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Ann Thorac Surg 1996;61:1805-1810
© 1996 The Society of Thoracic Surgeons

Original Article: Cardiovascular

Aortico–Left Ventricular Tunnel in Fetuses and Infants

Miguel Sousa-Uva, MD, Anita Touchot, MD, Laurent Fermont, MD, Dominique Piot, MD, Anne Lise Delezoide, MD, Alain Serraf, MD, Francois Lacour-Gayet, MD, Régine Roussin, MD, Jacqueline Bruniaux, MD, Claude Planché, MD

Hôpital Marie Lannelongue, Le Plessis Robinson, and Institut de Puériculture Brune, Paris, France

Accepted for publication February 20, 1996.


   Abstract
Top
 Footnotes
 Abstract
Introduction
 Patients and Methods
 Comment
 References
 
Background. Aortico–left ventricular tunnel is a rare congenital abnormal communication between the aorta and the left ventricle presenting in early childhood as aortic regurgitation and cardiac failure. This condition has rarely been reported in fetuses. Operation is the only treatment, and postoperative aortic incompetence could be related to the age or the type of repair.

Methods. We conducted a retrospective, two-institution review, from 1983 to 1995, of aortico–left ventricular tunnel diagnosed in utero and before 6 months of age.

Results. Three cases of aortico–left ventricular tunnel were diagnosed in utero by Doppler echocardiography between 22 and 24 weeks' gestation. Prenatal aortico–left ventricular tunnel was associated with severe left ventricular dysfunction, aortic valve anomalies, and fetal hydrops. One death occurred in utero and one immediately after birth, and in 1 case pregnancy was interrupted. In these 3 cases the diagnosis was confirmed by autopsy. Three neonates and 2 infants had the diagnosis of aortico–left ventricular tunnel made after birth and underwent successful surgical repair. At short and midterm follow-up all patients are alive and aortic valve regurgitation is absent or trivial.

Conclusions. This series shows that aortico–left ventricular tunnel covers an anatomic spectrum of lesions. Cases diagnosed in utero by Doppler echocardiography are characterized by severe ventricular dysfunction, associated aortic valve lesions, and poor outcome. Postnatal cases represent the more favorable end of the spectrum, with no associated lesions, and can be repaired without mortality and with good functional results.


   Introduction
Top
 Footnotes
 Abstract
 Introduction
Patients and Methods
 Comment
 References
 
Aortico–left ventricular tunnel is a congenital abnormal paravalvular pathway between the aortic root and the left ventricular cavity bypassing the aortic valve. This cardiac anomaly was first described by Levy and Lillehei in 1963 [1]. Since that time about 70 patients have been reported in the literature [2, 3]. The clinical picture is that of aortic regurgitation. Age of presentation and severity of symptoms depend on the size of the tunnel. Neonatal or infant presentation reflects a large communication with severe left ventricular volume overload. Operation is indicated as soon as the diagnosis is established to prevent rapid deterioration and death [4]. Little information is available on the outcome of this rare condition when symptoms are present in early life, and virtually none when the diagnosis is made in utero [5, 6]. The objective of this study is to report the prenatal and postnatal spectrum of aortico–left ventricular tunnel and to address some diagnostic and therapeutic issues.


   Patients and Methods
Top
 Footnotes
 Abstract
 Introduction
 Patients and Methods
Comment
 References
 
The files of two pediatric heart institutions were retrospectively reviewed from 1983 to 1995 to search for patients with a diagnosis of aortico–left ventricular tunnel made before 6 months of age. One of the institutions is also a referral center for fetal congenital heart disease diagnosis, whereas the other is a pediatric cardiac surgical unit. Three cases of aortico–left ventricular tunnel were diagnosed in utero and 5 during the neonatal or early infancy period.

