Ann Thorac Surg 2000;70:1704-1706
© 2000 The Society of Thoracic Surgeons
Case report
Coronary artery bypass grafting in adult coronary artery disease due to suspected Kawasaki disease in childhood
Guido Dohmen, MDa,
Manfred Dahm, MD, PhDa,
Mathias Elsner, MDb,
Wolfgang Kasper, MD, PhDb,
Hellmut Oelert, MD, PhDa
a Department of Cardiothoracic and Vascular Surgery, Johannes Gutenberg University Mainz, Mainz, Germany
b Department of Cardiology, St. Joseph Hospital, Wiesbaden, Germany
Address reprint requests to Dr Dohmen, Department of Cardiothoracic and Vascular Surgery, Johannes Gutenberg University Mainz, Langenbeckstrasse 1, 55131 Mainz, Germany
e-mail: dohmen{at}htg.klinik.uni-mainz.de
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Abstract
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Development of coronary artery aneurysms is one typical complication of Kawasaki disease and can cause coronary artery disease even in early childhood. Information about course and outcome in adults is rare. Here, we present a 49-year-old man with serious three-vessel coronary artery disease and giant coronary artery aneurysms following suspected Kawasaki disease.
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Introduction
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Apart from rheumatic fever, Kawasaki disease (KD; synonymous with mucocutaneous lymph node syndrome) is the most frequent cause of acquired heart diseases in childhood. It represents a systemic disease with generalized vasculitis primarily involving medium-sized muscular-type arteries [1]. The etiology is still unknown and suspected to be of infectious origin. Symptoms occur in subsequent stages, beginning with fever, cervical lymphadenopathy, oral and ocular manifestations, rash, and finally, cutaneous desquamation. Mortality is determined by cardiovascular manifestations; up to 25% of the (untreated) patients develop aneurysms of the proximal coronary arteries [2]. The inflammatory process of the vascular wall leads to accelerated atherosclerosis, often causing significantly calcified lesions. Even in the absence of other risk factors, manifestation of coronary artery disease (CAD) can be anticipated to early childhood. Treatment with intravenous gamma-globulin and high doses of aspirin in the acute stage is known to reduce significantly the incidence of coronary artery aneurysms, whereas corticoids are detrimental. Once symptomatic coronary artery disease has developed and cannot be managed conservatively, revascularization will be achieved by either percutaneous transluminal coronary angioplasty (PTCA) [1] or coronary artery bypass grafting (CABG) [3].
Until today, only little is known about the late outcome of patients with cardiac manifestation of KD. Here we describe the case of a 49-year-old man with assumed KD in childhood, presenting now with significant CAD requiring bypass surgery.
A 49-year-old man was referred for recurrent self-limiting ventricular tachycardia. At the age of 12, he experienced a febrile disease with a rash of unknown etiology and cutaneous desquamation, presumably Kawasaki disease. In the following years, he developed increasing chest pain on exertion which was diagnosed and treated as hypochondriac psychosis. At the age of 21, diagnosis of acute anteroseptal myocardial infarction was confirmed by electrocardiography. Current electrocardiographic and Holter monitoring showed an old anteroseptal infarction with ongoing ST-segment elevation, left ventricular hypertrophy and ventricular arrhythmia Lown IVb. Laboratory tests were negative for acute myocardial infarction. Echocardiography revealed an enlarged left ventricle with a 30% ejection fraction and an anteroapical aneurysm. Coronary angiography demonstrated a dilated left main coronary artery with a 50% stenosis, a proximal 90% stenosis of the left circumflex artery and a proximal left anterior descending artery break-off (Fig 1). The right coronary artery had a proximal diameter of 12 mm (Fig 2) and multiple dilative changes in the following course. The distal part of the left anterior descending artery was supplied by collaterals from the right coronary artery. An attempt of PTCA (left circumflex artery) failed even with a 30-second episode of 18 atmospheres pressure due to severe calcification of the stenosis. The patient therefore was accepted for coronary artery bypass surgery. During operation, the enormous dilated right coronary artery could be clearly visualized (Fig 3). Any manipulation on the markedly calcified vessel was avoided to prevent embolization. Surgery consisted of single saphenous CABG to the circumflex artery and a left internal mammarian artery bypass to the distal left anterior descending artery. The internal mammary artery was macroscopically not calcified and with good flow. The postoperative course was uneventful and the patient was discharged in good condition.
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Fig 1. Coronary angiogram of the proximal left coronary artery. Note the severe calcification of the aneurysmatic vessel.
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Fig 2. Coronary angiogram of the dilated proximal right coronary artery. Inner diameter was calculated to be approximately 12 mm.
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Fig 3. Operative situs of the right coronary artery (RCA). Outer diameter is approximately 2 cm. (Ao = aorta.)
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Comment
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Kawasaki disease was first described in 1967 [4]. Since 1975, it is known to be complicated by coronary artery aneurysms, but retrospective analyses in adult patients with aneurysmatic coronary artery disease revealed the incidence of KD long before 1967.
To our knowledge, the patient in this report may be the oldest patient described in the literature with CAD due to KD undergoing CABG-surgery. Primary symptoms in adolescence were deplorably diagnosed and treated as hypochondric psychosis, influencing the patients personal development. Significant CAD occurred with subsequent myocardial infarction and, after a period of 28 years, the patient again became symptomatic for CAD. Coronary angiography showed aneurysmatic coronary lesions, which already had been suspected on the plain chest x-ray films, where the typical calcifications could be demonstrated.
According to the literature, PTCA is known to be successful only in patients with a short interval—6 to 8 years—between onset of KD and manifestation of CAD [5]. In patients with longer intervals, namely adults, CABG surgery is the treatment of choice, whereas in children, the small caliber of the aortosaphenous vein grafts and their limited potential for growth may reduce long-term patency rates [5, 6]. This is in agreement with our findings, because all attempts of PTCA failed due to the heavily calcified coronary artery lesions, whereas CABG surgery could be performed without problems.
Due to the usually very young age of these patients, use of at least one arterial graft to the decisive coronary artery is crucial for long-term benefit [7], but considering the systemic character of KD, the favored arterial graft should be evaluated by preoperative angiography to rule out aneurysmatic lesions [1].
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References
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Accepted for publication January 24, 2000.
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Abstract
Introduction
