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Ann Thorac Surg 2005;80:1659-1665
© 2005 The Society of Thoracic Surgeons
a Division of Cardiovascular Surgery, St. Louis University School of Medicine/Cardinal Glennon Children's Hospital, St. Louis, Missouri
b Division of Pediatric Cardiology, St. Louis University School of Medicine/Cardinal Glennon Children's Hospital, St. Louis, Missouri
c Division of Pediatric Pathology, St. Louis University School of Medicine/Cardinal Glennon Children's Hospital, St. Louis, Missouri
d Division of Cardiothoracic Surgery, East Tennessee State University, Johnson City, Tennessee
Accepted for publication March 21, 2005.
* Address correspondence to Dr Fiore, St. Louis University Health Sciences Center, Cardinal Glennon Children's Hospital, 1465 S. Grand Blvd, St. Louis, MO 63104 (Email: fiorem2{at}slu.edu).
Presented at the Fifty-first Annual Meeting of the Southern Thoracic Surgical Association, Cancun, Mexico, Nov 24, 2004.
| Abstract |
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METHODS: A retrospective comparison between balloon angioplasty and surgery for the treatment of neonatal aortic coarctation was undertaken on 57 neonates younger than 40 days of age (angioplasty, 23 patients; surgery, 34 patients) treated between 1994 and 2004.
RESULTS: Cohorts were similar with respect to the preinterventional variables of age, weight, upper extremity systolic blood pressure, coarctation gradient, degree of aortic arch hypoplasia, associated conditions, and mean follow-up (angioplasty, 36 months; surgery, 38 months). Among the angioplasty group, 13 patients (57%) required surgery, and 8 required a second balloon dilatation, of whom 3 patients had an aortic aneurysm. Among the surgery cohort, 6 patients experienced recurrence (18%) after either SFA (3) or XETE anastomosis repair (3). All were successfully treated with balloon angioplasty. Actuarial freedom from any intervention was significantly greater in the surgery cohort as was the degree of aortic arch growth. At latest follow-up, antihypertensive medication was required in 3 of 9 angioplasty patients (33%) and 2 of 27 surgery patients (7%). No repeat intervention was required in the 13 patients who underwent angioplasty followed by surgery.
CONCLUSIONS: Primary angioplasty is palliative treatment for neonatal aortic coarctation, but it is the treatment of choice for recurrence after surgery. Surgery for neonatal aortic coarctation is associated with fewer reinterventions, improved aortic arch growth, no aortic aneurysm formation, and decreased need for antihypertensive medication when compared with neonates treated primarily with balloon angioplasty.
| Introduction |
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Balloon dilatation of congenital left-sided obstructive cardiac lesions has replaced surgery in some instances. The treatment of choice for neonatal congenital aortic stenosis is balloon valvuloplasty. Similarly, balloon valvuloplasty for pulmonary valve stenosis may have distinct advantages over surgery.
In contrast, the utility of balloon coarctation angioplasty for the initial treatment of NAC remains controversial. In this clinical setting, concerns include early restenosis, aneurysm formation, and the potential for early and late ischemic injury to the lower extremity.
To evaluate the efficacy and safety of these two modes of treatment, we retrospectively compared our current experience with balloon angioplasty (A) and surgery (S) for neonatal coarctation of the aorta.
| Patients and Methods |
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The diagnosis of coarctation was made by echocardiography or computed tomographic angiography in all patients. The maximum velocity across the coarctation site was recorded by continuous-wave Doppler measurement, and the peak instantaneous pressure gradient was calculated by the modified Bernoulli equation.
Patients were selected for angioplasty if the juxtaductal coarctation was discrete and not associated with significant aortic arch or isthmus hypoplasia. Angioplasty was performed according to the technique of Rao and associates [1], using umbilical artery cannulation for the first intervention in all patients.
The remaining neonates who had coarctation less amenable to angioplasty underwent operative correction for coarctation through a left thoracotomy. The operation performed was an SFA according to the technique of Waldhausen and associates [2] or coarctation resection with XETE anastomosis as described by Amato and coworkers [3].
Reintervention for recurrent coarctation was performed if the arm-to-leg gradient was greater than 20 mm Hg, the peak systolic echocardiographic gradient was greater than 25 mm Hg, or the upper extremity systolic blood pressure exceeded the 95th percentile for age.
