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Ann Thorac Surg 1995;59:1441-1447
© 1995 The Society of Thoracic Surgeons

Univentricular Heart With Systemic Outflow Obstruction: Palliation by Primary Damus Procedure

Heart Unit, Birmingham Children's Hospital, Birmingham, England

Accepted for publication February 10, 1995.

	Abstract

Top Footnotes Abstract Introduction Patients and Methods Results Comment References

 
In 24 consecutive infants (19 male and 5 female) with complex forms of single-ventricle physiology and systemic outflow obstruction, a modified Damus operation without the use of exogenous material was undertaken in conjunction with creation of an aortopulmonary shunt 3.5 mm in diameter. The median age at operation was 6 days (range, 1 to 170 days) and the median weight, 3.4 kg (range, 2.6 to 4.6 kg). There were nine early deaths. All 15 survivors (median follow-up, 6.5 months) were clinically well without major systemic ventricular dysfunction or atrioventricular or arterial valve regurgitation. Ten of them have undergone a superior vena cava-pulmonary shunt (one death), and 1 has required patch angioplasty of the aortic arch and innominate artery with revision of the aortopulmonary shunt. The 4 other survivors are awaiting a cavopulmonary shunt. Univariate analysis yielded the chronologic rank for an individual procedure (higher risk of death early in the series), presence of aortic arch atresia, and presence or absence of transposition of the great arteries as predictors of death. This aggressive surgical approach provides excellent early palliation, and because the operation prevents abnormal ventricular hypertrophy from pressure or volume overload, systemic ventricular function is optimally conserved for a future Fontan-type procedure.

	Introduction

Top Footnotes Abstract Introduction Patients and Methods Results Comment References

 
The optimal surgical approach for neonates with complex single-ventricle physiology and actual or potential systemic outflow obstruction (subaortic or supraaortic) is unclear. Early relief of subaortic stenosis and reduction of pulmonary blood flow are essential to preserve the option of a Fontan-type repair in the future. Pulmonary artery banding is suboptimal, as it accelerates the progression of subaortic stenosis and promotes ventricular hypertrophy, leading to decreased ventricular compliance and increased risk for the Fontan procedure [1–4]. In some cases of aortic atresia in association with a ventricular septal defect, conventional pulmonary artery banding is not possible. On the other hand, primary reconstruction of the outflow tract and aortic arch has met with increasing success in infants with hypoplastic left heart syndrome [5]. We report the results of a modified Damus-Kaye-Stansel procedure [6] as the initial palliation for this difficult group of infants.

	Patients and Methods

Top Footnotes Abstract Introduction Patients and Methods Results Comment References

 
Over a 3-year period to September 1994, 24 consecutive infants (19 male and 5 female) with complex forms of single-ventricle physiology associated with subaortic stenosis and unrestricted pulmonary blood flow but without classic hypoplastic left heart syndrome underwent a primary Damus type of anastomosis with concurrent repair of the aortic arch, atrial septectomy, and creation of a 3.5-mm Gore-Tex aortopulmonary shunt. The primary diagnoses are listed in Table 1. No patient was refused operation for any reason during this period.

On the basis of the various anatomic features shown in Table 1, 20 of the 24 patients may have been suitable for a more standard technique of palliation consisting of pulmonary artery banding associated with repair of the aortic arch and enlargement of the outlet foramen. In 4 patients, a primary Damus type of procedure was considered to be the only available surgical option. Three of them (patients 3, 5, and 19) had aortic atresia. For the other (patient 11), who had mitral valve stenosis associated with a ventricular septal defect, the alternative approach of mitral valve replacement, closure of the ventricular septal defect, and repair of the aortic arch was deemed too risky.

The median age at operation was 6 days (range, 1 to 170 days, with 16 patients being < 28 days of age) and the median weight, 3.4 kg (range, 2.6 to 4.6 kg). Preoperative risk factors included severe metabolic acidosis (pH < 7.1) in 7, established renal failure in 2, and electrocardiographic evidence of myocardial ischemia (with ST segment depression of > 3 mm) in 1 patient. Twelve patients were mechanically ventilated, and 9 required intravenous inotropic therapy prior to operation. The cardiothoracic ratio on the chest roentgenogram at admission to the hospital was greater than 0.55 in all instances. Informed consent for operation was obtained from the parents of all patients.

