Ann Thorac Surg 1999;67:1478-1480
© 1999 The Society of Thoracic Surgeons
Case Reports
Coronary ostial patch angioplasty in children
Kulvinder S. Lall, FRCSa,
Eccard Dombrowicz, MDa,
Thaseegaran M. Pillay, FRCSa,
James C.S. Pollock, FRCSa
a Department of Paediatric Cardiac Surgery, Royal Hospital for Sick Children, Yorkhill, Glasgow, Scotland, UK
Accepted for publication October 20, 1998.
Address reprint requests to Mr Lall, Department of Paediatric Cardiac Surgery, Royal Hospital for Sick Children, Yorkhill, Glasgow G3 8SJ, Scotland
e-mail: kulvinder1{at}aol.com
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Abstract
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Patch angioplasty for coronary artery ostial stenosis was first reported in adults in 1952 and only sparsely used until 1983 when Hitchcock reviewed the technique in adults. We present two cases that highlight our use of this technique in inflammatory diseases of the aorta in children. Patch angioplasty restores physiologic perfusion of the coronary artery tree, preserves conduit material for further procedures later in life, and allows subsequent percutaneous transluminal coronary angioplasty of stenosis. For these reasons we believe that patch angioplasty holds undoubted advantage over the available alternatives.
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Introduction
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Left main stem coronary artery stenosis is a rare condition in children, and the natural history of this condition leads to inevitable death. Patch angioplasty for coronary artery ostial stenosis was first reported in adults in 1952 and only sparsely used until 1983 when Hitchcock [1] revived the technique in adults. We present two cases that highlight our use of this technique in inflammatory diseases of the aorta in children.
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Patient 1
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A 9-year-old girl was referred with a 6-week history of severe central chest pain associated with tachycardia and rashlike flushes to her neck and face. Examination revealed hypertension and abdominal bruits. She continued to have chest pain of an anginal nature and on several occasions had evidence of anterior ischemia as demonstrated by electrocardiography. Cardiac catheterization revealed tight ostial stenosis of both left and right coronary arteries with a normal distal coronary artery tree. Despite maximal medical therapy her angina worsened and 6 days after admission she was taken to the operating theater for coronary revascularization.
A pericardial patch was taken and cardiopulmonary bypass established. The patient was cooled to 28°C, the cross clamp applied, and cardioplegic solution given. The aorta was opened obliquely and found to be markedly thickened with a grossly abnormal macroscopic appearance. The right coronary ostia could not be identified, and the left was a pinhole in size. Her right internal mammary artery was placed onto the right coronary artery. The main pulmonary trunk was retracted and not divided. The left ostial stenosis was identified and a transverse arteriotomy was made for 1.5 to 2 cm. The patch was sewn into place with a 7-0 polypropylene suture in a continuous fashion, and the aorta closed with 5-0 polypropylene. The left internal mammary artery was anastomosed to the left anterior descending coronary artery. The patient was rewarmed and weaned from cardiopulmonary bypass.
Histology revealed Takayasu’s disease. She was discharged 2 weeks later. Cardiac catheterization 2 months postoperatively showed excellent flow through both the patch angioplasty and the grafts.
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Patient 2
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A 9-year-old girl experiencing episodes of crushing central chest pain that was believed to be angina was referred for coronary arteriography. This confirmed a tight left ostial stenosis with a normal distal coronary artery tree (Fig 1). Because of ischemic changes, an operation was performed the following day.
A pericardial patch angioplasty to the left coronary ostium was performed, and the patient was weaned from cardiopulmonary bypass with ease. She was discharged home 7 days later. Cardiac catheterization at 6 months showed good flow (Fig 2). Histology showed middle aortic syndrome.
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Fig 2. Postoperative angiogram at 6 months in patient 2 showing patent left main stem coronary artery.
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Comment
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In children coronary ostial stenosis is rare with 99% mortality if untreated [2]. Congenital lesions such as membranous obstruction, hypoplasia, or atresia of the ostium or inflammatory disorders such as Takayasu’s, Kawasaki, and middle aortic syndrome cause these lesions. Takayasu’s arteritis is an inflammatory disorder of the aorta with unknown cause that in 8.5% of cases shows narrowing of the coronary artery ostium. Described by Dr Tomisaku Kawasaki in 1967, Kawaskai disease is an acute vasculitis of small- and medium-sized blood vessels, and involvement of the coronary arteries is common (9%). Middle aortic syndrome occurs with unknown origin and causes inflammation of the aortic media. These diseases tend to cause aneurysms of the artery, which can lead to stenosis although stenosis can occur as a primary event. The male to female ratio is 1:2.5, and age of onset is at 10 years. Fever is the most frequently noted clinical symptom, followed by abdominal, joint, and muscle pain. Examination reveals impaired circulation of the upper extremities, cardiac and vascular murmurs, hypertension, impaired cerebral circulation, and visual disorder. Murmurs are found over the chest wall, the cervical area, and abdomen. Absence of pulsation in the upper extremities occurs in 66% of patients [3].