Assessment for aortic arch hypoplasia was performed using the ratio of the distal arch diameter (diameter of the transverse aortic arch between the left common carotid artery and left subclavian artery) to the ascending aortic diameter (measured just proximal to the innominate artery) as measured echocardiographically. Ratios less than 0.5 were considered abnormal [4]. The growth of the distal aortic arch with time was assessed by comparing the diameter of the distal arch to the diameter of the ascending aorta using echocardiographic measurements at the time of presentation and after the first intervention.
Postintervention aneurysm formation was defined according to the criteria of Beekman and coworkers [5]. These included either a fusiform dilatation at the coarctation site with a diameter greater than 150% of the aortic diameter at the diaphragm or a discrete saccular dilation that was not present at the preangioplasty imaging study. If an aneurysm was detected on a follow-up aortogram, the preintervention aortogram was reviewed to ascertain that this irregularity of the aorta was not present before treatment.
Pathologic evaluation of the resected coarctation segments from patients having surgery alone or angioplasty followed by surgery was performed by a pediatric pathologist (D.D.) blinded as to the initial intervention undertaken. Specimens were oriented randomly and stained with hematoxylin and eosin or Masson-Trichrome. The four pathologic features quantitated in each specimen were fibrinoid necrosis, inflammation, intimal proliferation, and hemorrhage.
Early mortality was defined as death in the hospital or within 30 days of discharge.
The Statistical Package for Social Sciences (SPSS, Chicago, IL) was used for statistical analysis. Data are expressed as a mean ± standard deviation. For all statistical testing, a p less than 0.05 was considered significant and two-tailed tests were used or
2 analysis as applicable. Categorical data were compared using Fisher's exact test, whereas Student's t tests were used for continuous variables. Time-dependent variables were examined using Kaplan-Meier analysis. The Wilcoxon test was used to determine statistical significance.
| Results |
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Surgery
Thirty-four patients underwent operation as the initial treatment for NAC. Of these, 8 patients (14%) had SFA and 26 patients (76%) received coarctation resection with XETE anastomosis. Six of 34 patients (18%) experienced recoarctation at a mean of 3.4 ± 1.8 months (range, 0.85 to 5.3 months). There was no significant difference in the number of patients undergoing SFA and the number undergoing XETE repair (3 of 8, 38% SFA; versus 3 of 26, 12% XETE). The suture material used among these six recurrent coarctations was equally divided between polypropylene (Ethicon, Inc, Somerville, NJ) and absorbable polydioxanone (Ethicon, Inc). All six surgical recurrences underwent successful balloon dilatation with a reduction of the arm-to-leg gradient to 3 ± 4 mm Hg and peak echocardiographic gradient to 16 + 10 mm Hg.
One patient required a second operation after XETE to enlarge the proximal transverse aortic arch. The 13 patients who underwent balloon angioplasty followed by surgery did not require any further reintervention.
Actuarial freedom from reintervention is shown in Figure 1. Each curve compares both cohorts with respect to the initial intention to treat. Reintervention is defined as either angioplasty or surgery. Patients in both cohorts required reintervention within the first year, but neonates initially treated by operative therapy experienced significantly fewer reinterventions than neonates initially treated with angioplasty (p < 0.05).
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Among the 23 patients initially treated by angioplasty, 3 experienced saccular aneurysms. These 3 patients were among the 8 receiving a second angioplasty.
Growth of Aortic Arch
At admission, both cohorts had a mild degree of distal arch hypoplasia as evidenced by the ratio of the diameter of the distal arch to the diameter of the ascending aorta approaching 0.5. However, neonates receiving surgical intervention experienced improved arch growth as evidenced by a significantly greater diameter of the distal arch to the diameter of the ascending aorta ratio (Fig 2). The follow-up in both cohorts was similar (group A, 34.5 months; group S, 32 months).
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Patients receiving two balloon angioplasties with subsequent aneurysm formation were noted to have a thin media with focal fibrosis, intimal proliferation, and fracture of the internal elastic lamina in the aneurysm region (Fig 3).