Surgical Technique
Cardiopulmonary bypass was established using arterial return either by way of the ascending aorta if it was of good size or the pulmonary trunk. In neonates, a single venous cannula in the right atrium was used for venous return. Deep hypothermia to 18°C and periods of complete circulatory arrest were employed to perform the repair.

After the cross-clamping of the head and neck vessels, all ductal tissue was excised, including the segment of coarcted aorta. Having been divided, the isthmus was opened back under the arch to the ascending aorta. The descending thoracic aorta was anastomosed for about 50% of its circumference to the undersurface of the arch. The proximal pulmonary trunk was then anastomosed to the ascending aorta, arch, and remaining circumference of the descending aorta (Figs 1, 2). Care was taken not to distort either the aortic or the pulmonary valve annulus when the Damus anastomosis was done. The native aortic valve was left open in all patients. The distal pulmonary trunk was repaired with native or bovine pericardium, and a 3.5-mm Gore-Tex shunt was placed between the undersurface of the brachiocephalic artery and the proximal right pulmonary artery. It was possible to repair the aortic arch without the use of exogenous material in all except 1 patient. All patients underwent atrial septectomy.

View larger version (62K): [in this window] [in a new window]   Fig 1. . Steps in modified Damus procedure. (MPA = main pulmonary artery.)

View larger version (103K): [in this window] [in a new window]   Fig 2. . Follow-up angiograms demonstrating the surgical repair. (A) An unobstructed connection between the aortic (ao) and pulmonary trunks (pt) and the descending aorta (dao) is shown, with competent arterial valves. (B) The modified 3.5-mm Blalock-Taussig shunt (arrow) between undersurface of brachiocephalic artery (bca) and right pulmonary artery (rpa).

	Results

Top Footnotes Abstract Introduction Patients and Methods Results Comment References

 
There were nine early deaths (<30 days after operation); seven occurred in the first 9 patients to undergo the procedure. Two of the oldest patients (patients 1 and 5, see Table 1) had diffuse grade 3 to 4 pulmonary vascular disease at postmortem examination [9]. Another patient, who died 25 days after operation (patient 9), had disseminated arterial calcification also affecting the coronary arteries and evidence of severe myocardial ischemia.

Two patients with congenitally corrected transposition and Ebstein's malformation of the left atrioventricular valve died at a second surgical procedure. Patient 19 had an intact ventricular septum and aortic atresia. After the Damus procedure and atrial septectomy, there was echocardiographic evidence of progressive distention of the blind-ending morphologic right ventricle because of severe atrioventricular valve regurgitation (Fig 3). This was associated with a hypertrophied, hypercontractile but underfilled systemic ventricle and low cardiac output. At 18 days, patch closure of the left atrioventricular valve was performed, but the patient did not survive the procedure. In patient 4, who had a ventricular septal defect associated with subaortic stenosis and coarctation, the initial procedure was coarctation repair with closure of the ventricular septal defect and relief of subaortic stenosis. Low cardiac output after repair prompted on-table revision, with takedown of the ventricular septal defect patch, creation of a Damus anastomosis, and a superior vena cava-pulmonary shunt.

View larger version (159K): [in this window] [in a new window]   Fig 3. . (Patient 19.) Modified echocardiographic four-chamber views. (A) In the diastolic frame, the dilated and blind-ending morphologic right ventricle (RV) is seen, with Ebstein's malformation of the atrioventricular valve (arrows). The systemic (morphologic left) ventricle (LV) is hypertrophied with restricted filling. (B) In systole, the cavity of the left ventricle is virtually obliterated, confirming good systolic function but low stroke volume. The apically displaced left atrioventricular valve does not close completely, with resultant free regurgitation. (ARV = atrialized portion of right ventricle; LA = left atrium.)

