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Ann Thorac Surg 1996;62:442-448
© 1996 The Society of Thoracic Surgeons
Divisions of Cardiothoracic Surgery and Cardiology, Children's Hospital Los Angeles, and Departments of Surgery and Pediatrics, USC School of Medicine, Los Angeles, California
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Abstract |
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Methods. Between July 1992 and January 1996, we have performed 24 aortic root replacements with the pulmonary autograft in pediatric patients (<18 years). Of this group, 8 were infants and children with complex LVOTO aged 9 days to 22 months (mean, 8.6 ± 8 months) and weighing 3.3 to 10.2 kg (mean, 6.3 ± 2.6 kg). The diagnoses were interrupted aortic arch/ventricular septal defect/subaortic stenosis in 3, recurrent aortic stenosis in 2, aortic stenosis and subaortic stenosis in 1, and aortic stenosis/subaortic stenosis/mitral stenosis/regurgitation in 2. All patients had undergone one to three previous operative procedures (mean, 1.5 ± 0.8 procedures/patient). Preoperative echocardiographic peak LVOT gradient was 71.7 ± 25 mm Hg (range, 40 to 110 mm Hg) and aortic annulus size was 7.2 ± 2.3 mm (range, 4 to 10.6 mm). The surgical technique included replacement of the aortic root with the pulmonary autograft combined with incision of the conal septum to relieve subaortic stenosis or accommodate for size discrepancy between the aortic and pulmonary autograft root and a pulmonary homograft placed in the right ventricular outflow tract.
Results. There were no perioperative or late deaths at follow-up (range, 2 to 25 months; mean, 13.5 ± 8 months). Mean hospital stay was 15 ± 17 days (range, 4 to 53 days). Three children had the following complications: diaphragmatic paresis (1), delayed pericardial effusion (1), and atrioventricular block requiring a pacemaker (1). In follow-up, echocardiographic findings showed absent aortic regurgitation in 3 and trivial aortic regurgitation in 5, and no significant LVOTO (mean peak gradient, 6.2 ± 7.6 mm Hg; range, 0 to 16 mm Hg). Pulmonary homograft regurgitation was absent in 5, trivial in 2, and moderate in 1. Peak right ventricular outflow tract gradient by echocardiogram was trivial in 7, and a significant gradient of 55 mm Hg has developed in 1 infant. There were no infective or embolic complications during follow-up.
Conclusions. Our experience shows that aortic root replacement with the pulmonary autograft can be performed in children with excellent clinical results. The technique of root replacement combined with ventriculoplasty allows definitive and simultaneous relief of complex and multiple-level obstructive lesions. Considering the growth potential of the pulmonary autograft, this should be regarded as the optimal treatment modality in infants with complex LVOTO.
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Introduction |
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TopFootnotesAbstractIntroductionMaterial and MethodsResultsCommentReferences |
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Children presenting with complex left ventricular outflow tract obstruction (LVOTO) represent a challenging medical and surgical problem. The treatment modalities are often palliative because the initial therapy frequently leaves residual lesions. More definitive approaches have involved valve replacement with mechanical prostheses, which require anticoagulation, or bioprostheses (aortic homograft), which have limited durability and no growth potential. Based on our positive experience with the Ross procedure in older patients, we have extended the technique of aortic root replacement with the pulmonary autograft to the population of children less than 24 months of age with complex left heart obstruction. The early results with this procedure are presented.
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Material and Methods |
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TopFootnotesAbstractIntroductionMaterial and MethodsResultsCommentReferences |
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). Eight patients (33%) were children aged less than 24 months with complex left heart obstruction, defined as the coexistence of multiple-level or recurrent obstructive lesions of the left heart.
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Clinical Status
Patients were in New York Heart Association class III or IV before their aortic root replacement.
Anatomic Diagnosis
The primary disease condition was interrupted aortic arch/ventricular septal defect/subaortic stenosis (IAA/VSD/SAS) in 3, recurrent aortic stenosis on bicuspid valve in 3, and critical aortic stenosis with mitral regurgitation in 2 (Table 2). Two patients with IAA/VSD/SAS presented with aortic valve regurgitation, persistence of interventricular communication, and arch obstruction. The third infant with IAA/VSD/SAS had undergone one-stage neonatal repair at our institution [1]. This patient also had a mildly stenotic bicuspid aortic valve with hypoplastic annulus, which progressed toward a moderate-to-severe lesion within 12 months. The 3 infants with critical neonatal aortic stenosis had received balloon valvuloplasty without lasting relief of the obstruction. Two patients with complex LVOTO and mitral regurgitation had a previous balloon valvuloplasty before the aortic root replacement (see Table 2).
