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Eur J Cardiothorac Surg 2000;17:538-542
© 2000 Elsevier Science NL

Single-stage repair of aortic coarctation with ventricular septal defect using isolated cerebral and myocardial perfusion

Department of Cardiovascular Surgery, Okayama University Medical School, 2-5-1 Shikata-cho, Okayama 700-8558, Japan

Corresponding author. Tel.: +81-86-235-7359; fax: +81-86-235-7431
e-mail: ishino{at}tb3.so-net.ne.jp

	Abstract

Top Abstract 1. Introduction 2. Materials and methods 3. Results 4. Discussion Appendix A Conference discussion References

 
Objective: To avoid hypothermic circulatory arrest, we have repaired aortic coarctation with ventricular septal defect (VSD) in a one-stage procedure using an isolated cerebral and myocardial perfusion technique, and retrospectively compared this novel approach to the conventional two-stage approach. Methods: Between October 1991 and February 1999, 24 infants, aged 4–137 days (median, 27 days) and weighing 1.7–4.3 kg (median, 3.0 kg), underwent the repair of aortic coarctation with VSD either in one (group I, n=11) or two stages (group II, n=13). In Group I, an arterial cannula for cardiopulmonary bypass was inserted into the ascending aorta in six patients with coarctation only, or into a polytetrafluoroethylene (PTFE) graft which was anastomosed to the innominate artery in the remaining five who had hypoplastic arches. A cross-clamp was placed between the innominate and left carotid arteries. The bypass flow was reduced to 30–50% of full flow at 28°C, thereby maintaining a radial artery pressure of 30–45 mmHg. At this point, the aortic coarctation was repaired by an end-to-end arch anastomosis, while maintaining brain perfusion and with the heart still beating. In five patients with hypoplastic aortic arches, the innominate artery proximal to the graft was then secured down and the arch anastomosis was extended to the distal ascending aorta, while providing isolated cerebral perfusion and cardioplegic arrest. After arch reconstruction was performed, the clamp was moved onto the ascending aorta, and the VSD was closed with systemic perfusion. In contrast, for group II patients, coarctation repairs were performed through a posterolateral approach, and existing VSDs were closed as secondary procedures. Results: The mean isolated cerebral and myocardial perfusion time for group I was 13 min (range, 7–20 min). The myocardial ischemic time did not differ between groups I and II (43±4 vs. 42±5 min, not significant). There were no hospital mortalities or neurological complications in either group, but one late death in each group. Conclusion: Single-stage repair of aortic coarctation with VSD does not increase myocardial ischemic time compared to the traditional two-stage approach. The isolated cerebral and myocardial perfusion technique may offer substantial brain and myocardial protection during aortic arch reconstruction.

Key Words: Coarctation • Ventricular septal defect • Circulatory arrest • Cerebral perfusion

	1. Introduction

Top Abstract 1. Introduction 2. Materials and methods 3. Results 4. Discussion Appendix A Conference discussion References

 
The optimal management of aortic coarctation with ventricular septal defect (VSD) remains controversial [1]. Since the introduction of the preoperative use of prostaglandin E₁ to maintain ductal patency [2], most infants now survive until they are hemodynamically stable enough to undergo surgical repair. The traditional two-stage operation includes the repair of coarctation, with or without pulmonary artery banding, through a left thoracotomy, and closure of the concomitant VSD as a secondary procedure. Recently, encouraging results have been reported for a primary total correction procedure through a midline sternotomy [3,4]. With two different procedures now available, a debate exists concerning the relative benefit of the single-stage versus the two-stage repair for these lesions.

Deep hypothermia and circulatory arrest have been used by many surgeons to support vital organs during aortic arch reconstruction. However, circulatory arrest prolongs myocardial ischemic time and, regardless of its duration, may be associated with transient cerebral dysfunction [5,6] and delayed psychomotor development [7]. In order to avoid circulatory arrest, we have employed an isolated cerebral and myocardial perfusion technique for the single-stage repair of aortic coarctation with VSD, and retrospectively compared this technique with the conventional two-stage approach.

