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Ann Thorac Surg 2004;77:23-28
© 2004 The Society of Thoracic Surgeons

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Original article: cardiovascular

Management of univentricular heart with systemic ventricular outflow obstruction by pulmonary artery banding and Damus-Kaye-Stansel operation

^a Department of Cardiovascular Surgery, Osaka Medical Center and Research Institute for Maternal and Child Health, Osaka, Japan
^b Department of Pediatric Cardiology, Osaka Medical Center and Research Institute for Maternal and Child Health, Osaka, Japan

^* Address reprint requests to Dr Miura, Department of Cardiovascular Surgery, Osaka Medical Center and Research Institute for Maternal and Child Health, 840 Murodo-cho, Izumi, Osaka, 594-1101 Japan.
e-mail: miura{at}mch.pref.osaka.jp

Presented at the Poster Session of the Thirty-ninth Annual Meeting of The Society of Thoracic Surgeons, San Diego, CA, Jan 31–Feb 2, 2003.

	Abstract

Top Abstract Introduction Patients and methods Results Comment References

 
BACKGROUND: Some patients with univentricular hearts who are candidates for Fontan operation may develop ventricular outflow tract obstruction after pulmonary artery banding (PAB) or Fontan. However, the indication for Damus-Kaye-Stansel (DKS) operation for these patients has not been clear. To clarify the indication, the changes in the diameter of ventricular outflow tract and the feasibility of DKS operation before or with Fontan were investigated.

METHODS: Among the patients with univentricular heart who underwent PAB, 21 patients had probable ventricular outflow obstruction with an aorta arising from the morphologic right ventricle. Diameter of ventricular outflow tract was measured before and after PAB, Glenn, and Fontan operations with or without DKS, and indexed by normal value (%VOT).

RESULTS: Six patients died after PAB. In the surviving 15 patients, %VOT decreased significantly from 103% (median, range 75%–153%) to 75% (range 52%–153%) after PAB. Four with very small %VOT (52% to 63%) after PAB needed DKS with bidirectional Glenn or central shunt operation, and 5 with moderately small %VOT (67% to 109%) after PAB needed DKS concomitantly with Fontan. A patient with %VOT of 117% before Fontan required DKS after Fontan. A patient with %VOT of 153% underwent Fontan without DKS and obstruction did not develop after Fontan. The remaining 4 patients were under consideration for Glenn or Fontan operation.

CONCLUSIONS: The diameter of the ventricular outflow tract decreased after PAB and Fontan operations. DKS operations might be indicated before Fontan if the indexed diameter of ventricular outflow tract after PAB was below 70% and concomitantly with Fontan if it was below 120%.

	Introduction

Top Abstract Introduction Patients and methods Results Comment References

 
Although recent operative results in Fontan operation have been improving and many patients with univentricular hearts have been able to receive Fontan operations, the group of patients with potential systemic ventricular outflow tract obstruction (VOTO) still has relatively poor results due to this obstruction and subsequent ventricular problems [1, 2]. Although the classic risk factors described by Choussat and Fontan [3] did not include ventricular hypertrophy, presently, profound ventricular hypertrophy is regarded as one of the risk factors in Fontan operations because it may reduce ventricular compliance and may affect diastolic function. Diastolic function is considered important for Fontan-type circulation to maintain low left atrial pressure, and subsequent low pulmonary arterial and central venous pressure [4].

There were several papers that described close relationships between underlying ventricular morphology and development of systemic VOTO in univentricular hearts [5]. High correlation between pulmonary artery banding (PAB) and the development of systemic VOTO was also reported [6].

In these patients, systemic VOTO may progress even after the completion of a Fontan operation [7, 8]. But the indication for the surgery for the relief of systemic VOTO has been ambiguous and selection of the initial procedure in the neonatal period has been also controversial [9–12].

