(Circulation. 1997;96:1233-1239.)
© 1997 American Heart Association, Inc.
Articles |
Anatomic Features and Surgical Strategies in Double-Outlet Right Ventricle
From the Departments of Cardiology and Cardiac Surgery, Royal Children's Hospital, Melbourne, Australia.
Correspondence to Dr J.L. Wilkinson, Department of Cardiology, Royal Children's Hospital, Flemington Rd, Parkville 3052, Melbourne, Australia. E-mail WilkinsJ{at}cryptic.rch.unimelb.aed.au
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Abstract |
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Background The objective of this study was to review anatomic features and surgical strategies in children with double-outlet right ventricle (DORV) and to assess risk factors for early mortality.
Methods and Results Records were reviewed of all children with DORV undergoing surgery between 1978 and 1993. Noncomplex patients (group 1) had atrioventricular (AV) concordance, a single ventricular septal defect (VSD), balanced ventricles, no straddling AV valves, and no major pulmonary artery anomaly. Group 2 (complex) comprised all remaining patients. Independent risk factors analyzed included location of the main VSD, presence of additional VSDs, coarctation, ventricular outflow obstruction, ventricular hypoplasia, age at operation, operation before 1985, previous palliation, and type of definitive operation. Of 193 patients, 117 were in group 1 and 76 in group 2. In 148 patients, biventricular repair was undertaken, including 111 of 117 group 1 patients and 37 of 76 group 2 patients. Early mortality was higher among group 2 patients undergoing biventricular repair than among group 1 patients (8 of 37 versus 4 of 111, P<.005) and higher than group 2 patients undergoing a Fontan procedure (none of 29, P<.01). Aortic arch obstruction, operation before 1985, and multiple VSDs were significant risk factors for mortality. Age <1 month (P<.05) and multiple VSDs (P<.005) were independent risk factors after definitive repair. Up-to-date follow-up is available on 144 surviving patients, with 127 (88%) in New York Heart Association class I and the remaining 17 in class II. Overall 10-year survival probability was 81%, whereas probability of survival, free from reoperation (after definitive surgery), was 65% at 10 years.
Conclusions Biventricular repair can be achieved in most patients with DORV with low risk. In complex DORV, a Fontan procedure is associated with a lower surgical mortality.
Key Words: ventricles • surgery • risk factors • Fontan procedure
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Introduction |
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By describing a ventriculoarterial connection rather than a specific congenital malformation, double-outlet right ventricle (DORV) includes a broad spectrum of anatomic variants and associated malformations. In 1964, Kirklin et al1 reported successful correction in a child with DORV, subaortic ventricular septal defect (VSD), and concordant atrioventricular (AV) connections. Since then, complete correction through the use of a variety of surgical techniques has been achieved in more complex forms of DORV. Modified Fontan procedures have been advocated when biventricular DORV repair has been either impractical or extremely complex.2 With the improving short-term and medium-term outcome for Fontan procedures in recent years,3 4 5 this approach might also be extended to patients who are at increased operative risk with a conventional biventricular repair. This report reviews the anatomic findings, surgical strategies, and results among patients with DORV presenting for surgery in a single institution over a 15-year period.
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Methods |
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Patient Selection
The study population comprised all patients with a diagnosis of DORV who did not have a univentricular AV connection and who underwent a surgical procedure between 1978 and 1993. A diagnosis of DORV was made if both great arteries originated predominantly from the right ventricle with application of the "50% rule," which requires that one great artery arises completely and the second >50% from the right ventricle. The diagnosis and anatomic findings were based on a combination of angiography, echocardiography, and surgical inspection. Group 1 patients, with noncomplex anatomy, were defined as those with AV concordance, a single VSD, balanced ventricles, no straddling AV valves, and no major pulmonary artery anomaly. Group 2 patients, with complex anatomy, consisted of all the remaining patients. Complex anatomy was defined by the presence of atrial isomerism, AV discordance, atrioventricular septal defect, multiple VSDs, hypoplastic right or left ventricles, straddling AV valves, and major pulmonary artery anomalies including pulmonary atresia, pulmonary artery sling, aorto-pulmonary window, or discontinuous branch pulmonary arteries. Early mortality was defined as death on the same hospital admission as the operation, or within 30 days of operation for patients discharged from hospital. Follow-up information was obtained from hospital files for locally followed patients and from referring cardiologists for patients followed elsewhere.
