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Ann Thorac Surg 2000;70:1483-1488
© 2000 The Society of Thoracic Surgeons

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Original articles: cardiovascular

Coarctation of the aorta in adults: surgical results and long-term follow-up

^a Department of Thoracic and Cardiovascular Surgery, Rouen University Hospital Charles Nicolle, Rouen, France

Address reprint requests to Dr Bouchart, Service de Chirurgie Thoracique et Cardiovasculaire, CHU de Rouen, 1 rue de Germont, F76031 Rouen, France
e-mail: francois.bouchart{at}chu-rouen.fr

Presented at the Thirty-sixth Annual Meeting of The Society of Thoracic Surgeons, Fort Lauderdale, FL, Jan 31–Feb 2, 2000.

	Abstract

Top Abstract Introduction Material and methods Results Comment Acknowledgments References

 
Background. The aim of this retrospective study was to determine the impact of coarctation surgical repair on arterial blood pressure in adults more than 20 years of age.

Methods. Thirty-five adults (23 men), mean age 28.1 ± 5.7 years (range, 21 to 52 years), underwent coarctation surgical repair between 1977 and 1997. All patients had preoperative hypertension. Mean systolic blood pressure was 178 ± 37 mm Hg (range, 110 to 230 mm Hg). Thirty-three patients were taking at least one hypertension medication at the time of operation. All patients had preoperative catheterization and angiography (mean gradient across the coarctation was 62 ± 27 mm Hg [range, 32 to 130 mm Hg]). Operative technique was resection and end-to-end anastomosis for 30 patients, resection with Dacron (C. R. Bard, Haverhill, MA) graft for 4 patients, and a prosthetic bypass graft for 1 patient. There were no hospital deaths and no late morbidity.

Results. All patients were reviewed. Follow-up was 165 ± 56 months (range, 25 to 240 months). Of the 35 patients with preoperative hypertension, 23 were normotensive (systolic blood pressure 140 mm Hg, diastolic blood pressure 90 mm Hg) with no medication. Twelve patients were receiving medication: 6 required single-drug therapy and 6 patients required two drugs. Exercise testing was performed at an average of 6 ± 4 months after repair and revealed hypertensive response to exercise in 8 of the 23 patients who were normotensive at rest and without medication. There were no recoarctation or repeat operations. Six aortic valve diseases were observed: three aortic incompetences (two bicuspid valves) treated by two valve replacements and one Bentall procedure, and three aortic stenoses (two valve replacements). No patient had evidence of a cerebrovascular accident.

Conclusions. Surgical repair of coarctation in adults has proved to be an effective procedure and significantly reduces arterial hypertension. However, long-term surveillance is mandatory and should include exercise testing to identify patients with potential hypertension.

	Introduction

Top Abstract Introduction Material and methods Results Comment Acknowledgments References

 
The first clinical surgical repair of coarctation of the aorta was reported by Crafoord and Nylin [1] in 1945. Several series [2–4] have shown short-term success of coarctation repair in terms of decreased mortality and a decrease in hypertension. However, many questions regarding long-term survival and relief of hypertension remain unanswered. The late consequences of long-standing hypertension and the fate of the repaired aortic segment require further clarification. The highest probability of survival was observed in patients younger than 10 years at operation, and other series have shown that the incremental risk factor for recurrence of coarctation, late hypertension, and premature death was age. Furthermore, some authors [5] suggest that the best age for coarctation repair is approximately 1.5 years. There are, however, few data concerning the benefits of coarctation repair in adults [4, 6, 7]. The purpose of this retrospective study was to determine the impact of coarctation repair on blood pressure using exercise tests and ambulatory blood pressure measurements, to achieve satisfactory management of residual hypertension.

	Material and methods

Top Abstract Introduction Material and methods Results Comment Acknowledgments References

 
Between 1977 and 1997, 35 adult patients older than 20 years underwent repair of aortic isthmic coarctation. There were 23 men and 12 women with a mean age of 30.8 ± 5.7 years (range, 21 to 52 years). Preoperative findings are summarized in Table 1. Eleven patients (31%) presented with at least one symptom. For the other 24 patients, the diagnosis made by physical examination was prompted by the discovery of high blood pressure. Clinical findings were a systolic murmur in 4 patients, and femoral pulses were absent in 26 patients. One patient had Turner’s syndrome.