Follow-up and Further Surgical Procedures
All survivors (n = 15) have undergone regular outpatient follow-up (median follow-up, 6.5 months; range, 1 to 23 months); all are clinically well and in sinus rhythm. Serial two-dimensional color Doppler echocardiography has demonstrated good systemic ventricular function in all patients on qualitative estimation. Atrioventricular and arterial valve regurgitation was serially evaluated and classified into one of four grades of increasing severity on the basis of the extension of the color Doppler jet into the receiving chamber. In all patients who have subsequently undergone cardiac catheterization, angiographic estimation of regurgitation was also made. Only 1 patient has grade 2 aortic valve regurgitation. One patient has grade 1 pulmonary valve regurgitation, and 3 patients have grade 1 atrioventricular valve regurgitation. All other patients show no evidence of arterial valve regurgitation.

Ten of the 15 patients have undergone a superior vena cava-pulmonary shunt at 3 to 6 months of age with concurrent repair of branch pulmonary artery stenoses in 2. One patient (patient 22) died of an inadvertent overdose of sodium nitroprusside after the cavopulmonary shunt. At cardiac catheterization prior to the cavopulmonary shunt, none had a systemic ventricular end-diastolic pressure of greater than 10 mm Hg, a finding suggesting the absence of major diastolic dysfunction of this ventricle. The oldest patient (patient 6) has subsequently been restudied by cardiac catheterization and is currently awaiting completion of the Fontan procedure. One patient (patient 18) had echocardiographic and angiographic evidence of severe stenosis at the Damus anastomosis affecting the origins of the brachiocephalic and left common carotid arteries, but without recurrence of coarctation. At reoperation at the age of 3 months, patch angioplasty of the arch vessels was performed to relieve the stenosis, followed by insertion of a 4-mm aortopulmonary shunt. Finally, the 4 youngest survivors are awaiting cardiac catheterization prior to undergoing a cavopulmonary shunt.

	Comment

Top Footnotes Abstract Introduction Patients and Methods Results Comment References

 
The Fontan operation is often the only surgical option for long-term palliation of patients with a functional single ventricle and systemic outflow obstruction. Both ventricular hypertrophy and subaortic stenosis are adverse incremental risk factors for the Fontan procedure [10], and early relief of outflow obstruction has been advocated to diminish the risk of diastolic dysfunction of the systemic ventricle. Subaortic stenosis may be present at birth as the result of a restrictive outlet foramen. It may also occur as a consequence of progressive restriction of the outlet foramen, which has muscular margins [3]. Diagnosis of a restrictive outlet foramen in the neonate is difficult, and in the presence of a normal pulmonary outflow tract and patent duct, no gradient may be recorded between the single ventricle and the aorta. Previous studies have clearly demonstrated, however, that when the ratio of the outlet foramen diameter to the aortic root is less than 1, outlet foramen obstruction can be assumed to be present [11]. It is important to recognize that the outlet foramen is not circular, and diameter measurement in a single echocardiographic plane may be misleading. For this reason, biplane measurements were made when possible, and the largest diameter was chosen for comparison with the aortic root [12]. In addition, the presence of aortic arch obstruction in the neonate with a duct-dependent systemic circulation inevitably is predictive of rapid development of subaortic stenosis after the standard palliative approach of arch repair and pulmonary artery banding [2, 13].

By promoting muscular hypertrophy, pulmonary artery banding causes and accelerates the progression of subaortic stenosis, with consequent myocardial hypertrophy and ventricular dysfunction [1, 3]. Banding may also not prevent progression of pulmonary vascular disease in this subset of patients [14]. In view of these potential adverse effects of banding, alternative surgical approaches to this group of lesions have been proposed. Further, some neonates with similar physiology and aortic atresia are not candidates for conventional pulmonary artery banding.

Direct enlargement of the outlet foramen, particularly through the small aortic valve, is difficult in the neonate [15]. Relief of stenosis is often inadequate, and the risk of damaging the conduction system is high [16]. Valved conduits between the ventricle and aorta [17] invariably fail with somatic growth and require frequent replacement. A logical alternative is the modified Damus procedure, where relief of subaortic stenosis is achieved by creating two outlets to the systemic circulation, and pulmonary blood flow is regulated by placement of a small (3.5-mm diameter in this series) aortopulmonary shunt. It is then possible to proceed to a cavopulmonary shunt in early infancy once the pulmonary vascular resistance has diminished satisfactorily. In this manner, abnormal pressure and volume overload of the systemic ventricle can be circumvented and early staged Fontan repair, accomplished. Although a cavopulmonary shunt was combined with the Damus procedure in 1 patient, this is probably best avoided in the neonate seen with unrestricted pulmonary blood flow. Individual reports have attested to the efficacy of the Damus procedure for this group of lesions in the neonate [12, 18], and there is additional experience with this procedure in older children, the majority of whom had undergone prior pulmonary artery banding [11, 19, 20].