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One patient in the angioplasty group (4%) had acute tamponade secondary to wire perforation and required percutaneous pericardial drainage. Three additional patients (13%) experienced pulseless lower extremity that required heparin or tissue plasminogen activator before discharge. No neonate undergoing angioplasty required an operative procedure for limb ischemia.
Postoperative complications in the surgical group included reexploration for bleeding in 1 patient. Chylothorax occurred in 3 patients but responded to conservative dietary management and total parental nutrition. No patient experienced vocal cord paralysis or paraplegia.
| Comment |
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In this report, among 23 patients initially treated with angioplasty, only 4 patients (17%) required no further intervention. Eight patients required a second angioplasty procedure, which was effective in 75%, but 3 of these 8 patients had an aortic aneurysm. Nearly 60% of the angioplasty cohort required surgery, which is similar to reports by other investigators [79].
The mechanism for early restenosis in neonates after angioplasty may be related in part to elastic recoil constriction of ductal tissue surrounding the coarctation site. In addition, hypoplasia of the aortic isthmus proximal to the coarcted segment may result in impairment of blood flow into the dilated area and subsequently may induce late elastic recoil and recoarctation. Isthmus hypoplasia is associated with early restenosis and is a relative contraindication to balloon angioplasty for neonatal coarctation. It is likely that multiple factors including ductal tissue constriction or recoil, isthmus hypoplasia, intimal hyperplasia as a result of smooth muscle cell proliferation, and matrix protein production with arterial remodeling are involved in restenosis [10].
Femoral artery trauma and occlusion remains an additional concern especially in neonates. Although the umbilical artery was used for the initial intervention in the majority of our patients, subsequent angioplasty placed the femoral artery at increased risk. Thrombus formation not reversible with heparin or tissue-type plasminogen activator can result in irreversible occlusion of the femoral artery, potentially causing growth impairment of the ipsilateral leg. In our series, 3 patients experienced this complication, but fortunately all responded to anticoagulation and tissue-type plasminogen activator. Doppler echocardiography and magnetic resonance imaging are reliable noninvasive methodologies for detection of stenosis or occlusion, as clinical examination can be misleading in the presence of well-developed collateral circulation often observed in older children [11]. This complication may be underestimated because long-term analysis of lower extremity perfusion after balloon angioplasty was not undertaken in this series.
The frequency of aneurysm formation is another postinterventional complication that may be underestimated. In this report, significant aneurysms were only detected in the angioplasty population in 3 of 8 patients requiring a second balloon dilatation. However, none of our patients in either cohort have had late-term aortic imaging studies. Magnetic resonance angiography has proven to be an excellent noninvasive method for morphologic study of the aortic arch and is recommended for all patients after balloon angioplasty [12]. It allows examination in multiple planes and is therefore helpful in determining the presence of aneurysms and recurrent coarctation. Exactly how these patients with NAC should be imaged after their initial treatment has not been definitively established. From our experience, those having balloon angioplasty might require more asymptomatic imaging follow-up.
The surgical treatment of neonatal coarctation is highly effective and applicable to a wide variety of morphologic lesions, including discrete coarctation, long-segment coarctation, and coarctation associated with aortic arch hypoplasia. In this report, the 18% surgical recurrence rate was equally distributed throughout the 10-year study interval and is similar to other reported surgical series [1315]. The surgical technique (SFA or XETE) and the suture material used (polypropylene or absorbable polydioxanone) was equally distributed among the six recurrences. Balloon angioplasty was successful for the treatment of recoarctation in all patients, a finding consistent with other reports of balloon dilatation of recoarctation after surgery [16]. The hemodynamic result after balloon angioplasty was similar regardless of the initial surgical technique used. Periadventitial surgical scar had been thought to be protective against extensive vessel damage and aneurysm formation after balloon dilatation for postsurgical coarctation. However, this has not proven to be the case. Aneurysm formation has varied from 0% to 14% and emphasizes the need for close long-term follow-up. Other complications of balloon dilatation after recoarctation have included femoral artery trauma, 17%; neurologic damage, 2%; and postcoarctectomy syndrome, 2% [17]. The brief period of aortic interruption during dilatation did not result in neurologic damage to the spinal cord in any of our patients. The effectiveness and safety of this procedure has made balloon dilatation an attractive alternative to surgery, and it should be considered as the initial treatment option for the few patients with recoarctation after surgical intervention [18].