Only 1 patient was normotensive before operation, 23 patients received at least one hypertensive medication, and the remaining 11 patients were operated on soon after discovery of the aortic coarctation and thus did not receive antihypertensive medication. Antihypertensive medication included diuretics, vasodilators, ß-blockers, calcium-channel antagonists, and angiotensin-converting enzyme (ACE) inhibitors.

Left ventricular hypertrophy was found on electrocardiogram in 29 patients, rib notching was present in 31 patients, and a dilated ascending aorta was found in 4 patients (aortic diameter between 4 and 5 cm). Four patients had a bicuspid aortic valve with no regurgitation.

Thirty-three patients had a preoperative angiogram. Peak systolic gradient across the coarctation was 69 ± 32 mm Hg (30 to 117 mm Hg). Intrathoracic aneurysms were identified in 4 patients: three were intercostal artery aneurysms ranging from 15 to 40 mm. One patient had a descending aorta aneurysm measuring 6 cm that was treated at the same time as the coarctation.

Surgical technique
Coarctation repair was carried out through a left thoracotomy. Careful dissection and mobilization of the aortic arch and left subclavian artery, guided by the preoperative angiogram, allowed end-to-end anastomosis to be performed in 30 patients (86%). Great care was taken to the first intercostal arteries to avoid ligation or tearing. Interposition of a Dacron (C. R. Bard, Haverhill, MA) graft was necessary in the remaining 5 patients. In 1 patient with aortic arch hypoplasia, the coarctation was bypassed by a Dacron graft between the ascending and descending aortas. No bypass or shunts were used in any patient. Aneurysms of the intercostal artery and of the descending aorta were resected during the same procedure.

Follow-up
Patients were seen in outpatient clinics and data were obtained through questionnaires sent to the family physicians and cardiologists. Additional information was obtained from the cardiologist or from the physician if necessary. The last examination was performed between August and October 1997 for 32 patients, during 1996 for 2 patients, and 1 patient was lost to follow-up after 7 years.

All patients underwent an exercise stress test using the Bruce protocol. An abnormal hypertensive response to the exercise stress test in untreated patients was defined as DBP higher than the 95th percentile for age and sex as defined in the Framingham Heart Study by Singh and collaborators [9].

Ambulatory 24-hour blood pressure measurements were performed in all patients. The quality of the repair was evaluated by Doppler echocardiography. Doppler estimation of systolic peak flow velocity in the descending aorta and the presence or absence of a diastolic tail were observed [10]. Echocardiographic confirmation of a successful repair included a completely patent aortic lumen at the site of the repair, systolic peak flow velocity less than 3 m/s, and no diastolic flow in the descending aorta. Left ventricular wall thickness and systolic function were analyzed by M-mode echocardiography. Computed tomographic (CT) scans were performed in 28 patients. An angiogram was obtained in 8 patients early in our experience and excluded a significant residual stenosis at the site of repair.

Statistical analysis
Data are presented as the mean ± standard deviation. Systolic gradients across the coarctation before and after repair were compared using a paired t test. A value of p less than 0.05 was considered significant.

	Results

Top Abstract Introduction Material and methods Results Comment Acknowledgments References

 
Postoperative follow-up
There was no surgical mortality. Morbidity included two recurrent nerve palsies and one reoperation for bleeding. The mean length of follow-up was 13.8 ± 4.6 years (range, 1.4 to 22 years). One patient was lost to follow-up. At the most recent follow-up, all patients were alive. Thirty-two patients (94%) were symptom free (New York Heart Association class I) and leading a normal lifestyle; only 5 patients were unemployed. Two patients had angina pectoris and 1 patient had myocardial infarction.

At clinical examination, all patients had palpable femoral pulses with no significant femoral delay. A systolic murmur was found in 3 patients.

Hypertension
At the first postoperative evaluation, 25 patients (71%) were normotensive at rest and without any antihypertensive medication. The remaining 10 patients showed significant improvement of their SBP after the operation (178 ± 31 versus 140 ± 14 mm Hg, respectively, p < 0.0001). The evolution of SBP and DBP before the operation, 1 month after the operation, and at late follow-up is shown in Figure 1. There was a highly significant decrease in SBP (p < 0.0001), which remained significant at late follow-up (p < 0.0001). Between the postoperative period and late follow-up, the blood pressure did not change significantly. However, an increase in the number of drugs needed to maintain the blood pressure within normal range was observed during the same period. Figure 2 shows the number of patients who received from zero to three antihypertensive drugs to maintain their blood pressure within normal range before, just after, and late after the operation. Table 2 shows the evolution of blood pressure and treatment before and after the operation and at late follow-up. At last examination, 23 patients were normotensive at rest without any antihypertensive medication (compared with 25 patients at first examination). The other 12 patients received at least one antihypertensive drug. Three patients receiving medication remained hypertensive at rest.

