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Ho-Ming Su, Wen-Chol Voon, Chong-Chao Hsieh,1 Chaw-Chi Chiu,1 Tsung-Hsien Lin, Wen-Ter Lai, and Sheng-Hsiung Sheu
Division of Cardiology, Department of Internal Medicine, and
1
Division of Cardiovascular Surgery, Department of Surgery, Kaohsiung Medical University, Kaohsiung, Taiwan.
Coronary artery aneurysms are not uncommon. They are usually arteriosclerotic in origin, and may be congenital or secondary to injury, dissection, infection, inflammation, or Kawasaki disease (KD). Herein, we report a case involving a 25-year-old male smoker with acute myocardial infarction (AMI). Coronary angiography showed triple-vessel disease, coronary artery aneurysms, and diffuse ectasia. Coronary artery bypass grafting was performed without complications. Based on his history, serologic examinations, and angiographic findings, we suspected that his coronary artery aneurysms and ectasia were the adult sequelae of KD. This case is a good reminder that KD victims may suffer from young-onset AMI.
Key Words: acute myocardial infarction, coronary artery aneurysm, Kawasaki disease (Kaohsiung J Med Sci 2004;20:399–403)
Received: February 27, 2004 Accepted: June 1, 2004 Address correspondence and reprint requests to: Dr. Tsung-Hsien Lin, Division of Cardiology, Department of Internal Medicine, Kaohsiung Medical University, 100 Shih-Chuan 1st Road, Kaohsiung 807, Taiwan.
E-mail: [email protected]
Coronary artery aneurysms are not uncommon, being found in 1.4% of necropsies performed in patients over the age of 16 years [1]. They are usually arteriosclerotic in origin [2], and may be congenital or secondary to injury, dissection, infection, or inflammation. Coronary artery aneurysm is an important complication of Kawasaki disease (KD) [3]. Herein, we describe the case of a 25-year-old male smoker who suffered from acute myocardial infarction (AMI) and who was found by angiography to have coronary artery aneurysm and diffuse ectasia.
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RESENTATIONThis 25-year-old man presented with persistent chest pain.
There was nothing significant in his medical history and no previous episodes of chest pain. He could not recall any un-usual or severe childhood illnesses. There was no family history of ischemic heart disease. However, he was a smo-ker. He was sent to our emergency room due to chest pain. Twelve-lead electrocardiogram (ECG) showed dynamic ST-T changes in leads I, aVL, and V3–V6 (Figure 1), and there was also a series of changes in cardiac enzymes (peak levels: creatinine kinase/creatinine kinase myocardial bound, 2,785/97.8 U/L; troponin I, 40.4 ng/mL). As the patient was thought to have Killip I non-ST segment elevation myocar-dial infarction (NSTEMI), he was admitted to our cardiac care unit. On admission, echocardiography showed left ventricular (LV) dilatation, LV systolic dysfunction (fraction shortening, 18%), mitral valve B hump, and LV posterior and apical wall hypokinesis (Figure 2). He received standard NSTEMI medical treatment, which relieved his chest pain. Two days after admission, coronary angiography was performed due to depressed LV function. Selective left co-ronary angiography showed three aneurysms involving the distal left main artery and the diagonal branch of the left anterior descending artery (LAD) joined by non-aneurysmal
segments. The proximal LAD was occluded and its distal vessels filled backwards from the right coronary artery and the diagonal branch. The left circumflex artery (LCX) was occluded just behind its orifice and received collateral circulation from the diagonal branch. Selective right coro-nary angiography showed diffuse ectasia and 99% dis-crete stenosis over the posterior descending artery branch (Figure 3). Due to the occurrence of AMI and coronary aneurysms in a young patient, we performed a series of serologic tests, including hepatitis B surface antigen, vene-real disease, lupus anticoagulant antibody, anti-cardiolipin antibody, and antinuclear antibody, all of which were nega-tive. Serum lipid concentrations showed: total chole-sterol, 172 mg/dL; low-density lipoprotein cholechole-sterol, 93 mg/dL; high-density lipoprotein cholesterol, 62 mg/dL; and triglycerides, 71 mg/dL. Fasting sugar was 92 mg/dL. Since coronary arteriography showed three-vessel coronary artery disease (CAD), and echocardiography revealed depressed LV function, we performed coronary artery bypass grafting (CABG). The LAD was grafted to the left internal mammary artery, and the diagonal branch, right posterior descending branch, and left obtuse
margi-nal branch were grafted with vein grafts. After surgery, the patient gradually recovered without event.
