000;102(19 Suppl 3):III123-III129.
  6. Nollert G, Teddy F, Bouterwek S, et al. Long term survival in patients with repair of tetrology of Fallot: 36-year follow up of 490 survivors of the first year after surgical repair Am Coll Cardiol 1997;30:1374-1383.[Abstract]
  7. Stirling GR, Stanley PH, Lillehei CW. The effects of cardiac bypass and ventriculomy upon right ventricular function Surg Forum 1957;8:433-438.[Medline]
  8. d'Udekem Y, Ovaert C, Grand JF, et al. Tetralogy of Fallot: transannular and right ventricular patching equally affect late functional status Circulation 2000;102(Suppl 3):116-122.
  9. Dietl CA, Torres AR, Cazzaniga ME, et al. Right atrial approach for surgical correction of tetrology of Fallot Ann Thorac Surg 1989;47:545-552.
  10. Pacifico AD, Sand ME, Bargeron LM, et al. Transatrial-transpulmonary repair of tetrology of Fallot Thorac Cardiovasc Surg 1987;93:919-924.
  11. Kobabayashi J, Hirose H, Susumu N, et al. Ambulatory electrocardiographic study of the frequency and cause of ventricular arrhythmia after total correction of tetrology of Fallot Am J Cardiol 1984;54:1310-1313.[Medline]
  12. Kaushal SK, Radhakrishanan S, Dagar KS, et al. Significant intraoperative right ventricular outflow gradients after repair for tetralogy of Fallot: to revise or not to revise? Ann Thorac Surg 1999;68:1705-1712.[Abstract/Free Full Text]
  13. Kirklin JK, Kirklin JW, Blackstone EH, et al. Effect of transannular patching on outcome after repair of tetralogy of Fallot Ann Thorac Surg 1989;48:783-791.[Abstract]
  14. Kawashima Y, Kitamura S, Nakano S, Yagihara T. Corrective surgery for tetralogy of Fallot without or with minimal right ventriculotomy and with repair of the pulmonary valve Circulation 1981;64(Suppl 2):147-153.
  15. Edmunds Jr LH, Saxena NG, Friedman S, Rashkind WJ, Dodd PF. Transatrial repair of tetralogy of Fallot Surgery 1976;80:681-688.[Medline]
  16. Kirklin JK, Kirklin JW, Pacifico AD. Transannular outflow tract patching for tetralogy: indications and results Semin Thorac Cardiovasc Surg 1990;2:61-69.[Medline]
  17. Stewart RD, Backer CL, Luciana Y, et al. Tetrology of Fallot: results of a pulmonary valve sparing surgery Ann Thorac Surg 2005;80:1431-1439.[Abstract/Free Full Text]



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