Therefore, prediction of systemic VOTO may be a very important issue in this type of patient. An appropriate prediction enabled us to decide the proper application of the operation to relieve systemic VOTO in a suitable time. In this article, we examined the change in the size of the systemic ventricular outflow tract during staged procedures toward the Fontan operation to predict systemic VOTO and to decide the indication for the Damus-Kaye-Stansel (DKS) operation for the relief of systemic VOTO.

	Patients and methods

Top Abstract Introduction Patients and methods Results Comment References

 
Patients
Twenty-eight children who were candidates for a Fontan operation underwent PAB due to excessive pulmonary flow in our hospital since 1991. One patient who had undergone PAB in an another hospital but underwent a bidirectional Glenn (BDG) operation in our hospital was also a candidate for a Fontan operation. No patient was suitable for biventricular repair.

Among these 29 patients, 11 patients who had transposition of the great arteries and 10 patients who had double-outlet right ventricle were included in this study. These 21 patients were at a risk of ventricular outflow tract obstruction after PAB and Fontan because of subaortic stenosis or stenosis of bulboventricular foramen (BVF). Details of the diagnoses of these patients are shown in Table 1. Aortic arch obstruction was associated in 6 patients: coarctation of aorta in 4 and interruption of the aortic arch in 2.

Among the 15 surviving patients after PAB, 5 patients underwent a bidirectional Glenn (BDG) operation. Median age at operation was 1.9 years (range, 1.1 to 2.1 years), and median body weight was 10.8 kg (range, 3.5 to 12.3 kg). Associated operative procedures with BDG were atrial septal defect (asd) enlargement in 4 patients, DKS procedure in 3, repair of atrioventricular valve in 2, closure of atrioventricular valve in 1, and subaortic muscle resection in 1. Another patient underwent central shunt from right brachiocephalic artery to main pulmonary artery using a 4-mm expanded polytetrafluoloethylene graft with a DKS operation.

Finally, 9 patients underwent Fontan operations. Two of them had undergone previous BDG operations as a staged repair. Six patients underwent Fontan operations without staged procedures after PAB. And 4 of them underwent DKS concomitantly with Fontan operations. Median age at time of Fontan operation was 3.4 years (range, 1.4 to 5.2 years), and median body weight was 12.7 kg (range, 9.5 to16.4 kg). All patients underwent total cavopulmonary connection: 4 of them underwent lateral tunnel intraatrial baffling; 5 utilized an extraatrial conduit of expanded polytetrafluoloethylene graft from the inferior vena cava to the pulmonary artery.

The Damus-Kaye-Stansel operation was performed on 10 patients. Median age at the operation was 3.4 years (range, 0.9 to 6.0 years), and median body weight was 12.3 kg (range, 6.5 to16.4 kg). The Damus-Kaye-Stansel was done concomitantly with the Fontan operation in 5 patients, with BDG in 3, and with central shunt in 1. In 1 patient, DKS was performed 21 months after completion of the Fontan operation because of progression of subaortic stenosis. Details of the DKS procedure have changed during the study period. In 3 patients, the pulmonary trunk was divided and the proximal end of pulmonary trunk was connected to the side of ascending aorta (original DKS). In 7 recent patients, both the aortic and pulmonary arteries were divided above the sinus and sutured to each other in a side-to-side fashion, and the common orifice of both arteries was anastomosed to the divided distal ascending aorta [14].

Measurement of ventricular outflow tract
The usual follow-up was done by echocardiography for these patients, with 3- to 6-month intervals to decide the timing of precise cardiac catheterization. Cineangiography was performed before and after PAB, BDG, and Fontan operations. The diameter of subaortic stenosis or of BVF was measured at the end-systolic phase (Fig 1). After the DKS procedure, the ventricular outflow tract consisted of two outflow tracts: aorta and pulmonary artery. The original ventricular outflow tract (aortic route) was measured for this study because the purpose was to evaluate the change of the original ventricular outflow tract and feasibility of different operations. These values were indexed by normal diameter of aortic valve derived from an equation calculated by body surface area [15].