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Operative Technique
Cardiopulmonary bypass was established using one aortic and two caval cannulas in 6 patients. Two cases in which a period of deep hypothermic (18°C rectal temperature) circulatory arrest was used for repair of the aortic arch had a single right atrial cannula. The remaining patients (n = 6) were cooled to a rectal temperature of 22° to 24°C before aortic cross-clamping. An initial dose of cold (0° to 4°C) blood cardioplegia was administered in the aortic root, before transection of the aorta well above the valve commissures. Continuous cold (0° to 4°C) saline perfusion of the pericardial sac was performed during the operation to maintain myocardial temperature between 4° and 8°C. In the children having reconstruction of the aortic arch (Table 4), the continuity of the arch was reestablished with an end-to-end posterior anastomosis of the native aorta and anterior augmentation with a pulmonary homograft after resection of the stenotic segment (recurrent coarctation in 2 patients, stenotic polytetrafluoroethylene graft in 1). The aortic valve was then excised and the two coronary buttons harvested. The main pulmonary artery was transected at the bifurcation and completely separated posteriorly from the left main coronary artery (Fig 2). The anterior surface of the right ventricular infundibulum was then incised transversely 4 to 5 mm below the valve level, and dissection of the posterior aspect of the pulmonary root was completed with care to avoid large septal arteries. No excess portion of the RVOT muscle was harvested to accommodate the completion of the septal ventriculoplasty. The subaortic region was then assessed and any fibrous membrane resected. The incision of the conal septum, starting at the level of the intercoronary commissure, was carried leftward to avoid the conduction system. The size of the ventriculoplasty was adjusted according to the severity of the LVOTO and the degree of size mismatch between the aortic and pulmonary autograft root. The continuity of the interventricular septum was reconstructed by one of two techniques. If a relatively shallow septal incision was sufficient to open the subaortic area, reconstruction was completed with the subpulmonary conal muscle of the autograft (n = 4). When a deeper incision into the septum was required, a Dacron patch was used (n = 4). In both instances repair was accomplished using Teflon-pledgeted mattress sutures along the muscular septum (see Fig 2). Repair of coexistent outlet VSDs (n = 2) was carried out at this time. A second dose of cold blood cardioplegia was infused directly into the coronary ostia to disclose any significant bleeding source at the level of the incised pulmonary valve. The proximal anastomosis of the pulmonary autograft was then completed with three 5-0 Prolene (Ethicon, Somerville, NJ) monofilament sutures, starting at each commissure and anchoring the autograft root onto the crest of the ventricular septum and aortic annulus. Reimplantation of the coronary arteries was done as buttons onto the right and left sinuses of the pulmonary valve using a running 7-0 Prolene monofilament suture. The RVOT was reconstructed using pulmonary homografts (range, 10 to 21 mm; mean, 15 ± 3 mm) and running 6-0 and 5-0 Prolene sutures for the distal and proximal anastomoses, respectively (Fig 3). Ascending aortic continuity was then reestablished with an end-to-end anastomosis between the distal pulmonary autograft and the ascending aorta with a running 6-0 Prolene suture.
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Associated procedures included repair of arch obstruction in 3 infants, modification of the septal ventriculoplasty to accommodate for concomitant VSD repair in 2, removal of a pulmonary artery band at the time of autograft harvesting in 1, and mitral valvuloplasty in 2.
Postoperative Follow-up
All patients underwent predischarge and follow-up transthoracic echocardiographic assessment. Echocardiographic examinations included quantitative and qualitative estimates of ventricular function, semilunar valve diameters and function, anatomy of the ventricular septum, and left heart-aorta complex.
Statistical Analysis
Data were expressed as means ± standard deviation. A two-tailed, paired Student's t test was used to compare continuous variables. Discrete variables were compared using Pearson's 
2 test. A p value less than 0.05 was considered significant.
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Results |
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Postoperative Course
The postoperative recovery in the intensive care unit included an average of 2 days (range, 1 to 14 days) of inotropic support with dopamine and dobutamine infusion and an average of 2 days of mechanical ventilation (range, 1 to 13 days). Prolonged (>2 days) inotropic and ventilatory support were necessary in 2 infants, 1 with previously palliated IAA/VSD/SAS who had been intubated and receiving an intravenous catecholamine infusion since the last surgical procedure. In addition, 1 patient also with previously palliated IAA/VSD/SAS experienced left hemidiaphragm paresis, confirmed by fluoroscopic examination. Function of the hemidiaphragm recovered after 13 days of mechanical ventilation, without the need for surgical plication. All children recovered regular sinus rhythm except 1 infant, in whom complete heart block developed after arch reconstruction, autograft root replacement, and mitral valvuloplasty. In this case, a permanent pacemaker was required followed by a mitral valve replacement. Three patients showed transient ST-T wave abnormalities, which were not detected at subsequent follow-up electrocardiograms. Cumulative hospital stay averaged 15 days (range, 4 to 53 days). Length of stay was a mean of 8.8 days for 7 of the 8 patients. One patient required a prolonged hospitalization (53 days) because of the need for subsequent mitral valve replacement (17-mm St. Jude Medical [St. Paul, MN]). Predischarge echocardiographic examination demonstrated absence of semilunar valve regurgitation or stenosis with normal biventricular kinetics in all patients (see Table 3). Six patients were discharged on oral furosemide and digoxin therapy and 2 infants required no discharge medications. One patient with previously palliated critical aortic stenosis presented with a low output syndrome 10 days after extended aortic root replacement with the pulmonary autograft. Emergency echocardiographic examination showed a large pericardial effusion that caused cardiac tamponade. The infant underwent surgical drainage through a subxiphoid window with immediate improvement in the hemodynamic status and recovered uneventfully.