	2. Materials and methods

Top Abstract 1. Introduction 2. Materials and methods 3. Results 4. Discussion Appendix A Conference discussion References

 
2.1. Patient population
Between October 1991 and February 1999, 24 infants (13 boys, 11 girls), aged 4–137 days (median, 27 days) and weighing 1.7–4.3 kg (median, 3.0 kg), underwent the repair of aortic coarctation with VSD at Okayama University Hospital. The indication for operation in all cases was congestive heart failure. Eighteen patients (75%) received an infusion of prostaglandin E₁ to maintain ductal patency prior to surgery. Six patients (25%) necessitated inotropic support, and three (13%) required mechanical ventilation. Additionally, balloon aortoplasty was attempted in two patients before the definitive operation.

The 24 patients were divided into two groups, based upon the surgical approach employed. Eleven infants (group I) underwent a complete single-stage repair after 1996. Five of them had coarctation along with a hypoplastic arch [3]. The location of the VSD was perimembranous in seven patients, subpulmonary in three, and muscular in one. Associated anomalies included a critical aortic stenosis in one infant, Ebstein's anomaly in another, and atrial septal defects (ASD) in four patients. Thirteen infants (group II) underwent a two-stage repair before 1996. Hypoplastic arches were present in four patients. The VSD was perimembranous in seven patients, subpulmonary in four, and muscular in two. In addition, two patients had a subaortic stenosis, three had ASDs, and one had Ebstein's anomaly.

2.2. Operative technique
2.2.1. Single-stage repair
Arterial blood pressure monitoring lines were placed in the right radial artery and femoral artery in each patient preoperatively. Through a midline sternotomy, the thymus gland was excised. The aortic arch, its branches and the duct were dissected out. The left carotid and left subclavian arteries were looped with tourniquets, to allow blood flow control. An arterial cannula was inserted into the ascending aorta in patients with aortic coarctation only, or into a 3.5-mm polytetrafluoroethylene (PTFE) tube (Gore-Tex, WL Gore & Associates, Inc, Flagstaff, Ariz.) which was anastomosed to the innominate artery in patients with coarctation plus a hypoplastic arch (Fig. 1B). Cardiopulmonary bypass was commenced by bicaval cannulation with a flow rate of 150 ml/min per kg. During the cooling phase, the descending thoracic aorta was extensively mobilized by blunt dissection as far distally as possible without division of the intercostal arteries.

View larger version (19K): [in this window] [in a new window]   Fig. 1. An arterial cannula is inserted: (A), into the ascending aorta for repair of aortic coarctation; or (B), into a 3.5-mm PTFE tube which is anastomosed to the innominate artery for repair of coarctation plus a hypoplastic arch.

View larger version (19K): [in this window] [in a new window]   Fig. 2. Isolated cerebral and myocardial perfusion are established by clamping the aortic arch between the innominate artery and left carotid artery. (A) Coarctation repair is carried out with brain perfusion and the heart beating. (B) The clamp is repositioned onto the ascending aorta, and the VSD is closed with cardioplegic arrest.

View larger version (20K): [in this window] [in a new window]   Fig. 3. (A) In a case of coarctation plus hypoplastic arch, about two-thirds of the arch anastomosis is accomplished with isolated cerebral and myocardial perfusion. (B) The innominate artery just proximal to the PTFE tube is snared, the arch is unclamped, and the arch anastomosis is extended with isolated cerebral perfusion.

2.3. Statistical methods
The medical records, operative reports and echocardiographic results were reviewed. Follow-up was completed for all patients. The Mann–Whitney rank sum test was used for the comparison of continuous variables between the two groups. The level of statistical significance was set at P<0.05. The designation P=NS indicates ‘not significant’. The data are presented as means±standard deviations.

	3. Results

Top Abstract 1. Introduction 2. Materials and methods 3. Results 4. Discussion Appendix A Conference discussion References

 
3.1. Operative outcome
The isolated cerebral and myocardial perfusion time in group I was 13±4 min (range, 7–20 min), and the additional isolated cerebral perfusion time required in the five patients with hypoplastic aortic arches was 11±2 min (range, 10–13 min). Thus, the total cross-clamping time of the descending aorta was 17±4 min (range, 10–23 min). The myocardial ischemic time in group I did not differ from the time seen during the second operation in group II (43±4 vs. 42±5 min, P=NS). There were no hospital mortalities or neurological complications in either group.

