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Aortic Root Involvement in Congenital Heart Defects
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Etiology
Etiology of aortic-ventricular tunnel is unknown. However, the substrate for its formation
and that of the associated valvular and coronary artery anomalies may be inferred from
developmental anatomy. The cushions forming the facing aortic and pulmonary sinuses with
their respective valvar leaflets normally become separated by an extracardiac tissue plane.
The coronary arteries, also initially encased by this cuff of myocardium, grow through it to
connect with the aortic sinuses. Failure of this tissue plane development might result in a
tunnel above one of the facing aortic sinuses and explain also the potential involvement of the
proximal coronary arteries and the aortic leaflets [75, 76].
Pathophysiology and Clinical Description
The aortico-left ventricular tunnel, by virtue of its anatomic features, is a
pathophysiological model of massive aortic insufficiency.
In AVLT a loud “to and fro” murmur radiating over the entire precordium is typical,
usually with systolic and diastolic thrills. Bounding pulses indicate rapid aortic run-off. In
older patients, these signs may suggest aortic valve stenosis with incompetence, but the
second heart sound should have a normal aortic components in uncomplicated ALVT. Most
patients develop sympoms of heart failure during the first year of life, tipically in the first six
months. The onset, severity and progression of heart failure is, however quite variable,
ranging from many years of asymptomatic status [77-79] to a rapid decompensation [80],
sudden death or death in utero [81]. Generally is not possible to correlate clinical course to
specific morphology of the tunnel, but the wide clinical spectrum could reflect variable
degree of coronary artery involvement, left ventricular outflow tract obstruction or right
ventricular outflow obstruction.
Diagnosis
Echocardiography is the modality of choice for the diagnosis of ALVT [82, 83].
Transthoracic cross-sectional imaging in a parasternal long-axis view demonstrates the
tunnel, as well as its aortic origin and left ventricular opening (Figure 21). Both two
dimensional and real-time three dimensional echocardiography have also established reliable
fetal diagnosis [71, 84]. Color-doppler study typically shows diastolic flow passing laterally
from the aorta to the left ventricle (Figure 22). Left ventricle, best assessed in short axis cuts,
usually shows some degree of hypertrophy and dilatation. MRI is also used as a second-level
modality for diagnosis of ALVT. Cardiac catheterization is actually indicated only when
associated lesions or coronary artery origins cannot be evaluated on non-invasive studies.
Differential diagnosis is crucial, considering that ALVT must be distinguished from other
lesions which cause rapid diastolic run-off of blood from aorta into left ventricle and produce
cardiac failure. Among these must be mentioned:
Sinus of Valsalva fistula
Truncus Arteriosus with valvular regurgitation
Aorto-pulmonary window
Ventricular septal defect with aortic regurgitation
Coronary artery fistula
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Figure 21. Parasternal long-axis view showing aorto-left ventricular tunnel. Ao (aorta); LV
(left ventricle); T (tunnel). Reprinted from Kathare P et al. [85].
Figure 22. Modified Parasternal long-axis view showing aorto-left ventricular tunnel and its diastolic
run-off into left ventricle. Ao (aorta); LV (left ventricle); RVOT (right ventricular outflow tract);
T (tunnel). Reprinted from Kathare P et al. [85].
Methods
Principles of Surgical Management
Without intervention, most of the patients die early in life for congestive heart failure.
Optimal surgical timing is within the first six months of life, due to the evidence of
normalization of left ventricular size and function if correction is performed in that time
window. Lack of support to the right or left aortic leaflet can result in progressive aortic
regurgitation, although surgical technique may importantly influence the long-term aortic
valve function.
Principles of surgical correction include:
Closure of the aortic and ventricular openings
Restoration of aortic valve function
Ensuring normal coronary perfusion
Relief of left or right ventricular outflow tract obstruction
Aortic Root Involvement in Congenital Heart Defects
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Several techniques has been proposed in surgical management of ALVT, as direct closure
of tunnel openings eventually associated with additional measures like external tunnel
ligation or obliteration [86, 87]. Currently, the best surgical approach is a separate patch
closure of both the aortic and ventricular opening, through tran-aortic and trans-infundibular
approach, along with internal plication and reduction of the lateral tunnel wall [88]. The
rationale of this strategy lies in these keypoints:
Closing the ventricular opening in order to avoid an open blind-ending pouch with
high pressure. This condition might create a rightward buldging of the lateral tunnel
wall generating right ventricular outflow tract obstruction.
Restoring an adeguate anatomical support to the aortic valve and anulus by means of
two separate patches
Closing the ventricular opening of the tunnel from the right side excluding its thin
lateral wall.
Maintaining integrity of the aortic root avoiding external opening of the tunnel
The Two-Patch Technique
Surgery is performed on cardiopulmonary bypass (CPB) under mild hypothermia. The
tunnel is externally compressed during antegrade cold blood cardioplegia delivery in order to
avoid left ventricular run-off. If a coronary arise from the tunnel, so as to preclude external
compression, retrograde cardioplegia may be delivered.
A transverse aortotomy is performed. The aortic valve and coronary ostia are carefully
inspected to exclude anomalies. An additional dose of selective cardioplegia is delivered in
the coronary ostia.