View larger version (89K): [in this window] [in a new window]   Fig 1. Serial angiography in a patient with a double-inlet left ventricle with outlet chamber and ventriculoarterial discordance. Arrows indicate the portion of measurement of subaortic stenosis. (A) Left ventriculography before PAB: %VOT = 101%. (B) Left ventriculography after PAB: %VOT decreased to 82%. (C) Left ventriculography after Fontan and DKS procedures: %VOT in native aortic route was 62%. (Ao = ascending aorta; DKS= Damus-Kaye-Stansel operation; PA = pulmonary artery; PAB = pulmonary artery banding.)

	Results

Top Abstract Introduction Patients and methods Results Comment References

View larger version (24K): [in this window] [in a new window]   Fig 2. Courses of 21 patients in this study. (BDG = bidirectional Glenn operation; CS = central shunt; DKS = Damus-Kaye-Stansel operation; PAB = pulmonary artery banding.)

Central shunt.
There was no death after central shunt with DKS.

Fontan operation.
There was no death after completion of the Fontan operation.

DKS operation.
One operative death was recognized in a patient who underwent DKS and BDG operations, but the death was due to high pulmonary vascular resistance and not due to the concurrent DKS procedure.

Change of ventricular outflow tract
Initial %VOT before PAB was 52% to 153% (median, 99%) in 15 patients whose angiographic data were available. The patient with the smallest %VOT (52%) showed severe subaortic stenosis after PAB. An attempt was made to relieve subaortic stenosis by muscle resection shortly after PAB, but we could not save his life. In the 10 patients whose data were available both before and after PAB among surviving 15 patients, %VOT decreased significantly from 75% to 153% (median, 103%) to 52% to 153% (median, 75%) after PAB (Fig 3). Mean reduction rate in %VOT was 25% (range, 0% to 44%).

View larger version (27K): [in this window] [in a new window]   Fig 3. Change in %VOT before and after pulmonary artery banding (PAB). Bars indicate mean and standard deviation.

View larger version (28K): [in this window] [in a new window]   Fig 4. Serial change in %VOT: before and after PAB, after BDG or central shunt, and after Fontan operation in patients who underwent the DKS procedure (except group 4). Open circles indicate the %VOT in the patients who need DKS with BDG or central shunt (group 1). Closed circles indicate those patients who need DKS concomitantly with Fontan operation (group 2). Open squares indicate a patient who needs DKS after Fontan operation (group 3). Closed squares indicate a patient who did not develop systemic VOTO throughout the course (group 4). (BDG = bidirectional Glenn operation; CS= central shunt; DKS = Damus-Kaye-Stansel operation; PAB= pulmonary artery banding.)

In 5 patients with moderately small %VOT (67% to 109%) after PAB, DKS operations were done concomitantly with Fontan operations (group 2; Fig 4). In the 4 patients whose data were available both before and after Fontan and DKS operations, %VOT decreased significantly from 67% to 86% (median, 83%) before Fontan to 58% to 77% (median, 61%) after Fontan.

A patient with a %VOT of 117% had undergone a Fontan operation only, because the %VOT was larger than the normal value (group 3; Fig 4). But cardiac catheterization 15 months after a Fontan operation had revealed a pressure gradient of 10 mm Hg between the ventricle and aorta and a %VOT of 82%. In this patient, the pulmonary trunk had been divided in the Fontan operation. But the pulmonary valve had not been sutured and reserved for the possible DKS procedure in the future. Her pulmonary valve was pliable and well functioning, and she successfully underwent DKS 21 months after the Fontan operation [16]. Fifteen months after the DKS operation, repeated angiography revealed the %VOT had further decreased to 55%, but there was no pressure gradient between the ventricle and aorta because the bypass was created by DKS anastomosis.

A patient with a %VOT of 153% underwent Fontan without DKS, and the %VOT decreased to 139% and the patient did not develop systemic VOTO after Fontan (group 4; Fig 4).

