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Ann Thorac Surg 1998;66:1378-1382
© 1998 The Society of Thoracic Surgeons

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Original articles: cardiovascular

Repair of coarctation of the aorta during infancy minimizes the risk of late hypertension

^a Division of Cardiothoracic Surgery, New England Medical Center and Tufts University School of Medicine, Boston, Massachusetts, USA
^b Division of Pediatric Cardiology, New England Medical Center and Tufts University School of Medicine, Boston, Massachusetts, USA

Accepted for publication April 22, 1998.

Address reprint requests to Dr Warner, Division of Cardiothoracic Surgery, New England Medical Center, Box 266, 750 Washington St, Boston, MA 02111
e-mail: (kenneth.warner{at}es.nemc.org)

	Abstract

Top Abstract Introduction Material and methods Results Comment Summary References

 
Background. Recent surgical reports on coarctation of the aorta have primarily focused on the relative merits of various operative techniques. However, appropriate timing for elective repair remains unclear.

Methods. In a retrospective analysis we examined the surgical outcomes in 176 consecutive patients undergoing repair of coarctation of the aorta in our institution over a 25-year period. Ninety-nine percent of the patients had follow-up for a median of 7.5 years.

Results. A total of 13 patients have died (7.4% overall mortality). Nine of these patients had associated complex intracardiac anomalies. There was no mortality in the 113 patients with isolated coarctation. Residual or recurrent coarctation occurred in 27 patients (15.3%). The age at operation and the type of surgical repair did not have an effect on the incidence of recurrence. Persistent or late hypertension was identified in 18 of the 107 patients who have been followed up for more than 5 years (16.8%). A total of 48 patients operated on during infancy have been followed up for more than 5 years. Only 2 have developed late hypertension (4.2%). Both of these patients had recurrence. In contrast, 16 of the 59 patients operated on after a year of age had late hypertension (27.1%).

Conclusions. To minimize the risk of persistent hypertension, elective repair of coarctation should be performed within the first year of life.

	Introduction

Top Abstract Introduction Material and methods Results Comment Summary References

 
Proper timing for elective repair of coarctation of the aorta remains controversial. Repair during infancy has been associated with a higher rate of recurrence than later repair [1–4]. On the other hand, delaying the surgical procedure until later in childhood may fail to relieve systemic hypertension completely [1, 5–10]. Older studies dating from the 1970s and 1980s have suggested that relief of aortic coarctation should be performed before school age to reduce the incidence of persistent hypertension [7–10]. Recent reports from the surgical literature have primarily focused on various operative techniques including the increasingly popular extended end-to-end anastomosis [2, 4, 11–18]. However, there is little information on the appropriate age for elective repair of coarctation in the current era.

For the past 25 years we have repaired coarctation of the aorta in all age groups with a variety of surgical techniques. The objectives of this retrospective review were threefold. Our first intent was to analyze the results of the various operative approaches to determine whether one technique was superior to the others. The second goal was to establish, if possible, the optimal age for elective relief of coarctation by analyzing our short- and long-term results. The third and final objective was to establish the long-term results of a surgical procedure as a comparison with the balloon angioplasty technique, which is currently used with increasing frequency in our and other institutions as a preferred technique for the treatment of native coarctation.

	Material and methods

Top Abstract Introduction Material and methods Results Comment Summary References

 
The records of the 176 consecutive patients undergoing a surgical procedure for coarctation of the aorta between 1969 and 1995 were reviewed. This retrospective study reviewed operative notes, follow-up clinic visits, and postoperative echocardiographic and cardiac catheterization data. Ninety-nine percent of the patients have been followed up for a mean of 7.5 years (range, 6 months to 24 years).

The median age at operation was 11 months (range, 1 day to 33 years). The patients were grouped according to age: neonates (less than 1 month, n = 63), infants (1 to 12 months, n = 31), toddlers (1 to 4 years, n = 24), and children and adults (more than 4 years, n = 58).

Patients were stratified according to the presence of concomitant intracardiac anomalies [19]. Class 1 (n = 113) had isolated coarctation, class 2 had coarctation and a single ventricular septal defect (n = 27), and class 3 had coarctation and complex cardiac pathology (n = 36). Figure 1 shows the distribution of the classes according to the age at repair. The associated cardiac lesions for class 3 patients were as follows: important subvalvar aortic stenosis, 11; complete atrioventricular canal defect, 6; congenital mitral valve disease, 5; double-outlet right ventricle, 4; critical valvar aortic stenosis, 4; single ventricle, 3; and transposition of the great arteries, 3.