Fetal Aortico–Left Ventricular Tunnel
Three 27- to 30-year-old primigestation primipara women were refered to fetal echocardiographic examination after routine obstetric echography had shown fetal cardiomegaly or fetal hydrops. Fetal Doppler echocardiography performed between 22 and 24 weeks' gestation revealed severe ventricular dysfunction/dilatation and myocardial hypertrophy in the 3 cases (Fig 1Go). Dysplastic regurgitant aortic cusps and a preserved general cardiac architecture were constant features of these fetal hearts. In 2 cases Doppler examination also showed significant aortic valve stenosis. In 1 case paravalvular aortoventricular reflux was identified on color Doppler imaging, suggesting the diagnosis of aortico–left ventricular tunnel (see Fig 1Go). The sum of these findings clearly pointed toward the diagnosis of aortico–left ventricular tunnel in 2 cases, the third case being diagnosed as a poorly tolerated critical aortic stenosis. Serial echocardiographic examinations demonstrated the persistence in the 3 cases of cardiac failure with fetal hydrops and progressive deterioration of left ventricular shortening fraction. The parents were informed of the type of malformation, severity of the ventricular dysfunction, and the unpredictability of postnatal recovery. Two mothers wanted to proceed with interruption of pregnancy. According to French law, their medical records were submitted to an ethical committee, and permission for termination was granted in 1 case. The other case was spontaneously interrupted by fetal death at 27 weeks' gestation. The case diagnosed as critical aortic stenosis was regularly followed up until term, at which time a ceserean section delivered a 2.9-kg male child in severe heart failure. This baby died a few hours after birth before he could be transferred to a surgical unit.



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  		Fig 1. . Fetal Doppler echocardiography at 24 weeks. The left ventricle (LV) is dilated and there is a regurgitant flow (arrow) at the aortic valve level. (AO = aorta.)

 
Anatomic examination of the fetal hearts showed the characteristic features of aortico–left ventricular tunnel very similar to those observed in postnatal hearts (Fig 2Go). However, in contradistinction with postnatal aortico–ventricular tunnels, associated extensive myocardial fibrosis was present in all cases and 2 specimens displayed in addition severe aortic valve dysplasia with almost fixed aortic cusps.



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  		Fig 2. . Fetal autopsy heart specimen, frontal section through the ascending aorta and left ventricle. The stylet is in the aortico–left ventricular tunnel. The left ventricular wall is hypertrophied.

 
Neonatal and Infant Aortico–Left Ventricular Tunnel
Five children (4 boys and 1 girl) presented with symptoms soon after birth or during early infancy. Systolic and diastolic murmurs, bouncing peripheral pulses, an enlarged cardiac silhouette on chest roentgenograms, and electrocardiographic left ventricular hypertrophy were noted in all children. Symptoms of cardiac failure were moderate to severe in 2 neonates and mild in the remaining patients. The diagnosis of a left ventricular tunnel was formally made by Doppler echocardiography in 4 of the patients based on the disclosure of a paraaortic anterior anechogenic structure where color Doppler echocardiography showed antegrade systolic and retrograde diastolic regurgitant flow (Figs 3, 4GoGo). The diameters of the tunnels varied between 3 and 8 mm and left ventricular shortening fraction between 0.23 and 0.26. In 1 patient the diagnosis could not be formally ascertained preoperatively even after catheterization and angiography. In this patient, angiography showed an irregularly contoured, nonruptured aneurysm of the right sinus of Valsalva and grade 2 aortic incompetence (Fig 5Go). On two-dimensional Doppler echocardiography the proposed diagnosis in this patient was that of a large aneurysm of the sinus of Valsalva protruding into the pulmonary infundibulum and a prolapsed right aortic cusp with grade 2 aortic regurgitation (Fig 6Go). Among the 5 patients, this was the only patient with aortic regurgitation. Overall, left heart catheterization was done in only 2 patients and did not add any contributive information.



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  		Fig 3. . Two-dimensional echocardiogram, parasternal long-axis view, showing the aortico–left ventricular tunnel (arrow) bypassing the aortic valve. (AO = aorta; LV = left ventricle; RV = right ventricle; T = tunnel; triple arrows = aortic origin of tunnel.)

 


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  		Fig 4. . Doppler color-flow imaging in the parasternal long-axis view. Diastolic regurgitation is shown through the tunnel (T). (AO = aorta; LA = left atrium; LV = left ventricle.)