We believe that the primary treatment for NAC should be surgical. Of the 34 patients who received an optimal surgical repair, 28 have not required reintervention for coarctation and only 2 patients (7%) remain on antihypertensive medication as compared with 33% of the angioplasty cohort. Advances in surgical and anesthetic techniques and improved intensive care unit management have lowered hospital mortality for neonatal coarctation [19]. However, neonates have a 10% to 40% recoarctation rate, which is related to the migration of ductal tissue into the aortic wall longitudinally and circumferentially. Although not observed in this report, recoarctation is reported more often in neonates having SFA in which ductal tissue was retained [20]. However, we believe SFA still has an important place in coarctation surgery, particularly in certain critically ill neonates in whom extensive aortic dissection is not required and end-to-end repair might be associated with an increased operative risk. The XETE repair requires complete circumferential dissection of all aortic arch vessels and the descending thoracic aorta for three to five pairs of intercostal arteries to perform a tension-free anastomosis. The wider resection with the XETE anastomosis provides excellent coarctation relief with removal of all ductal tissue, and extending the incision into the concavity of the distal aortic arch relieves distal arch hypoplasia.
The incidence of distal aortic arch hypoplasia ranges between 50% and 80% [21]. Whether the arch grows after coarctation repair is debatable. Brouwer and associates [22] believe that the arch grows after simple coarctation resection using end-to-end anastomosis. Jahangiri and coworkers [23] believe SFA is an effective technique, and the aortic arch will grow in the majority of patients without additional procedures. Meyers and associates [24], however, demonstrated that a truly hypoplastic arch will not grow after SFA or simple end-to-end anastomosis. In this report, we demonstrate that the distal aortic arch does grow to a greater degree in those neonates having surgery as compared with balloon angioplasty. This is not surprising, as the majority of the surgery cohort received an XETE anastomosis, which is designed to relieve distal aortic arch hypoplasia. It is our belief that to avoid residual coarctation and recoarctation, the repair technique should remove all ductal tissue and relieve distal aortic arch obstruction. For this reason, the XETE anastomosis is our procedure of choice for neonatal coarctation.
We have compared balloon angioplasty and surgery for NAC. This comparison is inherently problematic because neonatal coarctation has a wide morphologic variability including proximal tubular isthmus and distal aortic arch hypoplasia, associated intracardiac defects that can influence initial treatment selection and early outcome. In our experience, surgical treatment is more often selected in patients with isthmus and distal aortic arch hypoplasia and long-segment coarctation. Similarly, available data in published reports make it difficult to objectively compare various methods of surgical therapy [25]. In addition to these weaknesses, the present study has certain limitations. The analysis between the two treatment modalities was retrospective, nonrandomized, and from a single institution. The cohorts were small, making statistical comparisons difficult, and mean follow-up was only 3 years.
In the neonatal population, cognizant of our study's limitations, our data suggest that the role of balloon angioplasty for NAC is palliative, with the majority of angioplasty patients requiring surgery within the first year of life. Balloon angioplasty, however, appears to offer excellent results for postsurgical coarctation and possibly as palliation in small, critically ill neonates with coarctation and associated medical conditions making them a nonsurgical candidate at the time of presentation.
| Requirements for Recertification/Maintenance of Certification in 2006 |
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The American Board of Thoracic Surgery will no longer publish the names of individuals who have not recertified in the American Board of Medical Specialties directories. The Diplomate's name will be published upon successful completion of the recertification/maintenance of certification process.
The CME requirements are 70 Category I credits in either cardiothoracic surgery or general surgery earned during the 2 years prior to application. SESATS and SESAPS are the only self-instructional materials allowed for credit. Category II credits are not allowed. The Physicians Recognition Award for recertifying in general surgery is not allowed in fulfillment of the CME requirements. Interested individuals should refer to the Booklet of Information for a complete description of acceptable CME credits.
Diplomates should maintain a documented list of their major cases performed during the year prior to application for recertification. This practice review should consist of 1 year's consecutive major operative experiences. If more than 100 cases occur in 1 year, only 100 should be listed.
Candidates for recertification/maintenance of certification will be required to complete all sections of the SESATS self-assessment examination. It is not necessary for candidates to purchase SESATS individually because it will be sent to candidates after their application has been approved.