View larger version (23K): [in this window] [in a new window]   Fig 1. Classes of patients stratified according to age at the time of repair. Nearly two thirds of the neonates and half of the infants had associated cardiac anomalies (classes 2 and 3). In contrast, the majority of patients over 1 year of age had isolated coarctation (class 1). (yrs = years.)

View larger version (35K): [in this window] [in a new window]   Fig 2. Various operative techniques grouped according to age. (E-EE = extended end-to-end anastomosis; G = graft interposition; PA = patch angioplasty; RETE = resection with end-to-end anastomosis; SFA subclavian flap angioplasty; yrs = years.)

Residual or recurrent coarctation was defined as a systolic blood pressure gradient between the right arm and either leg exceeding 20 mm Hg. Documentation of persistent coarctation was confirmed with echocardiography, angiography, or both.

Variables used in the univariate analysis included age at repair, class, type of operation, era (1969 to 1981 versus 1982 to 1995), and placement of a pulmonary artery band. Specific outcomes analyzed included mortality, recurrent coarctation, and development of late hypertension. Multivariate analysis was performed using the Cox proportional hazards model. Actuarial data were analyzed by the Kaplan–Meier method with 95% confidence limits. Categoric data were compared with the Fisher’s exact test. Statistical significance was defined when p was less than 0.05.

	Results

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Mortality
Six patients died within 30 days of the operation, for an operative mortality of 3.4%. An additional 7 patients died between 3 months and 6 years after the operation, for an overall mortality of 7.4%. Survival was in part determined by associated cardiac anomalies. There were no deaths in the 113 patients with isolated coarctation (class 1). Two of the 27 class 2 patients (7.4%) died. Eleven of the 36 class 3 patients (30.6%) died. Univariate analysis showed that determinants of mortality included age at repair (neonates), class 3 patients, and placement of a pulmonary artery band. Two of the 8 class 2 patients who received a pulmonary artery band (25%) died. Nine of the 17 class 3 patients who received a pulmonary artery band (53%) died. Multivariate analysis indicated that the only determinant of mortality was associated class 3 anomalies. The 10-year overall survival was 100% for class 1, 92% for class 2, and 67% for class 3 (Fig 3).

View larger version (17K): [in this window] [in a new window]   Fig 3. Kaplan–Meier survival curves indicate that class 3 had a significantly worse outcome than class 1 and class 2 patients.

View larger version (22K): [in this window] [in a new window]   Fig 4. Cumulative data for freedom from intervention for recurrence of coarctation. There was no statistical difference in the rate of recurrence for the various operative techniques. (NS = not significant; for other abbreviations see Fig 2.)

View larger version (14K): [in this window] [in a new window]   Fig 5. Percentage of patients with persistent or late hypertension listed according to age at operation. The percentage of patients with late hypertension was significantly less in the neonatal and infant groups (4.2%) than in toddlers and older children (27.1%; p < 0.002). (YR = years.)

	Comment

Top Abstract Introduction Material and methods Results Comment Summary References

Recurrence of coarctation
The 15% incidence of recurrence of coarctation in this study is similar to that reported by others [1–6, 11–14, 16, 18, 25–27]. Several investigators have shown a higher incidence of recurrence following repair of coarctation during infancy [1, 2, 5, 6, 10, 19, 28]. This higher rate of recurrence in very young patients may reflect inadequate relief of aortic arch hypoplasia commonly seen in this age group. In the current study, we did not find a significantly higher rate of recoarctation in neonates and infants. This may have reflected a more aggressive approach in recent years to augment the aortic arch with the extended end-to-end anastomotic technique.

Incidence of late hypertension
Of particular importance in this study is the finding that the incidence of late hypertension was directly related to the age at operation. Surgical relief of coarctation of the aorta before the age of 1 year was associated with a very low (4.2%) incidence of late hypertension. In contrast, relief of coarctation after 1 year of age resulted in a sixfold increase in the occurrence of late hypertension. The importance of the prevention of persistent elevation of blood pressure is illustrated in studies which have shown that late postoperative hypertension is an independent risk factor for premature death [1, 8, 10, 27].

The 50% incidence of late hypertension in our patients with recurrence of coarctation underscores the importance of identifying this complication at an early age [5–7]. Early relief of recoarctation with either balloon angioplasty or a reoperation before 1 year of age may prevent the onset of late hypertension. Close surveillance of all patients is thus warranted after repair of coarctation.

