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Ann Thorac Surg 1996;62:778-782
© 1996 The Society of Thoracic Surgeons
Division of Pediatric Cardiology, Children's Hospital, Berne, Switzerland, and Division of Thoracic and Cardiovascular Surgery, Hannover Medical School, Hannover, Germany
Accepted for publication May 2, 1996.
| Abstract |
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Methods. Between January 1987 and December 1993, 46 neonates and infants with functionally isolated aortic coarctation underwent operative resection and end-to-end anastomosis. For the patients included in the study, all hospital records, catheterization data, and operative protocols were evaluated for retrospective analysis. To obtain valid follow-up information, all patients were systematically seen on an outpatient basis during 1994.
Results. After a mean follow-up of 49 ± 24 months (range, 13 to 95 months), recoarctation (arm-leg blood pressure gradient >20 mm Hg) occurred in 5 of 26 patients with neonatal operations (19%) and in 1 of 20 patients with operations in infancy (5%, p= not significant). Four of these 6 children with recoarctation needed reintervention. The other 2 patients had only mild recoarctation (gradients of 22 and 30 mm Hg, respectively) and were not treated. In all 6 patients, recoarctation was diagnosed within the first 6 months postoperatively. During the whole follow-up period, right arm systolic blood pressures slightly above the 90th percentile of normal developed in 11 of the patients (24%) (7 in the group with neonatal operation and 4 after operation in infancy; p = not significant).
Conclusions. Resection with end-to-end anastomosis was shown to be an adequate therapeutic strategy for isolated aortic coarctation in neonates and infants. The results indicate that already beyond the neonatal age, there is a relatively low incidence of recoarctation.
| Introduction |
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Despite the advancement of catheter interventions, operation is still the treatment of choice for native aortic coarctation in most institutions [1]. Considerable debate continues to focus on the questions of the ideal timing of operative treatment and the optimal techniques in the different age groups [2]. Operative repair in the first year of life is associated with a considerable risk of recoarctation, with neonates carrying the highest risk independent of the operative technique used [3]. In both the neonatal period and early infancy, good results have been reported for operative repair with the subclavian flap angioplasty, as well as with resection and end-to-end anastomosis [4, 5]. Early repair has been shown to decrease considerably late postoperative cardiovascular morbidity and mortality [6].
The present study was intended to evaluate our institution's results with resection and end-to-end anastomosis in a homogeneous group of patients, operated on for functionally isolated coarctation during the first year of life by one surgeon. Patients with hemodynamically relevant associated cardiovascular anomalies were not included in this study.
| Material and Methods |
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To obtain conclusive follow-up data, all the children were seen in our outpatient clinic during 1994, at which time clinical examination, blood pressure measurements on all limbs, standard electrocardiogram, chest roentgenogram, and echocardiography (including color Doppler) were performed. Patients were excluded from the study if recent follow-up information could not be obtained (n = 4, all with successful repair).
For comparison of the operative results and outcome, the patients were grouped as to whether they were operated on in the neonatal period or during infancy.
Blood Pressure Measurements
Blood pressure was measured with the oscillometric method (Dinamap; Critikon) using an appropriate-sized cuff [7] and with the patient supine. For calculations, the mean of three measurements in the quiet patient was taken. The right arm systolic pressure was used to assess the presence of arterial hypertension. Right arm and right leg pressures were compared to assess the pressure gradient across the site of operation. Normal values for blood pressure were derived from the percentile charts established by the Task Force on Blood Pressure Control in Children [7].
Recoarctation was defined as a systolic blood pressure gradient of greater than 20 mm Hg between the right arm and right leg [8]. Echocardiographic pressure gradients were derived from the velocity in the descending aorta using continuous Doppler with the transducer positioned in the suprasternal notch. Pressure gradient was calculated using the modified Bernoulli formula: 4(V22 - V12), where V1 = flow velocity in the ascending aorta and V2 = velocity in the descending aorta.
The presence of a blood pressure gradient greater than 30 mm Hg with arterial hypertension was considered an indication for reintervention.
Operative Technique
All patients were operated on by the same surgeon. Moderate body surface hypothermia (31° to 33°C) was induced in all patients. Left lateral thoracotomy in the fourth interspace was performed in all patients. The descending aorta as well as the left subclavian and carotid arteries were exposed and mobilized. The ductus arteriosus was transected. Resection of the stenotic segment of the aorta and end-to-end anastomosis were performed in all children. During resection, care was taken to excise all ductal tissue. When ductal tissue was seen to extend into the proximal left subclavian artery, the artery was transected and reimplanted (end-to-side into the aortic anastomosis) after excision of all ductal tissue (n = 4). In the presence of mild aortic arch hypoplasia, a longitudinal incision was made in the undersurface of the aortic arch and extended proximal to the hypoplastic segment. The anastomosis then was made side-to-end between the descending aorta and the undersurface of the aortic arch (n = 15). The aortic anastomosis was performed using either a running 6-0 (infants) or 7-0 (neonates) absorbable suture (polydioxanone [PDS]; Ethicon, Somerville, NJ).
