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Ann Thorac Surg 2005;79:2153-2155
© 2005 The Society of Thoracic Surgeons
a Department of Pediatric Cardiac Surgery, Green Lane Hospital, Auckland, New Zealand
b Department of Pediatric Cardiology, Green Lane Hospital, Auckland, New Zealand
Accepted for publication December 2, 2003.
* Address reprint requests to Dr Solomon, Department of Cardiothoracic Surgery, Green Lane Hospital, Auckland, New Zealand (E-mail: nev_sheeba{at}yahoo.com).
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Abstract |
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Introduction |
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A male infant weighing 930 g was delivered by emergency cesarean section at 28 weeks gestation after intrauterine death of a twin secondary to twin to twin transfusion. The child was ventilated for 5 days and was on inotropic agents for 7 days. Early echocardiograms showed a patent ductus arteriosus that failed to close with a single course of indomethacin. A later echocardiogram showed a large patent ductus arteriosus with significant dilatation of the left atrium and left ventricle. There was mild SVAS with a peak velocity of 2.4 m/s, and the aortic valve was domed in systole, but was tricuspid and only mildly thickened. Moderate left ventricular hypertrophy was noted and left ventricular function was low to normal. There was associated mild pulmonary valvar stenosis with a peak velocity of 3.2 m/s and doming of the pulmonary valve leaflets. The baby had no features of Williams syndrome and no family history of SVAS. An electrocardiogram (ECG) at 6 weeks showed widespread mild segment elevation depression with T-wave inversion in leads V2 and V6 I and augmented ventricular left (AVL), (see Fig 1; ECG 1). The echocardiogram had shown some brightness of the papillary muscles and endocardium, and these features did give rise to concern about ischemia, although their full significance was not appreciated at this stage.
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The patient was immediately returned to the operating room. Through a median sternotomy, he was placed on cardiopulmonary bypass. The heart was dilated and cyanotic with impaired contraction. There was localized mild SVAS at the level of the sinotubular junction. The left aortic valve leaflet was small with a very short free edge, and the prominent supravalvular ridge combined to create a narrow slit-like entrance to the left coronary sinus. The left coronary ostium was normal. The right coronary ostium was likewise normal, but it was located close to the commissure between the right and noncoronary cusps that were of normal size. The leaflets were a little thickened. The supravalvular ridge was excised, and the free edge of the left leaflet was mobilized creating a wide entrance to the left coronary sinus. The aorta was widened with a single bovine pericardial patch extended into the noncoronary sinus. Pulmonary valvotomy was performed.
Postoperatively there was transient renal failure treated by peritoneal dialysis. Sternal wound closure was delayed until postoperative day 2, and the infant was weaned from inotropic agents and was extubated on postoperative day 5. Subsequently there has been progressive recovery of his left ventricular function. He is now 2 years old and is developmentally normal. The ECG shows no evidence of ischemia, and on echocardiography the patient has no residual SVAS and no aortic regurgitation. Left ventricular function appears normal, and there is no valvar pulmonary stenosis. Hyperechogenic areas persist in the left ventricular papillary muscles.
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Comment |
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Mobilization of the free edge of the left leaflet and excision of the supravalvular ridge gave excellent relief of narrowing without producing aortic regurgitation. To date there is no evidence of recurrent narrowing, and others have had similar experiences.
In view of our patient’s young age and mild aortic narrowing, we used a single patch extended into the noncoronary sinus to relieve the supravalvular stenosis. Other techniques such as the inverted, bifurcated patch described by Doty and colleagues [4] or the techniques involving enlargement of all three sinuses as described by Brom [5] and Chard and Cartmill [6] did not seem appropriate.
In retrospect, better appreciation of the significance of the ECG findings of segment elevation changes and the echocardiographic appearances suggesting endocardial injury should have led to a suspicion of coronary stenosis before surgery. However, myocardial ischemia, including papillary muscle infarction can occur in newborns with a structurally normal heart as a result of prenatal or perinatal stress [7, 8]. This was considered to have been likely in this pre-term infant, given the loss of the twin in utero and the early postnatal need for inotropic support typical of such infants. However, the persistence and even progression of the segment elevation changes in this case suggested an ongoing problem; we had incorrectly ascribed this to ductal steal.
This case report is meant to emphasize the possibility of coronary ischemia in the presence of mild supravalvular aortic stenosis. Myocardial ischemia can even occur in mild forms of SVAS and can result in major problems early in infancy.
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This article has been cited by other articles:
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  E. Arnaiz, D. Koolbergen, A. Adsuar, and M. G. Hazekamp Surgery for supravalvular aortic stenosis - the three-patch technique MMCTS, September 15, 2008; 2008(0915): 2329. [Abstract] [Full Text] [PDF]
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