Follow-up
There were no deaths or reinterventions during an average follow-up of 13.5 ± 8 months (range, 2 to 25 months). Clinical assessment of patients demonstrated appropriate weight gain in all infants and significant improvement in functional status (Table 3). No infective or embolic events were reported by the families or referring physicians following up the infants.
The echocardiographic data demonstrated lasting resolution of the LVOTO with peak pressure gradients ranging from 0 to 16 mm Hg (mean, 6.2 ± 7.6 mm Hg) (Fig 4; see Table 3). Aortic regurgitation was absent in 3 patients and trivial or mild in 5. Comparing the diameter of the aortic annulus at the early postoperative and follow-up examination, there was aortic root enlargement in proportion to somatic growth suggesting growth of the autograft (Table 5). Five infants had absent pulmonary regurgitation, 2 had trivial regurgitation, and 1 had moderate regurgitation. In 1 infant significant pulmonary homograft obstruction has developed during follow-up, with an estimated gradient of 55 mm Hg by echocardiogram. The remaining 7 patients have no demonstrable RVOT gradients. Left and right ventricular function were normal in all patients.
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Comment |
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Critical congenital aortic valve stenosis presenting in the neonate is far from a benign lesion. A recent review of multiinstitutional experience with the invasive treatment of neonatal aortic stenosis reported an early mortality exceeding 50% and late mortality approaching 20% of survivors [2]. Thus, only 2 of 5 neonates initially treated for this lesion will survive up to 15 years. Furthermore, nearly 100% of long-term survivors will require reintervention, in half of the patients consisting of aortic valve replacement with or without aortic root enlargement procedures [2]. Even more complex are the infants presenting with multiple levels of LVOTO, which may include the mitral valve, subaortic region, aortic valve, and aortic arch as represented by 3 infants in this series. These infants may have even higher mortality than infants with critical aortic stenosis.
Traditional operative strategies applied to recurrent aortic stenosis in babies have included multiple balloon valvuloplasty and open valvotomy procedures to postpone the problem of valve replacement to childhood or adolescence. Often this leaves the child with residual lesions resulting in a compromised left ventricle when definitive repair is undertaken. Repairs have included valve replacement techniques, which often require annulus enlargement [3–6]. However, analysis of the clinical results with such techniques, including the Nicks posterior patch annuloplasty, the Manougian mitral–aortic annuloplasty, and the Konno aortoventriculoplasty, when used in the child, has demonstrated disappointingly high mortality and morbidity [7]. Alternative operations have also been proposed for the older child but not yet successfully reported for the infant [8, 9]. In addition, all of the above-mentioned options maintain the disadvantage of tissue degeneration (bioprostheses), the devastating consequences of thromboembolic and hemorrhagic complications (mechanical prostheses), and the lack of growth potential requiring reoperation [7, 10]. Similar conclusions must be drawn when performing these operations for associated aortic valvular and subvalvular stenosis.
Alternative surgical strategies advocating extraanatomic bypass of the obstruction with left ventricle-aorta conduits have assumed primarily historical importance, given the precarious palliation offered and the unsolved problem of reoperation [11]. Bypassing the obstructed left ventricular outflow tract using the Norwood approach has gained some recent favor; however, this operation predisposes these infants to univentricular physiology [12]. In spite of the satisfactory early survival reported with this approach, justification of its application becomes difficult in the presence of two well-developed ventricular chambers, as in IAA/VSD. In fact, as the morbidity and intraprocedural mortality of the Norwood staged palliation is still very high, various techniques of biventricular one-stage repair seem justified [1, 13].
