C in its etiology but also in regard to the optimal

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1 Role of Extended Aortoplasty Related to the Definition of Coarctation of the Aorta Joseph J. Amato, MD, Ralph J. Galdieri, MD, and Joseph V. Cotroneo, MD Pediatric Cardiothoracic Surgery, Schneider Children s Hospital, Long Island Jewish Medical Center, Albert Einstein College of Medicine, New Hyde Park, New York, and Pediatric Cardiovascular Surgery, Children s Hospital of New Jersey, United Hospitals Medical Center, University of Medicine and Dentistry of New Jersey, Newark, New Jersey One hundred thirty-nine patients underwent operation for coarctation of the aorta. Age ranged from 1 day to 1 years and weight, from 1.5 to 7.4 kg. Numerous methods of repair were used. The operative mortality was low (1.3%), and 17 patients (11.3%) died late. Recoarctation occurred in 13 patients (9.4%). We attempted to correlate mortality and recoarctation with the surgical procedure. A review of the literature revealed no classifications of coarctation that applied to the anatomical and pathological variations we found at the time of operation. Therefore, we devised a surgical classification to separate the various entities in the spectrum of coarctation: type I = primary coarctation; type I1 = coarctation with isthmus hypoplasia; and type 111 = coarctation with tubular hypoplasia involving the isthmus and segment between the left carotid and left subclavian arteries. Each of these types has subtypes: A = with ventricular septal defect and B = with other major cardiac defects. We believe that rather than labeling one procedure as the procedure of choice, providing this classification will allow the surgeon to use a method of repair that is suited to the anatomical variation. ( 1991;5:15-) oarctation of the aorta remains controversial not only C in its etiology but also in regard to the optimal method of repair. This condition, especially in neonates, remains an enigma because of the high rates of recoarctation and mortality. Thus, a multitude of techniques including end-to-end anastomosis, subclavian flap turndown, subclavian angioplasty, and extended end-to-end anastomosis have been compared. It is our belief that the anatomical classification of coarctation, presented to the surgeon in this spectrum of pathological entities, has not been adequately addressed. The purpose of this report is threefold: to present a surgical classification of the coarctation spectrum; to review our data on coarctation repair and discuss the correlation of recoarctation and anatomical operative findings; and to discuss indications for the various operative techniques and some disadvantages of each method. Material and Methods From January 1979 to September 199, there were 151 procedures performed on 139 patients, 85 male and 54 female. Age ranged from 1 day to 1 years and weight, from 1.5 kg to 7.4 kg. Of the 139 patients, 88 had no associated anomalies, 7 had ventricular septal defect, and 4 had other major cardiac defects. Patent ductus Accepted for publication Dec 31, 199. Address reprint requests to Dr Amato, Schneider Children s Hospital-37, Long Island Jewish Medical Center, New Hyde Park, NY 114. arteriosus and atrial septal defect were not considered associated anomalies. Our series of 139 patients comprised newborns. Twenty-four of them were seen with only coarctation, 1 had coarctation with associated ventricular septal defect, and 1 had coarctation with other associated complex defects. Therefore, 4 (3.%) of the neonates had associated anomalies. Of the 151 procedures performed in the overall series, 43.7% (/151) were on neonates less than 1 month of age, 53.% (81/151) were on infants less than 3 months of age, and.3% (91/151) were on infants less than 1 year of age (Fig 1). The surgical repair included multiple techniques (Fig ). The end-to-end repair, as first described by Crafoord and by Gross, was performed on 7 patients (17.9%). The subclavian flap turndown repair, introduced by Waldhausen in 19, was done on 79 patients (5.3%) and the subclavian flap turnup, on 4 (.