Eur J Cardiothorac Surg 1998;14:214-217
© 1998 Elsevier Science NL
Repair of aortico-right ventricular tunnel
J.A.M. van Sona,
J. Hambschb,
P. Schneiderb,
F.W. Mohra
a Department of Cardiac Surgery, Herzzentrum, University of Leipzig, Russenstrasse 19, D-04289 Leipzig, Germany
b Department of Pediatric Cardiology, Herzzentrum, University of Leipzig, Russenstrasse 19, D-04289 Leipzig, Germany
Received 26 January 1998;
received in revised form 11 May 1998;
accepted 18 May 1998.
Corresponding author. Tel.: +49 341 8651421; fax: +49 341 8651452.
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Abstract
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Aortico-right ventricular tunnel was successfully corrected in a 15-month-old child. Both the aortic and right ventricular openings were closed with pledgeted sutures. The coronary artery anatomy was normal. At 12-month follow-up the patient is in excellent clinical condition. Before surgical intervention for aortico-right ventricular tunnel is undertaken, every effort should be made to diagnose the coronary artery anatomy, because failure to do so in the case of aberrant origin of a coronary artery may prevent successful surgical correction.
Key Words: Aortico-right ventricular tunnel
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Introduction
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Aortico-ventricular tunnel is a rare congenital malformation, in particular when the tunnel communicates with the infundibulum of the right ventricle. More than 50 cases of aortico-left ventricular tunnel have been reported
[1]
[2]
[3]
[4]. In contrast, thus far only 6 cases of aortico-right ventricular tunnel have been reported in the English literature
[5]
[6]
[7]
[8]
[9]
[10] (Table 1). Of these six patients, five were operated on; only two patients, a 3-month-old infant and a 10-year-old patient, were long-term survivors
[5]
[6]
[7]
[8]
[9]
[10]. We report a third case of successful surgical closure of this anomaly in a young child.
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Case
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A 15-month-old male child was referred to us with the diagnosis of a ventricular septal defect. The child failed to thrive in comparison with his sibling. At the age of 2 months, a continuous murmur was noted at the left sternal border together with bounding carotid and femoral pulses. Symptoms of mild congestive heart failure were treated with diuretics.
Two-dimensional echocardiographic and angiographic examinations on admission demonstrated a free communication between the right side of the aortic root and the right ventricular infundibulum (
Fig. 1
). The coronary arteries were identified as separate structures. The aortic valve was tricuspid and was competent. A ventricular septal defect and patency of the ductus arteriosus were excluded.
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Fig. 1. Angiogram of aortico-right ventricular tunnel. (A). Injection of dye into the ascending aorta demonstrates that the tunnel communicates with the right ventricular infundibulum. (B). Lateral projection shows the origin of the tunnel (black arrow) in close proximity to the right coronary ostium (white arrowhead). (C). Injection of dye into the infundibulum shows a `wash out' effect at the entrance site of the tunnel (arrow).
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The child was operated on without delay. Dissection to the left of the right coronary orifice showed a 10 mm long and 8 mm wide tunnel between the distal right sinus of Valsalva and the right ventricular infundibulum. Hypothermic cardiopulmonary bypass was instituted with an ascending aortic cannula and a single venous cannula in the right atrium. The aorta was cross-clamped and cardioplegic solution was administered into the aortic root while the tunnel was occluded with a forceps.
Through a standard oblique aortotomy a slit-like opening was seen to originate from the upper margin of the right sinus of Valsalva, 3 mm to the left and slightly cephalad to the right coronary ostium. The right sinus of Valsalva was moderately dilated. Both coronary arteries were normal in origin and distribution. Passage of a probe through the tunnel into the right ventricular infundibulum confirmed the diagnosis of an aortico-right ventricular tunnel.
The tunnel was divided 2 mm from its entrance site at the right ventricular infundibulum. Under direct vision of the aortic root structures, the aortic end of the tunnel was subsequently oversewn with 4 matress polypropylene sutures (Prolene, Ethicon, Somerville, NJ, USA.) supported with autologous pericardial pledgets. Closure of the aortic end of the tunnel did not result in distortion of the aortic leaflets or obstruction of the right coronary ostium. The opening in the infundibulum was similarly closed with four pledgeted sutures.
The post-operative course was uncomplicated. The child was discharged home on the sixth post-operative day. At 12-month follow-up the patient was thriving well. Echocardiographically there were no residual cardiac abnormalities; notably, there was no aortic regurgitation.