Statistical Analysis
Outcome measures included early survival and freedom from
reoperation. To assess potential risk factors for early mortality and
reoperation, univariate analysis with Fisher's
exact test was initially used for each variable. Variables with
a value of P<.1 were then entered into a stepwise logistic
regression analysis. A commercially available statistic program
was used (StataCorp, 1995. Stata Statistical Software: Release 4.0).
Variables were considered statistically significant in the
multivariate analysis at P<.05.
Odds ratios and 95% confidence intervals are stated. Survival and
freedom from reoperation probabilities were estimated with the
Kaplan-Meier method. t tests were used to compare normally
distributed continuous variables and Pearson's 
2
analysis or Fisher's exact test to compare discrete
variables. Values are expressed as mean±SD unless otherwise
stated.
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Results |
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A total of 193 patients who underwent surgical procedures comprised the study population. Four types of definitive repairs were performed: (1) intraventricular repair with a baffle from the left ventricle to the aorta (IVR), (2) arterial switch operation with a baffle from the left ventricle to the pulmonary artery (ASO), (3) atrial inversion using the Senning operation, and (4) cavopulmonary shunt/Fontan procedures including bidirectional cavopulmonary connection, modified Fontan procedures, total cavopulmonary connection, and the Kawashima operation. The mean age at definitive repair was 3.2±4.1 years and ranged from 1 day to 30.2 years, with a median of 1.9 years.
Anatomic Features
Complex versus noncomplex anatomy and associated
anomalies. Complex anatomic features among group 2 patients are
listed in Table 1
. Of the 76
(39%) patients in group 2, the most commonly present complex
anatomic feature was the presence of additional VSDs (24 patients),
followed by hypoplastic right or left ventricle (20 patients) and
atrioventricular septal defect (18 patients).
Associated noncomplex anomalies present in group 1 and 2 patients
are listed in Table 2.
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VSD type. The location of the primary VSD and the presence
of additional VSDs are shown in Table 3. Of the 90 patients in whom
the main VSD was subaortic, 50 had tetralogy of Fallot–type
anatomy. A subpulmonary VSD was present in 49
patients. The main VSD was not committed to either outflow tract in 31
patients ("noncommitted"). This designation was used in all cases
in which the VSD was anatomically distant from both
arterial valves and was not related more directly to one
outflow tract than to the other. An additional 18 patients had a
complete atrioventricular septal defect. In 5 patients,
all in group 1, the VSD was committed to both outflow tracts. There was
no statistical difference in the primary VSD location between patients
in group 1 and group 2. In three cases, all with subaortic VSDs, the
VSD was restrictive.
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Outflow tract obstruction. Pulmonary outflow tract obstruction (POTO) not associated with tetralogy of Fallot but including pulmonary atresia was present in 45 patients and was most prevalent in the group with subaortic VSDs (17 of the remaining 40 patients, 42%). Eleven of 31 (35%) of children with noncommitted VSD had POTO, as did 2 of 5 (40%) with doubly committed subarterial defects and 7 of 18 with atrioventricular septal defects (39%). Only 8 of 49 patients (16%) with subpulmonary VSD had valvar or infundibular pulmonary stenosis. The majority of patients (64%) with pulmonary outflow obstruction belonged to group 2. Aortic or subaortic stenosis was seen in 6 patients (3%), of whom 2 belonged to group 1 and 4 to group 2. Of the 6 patients with left ventricular outflow obstruction, 3 had subpulmonary VSDs, 2 had subaortic, and 1 a noncommitted VSD. Aortic arch obstruction coexisted with left ventricular outflow tract obstruction in 3 patients. Overall, aortic arch obstruction (aortic coarctation or interruption) occurred in 26 (13%) of patients, of whom 14 had subpulmonary, 7 had subaortic, and 5 had noncommitted VSDs. The majority (17 of 26, 65%) of patients with aortic arch obstruction had noncomplex anatomy (group 1).