%). An interposition graft for replacement of the entire coarctation segment was used in 3 adolescents (.%). Enlargement of the coarctate segment was accomplished with a patch, as described by Vosschulte in 1957, of either Dacron or polytetrafluoroethylene in 3 patients (15.%) or with a free segment of subclavian artery in 3 (.%). The technique of subclavian reimplantation, described by de Mendonca and colleagues [l] in 1985, was used in 4 patients (.%), 3 newborns and 1 -year-old child. Other angioplasties performed in conjunction with an end-to-end anastomosis included subclavian flap turnup in (4.%), subclavian flap turndown in 1 (.7%), and subclavian reimplantation in 1 (.7%). This last angioplasty procedure was presented 1991 by The Society of Thoracic Surgeons /91/$3.5

2 1 CONGENITAL HEART AMATO ET AL 1991;51% 38 PROCEDURES 7 AGE AT TIME OF REPAIR 43.7% ( procedures) less than 1 month 53.% (81 procedures) less than 3 months 5.% (91 procedures) les, than a year of age 4 ill LO 1 <lm 1-3m 4-m 7-1 >l-loy >1Oy Fig 1. Age of the 151 patients at the time of surgical repair. by one of us (J.J.A.) in 1977 []. The age group for each of these repairs is shown in Table 1. The suture material used for the anastomoses beginning in 1979 was 5- or - Prolene (Ethicon, Somerville, NJ). In 198, we converted to monofilament absorbable suture. At first we used polydioxanone (PDS; Ethicon), but technical difficulty with handling the memory of the suture and inability to tighten the suture line led to the use of polyglyconate (Maxon; Davis + Geck, Danbury, CT) in Continuous suture technique was used in all anastomoses. 37 Results Mortality The operative mortality rate was 1.3% ( patientdl51 procedures). One of the patients who died was a newborn not considered to be in the classification of hypoplastic left heart syndrome, although a hypoplastic aortic arch, unrecognized severe aortic stenosis, and mild hypoplasia of the left ventricle were present. This patient died of low cardiac output on the night of operation. The other patient who died was a newborn in whom coarctation of a right-sided aortic arch was diagnosed preoperatively. At postmortem examination, an associated vascular ring was discovered. During the end-to-end anastomosis, the patient became acidotic because of poor ventilation and died in the operating room. The late mortality rate was 11.3% (17 patientdl51 procedures). Eight patients (5.3%) died at a later operative correction. Causes of death not related to the initial operation in the other 9 patients (.%) included sepsis (4), meningitis (l), biliary infection (l), and respiratory problems (). Recoarcta tion Recoarctation, by definition, is recurrence of coarctation or restenosis. Some authors [3] have stated that recoarctation is defined as a resting gradient of 1 mm Hg or more between the arm and the leg, but others have chosen a gradient of mm Hg [4] or even 3 mm Hg [5]. An exact clinical determination of the gradient is difficult. At Schneider Children s Hospital, recoarctation has been END-END INTERPOSITION GRAFT PATCH REPAIR SUBCLAVIAN FLAP REPAIR - TURNDOWN SUBCLAVIAN FLAP TURNUP SUBCLAVIAN FLAP REPAIR + END-END SUBCLAVIAN REIMPLANTATION Fig. Methods of repairing coarctation of the aorta at Schneider Children s Hospital. SUBCLAVIAN REIMPLANTATION + END-END ANASTOMOSIS AMATO REPAIR 1977

3 1991;5:15- CONGENITAL HEART AMATO ET AL 17 Table 1. Types of Repair for Coarctation of the Aorta' SFTU SR SFTD Age E-E SFTD SFTU Patch IG E-E SR E-E E-E Total <1 Month Months 4- Months Months >1-1 Years 1 >1 Years 3 Total 7 (17.9) (5.3) 4 (.) Repair (17.) (.) 