Children with severe coronary stenotic lesions present with myocardial ischemia. At angiography, coronary ostial stenosis is easily missed. An aortic sinus injection should be performed to look for ostial stenosis if either no arterial pressure is recorded when the catheter is engaged or there is no reflux of contrast into the aortic sinus.
Patch angioplasty was first reported in adults in 1952 with unacceptably high mortality. The technique was superseded by conventional coronary artery bypass grafting. In 1983 Hitchcock revived this technique for adults with coronary ostial stenosis, with cardiopulmonary bypass facilitating the operation [1]. With Hitchcock’s strict criteria (New York Heart Association class III/IV angina; isolated ostial lesion more than 50%; absence of distal coronary disease; normal left ventricular function; no previous myocardial infarction), we believe that in children, the patch angioplasty has many advantages over the alternatives. It restores physiologic perfusion of the coronary artery tree by obviating the need for retrograde flow to some vessels with associated decrease in perfusion pressure—the so-called Prizometer principle. It preserves conduit material for further procedures later in life and also allows percutaneous transluminal coronary angioplasty of subsequent stenosis developing distally. The material for performing the repair is readily available and, like the patient’s own, will not in any way be immunogenic. The inflammatory process that affects the aorta and its ostia may also cause stenosis at the origin of either internal mammary artery. For these reasons we believe that patch angioplasty holds undoubted advantage over the alternatives. In our first case the patch was used in conjunction with the left internal mammary artery as an adjunct. Postoperative angiography revealed a widely patent ostia at the site of the patch angioplasty; thus, a patch alone was used in the second case.
Pericardial patches are well established in aortic root enlargement. Long-term results of these patches in adults have found the patch to be very stable with no evidence of either aneurysm formation or rupture. Polytetrafluoroethylene and Dacron grafting can cause aneurysmal dilatation at suture lines, which is even more pronounced in the inflammed aorta as in Takayasu’s and Kawasaki’s [4]. Other autologous tissues such as atrial appendage and saphenous vein graft have been used with as yet no long-term follow-up [5].
Other procedures for ostial stenosis are still controversial. Percutaneous transluminal coronary angioplasty has been performed on patients with coronary lesions that result from Kawasaki disease. However, the inherent risks of angioplasty to a left main stem coronary artery are high, with risk of dissection, infarction, embolism, and fatal arrhythmias. Restenosis rate is 50% at 1 year [6]. Transaortic endarterectomy has the additional hazard of dissection in coring out too much of the arterial wall in these often fibrotic and calcified vessels [7].
Coronary artery bypass grafting has been performed successfully in children since 1985. Indications until 1990 were strictly limited because of unknown natural history of these coronary artery lesions. Saphenous vein graft patency rate was considered to be low in younger children (less than 8 years). In addition, saphenous vein grafting to a small coronary artery seemed to require a very high level of surgical skill. Whether growth of the graft after bypass occurs has not been established. Recurrent stenosis at the origin of the vein graft is reported to be 60% at 10 years [8].
It must not be forgotten, however, that the underlying inflammatory process must be treated, and that successful outcome depends not only on the type of procedure, but the timing of that procedure and aggressive medical treatment of the aforementioned underlying process. We believe that although rare, coronary ostial stenosis in children poses a difficult problem for which pericardial patch angioplasty provides an effective and simple solution, making it a far more attractive procedure to perform over the alternatives available.
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References
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Hitchcock J.F. The return of surgical angioplasty of the left main coronary artery. Int J Cardiol 1985;7:311-314.[Medline]
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Epstein F.H. International trends in mortality and morbidity from ischemic heart disease. Ann Clin Res 1988;20:21-25.[Medline]
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Maeda M., Kobayashi M., Okamoto S., et al. Aortitis syndrome in children: clinical observation of 35 cases in Japan. Acta Paediatr 1997;39:280-284.
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Ascer E., Collier P., Gupta S.K., Veith F.J. Reoperation for polytetrafluoroethylene bypass failure: the importance of distal outflow site and operative technique in determining outcome. J Vasc Surg 1987;5:298-310.[Medline]
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Kitayama H., D’Orsogna L., Karl T. Isolated non-atherosclerotic coronary stenosis: surgical angioplasty of the left coronary ostium using an anterior approach. Asia Pac Heart J 1996;5:124-126.
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Ino T., Akimoto K., Ohkubo M., et al. Application of percutaneous transluminal coronary angioplasty to coronary arterial stenosis in Kawasaki disease. Circulation 1996;93:1709-1715.[Abstract/Free Full Text]
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Ohara K., Kasegawa T., Ando T. Surgical treatment for coronary artery disease associated with aortitis syndrome. Kyobu Geka 1986;39:423-428.[Medline]
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Suma K., Takeuchi Y., Shiroma K. Early and late post operative studies in coronary artery lesions resulting from Kawasaki disease in children. J Thorac Cardiovasc Surg 1982;84:224-229.[Abstract]
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Abstract
Introduction