3.2. Follow-up
The follow-up periods in groups I and II were 20±12 (range, 2–44 months) and 67±25 months (range, 13–97 months), respectively. There were two late deaths; one group I infant died from infective endocarditis 2 months after surgery and one group II patient died from dilated cardiomyopathy 4 months after VSD closure. Recurrent coarctation, defined as a peak gradient exceeding 20 mmHg across the arch, has not developed in any group I patient, but one group II patient required a balloon angioplasty due to an arch gradient of 33 mmHg. Re-operation following repair of coarctation and VSD was performed in two group II patients. Specifically, one patient required repair of the mitral valve, while the other necessitated resection of subaortic stenosis.

	4. Discussion

Top Abstract 1. Introduction 2. Materials and methods 3. Results 4. Discussion Appendix A Conference discussion References

 
Hypothermic circulatory arrest is a commonly used perfusion strategy during aortic arch reconstruction. Although deep hypothermia provides significant protection from brain damage, substantial evidence has accumulated that neurologic injury can occur from circulatory arrest, and can lead to both early and late sequelae [6,7]. Another disadvantage of circulatory arrest is the prolongation of myocardial ischemia. This correlation between circulatory arrest time and myocardial ischemia was borne out by Karl et al. [3], who achieved a better outcome in infants undergoing aortic arch repair with circulatory arrest, in whom myocardial ischemic time was reduced by employing isolated myocardial perfusion [8]. Previous reports [9,10] have recommended one-stage repairs of aortic coarctation and VSD without circulatory arrest, using a left thoracotomy followed by a midline sternotomy performed during the same anesthesia period. By using the isolated cerebral and myocardial perfusion technique, aortic coarctation and VSD could be repaired more easily during a single operation through a single incision, without interruption of the brain perfusion and without increasing the myocardial ischemic time.

The isolated cerebral and myocardial perfusion technique was first reported by Asou et al. in 1996 [11]. Their group perfused the innominate artery either through a PTFE shunt for Norwood operation or by a direct cannulation using a specially designed thin-walled cannula. In the present series, the ascending aorta, when large enough, was cannulated in patients with coarctation only, while a long PTFE tube on the innominate artery was the arterial perfusion route in patients with coarctation plus a hypoplastic arch. Because of the flexibility of the tube, the extended aortic arch anastomosis was carried out without interference from the cannula and without any undue tension around the anastomotic site. This arterial perfusion route is currently our routine technique for complex aortic arch reconstructions, and has been applied in two babies with hypoplastic left heart syndrome who weighed less than 2 kg; both infants survived.

The appropriate perfusion rate for the brain and heart under hypothermic conditions remains unknown. Asou et al. [11] reported their experiences with isolated cerebral perfusion in neonates, in which they selected a flow rate of 50 ml/kg per min at 22°C, with a resultant right radial artery pressure of 20 mmHg. Pigula et al. [12] employed the same perfusion technique except with flow rates of 15–20 ml/kg per min at 18°C, and documented sufficient brain perfusion based on cerebral oxygen saturation and relative cerebral blood volume. In terms of investigating adequate cardiac blood supply, Sano and Mee [8] maintained perfusion pressures at 30–45 mmHg for isolated myocardial perfusion through the ascending aorta. In our study, the innominate artery (at 28°C) supplied both cerebral and myocardial perfusion. Given the uncertainty of flow distribution to these two vital organs, we adjusted the flow rates to keep perfusion pressures at 30–45 mmHg during isolated cerebral and myocardial perfusion.