View larger version (27K): [in this window] [in a new window]   Fig 1. Evolution of systolic and diastolic blood pressure before coarctation repair, within 1 month after the repair, and at late follow-up. *p < 0.0001 versus preoperative (Preop) value. (postop = postoperative.)

View larger version (36K): [in this window] [in a new window]   Fig 2. Evaluation of hypertension by the number of different drugs required to maintain blood pressure within normal range before coarctation repair, within 1 month after the repair, and at late follow-up. (postop = postoperative; Preop = preoperative.)

All patients available for follow-up underwent ambulatory blood pressure measurements within 1 year to last follow-up. Nine patients showed abnormal profiles: 6 already received antihypertensive medications and the other 3 had abnormally high profiles during daytime but showed a normal blood pressure decrease at night.

Recoarctation
Echocardiography, CT scan, and angiography showed evidence of mild stenosis at the distal anastomosis of a Dacron graft in 1 patient. Nevertheless, 3 patients operated on 11 and 12 years ago have a peak gradient respectively more than 26 mm Hg requiring close follow-up.

Aortic valve disease
A bicuspid aortic valve was associated with the coarctation in 4 patients and their valves were functional with no significant gradient at first examination. Six patients developed significant aortic valve disease on long-term follow-up (only 1 patient had mild aortic incompetence before operation). Three patients had significant aortic regurgitation requiring aortic valve replacement in 2 patients (two bicuspid valves) and aortic root replacement (Bentall procedure) in 1 patient. Three patients had aortic valve stenosis requiring aortic valve replacement in 2 patients (one bicuspid valve).

No patient had a significant aneurysm at late angiography or CT scan requiring operation, but slight abnormalities were noted in 11 patients. A moderate enlargement of the ascending aorta was present in 4 patients, the left subclavian artery was dilated in 4 patients, and a slight narrowing was present at the site of repair in 3 patients.

Ischemic disease
Only 1 patient in this series presented ischemic heart disease treated by oral medication. One patient underwent a carotid endarterectomy. None of the patients presented a significant neurologic event during the follow-up period.

	Comment

Top Abstract Introduction Material and methods Results Comment Acknowledgments References

 
Natural history
The natural history of coarctation has been well documented by Campbell [11], who demonstrated that 50% of untreated patients were dead by the age of 30 years, 75% at 46 years, and 90% at 58 years. Comparing these data with normal life expectancy tables we observed that there was a high increase in mortality during the third to fifth decades of life. Presbitero and coworkers [3], whose postoperative follow-up ranged from 15 to 30 years, reported improved life expectancy after operation.

Operative procedure
In this series, reconstruction of the aorta with end-to-end anastomosis remained the procedure of choice. A graft interposition was used whenever adequate resection of the narrowed segment resulted in too great a discrepancy to permit direct suture of the aorta. In one case, we used a bypass between the proximal and distal aortas.

Coarctation repair in adults presents technical difficulties: thickness of the aortic wall, difficulties in aortic arch mobilization, severe calcifications, and large collateral arteries with aneurysmal dilatation. In these cases, a prosthesis should be used to avoid traction on aortic ends. In some cases, bypass grafting between the subclavian artery or the proximal aorta and the distal aorta should be the procedure of choice. Aris and associates [7] performed operations on 8 patients over 51 years using this technique and achieved good results. This procedure requires less aortic dissection, can be performed with a partially occluding clamp, and does not compromise the spinal cord vascularization. This procedure seems to be as effective as any other type of repair, as shown by Wells and coworkers [4], who used it in 11 of 26 patients and achieved results comparable with other procedures. On the other hand, some authors [10, 11] have concluded that patch aortoplasty repair of aortic coarctation should be abandoned in adults, because of the high incidence of aneurysm formation after the procedure. Bypass grafting is an effective procedure if end-to-end anastomosis with or without prosthesis cannot be performed [6–8].

