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Ann Thorac Surg 2004;77:1691-1696
© 2004 The Society of Thoracic Surgeons

Original articles: cardiovascular

Selective timing for the arterial switch operation

Brian W. Duncan, MDa*, Nancy C. Poirier, MDa, Roger B. B. Mee, CHB, FRACSa, Jonathan J. Drummond-Webb, MDa, Athar Qureshi, MDb, C. Igor Mesia, MDb, June A. Graney, RN, BSNa, Cheryl L. Malek, RN, CPNPa, Larry A. Latson, MDb

a Department of Pediatric and Congenital Heart Surgery, The Children's Hospital at The Cleveland Clinic, Cleveland, Ohio, USA
b Department of Pediatric Cardiology, The Children's Hospital at The Cleveland Clinic, Cleveland, Ohio, USA

Accepted for publication October 16, 2003.

* Address reprint requests to Dr Duncan, The Cleveland Clinic Foundation, Pediatric and Congenital Heart Surgery/M41, 9500 Euclid Ave, Cleveland, OH 44195, USA
e-mail: duncanb{at}ccf.org


   Abstract
Top
 Abstract
Introduction
 Material and methods
 Results
 Comment
 References
 
BACKGROUND: To determine outcomes for the arterial switch operation individualized according to the underlying anatomy and clinical status.

METHODS: A retrospective review of a consecutive series of infants less than 90 days of age who underwent the arterial switch operation at a single institution.

RESULTS: From July 1993–April 2001, 117 infants underwent an arterial switch operation before 90 days of age. Seventy-five patients (64%) had transposition of the great arteries with intact ventricular septum with the aim of operation before 14 days of age; however, 8 of these patients had delayed presentation (range 15–46 days). Thirty-five patients (30%) had transposition with a ventricular septal defect (30 patients) or double outlet right ventricle (5 patients) and normal arch anatomy and were repaired within the first 90 days of life depending on the severity of heart failure at a median of 12 days of age (range 3–83 days). Seven patients (6%) had associated aortic coarctation (5 patients) or interrupted aortic arch (2 patients). One patient died during hospitalization (0.85% hospital mortality) and one patient died from noncardiac causes during a median follow-up of 35 months (1.7% total mortality). Four patients required intervention during follow-up (3.4%) for new aortic coarctation (2 patients), supravalvar pulmonic stenosis (1 patient), or right hemi-diaphragm paralysis (1 patient).

CONCLUSIONS: Individualized timing for the arterial switch operation within the first ninety days of life produces excellent survival rates for all types of transposition physiology with the expectation of a satisfactory course during follow-up.


   Introduction
Top
 Abstract
 Introduction
Material and methods
 Results
 Comment
 References
 
The arterial switch operation is the treatment of choice for transposition of the great arteries (TGA) and other physiologically similar entities such as double outlet right ventricle (DORV) with a subpulmonic ventricular septal defect (VSD). The hospital mortality for transposition of the great arteries with an intact ventricular septum (TGA-IVS) is now routinely reported to be less than 2% at many centers; however, anatomic features that add greater complexity to this condition, such as abnormal coronary artery patterns, the presence of a VSD (TGA-VSD), or DORV and associated aortic arch abnormalities, may increase this mortality [1]. We have developed a treatment protocol that matches the timing of the arterial switch operation with each child's underlying anatomy and physiology. According to this approach all infants undergo corrective surgery within the first 90 days of life. The utility of this treatment protocol is especially important for cases of "complex" TGA with unusual coronary artery patterns, VSD, or arch abnormalities. Using the treatment protocol presented below, we have used the arterial switch operation to successfully treat all forms of transposition of the great arteries regardless of anatomy or associated conditions.


   Material and methods
Top
 Abstract
 Introduction
 Material and methods
Results
 Comment
 References
 
Patients
A retrospective chart review was performed for all children who underwent an arterial switch operation from July 1993–April 2001. The hospital record was utilized to determine the in-hospital course for these children and office records of correspondence with referring physicians was used to establish follow-up data. This study was approved by the Institutional Review Board of the Cleveland Clinic Foundation.