A recent advancement in the surgical management of complex LVOTO in the child has come from the idea of combining the extended aortic root replacement offered by the Konno aortoventriculoplasty with the implantation of aortic homografts [14]. Although this is effective in relieving the obstructive lesions, the vulnerability of the aortic homograft to early degeneration predisposes the child to the need for repeat operations [15]. With the expected longer duration of the pulmonary autograft in the aortic position, we, like others, have combined the aortoventriculoplasty technique with pulmonary autograft insertion [16–20]. Encouraged by our preliminary results and faced with a growing population of infants with complex LVOTO for whom alterative options have shown dismal outcome, we have applied the extended aortic root replacement with the pulmonary autograft to this unusual patient population.
Pulmonary Autografts in Infants
Clinical experience with the use of pulmonary autografts in children is currently limited. Preliminary results have been encouraging except in patients with important endocardial fibroelastosis [19–24]. Our data suggest that aortic root replacement with the pulmonary autograft is a safe operation even in young infants with body weights between 3 and 10 kg.
Although our current approach to critical aortic stenosis in the neonate includes attempts at percutaneous or surgical valvotomy, and the approach to IAA/VSD/SAS complex involves one-stage primary repair [1], we have been largely satisfied with the present technique of aortic root replacement when valvular and subvalvular lesions recur.
Several technical aspects are important to the intraoperative and perioperative clinical course, including routine administration of aprotinin and myocardial protection using cold induction and maintenance followed by warm reperfusion blood cardioplegia. Despite relatively long duration of aortic cross-clamp and cardiopulmonary bypass, especially in patients undergoing associated procedures, recovery of mechanical and electrical cardiac activity was prompt in all cases.
Ventricular septoplasty by means of incision and resection of the conal septum and any other obstruction at the aortic subvalvular level may be completed by reconstruction of the septum either with Dacron or by implanting the autograft lower down in the aortic root. Unlike other authors [17, 24], we have avoided harvesting additional muscle from the RVOT free wall to accommodate the septoplasty. Although the use of prosthetic material potentially exposes the patient to infectious complications, we believe the untoward effects of extended right ventricular resection in a small child in terms of contractility far outweigh its expected benefits. Our data also suggest that aortoventriculoplasty using pulmonary autografts can be performed in children with a very low rate of complications, including postoperative conduction disturbances (12% in our series) previously reported as 7% to 8% [17, 24].
Functional Results
Analysis of midterm results in this series of infants and children undergoing aortic root replacement with pulmonary autografts parallels previous reports on older children and adolescents [16, 17, 21]. Both the autograft and the homograft valves are functioning well with the exception of one pulmonary homograft with a demonstrable stenosis by echocardiography. Contrary to other authors [17], we have not observed any tendency toward progressive autograft root dilatation in our patients, although we recognize the short-term follow-up in this group of infants.
Furthermore, the aortoseptoplasty operation has resulted in lasting normalization of left ventricular outflow tract anatomy and flow. In spite of the complexity of the procedure, postoperative and follow-up ventricular function have shown prompt recovery, as previously reported by others [20].
Finally, the present surgical approach would represent the most physiologic procedure if appropriate autograft valve growth were clearly demonstrated. The major criticism to the pioneering experience of Ross and associates in children is that autograft growth has never been proven [19]. More recent experience by Elkins and coworkers [21] has shown enlargement of the autograft root with time, which is proportional to somatic growth. Once again, the infant and young child represent an unusual subset of patients in that the somatic growth is rapid in the baby and proportional autograft enlargement should be demonstrable. The preliminary data of our experience would tend to confirm autograft enlargement that parallels somatic growth (see Table 5). We are therefore looking with great expectations at this population as the most dependable clinical model to prove or disprove the growth of autograft valves.
We recognize there are limitations of this study. The follow-up is short, and meaningful conclusions are yet to be determined. The use of a pulmonary homograft for RVOT reconstruction is likely to expose the patient to at least one reoperation to accommodate for lack of growth of the cadaveric implant. However, as the choice of slightly oversized pulmonary homografts (average diameter, 15 mm) may delay the timing of reoperation, the durability of pulmonary homografts has yet to be determined. Finally, as with all clinical applications of the autograft valve, one should remember that our operation exposes two valves to risk during follow-up.
Conclusions
The population of young children with complex LVOTO have a uniform lack of valid therapeutic alternatives. The present experience with aortoventriculoplasty using pulmonary autografts offers excellent early survival and functional outcome. Considering the freedom from prosthetic valve-related complications, wider use of this technique is recommended. If growth of the autograft valve were to be confirmed by longer follow-up, this operation could become the ultimate surgical therapy for left-heart obstruction in infants and young children.
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Footnotes |
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TopFootnotesAbstractIntroductionMaterial and MethodsResultsCommentReferences |
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Address reprint requests to Dr Starnes, Division of Cardiothoracic Surgery, USC School of Medicine, Childrens Hospital Los Angeles, 4650 Sunset Blvd MS 66, Los Angeles, CA 90027.
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TopFootnotesAbstractIntroductionMaterial and MethodsResultsCommentReferences |
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