Hypertension
Persistent hypertension or hypertension recurring or developing after coarctation repair is the most important factor of morbidity and mortality associated with advancing age in patients. Some series [12–14] found that the younger the age the operation was performed the greater the postoperative reduction in blood pressure. Also late hypertension was more common when the operation was delayed until after the age of 20 years. Maron and colleagues [6] suggested that prolonged preoperative hypertension and coarctation repair after age 25 years increased the risk of premature cardiovascular death (mean follow-up of 16 years in 248 patients who were operated on at a mean age of 20 years). Some authors [16] have emphasized an association between preoperative hypertension and an increasing risk of late mortality, however, this was not confirmed by our study. Patients older than 20 years at the time of operation develop hypertension affecting SBP and DBP 10 to 20 years after the repair [4, 13, 15]. Persistent hypertension after aortic coarctation repair is multifactorial [8, 16–20]. In adults, these factors include anatomic aortic alterations, functional and structural wall alterations of thoracic and peripheral vessels, poor compliance of the arterial tree, endocrine factors, and altered renin-angiotensin system [21]. After repair, patients have an increased sympathetic discharge at peak exercise leading to increased peripheral resistances. Gardiner and colleagues [20] also documented an exercise-related hypertension in operated patients caused by significant impairment of arterial dilatation in the precoarctation vascular bed compared with healthy young adults. Our series demonstrated that persistence of vessel abnormal reactivity after coarctation repair should be investigated by exercise tests. Among normotensive patients shortly after operation, only 2 patients (8%) required antihypertensive medication at late follow-up. However, 8 patients were normotensive at rest and were hypertensive after exercise test. Most long-term series lack information regarding the evolution of SBP during exercise stress tests but also regarding antihypertensive therapy. In patients with hypertension at rest or during exercise, it could be useful to carry out repeated exercise tests, on a yearly basis, to be able to implement antihypertensive medications, ß-blockers, or ACE inhibitors [18]. This series also revealed a high number of patients presenting residual hypertension after repair who required an increase in their medications, but these patients had a relatively short follow-up compared with other series [12, 13, 16]. All patients operated on at 40 years or older remained hypertensive after operation and at late follow-up.

According to the literature, the incidence of hypertension during exercise despite normal blood pressure at rest varies between 25% and 56% [17, 22, 23]. In these patients it seems necessary to carry out frequent and regular tests to administer effective antihypertensive medication [17, 22].

Our study did not show any correlation between residual gradient present in 3 patients and late hypertension. Of the 25 available patients who were normotensive at discharge, 23 remained normotensive at late follow-up. Nevertheless, no relationship was found between perioperative hypertension and subsequent hypertension at late follow-up, although other authors have reported the contrary [2, 6].

Cerebrovascular accidents
In agreement with other authors [8, 25], we report no deaths from cerebrovascular accidents, which accounted for 11.8% in earlier autopsy results [2]. However, relief of distension and hypertension in the upper segment, combined with improvement in antihypertensive therapy, may provide an explanation [3, 24, 25].

Associated heart disease
In our study, 4 patients had bicuspid aortic valve associated with coarctation. Five patients with aortic valve disease underwent operation. One patient may require operation in the forthcoming years. Calcifications of the valve become more common in the third or fourth decade. A high percentage of patients who undergo aortic valve replacement have bicuspid valves that are stenotic (2 patients in our series), incompetent (1 patient in our series), or damaged by endocarditis [24].

Conclusion
The results of this study confirm that surgical repair of aortic coarctation in patients older than 20 years of age reduces systolic hypertension. However, the incidence of ischemic heart disease and recurring or developing postoperative hypertension emphasizes the fact that accurate follow-up, including an exercise test, is mandatory to control residual hypertension and to avoid late cardiovascular complications.

	Acknowledgments

Top Abstract Introduction Material and methods Results Comment Acknowledgments References

 
We thank Richard Medeiros for his valuable advice on editing the manuscript.

	References

Top Abstract Introduction Material and methods Results Comment Acknowledgments 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): François Bouchart Arnaud Dubar Robert Soyer Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Bouchart, F. Articles by Soyer, R. Search for Related Content PubMed PubMed Citation Articles by Bouchart, F. Articles by Soyer, R. Related Collections Related Article