Appropriate timing for the surgical procedure
Previous investigations which have attempted to determine the appropriate age for repair often failed to include significant numbers of patients from all age groups, thus precluding accurate statistical comparisons [5, 6, 8–10, 28]. Clarkson and colleagues [8] concluded that elective repair of coarctation should be performed during the second decade of life; infants and small children were not included in the series. Bergdahl and coworkers [9] recommended surgical repair before school age; however, no patient less than 5 years of age was included in the study. Campbell [28] suggested that the optimal age for surgical correction was during infancy; however, children older than 1 year of age were not included in the study. Three additional studies [7, 10, 27] reported that the appropriate age of relief of coarctation was during the second year of life; however, the number of neonates and infants included in these studies was comparatively few.

Two studies from Great Britain reported in the 1970s suggested that the optimal age for coarctation repair was during the first year of life [5, 6]. However, the conclusions of both studies were not supported by the data. In the study by Shinebourne and associates [5], the incidence of late hypertension was nearly identical in patients operated on either before or after 1 year of age (29% versus 32%). Disconcerting was the observation that the operative mortality was actually greater in the neonatal and young infant groups when compared with older children (21% versus 4%). Similarly, in the study published by Patel and coworkers [6], the rates of persistent hypertension were virtually the same for the infant and older age groups. More important, both operative mortality and the development of recoarctation were significantly greater in the infant age group. Hence, contrary to the stated conclusions, the results from these two studies suggest that safe repair of coarctation should be performed in children after, not before, 1 year of age.

In contrast to these previous studies, statistical analysis of the data from the current study does support our conclusion that repair of coarctation should be performed during the first year of life. Using a variety of surgical techniques we demonstrated that the surgical mortality and freedom from recurrence in the neonatal and infant age groups were equivalent to those of toddlers and older patients. Thus, repair of coarctation during infancy in our series of patients was performed safely and with minimal morbidity. The benefit of early repair was clearly shown in the analysis of the long-term results. The relatively low incidence of late hypertension in the neonatal and infant repairs compared to the sixfold increase in the toddler and older age groups provides compelling evidence that repair of coarctation of the aorta should be performed during the first year of life to avoid this serious lifelong complication.

Associated mortality with pulmonary artery band
Of particular concern was the 30% mortality in class 3 neonates undergoing coarctation repair in our series. Other investigators have reported similar results in this subset of patients [6, 15]. Placement of a pulmonary artery band was associated with particularly high mortality in our series. The relatively high morbidity and mortality resulting from pulmonary artery banding are well recognized [29] and suggest that a more aggressive approach to this group of patients may provide a more favorable outcome. A recent study has suggested that either the Damus–Kaye–Stanzel procedure or the Norwood operation may improve short-term and long-term survival compared with placement of a pulmonary band in patients with single ventricle anatomy and multiple levels of left-sided obstruction [30].

Similarly, the relatively high operative mortality (7%) in the class 2 patients with isolated ventricular septal defects also suggests that an alternative surgical approach may be appropriate in this group of patients. A single-stage approach with simultaneous ventricular septal defect repair and relief of the aortic arch obstruction through a midline approach has gained increasing support [31]. However, a recent large multicenter study failed to demonstrate an advantage for infants undergoing a single-stage repair of isolated ventricular septal defect and coarctation [15].

Limitations of study
A recognized limitation in this study is that the data were collected and analyzed in a retrospective manner. It is possible that important determinants for the stated outcomes of the study (mortality, recurrence, and late hypertension) were either not appreciated or not properly analyzed. We also realize that because this was not a prospective randomized study the inferences made may be subject to further statistical scrutiny. However, we believe that the relatively large number of patients in our study accrued over a quarter of a century has established a sufficient database for proper statistical analyses to support our conclusions.

Comparison with balloon angioplasty
In recent years, percutaneous balloon angioplasty has become an alternative method for the treatment of native coarctation [32, 33]. Although the short-term results are encouraging, the long-term outcome including recurrence, the occurrence of aneurysm formation, and the incidence of late hypertension are unknown. The results of this surgical series establish a baseline for comparison for the balloon angioplasty technique.

	Summary

Top Abstract Introduction Material and methods Results Comment Summary References

 
In summary, the findings in this study indicate that the optimal age for elective surgical relief of coarctation of the aorta is during the first year of life. Although the incidence of recurrence was relatively low and unrelated to the surgical approach, identification and proper treatment of recoarctation at an early age are imperative to minimize the development of late hypertension.

	References

Top Abstract Introduction Material and methods Results Comment Summary References

 

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