5 1 (4.) (.) (.7) (.7) (1) a Numbers in parentheses are percentages. E-T = end-to-end anastomosis; IG = interposition graft: SFTD = subclavian flap turndown; SFTU = subclavian flap turnup; SR = subclavian reimplantation. defined as a gradient of mm Hg or greater between the right arm and the legs. Thirteen (9.4%) of the 139 patients in this series had recoarctation (Table ). When we analyzed recoarctation by type of repair (Table 3), only 1 of the 7 patients with end-to-end anastomosis had recurrence of coarctation. Five of the 79 patients with subclavian flap turndown needed a second procedure. In 3 patients whose anatomy did not lend itself to a subclavian flap turndown, a free patch graft of subclavian artery was used. There was Table. Patients with Restenosis of Coarctation Repair Age at First Patient Repair Interval Second No. Original Repair (days) (mo) Repair 1 E-E 13 8 SFTD SFTD 4 Dacron patch 3 SFTD 1 15 graft Dacron patch graft 4 SFTD 1 14 E-E 5 SFTD 4 E-E SFTD 15 E-E 7 SFTU Gore-Tex 8 SFTU + E-E 3 patch Gore-Tex patch 9 SFTU + E-E 5 Dacron patch 1 Free subclavian 11 5 E-E artery patch 11 SR 9 9 SFTD 1 SR 14 Unknown" 13 SR 1... Late death a This repair was done at another center. E-E = end-to-end anastomosis; SFTU = subclavian flap turnup; SFTD = subclavian flap turndown; SR = subclavian reimplantation. recoarctation in 1 patient. Six patients had a long hypoplastic segment between the subclavian and left carotid arteries for which a subclavian turnup with end-to-end anastomosis was performed; (33%) had recoarctation. The most devastating results were seen with the subclavian reimplantation technique; 3 (75%) of the 4 patients had restenosis. The 3 affected were newborns, whereas the fourth was a -year-old child who did well. Of the 4 patients having a subclavian flap turnup, 1 infant (5%) had recoarctation. No recoarctation occurred in the 3 patients having patch repair with Dacron or polytetrafluoroethylene or in the 3 with an interposition graft. No patient in the patch repair group had aneurysm formation. The l patient who had subclavian reimplantation with end-to-end repair, as reported in 1977 [], did not Table 3. Restenosis by Type of Repair" No. of Repair Procedures Restenosis E-E 7 1 (3.7) SFTD 79 5 (.3) SFTU + E-E (33.3) Patch 3 () SR + E-E 1 () IG 3 () Free subclavian graft 3 l(33.3) SR 4 3 (75) SFTU 4 1 (5) SFTD + E-E 1 () Total procedures Total patients (9.4) a Numbers in parentheses are percentages. E-E = end-to-end anastomosis; IG = interposition graft; SFTD = subclavian flap turndown; SFTU = subclavian flap turnup; SR = subclavian reimplantation.

4 18 CONGENITAL HEART AMATO ET AL 1991;5:15- have recoarctation, nor did the patient who had subclavian flap turndown with end-to-end anastomosis. Comment Since the first repair of coarctation by Crafoord and by Gross, there has been considerable controversy about the choice of operation for coarctation, particularly in newborns. We analyzed our data to try to determine critical factors for low operative mortality and low incidence of recurrence. In the last two decades, numerous authors judged the end-to-end anastomosis to be less favorable than the Waldhausen subclavian flap turndown. Within the last 5 years, however, there has been a resurgence of favor toward the end-to-end anastomosis. To compare results, numerous methods have been used to determine the "success" of the operations. In some reports [, 71, patients were placed into three anatomical groups based on the presence of "pure" coarctation, coarctation with ventricular septal defect, and coarctation with associated anomalies [8, 91. However, these groups addressed mortality statistics rather than recoarctation. Bergdahl and colleagues [lo] chose to place their patients into three anatomical groups and three functional groups. The anatomical groups again were divided into pure coarctation, coarctation with ventricular septal defect, and coarctation with major anomalies. Ziemer and associates [8], after analyzing the data on patients with either end-to-end anastomosis or subclavian flap turndown repair, came to no conclusion as to the merits of either technique. Others grouped patients according to age to determine the procedure of choice: the first month [9], the first 3 months [ll], the first months [1], and the first year [13]. In reviewing the literature, we began to question methods that grouped patients by age or anatomy, namely, pure coarctation, coarctation