Timing of operation
Children with TGA-IVS undergo the arterial switch operation within the first 14 days of life. In cases where diagnosis is delayed or concomitant conditions preclude cardiopulmonary bypass (necrotizing enterocolitis, intracranial hemorrhage) we perform the arterial switch operation in children with TGA-IVS or a restrictive VSD up to 60 days of age without obtaining preoperative cardiac catheterization to measure the left ventricular pressure. Children with TGA-VSD or DORV and normal arch anatomy undergo an arterial switch operation within the first 90 days of life depending on the severity of congestive heart failure. Patients who have congestive heart failure that is easily controlled medically may have the arterial switch operation at any point up to 90 days of age which we believe improves results by deferring a procedure of this magnitude beyond the neonatal period. Patients with systemic or near systemic left ventricular pressure levels from left ventricular outflow tract obstruction or a large patent ductus arteriosus may also have repair deferred beyond the newborn period. Patients with TGA-VSD or DORV and associated aortic coarctation or interrupted aortic arch undergo repair within the first 1–2 weeks of life.

Technique of operative repair
The pump is primed with fresh (less than 24-hour-old) heparinized blood. Repair of TGA-IVS is performed at a nasopharyngeal temperature of 22°C whereas forms of TGA that require arch reconstruction are repaired at 18°C. Phenoxybenzamine (1 mg/kg) is administered as cardiopulmonary bypass is instituted. Pump flows are maintained at 150–220 ml · kg–1 · min–1 to attain a mean perfusion pressure of approximately 35–40 mm Hg.

The operative approach is routine and has been previously described [2]. Briefly, after cannulation and aortic transection coronary buttons are fashioned by excising a D-shaped cuff of coronary sinus wall leaving only a 0.5–1 mm rim above the line of attachment of the valve leaflet. The pulmonary artery is transected and medially hinged trapdoor flaps are created in the main pulmonary artery at or above the sino-tubular junction. The coronary artery cuffs are then sewn into the defects using 7-0 polypropylene sutures taking great care to avoid axial rotation of the buttons. At the completion of the suture line, the medially based flaps create a conical extension of the proximal neo-aorta which reduces the angle through which the transposed coronary artery must rotate after implantation [2].

The coronary sinus defects in the neo-pulmonary artery are filled with individual patches of fresh autologous pericardium that are sewn together using 7-0 polypropylene sutures over their posterior extent [2]. The neo-pulmonary artery is then sewn to the distal pulmonary artery with a running suture of 7-0 polypropylene.

After completion of the repair, monitoring lines are placed in the left atrium and pulmonary artery. We routinely place peritoneal catheters after infant cardiac surgery to serve as a continuous drain of the abdominal cavity which may also be used for mini-dialysis for persistent low urinary output, hyperkalemia, or to remove fluid. In the immediate postoperative period systemic hypertension is avoided and left atrial pressure is ideally maintained at 10 mm Hg or less. Phenoxybenzamine is continued during the first several days after surgery.

Ventricular septal defect
VSD closure is performed through a right atriotomy without interruption of cardiopulmonary bypass. VSD closure is usually performed before the arterial switch with an additional dose of cardioplegia administered before transection of the aorta.

Unusual coronary artery patterns
The circumflex coronary artery travels behind the pulmonary artery if the circumflex arises from the right coronary artery in sinus 2. Placing the right coronary implantation 2–3 mm higher on the neo-aorta than is usually done allows the circumflex to assume a less angulated course [2]. Intramural coronary arteries often pass behind a commissure of the aortic valve with the sinus opening very close to the other coronary artery. In this situation, the commissure is taken down, the intramural coronary artery is unroofed on the aortic luminal side, the ostia are separated, and buttons are fashioned in the usual way [3]. The commissure is then resuspended onto the pericardial patch used to repair the aortic sinus defects.

Aortic arch abnormalities
If the arch is interrupted or very small the ductus arteriosus and ascending aorta are cannulated. At a nasopharyngeal temperature of 18°C isolated coronary perfusion may be instituted by first reducing cardiopulmonary bypass flow to approximately 10% of a predicted normal cardiac output. Next, the arch vessel snares are tightened, the ductal cannula is removed, a cross-clamp is placed obliquely just distal to the aortic cannula, and pump flows are adjusted to achieve an arterial bypass line pressure of 30 mm Hg. All ductal tissue is then excised and the descending aorta is anastomosed to the underside of the proximal arch with a running 7-0 polypropylene suture. When aortic reconstruction is complete the clamp is shifted proximal to the aortic cannula, cardioplegia is administered, full bypass is resumed, and the remainder of the operation is performed in a standard fashion.

Statistical analysis
The Fischer exact test was used to evaluate the statistical association with mortality and the following clinical parameters: operation beyond 14 days of age, the presence of unusual coronary artery patterns, the presence of DORV, and abnormal arch anatomy. Survival curves were constructed by the Kaplan–Meier method. Statistical analysis was performed using JMP Statistical Discovery Software (SAS Institute, Cary, NC); 95% confidence intervals (95% CI) were determined where appropriate.