Diplomates may recertify the year their certificate expires, or if they wish to do so, they may recertify up to two years before it expires. However, the new certificate will be dated 10 years from the date of expiration of their original certificate or most recent recertification certificate. In other words, recertifying early does not alter the 10-year validation.
Recertification/maintenance of certification is also open to Diplomates with an unlimited certificate and will in no way affect the validity of their original certificate.
The deadline for submission of applications for the recertification/maintenance of certification process is May 10 each year. A brochure outlining the rules and requirements for recertification/maintenance of certification in thoracic surgery is available upon request from the American Board of Thoracic Surgery, 633 N St. Clair St, Suite 2320, Chicago, IL 60611; telephone: (312) 202-5900; fax: (312) 202-5960; e-mail: mailto:info{at}abts.org. This booklet is also published on the website: www.abts.org.
| Discussion |
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I think that these data are reflective of other experiences and are stark support for condemning balloon dilatation in a neonate for the repair of neonatal coarctation. I don't think that many institutions around the country are actually doing this nowadays. And I think that we should be doing exactly what you are doing, publishing these results and reiterating the fact that this is not the intervention to perform for babies with neonatal coarctation.
I wonder if you can tell us, have your cardiologists abandoned this technique in neonatal coarctation? If they haven't, why haven't they? Also how hard was it to operate on these patients who have had aneurysm formation? Were there any special considerations that you would like to alert us to?
Thank you.
DR FIORE: Thank you very much, Dr Mavroudis. This particular intervention has been discontinued for neonatal coarctation at St. Louis University, but balloon dilation continues to have an important role in the treatment of older children and adolescents with coarctation of the aorta.
The aneurysms we have operated on have been 1 to 2 cm in diameter or less. Usually, they occur posteriorly or medially on the descending thoracic aorta at or above the ductal entry site. Applying clamps has not been difficult, but one needs to mobilize more aorta from each end to ensure an optimal anastomosis.
DR ROSS M. UNGERLEIDER (Portland, OR): Taking care not to err too much into the other extreme, there is selection bias here, and cardiologists perhaps have selection bias in thinking that all patients should be treated with interventional angioplasty techniques and surgeons need to be careful not to have the opposite selection bias that says that we should treat all patients with surgery. Forty-three percent of the patients with angioplasty ended up with an acceptable result, and I am curious if there are patients, you intimated this at the end of your talk with the patients that are critically ill, but could you go back and look at the patients that had a successful outcome and give us some idea of which patients might be considered to be good candidates for angioplasty?
The second question you just alluded to. In the patients who get angioplasty you saw aneurysms. We just talked about with the preceding paper that disruption of the intima can lead to aneurysm formation, and since that is the mechanism of effectiveness of balloon angioplasty, disruption or tearing of the vessel intima, perhaps you can tell us a little bit about what the aneurysms looked like in these patients as contrasted to the patients who have angioplasty for recurrent coarctation after surgery where you weren't seeing aneurysms. So is there a difference in what you were seeing, at least histologically, in these patients when you have operated on them?
A very important study, though, Andy, and you are to be congratulated for presenting these data.
DR FIORE: Thank you, Ross. In general, we do not feel that angioplasty is an appropriate procedure for neonates with aortic coarctation. However, as you point out, 43% of the patients required one or more angioplasties to yield an acceptable result. If a patient was considered for angioplasty, it should be beyond the neonatal period. The transverse aortic arch and isthmus segment should not be hypoplastic, and the coarctation site should be discreet.
With respect to your second question, let me show you a slide comparing the histology of a coarctation site in a patient who underwent surgery alone and compare that to a patient who underwent balloon angioplasty followed by surgery.
DR JOHN W. BROWN (Indianapolis, IN): Very nice study, Andy. In your institution is there a role for balloon angioplasty in the primary treatment of coarctation at any age?
DR FIORE: Well, not in neonates, but if you had a discrete coarctation in an older child, in whom there was no isthmus hypoplasia, which as you know is a component in restenosis after balloon angioplasty, and in whom no significant distal arch hypoplasia was present, you should consider balloon dilatation as a treatment option.
| Acknowledgments |
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| References |
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