with ventricular septal defect, or coarctation with other associated cardiac anomalies. We searched the literature for various classifications of coarctation of the aorta. It became apparent that, from the first description of coarctation in 17 by Morgagni to that of Ho and Anderson [14] in 1979, the classifications were ideal to fit the needs of the anatomist, pediatrician, or pathologist. Waldman and co-workers [13] stated in 1983 that the choice of operation should be based on anatomy rather than personal preference. In 1985, Pellegrino and colleagues [ 151 defined an anatomicopathological study with respect to treatment. Several types of obstructive lesions, which increased in severity from a shelflike lesion in coarctation to tubular hypoplasia, were noted. The feasibility of correlating surgical repair with the anatomy was discussed, and it was stated that such a study would have value only if the information obtained was applicable to clinical practice. It seems logical that repair of coarctation be based primarily on the anatomical variations seen in the aorta. Therefore, we composed a classification for coarctation of the aorta that allows the surgeon to choose a suitable operation based on the anatomy with which he or she is presented at the time of operation. We also determined A B C Fig 3. (A) Surgical classification of coarctation: type I = primary coarctation with or without patent ductus arteriosus with no other cardiac dejects; 1A = with ventricular septaal defect; and 1B = with other major cardiac defects; (B) type 11 = coarctation with isthmus hypoplasia with or without patent ductus arteriosus; ZIA = with ventricular septal defect; and IIB = with other major cardiac defects; (C) type Ill = coarctation with tubular hypoplasia involving isthmus and segment between the left carotid and left subclavian arteries with or without patent ductus arteriosus; IIlA = with ventricular septal defect; and IIlB = with other major cardiac defects. that together with the anatomical variations, the classification should be amplified to consider the associated lesions and thus the increased risk of death. Our pro-

5 1991;5:15- CONGENITAL HEART AMATO ET AL 19 I Table 4. Analysis of Late Deaths in Terms of Proposed Classification" Deaths At Later No. of Open Heart Other Classificationb Procedures Operation Causes I IA IB I1 IIA IIB 111 IIIA IIIB Total a Numbers in parentheses are percentages. of classification. See text for explanation posed surgical classification of coarctation of the aorta is as follows: IA IB Type I1 IIA IIB Type I11 IIIA IIIB Primary coarctation with or without patent ductus arteriosus (Fig 3A) With ventricular septal defect With other major cardiac defects Coarctation with isthmus hypoplasia with or without patent ductus arteriosus (Fig 3B) With ventricular septal defect With other major cardiac defects Coarctation with tubular hypoplasia involving isthmus and segment between left carotid and left subclavian arteries with or without patent ductus arteriosus (Fig 3C) With ventricular septal defect With other major cardiac defects With this classification in mind, we analyzed our 151 procedures with regard to mortality. Our early mortality of 1.3% comprised patients who would be considered type IB. The classification of the 17 patients who died late Fig 4. Comparison of (A) Amato repair of 1977 and (B) proposed Meier repair of is described in Table 4. We also retrospectively reviewed the cases of our patients with recoarctation in terms of our classification. The 13-day-old neonate with the end-to-end repair belonged in type I1 and most likely should have had a, subclavian flap turndown. The 5 patients who had an initial subclavian flap turndown belonged in type 11, and should have had either a longer segment of the subclavian flap turned down or, preferably, an end-to-end resection combined with subclavian flap turndown. The patient with a subclavian flap turnup and the patients with a subclavian flap turnup yith end-to-end anastomosis belonged in type I11 and, perhaps, needed