Performance of the repair without synthetic patch augmentation of the aorta may also be potentially advantageous in reducing the risk of subsequent recurrence of coarctation [21]. Apart from 1 patient in whom aortic narrowing developed at the takeoff of the head and neck vessels, none of the others have clinical, echocardiographic, or angiographic evidence of recoarctation. Semilunar valve incompetence is another potential risk factor in the long term [22], but the early follow-up has been encouraging. The presence of a morphologic left ventricle as the systemic ventricle may also favorably modify the natural history of these patients compared with patients undergoing a Norwood type of operation for classic hypoplastic left heart syndrome. Although age did not emerge as a risk factor in this series, it is recommended that the operation be performed early.

In 2 patients (aged 75 and 170 days), there was histologic evidence of pulmonary vascular disease at postmortem examination. Such early development of critical pulmonary vascular disease is unusual, but it highlights the importance of early intervention to restrict pulmonary blood flow and conserve the pulmonary vascular bed to allow a later Fontan procedure. Ebstein's malformation of an atrioventricular valve was the cause of death of 1 patient, and in such patients, the valve should probably be closed at the initial operation to reduce the risk of creating a large ``dead space'' and consequent underfilling of the systemic ventricle.

On statistical analysis, improvement in survival with increased experience outweighed the increased risk of death for infants without transposition of the great arteries, which was the predominant risk factor in the early part of the series (see Table 3). The small numbers of patients considered in each of the strata in Table 3 precluded demonstration of significant variation, but it can be appreciated that 100% of patients without transposed great arteries died during all but the most recent experience. It is not clear why patients with transposition of the great arteries appeared to do better than those with other types of ventriculoarterial connection (including congenitally corrected transposition). It is possible that the type of ventriculoarterial connection is a surrogate for another risk factor possessed by all patients without transposition, but this was not assessed in the study. The 2 patients in whom survival was incorrectly assessed using the two criteria of chronologic sequence of the procedure and presence or absence of transposition merit further consideration. One of them (patient 8), who also had development of postoperative paraplegia, died of necrotizing enterocolitis. The other (patient 19) has been discussed already.

Large heart size at presentation or evidence of myocardial ischemia (which was seen in 1 patient who survived the operation) did not appear to be risk factors for the procedure [23].

Despite the significant mortality early in the series, all survivors are clinically well. At follow-up, all but 1 of the patients who to date have undergone cardiac catheterization have been suitable candidates for a cavopulmonary shunt. The only patient with arch obstruction has had a further staging procedure with arch repair and revision of the aortopulmonary shunt. The crucial question is whether early conversion to a Norwood type of physiology with its associated mortality is a better option than initial pulmonary artery banding, which can be performed at a lower surgical risk. Analysis of only those patients in whom banding might have been possible as initial palliation shows that procedure-related mortality diminished from 38% (9/24 patients) to 25% (5/20), with all five deaths occurring among the first 7 patients to undergo operation. Documentation of a low ventricular end-diastolic pressure at follow-up cardiac catheterization further supports the potential benefits of early reduction of pressure and volume overload of the single ventricle. These results encourage the pursuit of an aggressive surgical approach in neonates with nonhypoplastic left heart syndrome variants of single-ventricle physiology with systemic outflow obstruction.

	Footnotes

Top Footnotes Abstract Introduction Patients and Methods Results Comment References

 
Address reprint requests to Dr Sreeram, Heart Unit, Birmingham Children's Hospital, Ladywood Middleway, Birmingham B16 8ET, England.

	References

Top Footnotes Abstract Introduction Patients and Methods Results Comment References

 

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This Article Abstract 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 Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Brawn, W. J. Articles by Sreeram, N. Search for Related Content PubMed PubMed Citation Articles by Brawn, W. J. Articles by Sreeram, N.