Surgical Treatment
Prior palliative procedures. Definitive repair was the
primary procedure in 92 of 193 (48%) patients. Table 4 summarizes the
details of prior palliative procedures undertaken in the remaining 101
patients. Of these, 77
patients required only one palliative procedure, 19 had two procedures,
4 patients had three, and 1 patient required four palliative
procedures. The proportion of group 2 patients requiring palliation
before definitive surgery (50 of 76 or 65.8%; CI, 54.0% to 76.3%)
was significantly higher than that of group 1 patients (51 of 117 or
43.6%; CI, 34.4% to 53.0%; P<.005).
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Definitive surgery. One hundred seventy-nine patients have
so far undergone definitive surgery. Of the remaining 14 patients, 5
await definitive surgery and 9 died before repair could be undertaken.
At definitive surgery, a biventricular repair was attempted
if considered possible. Table 5 summarizes the details of the
definitive surgical procedure according to the VSD
site. In 148 patients a
biventricular repair was undertaken, with an
intraventricular baffle in 107 cases, the
arterial switch operation as a primary procedure in 31, and
a Senning procedure in 10 (all in the early part of the series-before
introduction of the arterial switch). In 4 of these Senning
patients conversion to an arterial switch operation was
subsequently performed because of increasing effort intolerance late
after surgery because of either right ventricular
dysfunction or tricuspid incompetence. Three of these four patients are
long-term survivors, two being in New York Heart Association class I
and the third in NYHA class II. In 3 of the 6 other patients undergoing
a Senning procedure, the VSD was not closed because of the presence of
pulmonary vascular disease. One patient died after his Senning
operation because of intractable problems related to multiple VSDs
(being converted to a Fontan shortly before he died). Excluding this
latter patient, 31 modified Fontan-type procedures were performed for
definitive palliation. These included 29 group 2 patients who had
anatomic features (eg, severely hypoplastic ventricle or straddling AV
valve) precluding a biventricular repair. Two patients in
group 1, both with noncommitted VSDs, also underwent modified Fontan
procedures.
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Children underwent the ASO at a significantly younger age than the intraventricular baffle operation (1.4±2.9 years versus 2.9±3.4 years; P<.05) and the modified Fontan procedures (5.7±5.8 years; P<.005).
Surgical Outcome
Early mortality. Table 6 summarizes the details of 18
(9.3%; CI, 5.6% to 14.4%) hospital deaths, 6 of which occurred after
a palliative procedure and 12 after definitive
surgery. A significantly
higher proportion of group 2 patients (8 of 37 or 21.6%; CI, 9.8% to
38.3%) died after a biventricular repair than did group 1
patients (4 of 111 or 3.6%; CI, 1.0% to 9.0%; P<.005).
In contrast, there were no hospital deaths among the 29 group 2 or the
two group 1 patients undergoing a cavopulmonary shunt or
modified Fontan procedure (0%; CI, 0% to 11.2%). Table 7
presents the results of univariate and
multivariate risk factor analysis for early
mortality. Of the potential
risk factors analyzed in all 193 patients (including those
undergoing palliative procedures), only the presence of multiple VSDs,
aortic arch obstruction, and operation before 1985 reached near
significance in univariate analysis (Table 7, A)
and were significant risk factors for early mortality in
multivariate analysis (Table 7, B). In the
analysis of the 179 patients who survived to definitive repair,
the presence of multiple VSDs and age at operation <1 month were
significantly associated with early mortality, both in
univariate and multivariate
analysis (Table 7).
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Late deaths. Table 8 summarizes the details of late
deaths. There were 9 late
deaths, 2 in group 1 and 7 in group 2 at a mean follow-up time of
4.7±3.8 years. Three of the 9 patients had undergone only a palliative
procedure, 5 had undergone biventricular repair, and 1
patient a Fontan-type procedure. Fig 1 shows the overall survival
probability with 90% confidence intervals for the entire study cohort,
with an estimated 10-year survival probability of
81%.