   Results
Top
 Abstract
 Introduction
 Material and methods
 Results
Comment
 References
 
From July 1993–April 2001, a consecutive series of 117 children underwent an arterial switch operation before 90 days of age. The median age at the time of repair for the entire group was 7 days (range 1–83 days) and the median weight was 3.4 kg (range 1.5–6.1 kg). Eighty-four of these patients (72%) had a balloon atrial septostomy performed and prostaglandin was safely discontinued in 80 (68%) of all patients. The coronary artery pattern and the relationship of the great arteries found at the time of surgery are listed in Tables 1 and 2 respectively. Sixty-nine percent of patients with TGA-IVS had usual coronary artery patterns and 82% of patients with TGA-VSD demonstrated usual coronary artery patterns whereas only 56% of patients with DORV had usual coronary artery patterns.


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  		Table 1. Coronary Patterns

 

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  		Table 2. Great Vessel Relationship

 
Seventy-five patients (64%) had TGA-IVS: 65 of these patients had no associated lesions and underwent arterial switch operation at a median age of 5 days (range 1–14 days). Two patients with TGA-IVS had significant associated lesions that maintained systemic left ventricular pressures and were treated beyond 14 days of age: one premature infant had a large patent ductus arteriosus and was managed medically until the operation was performed at 30 days of age at which time the patient weighed 1.7 kg; one patient with TGA-IVS, left ventricular outflow tract obstruction, and significant cyanosis underwent a modified Blalock–Taussig shunt through a right thoracotomy and was repaired at 48 days of age. Eight additional patients with TGA-IVS and no additional cardiac lesions had the operation beyond 14 days of age (median 23 days; range 15–46 days) due to significant comorbid conditions in other organ systems (one patient with necrotizing enterocolitis, one patient with necrotizing enterocolitis and a grade 2 intraventricular hemorrhage) or delay in referral from other institutions (6 patients).

Forty-two patients (36%) had a hemodynamically significant VSD (TGA-VSD 33 patients, DORV 9 patients). Seven patients, with TGA-VSD (1 patient) or DORV (6 patients), had associated arch abnormalities with aortic coarctation (5 patients) or interrupted aortic arch (2 patients). One of these patients had DORV with multiple VSDs and a type B interruption whereas one patient with DORV had a type A interruption. One patient with DORV and aortic coarctation had significant obstruction of the systemic ventricular outflow tract as well. Of the 35 patients with TGA-VSD or DORV and no arch abnormality, the median age of repair was 12 days (range 3–83 days). Six of the 7 patients with arch abnormalities had repair within the first 14 days of life (median 7 days [range 2–12 days]). One patient with DORV and mild coarctation was referred late and underwent combined repair at 53 days of age. A total of 6 patients had significant obstruction of the systemic ventricular outflow tract (1 patient with TGA-IVS, 1 patient with DORV, 1 patient with DORV-coarctation, and 3 patients with TGA-VSD) necessitating transpulmonic resection.

Two patients had undergone previous operations: the patient with TGA-IVS and left ventricular outflow tract obstruction had undergone a modified Blalock–Taussig shunt and another patient with DORV and obstruction of the systemic ventricular outflow tract had undergone pulmonary artery banding. These patients were repaired at 48 and 83 days of age respectively.

Table 3 gives the cardiopulmonary bypass times, aortic cross-clamp times, and the duration of circulatory arrest for all patients. Postoperatively, patients required a median of 3 days of ventilatory support (range 1–46 days), 4 days in the intensive care unit (range 1–46 days), and 8 days of hospitalization before discharge (range 4–46 days). The median duration of ventilatory support [4.5 days (range 1–32 days)], intensive care unit stay [6 days (range 1–32 days)], and hospital stay [11 days (range 6–32 days)] was slightly longer for the 8 patients with TGA-IVS who underwent repair beyond 14 days of age. One premature infant who underwent delayed operation at 16 days of age due to multiple abdominal operations for necrotizing enterocolitis and who was also the smallest patient in the series (1.5 kg at the time of operation), was transferred intubated to the referring institution 32 days after surgery.


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  		Table 3. Cardiopulmonary Bypass Times

 
No patient in the series required delayed sternal closure or mechanical circulatory support. As discussed in the Methods section, we routinely place a peritoneal dialysis catheter at the time of operation; 12 patients (10%) were treated with peritoneal dialysis for low urine output or hyperkalemia in the immediate postoperative period.