Statistics
Values are presented as mean ± 1 standard deviation. Mean values of independent samples were compared using the unpaired t test. Differences in proportions were analyzed based on contingency tables using the
2 test. A p value less than 0.05 was considered statistically significant.
| Results |
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Preoperative Status
Comparison of important patient data is shown in Table 1
. Of the 46 patients, 28 showed signs of cardiac failure: 21 of 26 (80%) in the neonates and 7 of 20 (35%) in the infants (p < 0.01). Inguinal pulses were absent in all patients. Arterial hypertension as a leading symptom was noted in 34 of 46 patients (74%). Fourteen of 26 neonates (54%) were hypertensive, with systolic blood pressures beyond the 90th percentile of normal (range, 67 to 140 mm Hg; mean, 99 ± 20 mm Hg). All of the infants were hypertensive, with a mean systolic blood pressure of 137 ± 17 mm Hg (range, 116 to 165 mm Hg) (p < 0.01) (Fig 1
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Patent ductus arteriosus was noted in 13 neonates (50%) and in 3 infants (15%) (p = 0.01). Bicuspid aortic valve was seen in about one third of the patients (38% of neonates and 35% of infants). Mild aortic arch hypoplasia was observed in 12 neonates (46%) and in 3 infants (15%) (p = 0.025). In these patients, the aortic arch diameter (between the left carotid and left subclavian arteries) was reduced by a mean of 28% as compared with the diameter of the proximal descending aorta (angiographic measurements).
Surgical Procedure and Postoperative Period
Pure resection of the stenotic segment and end to end anastomosis were performed in 27 patients: in 10 of 26 neonates (38%) and in 17 of 20 infants (85%). In addition to pure resection, enlargement of the mildly hypoplastic aortic arch by proximal extension of the anastomosis was performed in 15 patients: 12 of 26 (46%) neonates and 3 of 20 (15%) infants (p = 0.025). In addition to resection and reanastomosis of the aorta, the left subclavian artery was transected and reimplanted in the remaining 4 neonates to excise all visible ductal tissue, which extended into the proximal part of that vessel. Reimplantation did not lead to appreciable stenosis of the left subclavian artery (mean systolic left arm pressure of 84 mm Hg, as compared with a mean of 100 mm Hg for the right arm). Except for 1 case with postoperative chylothorax (necessitating repeat thoracotomy), no operative complications were noted. The mean aortic cross-clamp time was 18 ± 6 minutes (range, 10 to 28 minutes) in the neonates and 14 ± 3 minutes (range, 10 to 21 minutes) in the infants (p = 0.02). The mortality rate was zero for the population studied. The mean postoperative blood pressure gradient (at hospital discharge) was 10 ± 7 mm Hg in the neonates and 7 ± 9 mm Hg in the infants (p = not significant) (Fig 2
). In both groups, the residual gradient exceeded 20 mm Hg in 2 patients, respectively. Transient postoperative hypertension was seen in 1 neonate (4%), but did not necessitate treatment. In 8 infants (40%), transient postoperative hypertension occurred, requiring drug therapy (between 1 day and 7 months) in all of them (p < 0.01).
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Follow-Up
A comparison of the mean blood pressure gradients between the groups of patients at different periods during follow-up is shown in Figure 2
. At the last follow-up visit, the blood pressure gradient did not differ statistically between the groups: 9 ± 10 mm Hg (range, 0 to 30 mm Hg) in the group with neonatal operation versus 4 ± 6 mm Hg (range, 0 to 20 mm Hg) in the group with operation during infancy. All children had good femoral pulses.
Although no patient experienced arterial hypertension necessitating drug therapy at last follow-up, there were a total of 11 children (24%) with systolic right arm pressures above the 90th percentile of normal (7 after neonatal operation and 4 after operation in infancy; p = not significant). Hypertension was associated with an arm to leg gradient greater than 20 mm Hg in only 1 patient.
Clinically, all children were well and showed normal growth as well as normal activity for age. No patient was receiving medication. In the patients with associated bicuspid aortic valve, the transvalvar gradient varied between 9 and 55 mm Hg at the last follow-up (mean, 16 mm Hg).
Recoarctation
During a mean follow-up period of 49 months (range, 13 to 95 months), 6 patients (13% of the whole population) demonstrated recoarctation according to the definition used. Of these 6 patients, 5 had primary operation as neonates, accounting for a 19% incidence of recoarctation in that patient group. Only 1 of the patients with recoarctation had operation during infancy (at 4 months of life), resulting in a 5% incidence of restenosis in that age group (Table 2). In all 6 cases, recoarctation developed within the first 5 months after the initial repair.