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Reoperations and follow-up information. Follow-up
information was available in 144 of 166 surviving patients. During the
period of follow-up, 25 patients underwent 29 reoperations at a mean
time interval of 3±3.6 years (range, 6 days to 14 years) after
definitive repair. Of these, 14 patients belonged to group 1 and 11 to
group 2. The initial anatomic features and the type of reoperation are
summarized in Table 9. Risk
factor analysis for reoperation is presented in Table 10. There were no
statistically significant variables associated with reoperation,
with only the presence of left ventricular outflow tract
obstruction present having a value of P<.10. The
probability of freedom from death and reoperation with 90% confidence
intervals is depicted in Fig 2. The estimated freedom from
death and reoperation at 10 years was 65%.
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Additionally, 10 patients underwent pacemaker implantation, 8 for postoperative complete heart block, 1 as prophylaxis, and the remaining patient because of frequent episodes of atrial flutter after a previous Senning operation and a subsequent arterial switch conversion.
Of the 144 patients available for long-term follow-up, 127 (88%) were in NYHA class I and 17 (12%) in class II. Twenty-three had mild and 4 moderate residual right ventricular outflow tract obstruction. Residual left ventricular outflow tract obstruction was present in 10 patients. In 7 of these it was classified as mild and in 3 as moderate.
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Discussion |
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This study confirms that a biventricular repair can be achieved in the great majority of patients with DORV with an acceptably low perioperative mortality. Previous reviews have reported hospital mortality figures of up to 27%, especially in cases with noncommitted or subpulmonary VSDs.6 7 8 9 Since then, new surgical techniques including the arterial switch operation10 11 have significantly improved the outcome in noncomplex forms of DORV.8 11 12 13
VSD Site and Choice of Surgical Procedure
Although the classification of DORV by VSD site as introduced by
Lev and coworkers14 remains useful for surgical planning,
the present study has not demonstrated a significantly higher risk
in patients with noncommitted, subpulmonary, or
atrioventricular septal defects, as has been reported
in earlier series.6 9 15 In our institution, the
arterial switch operation is now the procedure of choice
for patients with subpulmonary VSD without significant right
ventricular outflow obstruction as well as in other cases
in which baffling from the left ventricle to the pulmonary
artery is technically easier than baffling to the aorta. In the
present series, the arterial switch procedure for DORV
with subpulmonary VSD was associated with a low hospital
mortality. On the other hand, an intraventricular
baffle repair without arterial switch could only be
performed in a small number of cases with subpulmonary VSD.
When technically possible, this approach has led to good results with
low operative mortality and no increased need for
reoperation.13 16 17 The tricuspid–to–pulmonary
annulus distance has been shown to be a useful predictor for the
feasibility of this type of repair.12 13 17
Among patients with noncomplex forms of DORV and noncommitted VSD, an intraventricular baffle repair was still possible in the majority of cases. All VSDs in this group were "anatomically" rather than "surgically" noncommitted,18 with no case in which interposed tension apparatus of the atrioventricular valves prevented biventricular repair. In this series, none of the patients with complex forms of DORV who had an associated atrioventricular septal defect died after definitive repair. However, in more than half of such cases a cavopulmonary shunt or modified Fontan operation was chosen because of associated atrial isomerism with some degree of ventricular imbalance.
Risk Factors for Hospital Mortality
Aortic arch obstruction. Aortic arch obstruction was a
significant risk factor for hospital mortality in the analysis
of all patients combined and was an associated finding in more than
half of patients who died after palliative procedures. The majority of
patients with aortic arch obstruction underwent surgical arch repair as
a separate palliative procedure. Recently, better survival probability
has been reported in children with aortic arch obstruction and
associated complex intracardiac lesions if repair has been undertaken
as a one-stage procedure19 20 21 ; this is now our preferred
approach in noncomplex forms of DORV with associated aortic arch
obstruction that includes hypoplasia of the aortic arch proximal to the
left subclavian artery or complete interruption. When a localized
coarctation is present, this would normally be dealt with through a
left thoracotomy as a separate procedure.