Pressure gradient and the size of the ventricular outflow tract
The relationship between the pressure gradient through the ventricular outflow tract and %VOT just before the DKS operation is shown in Figure 5. A significant pressure gradient of more than 10 mm Hg was demonstrated between the ventricle and aorta, if %VOT was below 60%.

View larger version (65K): [in this window] [in a new window]   Fig 5. Relationship between %VOT and pressure gradient before DKS. Pressure gradient between dominant ventricle and aorta becomes apparent if the %VOT becomes smaller than 60% (shaded area). Open circles represent the patients in group 1. Closed circles represent the patients in group 2. The open square represents the patient in group 3. (DKS = Damus-Kaye-Stansel operation.)

	Comment

Top Abstract Introduction Patients and methods Results Comment References

Pressure gradient is one of the quantitative measurements for systemic VOTO. But, in some neonatal cases, it is difficult to detect a pressure gradient between the ventricle and ascending aorta although they apparently have small ventricular outflow tracts. The patients with large patent ductus arteriosus had almost the same systolic pressure in the ventricle, aorta, and pulmonary artery. The patients with interrupted aortic arch sometimes had no pressure gradient between the ventricle and aorta because the amount of blood flow through the aortic valve was less than half of the cardiac output necessary for the whole body. Freedom and colleagues also described that the absence of a pressure gradient between the ventricle and aorta could not exclude subaortic stenosis [6]. In our experience, resting pressure gradient became apparent if the diameter of the subaortic stenosis was less than 60% of the normal aortic valve diameter (Fig 5). These results indicate that apparent pressure gradient may suggest a significant narrowing of ventricular outflow tract. Therefore, we consider any pressure gradient through the ventricular outflow tract as evidence of VOTO.

In this study, we quantitatively evaluated the size of the systemic ventricular outflow tract, and found that 60% of ventricular outflow tract, compared with normal aortic diameter, is a critical value for the development of pressure gradient. Therefore, %VOT less than this value may be an indication for the surgical relief of systemic VOTO. If the initial %VOT in the neonatal period is less than 60%, relief of systemic VOTO should be necessary in the initial operation. In our cases, the patient with a %VOT of 52% before PAB developed critical systemic VOTO after PAB, and this patient should undergo relief of systemic VOTO at the initial operation.

After PAB, the patients with %VOT less than 70% were treated with DKS with BDG or central shunt, before the Fontan operation (group 1). Some of them had apparent pressure gradient between the ventricle and aorta. In these patients, %VOT in the systemic ventricular outflow tract had decreased to below 60% after a final Fontan operation. Therefore, a DKS procedure before the Fontan operation was feasible for these patients.

The patients with %VOT more than 70% but less than 110% after PAB underwent DKS concomitantly with the Fontan operation (group 2). In these patients, apparent pressure gradient was absent before the Fontan operation. Late after the DKS procedure with concomitant Fontan operation, %VOT in the systemic ventricular outflow tract decreased to 58% to 77%, which might develop a pressure gradient if not treated with the DKS procedure. Moreover, a patient with a %VOT of 117% had developed apparent systemic VOTO with a pressure gradient after a Fontan operation without a DKS procedure. Therefore, it is feasible to relieve systemic VOTO with the DKS procedure concomitantly with the Fontan operation if %VOT is below 120% of normal value before the Fontan operation.