Eleven patients (9%) suffered significant complications during hospitalization which is demonstrated in Table 4. Three patients had postoperative seizures including 1 patient who suffered cardiac arrest preoperatively during balloon atrial septostomy and who subsequently demonstrated a cortical infarction and seizures postoperatively. A second patient with a family history of antithrombin III deficiency developed a sagittal sinus thrombosis and seizures postoperatively. Finally, a third patient experienced postoperative seizures with normal cerebral anatomy. Septic complications occurred in 3 patients, urinary tract infection occurred in 2 patients, and gram-positive sepsis occurred in 1 patient.


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  		Table 4. In-Hospital Complications

 
There was one in-hospital death (0.85%, 95% confidence interval [CI] 0.2–4.7%) in this group of patients. This child had DORV with significant obstruction of the systemic ventricular outflow tract, a small right ventricle, multiple VSDs, hypoplasia of the aortic arch, and aortic coarctation. The patient died ventilated in the intensive care unit 46 days after surgery. An autopsy was performed which demonstrated thrombotic occlusion of the right coronary artery with extensive infarction of both the right and left ventricles. No patient died before surgery. There were no deaths in patients who underwent operation beyond 14 days of age or patients with unusual coronary artery patterns. The single in-hospital death in a patient with DORV and abnormal aortic arch anatomy resulted in p values of 0.06 and 0.07 respectively for the possible association of these two anatomic features with hospital mortality.

Follow-up is available for 105 of the surviving 116 patients (91%) at a median of 35 months (range 6–109 months) postoperatively. There was one noncardiac death 33 months after surgery due to a pedestrian motor vehicle accident resulting in a total mortality for these patients of 1.7% (95% CI 0.5%–6.0%). Four patients required intervention during follow-up (3.4%; 95% CI 1.3%–8.5%) for new aortic coarctation [2], supravalvar pulmonic stenosis [1], or right hemi-diaphragm paralysis [1] (median of 4.4 months after surgery until intervention; range 1.2–16.3 months). The actuarial survival and intervention free survival are demonstrated in Figure 1. Echocardiograms were obtained in the 104 long-term survivors with available follow-up a median of 26 months after repair (range 3 days–109 months); results are demonstrated in Figure 2 for postoperative aortic insufficiency, pulmonic stenosis, and left ventricular dysfunction.



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  		Fig 1. Kaplan–Meier curves for survival (open circles) and intervention-free survival (solid diamonds) after arterial switch operation. Note survival probability (y-axis) scale begins at 0.6.

 


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  		Fig 2. Follow-up echocardiography for 104 patients after arterial switch operation: (A) aortic insufficiency, (B) pulmonic stenosis, (C) left ventricular dysfunction.

 

   Comment
Top
 Abstract
 Introduction
 Material and methods
 Results
 Comment
References
 
Results for the arterial switch operation continue to improve; several recent reports have emphasized that refinements in operative and perioperative management have mitigated the effects of many features previously thought to increase operative risk [4–8]. Most notably, unusual coronary artery patterns confer no increased risk of death after the arterial switch operation in multiple studies [1, 4, 8–10]. Good outcomes may also be achievable in other high risk groups such as infants with TGA-IVS and late presentation, premature or low birth weight patients, patients with DORV and subpulmonary VSD, and patients with associated arch abnormalities [11–16]. The results in the present series also support the expectation of successful treatment for these groups; however, many of these complex forms of TGA undoubtedly confer higher absolute risks for perioperative morbidity and mortality. The absence of statistically significant differences in outcome between "simple" and "complex" TGA arise in part from small numbers of the more complex forms. A successful program for the treatment of all forms of TGA requires an individualized approach to each case based on the existing anatomy and physiology. The following summarizes the importance of individualized timing of repair and our approach to difficult anatomic issues such as unusual coronary artery patterns, DORV, and associated aortic arch abnormalities.

Timing of operative repair
The timing of operative repair for TGA should be individualized; patients with TGA-IVS should undergo an arterial switch operation within the first 2 weeks of life. Concomitant pathology such as necrotizing enterocolitis or intracranial hemorrhage may delay the arterial switch operation or, rarely, the diagnosis itself may be delayed. In these cases we perform the arterial switch operation in children with TGA-IVS up to 60 days of age without obtaining preoperative cardiac catheterization to measure the left ventricular pressure [17]. Similarly we do not perform preoperative conditioning of the left ventricle with pulmonary artery banding in these children but would perform an arterial switch operation and provide postoperative support with a left ventricular assist device if necessary [18]. We perform cardiac catheterization in infants presenting beyond 60 days of age to assess left ventricular pressure. Patients who demonstrate a left ventricular to right ventricular pressure ratio of less than 0.65 will likely require left ventricular conditioning by placement of a pulmonary artery band before the arterial switch operation or by postoperative support with a left ventricular assist device. Infants with TGA and a restrictive VSD may be expected to follow a similar clinical course as patients with TGA-IVS and should undergo early repair.