 


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  		Fig 5. . Aortogram showing an irregularly contoured "aneurysm" or pouch (P) of the right sinus of Valsalva with filling of the left ventricle (LV). (AO = ascending aorta.)

 


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  		Fig 6. . Echocardiographic short-axis parasternal view above the aortic leaflets showing the aortic origin of an aortico–left ventricular tunnel (T) misdiagnosed as an aneurysm of the sinus of Valsalva (same patient as in figure 5Go). (AO = aorta; RV = right ventricle.)

 
Three patients were operated on during the neonatal period (3, 19, and 28 days) and 2 at 5 months of age. In all patients there was an external anterior bulging of the aortic root extending over the origin of the pulmonary artery with a thrill that was abolished by firm pressure on the bulge. On cardiopulmonary bypass and aortic cross-clamping the aortic orifice of the tunnel was always found above the right coronary sinus or the right coronary–left coronary commissure. The ventricular orifice was closed by a polytetrafluoroethylene or Dacron patch in 3 patients. In 2 of them the right coronary ostium originated from the tunnel itself, preventing the closure of the aortic end of the tunnel. In 1 patient a simple closure of the aortic end of the tunnel was performed. The remaining patient had a large defect of the right sinus from the level of the commissure to the aortic "annulus," resulting in a right coronary leaflet arising from a fibrous bar detached from the aortic wall. A large polytetrafluoroethylene patch was used, attaching the lower border of the tunnel to the upper limit of its aortic orifice with reattachment of the "floating" right aortic cusp to this patch.

There were no operative deaths or significant morbidity. The follow-up ranges from 6 months to 11 years (median, 37 months). All patients are alive and developing normally. Trivial or no aortic leak and a tightly closed aortico–left ventricular tunnel was noted in all patients at follow-up on two-dimensional Doppler echocardiography. Left ventricular dimensions and shortening fraction were normal.


   Comment
Top
 Footnotes
 Abstract
 Introduction
 Patients and Methods
 Comment
References
 
Aortico–left ventricular tunnel is a rare malformation of unknown origin. Many pathogenic theories have been proposed including an abnormal coronary artery, a malformation of the bulbus cordis, or an aortic wall weakness allowing communication with ventricular sinusoids [1, 7, 8]. A common origin with ruptured aneurysm of the sinus of Valsalva has been suggested by some authors but denied by others [9–11]. Operation is the well-established form of treatment, but late aortic valve competence depends on the type and timing of the repair. This study has collected both prenatal and postnatal cases of aortico–left ventricular tunnel. It shows that aortico–left ventricular tunnel detected in utero is a severe and usually lethal form of this congenital malformation as opposed to the classical postnatal form, which has an excellent vital and functional prognosis after operation.

In Utero Diagnosis
An increasing number of congenital heart lesions are being detected during fetal life by systematic ultrasound imaging [12]. However, until very recently, in utero diagnosis of aortico–left ventricular tunnel had seldom been reported in the literature [13, 14]. The present study reports in utero diagnosis of aortico–left ventricular tunnel with serial fetal echocardiographic examination and correlation between echocardiographic findings and postnatal or autopsy findings. In utero diagnosis of aortico–left ventricular tunnel was made possible by the finding of fetal ventricular dysfunction and dilatation on routine obstetric ultrasound screening as early as the 18th to 20th gestational week coupled with systematic referral to a fetal cardiology unit.

The association of a normal fetal cardiac architecture and identification of color-Doppler echocardiography–documented aortic regurgitation should suggest the diagnosis of aortico–left ventricular tunnel because significant regurgitation at the sigmoidal level is extremely rare in the fetus. A reasonably sure diagnosis of aortico–left ventricular tunnel could be made in 2 of our 3 cases because either an anechogeneic paraaortic structure was seen or the regurgitation color jet was located inside the left ventricular cavity. Otherwise, aortico–left ventricular tunnel can only be suspected among other possible diagnoses such as aneurysm of the sinus of Valsalva, aortic stenosis, and aortic incompetence. Another erroneous diagnosis is tetralogy of Fallot as reported by Cook and associates [14], who made a correct diagnosis in utero in only 1 of 4 cases.