a more extensive resection of the coarctation segment before the end-to-end anastomosis. We believe in the theory of Skoda as described by Wielenga and Dankmeijer [1] and advanced by others [14, 171. The patient with a free subcla1jan artery graft belonged in type I1 and should have had a subclavian flap turndown with an end-to-end anastomosis. The 3 patients with subclavian displacement require discussion. Isthmosubclavioplasty was described by de Mendonca and associates [l] in 1985 in a case report. In 198, Meier and co-workers [18] reported that this technique was used in 8 patients with good results, but only patients were less than 3 months old. The 3 neonates in our series, who were 9, 1, and 14 days old, all had recoarctation. Other authors [ 19-1] have also reported recoarctation when the de Mendonca technique was used. Meier [] subsequently presented a modification of this technique to prevent recoarctation in newborns by suggesting end-to-end resection combined with reimplantation of the subclavian artery. This type of subclavian angioplasty was described by one of us (J.J.A.) in 1977 [] (Fig 4); and Binet [3], in 198, referred to this operation as the Amato technique. Several other techniques followed the principles of the Amato technique, namely, by enlarging the hypoplastic segment combined with an extended end-to-end anastomosis [4]. A similar procedure was described as a new technique by Diet1 and Torres [5] in We believe that the Amato technique or its modification should be used in infants with type I11 coarctation with tubular hypoplasia. Conclusion We have presented a surgical classification for coarctation of the aorta by which the surgeon can correlate the anatomy of each patient with the operation performed. There should be no preconceived ideal or classic procedure. In many infants, especially newborns, type I-dis- Crete coarctation4an be repaired equally well with an end-to-end anastomosis or a subclavian flap turndown or a patch repair. We do not recommend the free patch graft of subclavian artery, as the patch has a tendency to retract and cause recoarctation. Type 11-coarctation with isthmus hypoplasia-can be repaired preferably and easily by a subclavian flap turndown or a subclavian flap turndown with end-to-end anastomosis; or with extended resection, it can be managed with an end-to-end anastomosis. If a patch is used, we recommend that the posterior ridge of I

6 CONGENITAL HEART AMATO ET AL 1991;5:15 the coarctation not be resected because this could weaken the wall and lead to aneurysm formation []. Type 111-coarctation with tubular hypoplasia involving the isthmus and segment between the left carotid and the left subclavian arteries-is the most difficult to repair, especially as there are some [7] who believe that the hypoplastic segment might grow; there is no evidence to establish this. We agree that some hypoplastic segments will grow, but the likelihood of occurrence cannot be predicted. As the best repair, we believe that an extended angioplasty using the left subclavian artery as part of the patch repair or the technique of resection and extended anastomosis of the transverse arch, which was derived from our technique and endorsed by others [3], could be used. We are concerned, however, about the subclavian turnup with end-to-end anastomosis because of patients had recoarctation. Our conclusion is that subclavian reimplantation should not be used without an end-to-end resection in newborns. This technique, however, might be feasible in children more than 1 year of age. We propose that the anatomicopathological condition or aortic arch in each patient should dictate the method of repair. There is no one procedure that we would label the procedure of choice. References 1. De Mendonca JT, Carvalho MR, Costa RK, Filho EF. Coarctation of the aorta. A new surgical technique. J Thorac Cardiovasc Surg 1985;9: Amato JJ, Rheinlander HF, Cleveland RJ. A method of enlarging the distal transverse arch in infants with hypoplasia and coarctation of the aorta. 