There were a few papers that described a quantitative analysis of systemic VOTO in a univentricular heart. Matitiau and colleagues measured the area of BVF and reported that the initial BVF area indexed to body surface area predicted late obstruction [18]. They have shown that all patients with an initial BVF index less than 2 cm²/m² without early BVF bypass procedure develop late obstruction. If we compare the results of Matitiau's study to ours, 2 cm²/m² of cross-sectional area of BVF is equal to a %VOT of 113% in our study, if the body surface area of the patient is 0.2 m² (usual body surface area in a neonate). In our study, almost all patients whose initial %VOT was less than 140% needed surgery for late systemic VOTO. They also showed that the patients whose mean BVF area index was 0.94 cm²/m² needed bypass surgery of BVF in their initial palliation in early infancy, and that the patients whose initial mean BVF area index was 3.95 cm²/m² did not develop late obstruction. The BVF area index of 0.94 and 3.95 cm²/m² in their study almost coincided with %VOT of 77% and 159% in our study, respectively. In our study, a patient with initial %VOT of 52% might need bypass surgery in initial palliation, and a patient with initial %VOT of 153% did not develop late systemic VOTO. Although the expression of the stenosis was different, the indication of the operation to relieve systemic VOTO might not be so different between these two studies.

There is a controversy regarding the selection of operative procedure for neonatal patients with univentricular hearts and systemic VOTO. Some authors reported that initial PAB and early relief of systemic VOTO was a safe approach [9, 10], and the others reported that the Norwood-type operation was effective for the relief of systemic VOTO subsequently for the avoidance of ventricular hypertrophy [11, 12]. Although recent results of the Norwood-type operation have been improving and the concept of early relief of systemic VOTO is reasonable for the preservation of ventricular performance, this procedure requires cardiopulmonary bypass and the mortality and morbidity remain high in most hospitals. If the initial PAB was tolerable for the patient, initial PAB and early relief of systemic VOTO was a reasonable and safe approach. If the systemic VOTO was too severe to support systemic output, the Norwood-type procedure or other surgical relief of systemic VOTO may be necessary. The selection of procedure should depend on the size of the ventricular outflow tract. Therefore, quantitative measurement of the ventricular outflow tract may be necessary to select the surgical approach. From the results of our study, the neonatal patient with initial %VOT less than 60% may need a relief of systemic VOTO at the initial operation, including the Norwood-type operation.

Gates and associates[19] and Karl and associates[20] described independently that a diameter of the ventricular outflow tract less than half the size of aortic annulus was a definition of significant systemic VOTO. Hiramatsu and colleagues [21] described that the presence of pressure gradient or anatomical narrowing less than the aortic valve diameter might indicate DKS procedure. Even if the initial %VOT is more than 100%, it may decrease during serial staged surgeries and the relief of systemic VOTO may become necessary. In our study, final %VOT after Fontan operation was below 80%, even in the patients whose initial %VOT was more than 120% (Fig 4). The current study indicates that DKS may be necessary at final Fontan operation if %VOT after PAB was less than 120%.

Although the DKS operation is a good alternative for a narrow systemic outflow tract, the occurrence of regurgitation of both semilunar valves should not be neglected. Chin and colleagues reported that pulmonary regurgitation was recognized in one-fourth of the patients, although most of the regurgitation was mild [22]. They also reported that this frequency of regurgitation was higher in the patients long after the DKS procedure. Although the prophylactic DKS operation for possible later systemic VOTO was recommended by some authors [21], DKS should not be performed in patients who will not develop systemic VOTO because of possible later regurgitation.

In conclusion, we have made a quantitative analysis of the change of the original systemic ventricular outflow tract in univentricular hearts and revealed that the diameter of ventricular outflow tract decreased with PAB, BDG, or Fontan operations. Narrowing of systemic VOTO below 60% of normal aortic valve diameter may cause a pressure gradient and may indicate the surgical relief of systemic VOTO. PAB was safely possible in our patients with initial %VOT more than 60%. If the indexed diameter of ventricular outflow tract after PAB was below 70%, a DKS operation might be indicated at the next operation, usually with BDG before a Fontan operation. If the indexed diameter of the ventricular outflow tract after PAB was below 120%, DKS should be performed concomitantly with Fontan operation.

	References

Top Abstract Introduction Patients and methods Results Comment References

 

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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Takuya Miura Hidefumi Kishimoto Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Miura, T. Articles by Nakajima, T. Search for Related Content PubMed PubMed Citation Articles by Miura, T. Articles by Nakajima, T. Related Collections Congenital - cyanotic