A transverse ventriculotomy is carried in the sub-pulmonary infundibulum around 1 cm
below the ventriculo-arterial junction.
The aortic and ventricular openings of the tunnel are identified. A right angled clamp is
then passed through the aortic opening of the tunnel and an incision is made into its lateral
wall, thus creating a tunnel to right ventricular communication which is essentially a
iatrogeninc sub-aortic ventricular septal defect (Figure 23).
Figure 23. Schematic view of ALVT showing aortic and left ventricular openings and the tunnel course
(right panel). Right-angled clamp passed through the tunnel (left panel). Reprinted from Mueller C
et al. [88].
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Figure 24. Two-patch technique for ALVT correction: The aortic opening is closed through the
aortotomy and the ventricular opening through the right ventriculotomy The latera tunnel wall is
plicated and deduced (left panel). Reprinted from Mueller C et al. [88].
Figure 25. Final result of two-patch technique. Note the direct closure of aortotomy and right
ventriculotomy. Reprinted from Mueller C. et al. [88].
The opened lateral wall of the tunnel is then exposed from the right ventricular side.
The aortic opening is closed through the aortotomy using xenopericardial patch secured
with polipropilene running suture.
The ventricular opening is closed through the infundibular approach using the same
material and technique of the aortic opening (Figure 24). The thin lateral wall of the tunnel is
then plicated and reduced by means of direct suture.
Aortotomy and right ventriculotomy are closed by direct suture (Figure 25).
The aortic cross-clamp is removed and surgical result is checked by echocardiography.
Associated Anomalies
Coronary origin from the tunnel pose a surgical challenge. The orifice of a coronary
artery can be displaced above or below the origin of the tunnel, or it may lie within the tunnel.
When the coronary ostium arise proximally within the tunnel a patch closure of the aortic
opening distally so as to ensure coronary pefsusion of the aortic root has been described [87].
More distal origin of the coronary artery from the tunnel needs detachment of coronary botton
and its reimplantation in higher position into the ascending aorta.
Atresia of the left or right coronary artery has also been reported. More than two orifices,
single coronary ostium, and intramural course in the posterior wall of the tunnel have all been
observed [89].
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Bicuspid aortic valve, with or without stenosis, is present in about 20% of cases.
Pulmonary valve stenosis is reported in 5% of cases [89].
Discussion
Outcome and Current Perspectives
Operative outcome, overall survival and freedom from reintervention after ALVT have
been recently elucidated in a multicentric study performed by the European Congenital Heart
Surgeons Association and the World Society for Pediatric and Congenital Heart Surgery
(ECHSA – WSPCHS) [89]. Data were collected from 15 participating centers. The cohort
was represented by 42 patients (85% with ALVT) who underwent surgical correction of
aortic-left/right ventricular tunnel between 1987 and 2018.
Median age at diagnosis and operation in ALVT group was 25 days, with 77.8% of
patients undergoing surgery before six months of age. In 30% of ALVT patients, preoperative
moderate to severe aortic regurgitation (AR) was present.
In patients with right ventricular tunnels the median age at diagnosis and correction was 6
years.
Perioperative (30 days) mortality for the ALVT group was 8.3%. The median follow-up
of the ALVT survivors was 22 years and the freedom from reintervention was 90%. 10%
underwent reoperation for residual aortic stenosis (AS). The overall mortality of the entire
cohort of patients was 9.5%: The cause of death was mainly related to residual AS, thus
emphasizing the importance of fixing the aortic valve at the time of initial repair.
Surgical technique employed was not uniform in the ALVT group, with only 33% of
patients who underwent closure of both aortic and ventricular ends of tunnel (two-patch
technique).
Figure 26. Graphic representation of preoperative prevalence and postoperative evolution of AR in
ALVT patients. AR degree remains stable at late follow-up in 85% of patients, with only three patients
showing severe aortic regurgitation. AI (aortic regurgitation); AoLVT (aortico-left ventricular tunnel).
Reprinted from Protopapas ME et al. [89].
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Aortic regurgitation, probably due to turbulence-related damage of the leaflets, is a major
concern in ALVT management. In this cohort preoperative AR, ranging from mild to severe,
was present preoperatively in 42% of patients. Interestingly, at late follow-up, 85% of patients
had stable or improved AR with only three patients showing severe regurgitation (Figure 26).
The simple ALVT repair seems to fix aortic regurgitation in most cases. However, this is in
contrast with the results of Martins et al. who found that half of the ALVT patients required
late aortic valve replacement [71].
The ECHSA-WSPCHS study [89] is actually the large retrospective multicentric series
on surgical management of aorto-ventricular tunnel, and three important “take home
messages” can be extrapolated from it:
1. Early surgical repair, immediately after diagnosis, is recommended even in
asymptomatic patients.
2. Coexistence of significant aortic stenosis requires aggressive treatment, even with
Ross procedure if needed, since a more conservative approach is associated with high
early mortality and higher reoperation rate
3. Preoperative AR is a frequent finding in ALVT but remains stable postoperatively in
most cases later in life. ALVT repair alone seems sufficient to prevent evolution of
valvular regurgitation.
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