1977;3: Williams WG, Shindo G, Trusler GA, Dische MR, Olley PM. Results of repair of coarctation of the aorta during infancy. J Thorac Cardiovasc Surg 198;79: Hartmann AF Jr, Goldring D, Hernandez A, et al. Recurrent coarctation of the aorta after successful repair in infancy. Am J Cardiol 197;5: Beekman RH, Rocchini AP, Behrendt DM, Rosenthal A. Reoperation for coarctation of the aorta. Am J Cardiol 1981; 48: Penkoske PA, Williams WG, Olley PM, et al. Subclavian arterioplasty. Repair of coarctation of the aorta in the first year of life. J Thorac Cardiovasc Surg 1984;87: Harlan JL, Doty DB, Brandt B 111, Ehrenhaft JL. Coarctation of the aorta in infants. J Thorac Cardiovasc Surg 1984;88: Ziemer G, Jonas RA, Perry SB, Freed MD, Castaneda AR. Surgery for coarctation of the aorta in the neonate. Circulation 198;74(Suppl 1): Nair UR, Jones, Walker DR. Surgical management of severe coarctation of the aorta in the first month of life. Review of 48 consecutive cases. J Thorac Cardiovasc Surg 1983;8: Bergdahl LAL, Blackstone EH, Kirklin JW, Pacific AD, Bargeron LM Jr. Determinants of early success in repair of aortic coarctation in infants. J Thorac Cardiovasc Surg 198; 83: 73& Cobanoglu A, Teply JF, Grunkemeier GL, Sunderland CO, Starr A. Coarctation of the aorta in patients younger than three months. A critique of the subclavian flap operation. J Thorac Cardiovasc Surg 1985;89: Moulton AL, Brenner JI, Roberts G, et al. Subclavian flap repair of coarctation of the aorta in neonates. Realization of growth potential? J Thorac Cardiovasc Surg 1984;87: Waldman JD, Lamberti JJ, Goodman AH, et al. Coarctation in the first year of life. Patterns of postoperative effect. J Thorac Cardiovasc Surg 1983;8: Ho SY, Anderson RH. Coarctation of the aorta. In: Godman MS, Masquis RM, eds. Paediatric cardiology; vol : Heart disease in the newborn. Edinburgh: Churchill Livingstone, 1979: Pellegrino A, Deverall PB, Anderson RH, et al. Aortic coarctation in the first three months of life. An anatomopathological study with respect to treatment. J Thorac Cardiovasc Surg 1985;89: Wielenga G, Dankmeijer J. Coarctation of the aorta. J Pathol Bacteriol 198;95: Elzenga NJ, Gittenberger-de-Groot AC. Localised coarctation of the aorta. An age dependent spectrum. Br Heart J 1983; 49: Meier MA, Lucchese FA, Jazbik W, Nesralla IA, Mendonca JT. A new technique for repair of aortic coarctation. Subclavian flap aortoplasty with preservation of arterial blood flow to the left arm. J Thorac Cardiovasc Surg 198;9: Nawa A, Nakayama Y, Teramoto S, Mori K, Dohi T. Coarctation restenosis after isthmosubclavioplasty. A consideration on operative procedure and intraluminal balloon angioplasty. Chest 1989;95: Ladusans EJ, Campalani G, Parsons JM, et al. Recurrence of aortic coarctation following repair by reimplantation of the subclavian artery. Int J Cardiol 1989;3: Messmer BJ, Minale C, Muhler E, v. Bernuth G. Surgical correction of coarctation in early infancy: does surgical technique influence the result? 1991;5: Meier MA. Discussion of DeSanto A, Bills RG, King H, Waller 8, Brown JW. Pathogenesis of aneurysm formation opposite prosthetic patches used for coarctation repair. An experimental study. J Thorac Cardiovasc Surg 1987;94:7G3. 3. Binet JP. Discussion of: Meier et a1 [17]. 4. Lacour-Gayet F, Planche C, Bruniaux J, et al. Surgical repair of coarctation in 1 infants less than three months of age by resection and extended anastomosis of the transverse aortic arch. In: Crupi G, Pavenzan L, Anderson RH, eds. Perspectives in pediatric cardiology; vol, pt I. Mt. Kisco, NY: Futura, 1989:8P7. 5. Diet1 CA, Torres AR. Coarctation of the aorta: anastomotic enlargement with subclavian artery: two new surgical options. 1987;43:4-5.. Hehrlein FW, Mulch J, Rautenburg HW, Schlepper M, Scheld HH. Incidence and pathogenesis of late aneurysms after patch graft aortoplasty for coarctation. J Thorac Cardiovasc Surg 198;9:&3. 7. Siewers RD, Ettedgui J, Pahl E, Tallman T, del Nido PJ. Coarctation and hypoplasia of the aortic arch: will the arch grow? 1991;5:8-14.