An important finding of this study was that aortico–left ventricular tunnel can be a severe fetal cardiac malformation when the communication is large enough to produce major chronic volume overload. Associated aortic valve stenosis or incompetence, present in all prenatal cases, further aggravated the clinical picture, leading to severe ventricular hypertrophy, dilatation, and myocardial fibrosis. Although Cook and associates [14] did not note significant aortic valve malformations, all 4 of their fetuses had dilated thick left ventricles with decreased shortening fraction. Prenatal diagnosis of congenital heart defects should result in important benefit for both the patient and the family through immediate stabilization and treatment upon birth in appropriately equipped centers. Unfortunately, for logistic reasons, this could not be achieved in 1 of our cases. However, it is uncertain whether this child would have survived even with immediate repair at birth. Although only a few therapeutic interventions are possible in utero at present, one can speculate that a fetus with poorly tolerated aortico–left ventricular tunnel and no associated aortic valve anomalies could, in the future, become a candidate for in utero repair [15, 16].

Postnatal Presentation
DIAGNOSIS.
In most cases in the literature, as in the 5 patients presenting after birth reported herein, aortico–left ventricular tunnel was not detected prenatally. This could be due to the fact that the milder, more common forms of aortico–left ventricular tunnel (smaller communication without major aortic valve anomalies) that reach postnatal life produce moderate ventricular dysfunction and therefore could remain difficult to detect in utero. Generally aortico–left ventricular tunnel presents in childhood or infancy and the natural course depends on the degree of aortic regurgitation. In this series, the diagnosis of aortico–left ventricular tunnel was made by Doppler echocardiography, cardiac catheterization being performed in only 2 patients. Recognition of aortic incompetence in a neonate or young infant should suggest aortico–left ventricular tunnel as a possible diagnosis. Other lesions that must be differentiated are ruptured aneurysm of the sinus of Valsalva, coronary cameral fistula, congenital isolated aortic incompetence, and aortic incompetence with ventricular septal defect. Color-Doppler echocardiography has been recognized as the most useful technique in identifying aortico–left ventricular tunnel and distinguishing between regurgitation through both the tunnel and the aortic valve, which angiography fails to do [17–19]. In our experience, Doppler echocardiography correctly made a diagnosis in all but 1 patient (patient 5). In this patient, echocardiography showed the presence of a large aneurysm at the level of the right sinus of Valsalva with aortic incompetence due to prolapse of the right aortic cusp that occluded the ventricular orifice of the tunnel during diastole (see Fig 6Go).

TREATMENT.
Operation was performed on a semiurgent basis in the 3 neonates, all of whom had depressed left ventricular function. Several authors have reported repair of this malformation in neonates and infants with good results [5, 6, 20–22]. Eventually, all patients had complete recovery of their left ventricular function. Several techniques have been proposed to repair aortico–left ventricular tunnel without inducing aortic valve distortion. Hovaguimian and associates [23] devised a surgical classification of aortico–left ventricular tunnel and suggested that repair should be individualized according to the anatomic type. Ideally, both ends of the tunnel should be closed with a polytetrafluoroethylene or Dacron patch providing support to the right aortic leaflet, which is critical to the preservation of aortic valve competence and avoidance of subpulmonary obstruction [24, 25]. However, as demonstrated by 2 of our cases (patients 2 and 3), the right coronary ostium can arise from the tunnel itself, and closure of the tunnel's aortic orifice would compromise the patency of the right coronary artery. Bharati and colleagues [26] have observed an autopsy case of aortico–right ventricular tunnel where the right coronary artery arose from the distal part of the tunnel, and in a literature review by Hovaguimian and associates [23] the right coronary ostium could not be found in 6 cases. Horvath and associates [3] have also reported 2 cases of an abnormal origin of the left coronary artery in 1 patient and the right coronary artery in the other arising from the tunnel itself. These authors excised and reimplanted the coronary artery button before closing the aortic orifice. We preferred to simply close the ventricular end of the tunnel when careful inspection of the right aortic sinus did not identify a coronary ostium.