Infants with TGA and prostaglandin dependent circulations due to associated aortic arch abnormalities such as interrupted aortic arch or aortic coarctation should undergo early arterial switch operation and concomitant arch reconstruction. Children with TGA and an unrestrictive VSD should undergo individualized management based on the severity of congestive heart failure. Children with TGA-VSD who are ventilator bound due to severe congestive heart failure should undergo an early arterial switch operation. Children with TGA-VSD in whom symptoms of congestive heart failure are easily managed with oral medications may have repair deferred beyond the neonatal period. This approach to patients with TGA-VSD or DORV with a subpulmonic VSD is derived from more than 20 years of experience at the Royal Children's Hospital (Melbourne, Australia) and now here at the Cleveland Clinic (Cleveland, OH) and is based on the observation that children with these complex anatomic subtypes fare better if they can be stably managed to allow surgical correction beyond the newborn period [2, 19, 20]. The surgical approach is technically easier in a slightly larger heart and it is our belief that greater maturity of the myocardium and overall physiology beyond the newborn period results in a smoother postoperative course. However all of these children should undergo repair within the first 90 days of life as the development of pulmonary vascular obstructive disease becomes an increasing risk beyond this period.

Associated anatomic conditions—VSD or DORV
Aside from its impact on the timing of the arterial switch operation, there is little difference in the management of patients with TGA-VSD from that of TGA-IVS. After bi-caval cannulation, VSD closure is performed through a right atriotomy without interruption of cardiopulmonary bypass. VSD closure was performed via the right atrium in all cases in this experience which is technically easier than approaches that attempt to access the VSD through a small aortic root. VSD closure can be performed before or after the arterial switch operation with additional doses of cardioplegia.

Associated anatomic conditions—unusual coronary artery patterns
In this series more than 85% of cases had either a usual coronary artery pattern or one in which the left anterior descending came from sinus 1 whereas the right and circumflex arose from sinus 2. Of the remainder, the majority (6% of total) had the right coronary artery and left anterior descending coronary artery arising from sinus 1 and the circumflex coronary artery arising from sinus 2 or demonstrated single right or left coronary artery patterns (5% of total).

One rare pattern encountered in the present series was a common orifice of the left and right coronary arteries that arose high from sinus 2 with the aorta anterior and to the right of the pulmonary artery. The left anterior descending and circumflex coronary arteries coursed anteriorly between the great vessels whereas the right coronary artery followed a usual posterior path. It was suspected that the left coronary system was intramural and that it would be possible to incise the aortic wall over its proximal course effectively moving the orifice of the left system anteriorly to allow separation of the right and left systems into two coronary buttons. However, at operation, it was found that there was no intramural portion of the left coronary artery and that a common orifice of both the left and right systems was present. The common orifice of the coronaries was retained, the superior margin of the button was "leaned back" and sewn to a cutback down from the proximal margin of the transected pulmonary artery, and the remainder of the coronary button was sewn to a pericardial patch which was sewn to the remainder of the opening in the neo-aorta. An extra long patch of pericardium was used to reconstruct the neo-pulmonary artery to raise the pulmonary artery bifurcation off of the coronary artery reconstruction.

Associated anatomic conditions—aortic coarctation or interrupted aortic arch
Significant abnormalities of the aortic arch should be dealt with at the same time the arterial switch is performed [14, 16, 21–23]. We encountered 7 such patients in this series; 5 of the 7 had aortic coarctation and 2 of the 7 had interruption of the aortic arch (one type A, one type B interruption). All 7 patients had an associated large VSD; 6 of the 7 patients had DORV. Although often associated with obstruction of the right ventricular outflow tract only 1 of the 6 patients with right ventricular outflow tract obstruction severe enough to require resection had an arch abnormality (coarctation). Bicaval cannulation with cannulation of both the aorta and ductus and isolated coronary artery perfusion allow VSD closure, the arterial switch, and arch reconstruction to be done with a minimum of circulatory arrest time and myocardial ischemia.


   References
Top
 Abstract
 Introduction
 Material and methods
 Results
 Comment
 References
 

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