The management of a hypoplastic aortic arch is a surgical challenge. Such a lesion has been found in 60–70% of infants undergoing coarctation repair [13,14]. Although the extended arch anastomosis may be feasible through the left thoracotomy in the majority of patients [13,14], it is sometimes difficult to place the upper aortic clamp proximal to the hypoplastic segment without seriously compromising flow to the innominate artery, at least with this exposure. However, the midline sternotomy, used for the single-stage repair in this series, obviously provides better exposure of the proximal arch. In the five patients with coarctation plus a hypoplastic arch, a large anastomosis extending to the distal ascending aorta was possible without the proximal aortic clamp, but with brain perfusion through the innominate artery. None of these five patients have developed a recoarctation in the medium-term follow-up. Thus, the repair of a hypoplastic arch by sternotomy may compare favorably in this respect with the same repair performed via thoracotomy.

Isolated cerebral and myocardial perfusion may offer significant brain and myocardial protection during aortic arch reconstruction, although we have not provided any direct evidence of organ perfusion. This perfusion technique has now been standardized and the results are reproducible for a variety of surgical lesions, further verifying the applicability of the procedure. In particular, we have performed many more complex aortic arch repairs than reported here using this perfusion method, mainly for infants with interruption of the aortic arch and VSD, Taussig–Bing type double-outlet right ventricle, or hypoplastic left heart syndrome.

	Footnotes

 
Presented at the 13th Annual Meeting of the European Association for Cardio-thoracic Surgery, Glasgow, Scotland, UK, September 5–8, 1999.

	Appendix A Conference discussion

Top Abstract 1. Introduction 2. Materials and methods 3. Results 4. Discussion Appendix A Conference discussion References

 
Dr A. Corno (Lausanne, Switzerland): Since you have a report from the North American Congenital Heart Surgeons Society showing that there is a significantly better survival at 2 years after a staged approach versus primary repair, based on what kind of hypothesis do you start your study with a primary approach?

Are your series of consecutive patients or, during the same period, did you offer a staged approach to some patients?

Dr Sano: The results of our series were also the same in a single-stage repair group and a two-stage repair group. Therefore, I think it depends on the surgeon's policy. We used a two-stage repair until 1995, and have changed to a single-stage repair because we have developed a cerebral perfusion technique. If you adopt a single-stage repair, then you need to worry about the brain ischemia and brain damage, because of circulatory arrest. Although the arrest time is relatively short at coarctation repair, I think there may have some brain damage even in the subclinical level. As we have developed a cerebral perfusion technique, we don't need to worry about brain ischemia. This is why we have changed to a single-stage repair. There is also no difference in myocardial ischemic time between the two groups.

Dr D. Anderson (London, UK): I noticed that one of your techniques of cerebral perfusion is to apply a tube graft to the innominate artery for perfusion. Did you find that there were any problems related to that, either at the completion of the operation, or in the perfusion through the innominate artery or the right arm as a consequence of that technique?

Dr Sano: We adopted this technique in 1995 and used this technique more than 50 cases, including the Norwood procedure, without any complication so far. We put a shunt before going on bypass and measure a right radial artery pressure and a femoral artery pressure. Unless the patient has very severe hypoplasia of the aortic arch, the pressures in the two sites are almost always equal.

Dr H. Uemura (Osaka, Japan): Recently, I prefer to maintain perfusion, not only to the brain, but also to the organs of the lower body. When establishing cardiopulmonary bypass, two arterial cannulae are inserted, one to the ascending aorta and the other directly to the descending aorta. With this option, the postoperative urine output and the findings in liver enzymes were much better compared with those in the group of patients undergoing ischemia of the lower body. In this respect, I think that maintenance of perfusion to the lower body should be less invasive as well. What do you think about this option?

Dr Sano: I think that is a very good idea. Although, in a coarctation or interruption group, the mean circulatory arrest time to the lower body is only 10–20 min. Therefore, there is not much difference in postoperative urine, liver function and kidney function. Certainly, in the Norwood procedure, the circulatory arrest to a lower body takes more than 30 min, even in a good hand. In these cases, perfusion to a lower body may help more. I agree with you.

	References

Top Abstract 1. Introduction 2. Materials and methods 3. Results 4. Discussion Appendix A Conference discussion References

 

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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Shunji Sano Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Ishino, K. Articles by Sano, S. Search for Related Content PubMed PubMed Citation Articles by Ishino, K. Articles by Sano, S.