OUTCOME.
Our 5 patients have been followed up between 3 months and 11 years, and no significant aortic incompetence has been detected. Late reoperation for aortic regurgitation in 50% of patients 4 to 5 years after repair has been reported by Serino and colleagues [27]. These authors believe that early operation did not seem to prevent late deterioration of the aortic valve competence, but the youngest patient was operated on at 1 year of age, and all but 1 had the defect closed by direct suture, which, we believe, could have caused distortion of the aortic cusps [27]. The occurrence of late aortic incompetence after tunnel closure is due to preexisting aortic valve anomalies, lack of support of the aortic "annulus," and annuloectasia. At least the last two factors can be prevented by early tunnel closure with reestablishment of annuloseptal attachment. Whether or not late aortic insufficiency will develop in our patients operated on very early in life remains to be seen by serial follow-up.

Pathogenesis
Although it is commonly admitted that aortico–left ventricular tunnel is an entity separate from ruptured aneurysm of the sinus of Valsalva, confusion may arise in some cases because of overlapping anatomic features. The fact that some aneurysms can rupture in the left ventricle, the aneurysmal morphology of some aortico–left ventricular tunnels, and the fact that the aortic origin of aortico–left ventricular tunnel is not always located above the right coronary ostium make the distinction between tunnel and aneurysm sometimes difficult [5, 9, 18]. A developmental origin is also confirmed for aneurysms of the sinus of Valsalva by the fact that rupture can also occur in utero. Indeed, we have operated on 2 neonates soon after birth with aneurysm of the right sinus of Valsalva ruptured in the right ventricle (unpublished data), who presented a close anatomic resemblance to the intracardiac aneurysm-like type of tunnel (Hovaguimian type IV) [23]. A common etiologic factor between these two different entities could be the separation of the aortic wall from the "fibrous skeleton" of the aorta or a weakness herniation [10, 27]. In aortico–left ventricular tunnel, this process would take place in a more cephalad position, whereas in case of aneurysm of the sinus of Valsalva it would be located at a lower level, making the right ventricle the direct anatomic relationship (Fig 7Go). The cause of this right sinus weakness is unknown, but slight variations of the same basic defect during the development of the conotruncal junction could lead to either aortico–left ventricular tunnel or aneurysm of the sinus of Valsalva.



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  		Fig 7. . Right coronary sinus "weakness." (a) Projection of the interventricular septum. (b) Cephalad "weakness" in the aortic wall producing an aortico–left ventricular tunnel. (c) Caudal "weakness" of the aortic wall producing an aneurysm of the sinus of Valsalva rupturing in the right ventricle (RV). (d) Sagittal section through the ventricular–aortic junction showing the relationship between the right sinus of Valsalva and the ventricular cavities. (LV = left ventricle.) (Reproduced with permission from Lutfalla G, Binet JP, Corone P. Tunnel aorto-ventriculaire gauche et anévrisme du sinus de Valsalva rompu dans le ventricule gauche: une même malformation. Coeur 1971;2:105–27.)

 
Conclusion
This study reports a spectrum of aortico–left ventricular tunnel from the severe fetal lesion to the various neonatal or infant tunnel forms. In utero diagnosis may well improve the neonatal management of aortico–left ventricular tunnel through assessment of its prognosis and programmed assisted delivery. The neonatal or infant presentations may reflect intermediate, more favorable forms, due to smaller tunnel cross-sectional area or absence of severe aortic valve anomalies. Neonatal aortico–left ventricular tunnel can be successfully repaired with a very low operative risk and expected good late functional results.


   Footnotes
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Abstract
 Introduction
 Patients and Methods
 Comment
 References
 
Address reprint requests to Dr Planché, Pediatric Cardiac Surgery, Hôpital Marie Lannelongue, 133 Ave de la Résistance, 92350 Le Plessis Robinson, France.


   References
Top
 Footnotes
 Abstract
 Introduction
 Patients and Methods
 Comment
 References
 

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  10. Lutfalla G, Binet JP, Corone P. Tunnel aorto-ventriculaire gauche et anévrisme du sinus de Valsalva rompu dans le ventricule gauche: une même malformation. Coeur 1971;2:105–27.
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