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Discussion
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Aortico-right ventricular tunnel is an abnormal pathway that has its orifice above, or in, the upper portion of the right sinus of Valsalva, just to the left of the orifice of the right coronary artery, and that enters the infundibulum of the right ventricle. This anomaly, similar to the more common aortico-left ventricular tunnel, is believed to be related to a congenital weakness in the region of the right sinus of Valsalva. Associated abnormal origin, or absence, of the right or left coronary ostium has been reported
[5]
[6]
[8]
[10]. Hemodynamically, the anomaly produces a left-to-right shunt with resulting pressure and volume hypertrophy of the right ventricle.
Before surgical intervention is undertaken, every effort should be made to diagnose the coronary artery anatomy, because failure to do so in the case of aberrant origin of a coronary artery may prevent successful surgical correction
[8]. Echocardiography may establish the correct diagnosis by demonstrating the two ends of the tunnel connecting the aorta and the (often dilated) right ventricle and by obtaining high-velocity systolic and diastolic Doppler flow signals in the tunnel. Angiography remains an important adjunctive diagnostic technique, particularly to exclude coronary artery abnormalities.
It is our belief that repair of aortico-right ventricular tunnel should be undertaken as early in life as possible to minimize damage to the right ventricle and to avoid the development of obstructive pulmonary vascular disease. The repair should be individualized. As in our case, in the presence of a relatively long tunnel and sufficiently available autologous aortic wall, the aortic end of the tunnel may be closed without the use of a patch. Using this technique, the tunnel is divided close to its entrance site at the infundibulum and the aortic end of the tunnel is closed with pledgeted matress sutures or a continuous suture. This is performed under continuous observation of the aortic root structures, thus avoiding distortion of the aortic cusps or obstruction of the right coronary orifice. The advantage of this technique is that it may result in a slight reduction of the (often increased) aortic root size. In the presence of a short tunnel, a large aortic orifice, or aberrant origin of a coronary artery from the tunnel, patch closure of the aortic end of the tunnel may be preferred to prevent the aforementioned complications
[9]
[10]. Direct closure of the opening in the right ventricular infundibulum has not posed a problem in our patient. Alternatively, especially in the presence of a large infundibular opening, patch closure may be performed
[9]
[10]. Although interventional closure of an aortico-right ventricular tunnel may constitute an alternative therapeutic option, we believe that the proximity of the ostium of the tunnel to the right coronary ostium and the aortic valve leaflets would compromise the safety of such procedure. For the same reason surgical ligation of the tunnel without the use of cardiopulmonary bypass is a less ideal option.
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References
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- Levy M.J., Lillehei C.W., Anderson R.C., Amplatz K. Edwards, J.E. Aortico-left ventricular tunnel. Circulation 1963;27:841-853.[Abstract/Free Full Text]
- Levy M.J., Schachner A., Blieden L.C. Aortico-left ventricular tunnel: collective review. J Thorac Cardiovasc Surg 1982;84:102-109.[Abstract]
- Hovaguimian H., Cobanoglu A., Starr A. Aortico-left ventricular tunnel: a clinical review and new surgical classification. Ann Thorac Surg 1988;45:106-112.[Abstract]
- Duveau D., Baron O., Michaud J.L., Lefèvre M., Laboux L., Dupon H. Aorto-left ventricular tunnel. Long-term follow-up and therapeutic implications. Arch Mal Coeur 1989;82:785-789.
- Bharati S., Lev M., Cassels D.E. Aortico-right ventricular tunnel. Chest 1973;63:198-202.[Abstract/Free Full Text]
- Saylam A., Tuncali T., Ikizler C., Aytac A. Aorto-right ventricular tunnel. A new concept in congenital cardiac malformations. Ann Thorac Surg 1974;18:634-637.[Medline]
- Jureidini S.B., de Mello D., Nouri S., Kanter K. Aortico-right ventricular tunnel and critical pulmonary stenosis: diagnosis by two-dimensional and Doppler echocardiography and angiography. Pediatr Cardiol 1989;10:99-103.[Medline]
- Kleikamp G., Minami K., Thies W.R., Dohmann R., Raute-Kreinsen U., Meyer H., Körfer R. Aorta-right ventricular tunnel with a rudimentary valve and an anomalous origin of the left coronary artery. J Thorac Cardiovasc Surg 1992;104:1759-1760.[Medline]
- Westaby S., Archer N. Aortico-right ventricular tunnel. Ann Thorac Surg 1992;53:1107-1109.[Abstract]
- Rosengart T.K., Redel D.A., Stark J.F. Surgical repair of aorto-right ventricular tunnel in an infant. Ann Thorac Surg 1993;55:520-522.[Abstract]
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Abstract
Introduction