Multiple ventricular septal defects. By multivariate risk factor analysis of all patients surviving to definitive surgery, the presence of multiple ventricular septal defects was the only factor related to early mortality. Similar results were reported by Aoki et al13 in an analysis of patients with DORV undergoing biventricular repair. In most cases the presence of additional VSDs does not preclude successful biventricular repair. However, complex forms of DORV remain a challenge, and the optimal surgical approach needs to be individualized according to the specific anatomic features present, including the number and site of any additional VSDs. The advent of newer surgical and interventional catheter techniques22 23 may further facilitate biventricular repair and improve the results in this group of patients.
Early surgical repair. Patients who underwent definitive repair before 1 month of age were also at significantly higher risk in the present series, but this group tended to include patients with unfavorable anatomic variants. Early definitive repair is seldom required in most patients with DORV and is probably best avoided in patients with complicating anatomic features.
Role of the Modified Fontan Procedure
The use of a cavopulmonary shunt or modified Fontan
procedure has previously been advocated as the surgical procedure of
choice in the presence of complex anatomic features such as straddling
AV valves or ventricular imbalance. In recent years,
surgical modifications with staged procedures and atrial baffle
fenestration have been associated with a considerable improvement in
the short-term and medium-term results for the modified Fontan
operation.3 4 5 In the present series there was no
hospital mortality among any patients undergoing a
cavopulmonary shunt or modified Fontan procedure as the
definitive surgical procedure for complex forms of DORV. The difference
in early mortality between patients with complicating anatomic features
undergoing biventricular repair and those subjected to some
form of Fontan procedure strongly suggests that the latter is the
procedure of choice, particularly among those with surgically
inaccessible multiple VSDs.
Late Results and Requirements for Reoperation
Arrhythmias have been recognized as a cause for late
deaths in patients after DORV repair with increasing surgical age,
postoperative ventricular tachyarrhythmias,
and complete heart block as significant risk factors.7
Indications for permanent cardiac pacing should therefore include the
development of complete or prolonged postoperative heart block, which
is often poorly tolerated in the presence of even a minor residual
hemodynamic disturbance.
The 5- to 10-year survival and freedom from reoperation in the present series compare favorably with other reported series in which patients with complex DORV have not always been included in the analysis.13 24 It has been suggested that patients with subaortic and noncommitted VSD are at low and high risk, respectively, for reoperation.13 In the present series there were no statistically significant associations with reoperation, including the site of the primary ventricular septal defect and other complicating anatomic features.
Study Limitations
The classification of varying anatomic DORV subtypes into simple
and complex forms has, to some extent, been arbitrary. However, this
had no bearing on risk factor analysis, in which anatomic
variables were considered separately, regardless of whether the
anatomy was regarded as complex or noncomplex. This study has
the inherent limitations of a retrospective review in which patients
were not randomly allocated to varying treatment groups. Risk factor
analysis is, by its nature, predicated on outcome, which may in
turn be influenced by varying treatment options. Despite this, the
lower hospital mortality among patients with complex DORV who underwent
a modified Fontan procedure rather than an attempt at
biventricular repair indicates that the latter is a
worthwhile option in this difficult subset of patients.
Conclusions
The surgical procedures in a cohort of consecutive patients with
DORV and a wide range of anatomic features have been examined. Early
mortality was low among patients with noncomplex forms of DORV
undergoing biventricular repair but was higher in patients
with complicating anatomic features undergoing similar surgery. Risk
factors for early mortality identified from
multivariate analysis include aortic arch
obstruction, the presence of multiple VSDs, and definitive surgical
repair before 30 days of age. A modified Fontan procedure is the
treatment of choice for many patients with complex forms of DORV,
particularly in the presence of multiple ventricular septal
defects.
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Acknowledgments |
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The authors wish to thank Dr John Carlin for assistance with statistical analysis.
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Footnotes |
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Presented in part at the 44th Annual Scientific Session of the American College of Cardiology, New Orleans, La, March 1995.
Received October 2, 1996; revision received March 12, 1997; accepted March 18, 1997.
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References |
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TopAbstractIntroductionMethodsResultsDiscussionReferences |
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