Of the 6 patients with restenosis, 4 (9% of the whole study population) needed reintervention between 4 and 5 months after the initial repair. Of these, 3 (2 neonates) had successful balloon dilatation of their recoarctation and 1 neonate had a successful reoperation. None of these 4 patients had signs of recurrence of stenosis at the last follow-up visit. In the other 2 patients (neonates), only mild recoarctation developed (gradients of 22 and 30 mm Hg, respectively); this did not require treatment.
Of the 11 children operated on between 6 and 12 months of age, none showed signs of recoarctation during the entire follow-up period.
| Comment |
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As for operative therapy, especially in neonates and infants, there is an ongoing debate about the ideal operative technique to achieve the lowest rate of recoarctation. In addition, except for emergency interventions in the neonatal period, it is not clear what would be the optimal timing for operation, resulting in not only the lowest incidence of restenosis, but also a low rate of late postoperative hypertension and consequent cardiovascular morbidity and mortality. It has been shown that early repair positively affects the incidence of late postoperative hypertension [6, 10, 11].
Much of the literature dealing with coarctation repair published during recent years has focused on the issue of the most appropriate operative technique for repair in neonates and infants [2]. It has been concluded in several studies that subclavian flap angioplasty should be favored [4, 12, 13]. Other authors have found that subclavian flap angioplasty was advantageous over resection and end-to-end anastomosis in infancy, but not in the neonatal period [14]. A combined resection and flap procedure also has been recommended as especially suited for repair in neonates [3]. Several publications have shown that resection and end-to-end anastomosis in neonates and infants gave results at least as good as the subclavian flap angioplasty [1418]. On the other hand, it has been reported unanimously that patch graft aortoplasty in infancy was associated with the highest rate of recoarctation [3, 19, 20].
It has been reported earlier that the results of resection and end-to-end anastomosis could be improved with the use of new suture materials [21, 22]. That the growth of a circumferential suture line in a great artery of a neonate does not necessarily lead to stenosis has already been learned with the arterial switch operation performed in newborns with transposition of the great arteries [18]. Based on these experiences and on earlier data [15], all patients in the present series had their anastomosis sutured with continuous absorbable monofilament polydioxanone. The overall incidence of restenosis (13% at a mean follow-up of 49 months) and the rate of reinterventions (9%) in the patients reported compare favorably with published rates of reintervention of up to 23% [16, 20] and of restenosis of up to 54% [23]. Comparison is difficult, as most other studies had a longer follow-up; end to end anastomosis has been reported also to carry a high risk of late postoperative recoarctation [20]. In our study, however, all instances of restenosis occurred early, within the first 6 postoperative months only. Comparison is further rendered difficult by the fact that most other studies also included patients with hemodynamically relevant associated cardiac lesions. In addition, studies with longer follow-up were based on operative series starting in the early 1970s.
Another important issue in the development of recoarctation is the presence of residual ductal tissue within the aorta. Residual ductal tissue after coarctectomy has been implicated in the formation of restenosis [15, 18, 24] and was especially considered to be involved in the high rate of recoarctation after the subclavian flap operation in neonates [15, 20]. In the present series, special care was always taken during operation to excise all visible ductal tissue; this included transection and reimplantation of the left subclavian artery if ductal tissue was seen to extend into that vessel. We consider this approach to have certainly contributed to the good results in the children reported.
There were no operative deaths in the present series of infants and neonates with isolated coarctation. This is in accordance with previous reports in which operative risk increased with the presence of hemodynamically relevant associated cardiovascular lesions, and with a recent study in which operative mortality was 3% in a large multicenter series of seriously ill neonates who underwent end to end anastomosis [19].
The two patient groups (operation either in the neonatal period or during infancy) in the present study did not differ statistically in the incidence of arterial hypertension as measured at the last follow-up visit; almost one fourth of the patients had blood pressure values slightly above the 90th percentile, but no patient required medication for systemic hypertension. One limitation of the present study is the relatively short follow-up period, which does not yet allow specific comments on the incidence or severity of late postoperative systemic hypertension in the population studied. Longer follow-up of these patients will show whether early elective coarctation repair might positively influence late cardiovascular morbidity.
The present series showed that coarctation repair in newborns was associated with the highest risk of recurrence of stenosis; the incidence of restenosis was already lower when operation was done even in early infancy (although this difference did not reach statistical significance because of the relatively small number of patients studied). This finding must be confirmed by studies with larger patient numbers.
The results achieved with resection and end-to-end anastomosis in the patients reported demonstrate that this operative technique should be considered as an option of great value in the treatment of coarctation in infants and neonates.
| Footnotes |
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