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ISCUSSIONIt was difficult to determine the cause of our patient’s coronary artery aneurysms and ectasia. Trauma, mycotic aneurysm, embolism, syphilis, dissection, inflammation, and congenital abnormalities could be excluded by his medical history and the results of serologic testing. Except for smoking, the patient had no other known risk factor for atherosclerosis. In addition, when aneurysms are associated with atherosclerosis, the overwhelming majority of cases have pronounced coronary artery stenoses [4]. Takahashi et al defined coronary artery aneurysms in patients with KD as either localized or extensive [5]. Based on their defini-tion, extensive aneurysms involve more than one segment, and may be either ectatic (uniformly dilated) or segmented (having multiple dilated segments joined by normal or stenotic segments). In our patient, left coronary angiogra-phy showed segmented aneurysms (three aneurysms
Figure 1. Electrocardiograms in the: (A) emergency room, and; (B) on admission, show dynamic ST-T changes in leads I, aVL, and V3–V6.
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Figure 2. Echocardiograms on admission show: (A) left ventricular dilation and depressed left ventricular function, and; (B) mitral valve B hump.
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joined by non-aneurysmal segments), and right coronary angiography showed ectatic aneurysms. Such angiogra-phic appearance is consistent with Takahashi et al’s find-ings [5]. Although we found no childhood history of a Kawasaki-like illness in our patient, we suspected that KD, which started in childhood, was the cause of his coronary
Figure 3. Coronary angiograms. (A) 35° cranial angulation view shows three aneurysms (arrows) involving the distal left main artery and the diagonal branch of the left anterior descending artery. The left anterior descending artery and left circumflex artery are occluded. (B) 30° left anterior oblique view shows diffuse right coronary artery ectasia and 99% discrete stenosis over the posterior descending branch (arrow).
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artery aneurysms and ectasia. Our suspicion is partlysupported by the appearance of our patient’s aneurysmsand ectasia, which do not typically occur in atherosclerosis [6], but bear a notable resemblance to some of the lesions found in KD [3,5,7,8].
KD is an acute vasculitis of unknown etiology that oc-curs predominantly in infants and young children. Kawa-saki first described the illness in Japan in 1967 [9]. Symp-toms of KD include fever, bilateral non-exudative con-junctivitis, erythema of the lips and oral mucosa, changes in the extremities, rash, and cervical lymphadenopathy. Approximately 15% to 25% of untreated children with KD develop coronary artery aneurysms or ectasia, which may lead to myocardial infarction, sudden death, or chronic coronary artery insufficiency [10]. Treatment with intra-venous gamma globulin in the acute phase reduces the risk of coronary artery aneurysm by three- to fivefold [11]. KD sometimes remains subclinical in the acute phase or may not be recognized because of the nonspecific fea-tures, and may present at a later date with cardiac sequelae [12].
We suggest that what happened in our patient was that, after the unrecognized childhood occurrence of KD, coronary artery aneurysms and ectasia were formed, lead-ing to the formation of thrombus and occlusion within his coronary arteries and a silent myocardial infarction. This sequence of events is supported by the echocardio-graphic findings of depressed LV function and dilated LV chamber. This time, thrombus occlusion probably occurred in his LCX, as suggested by 12-lead ECG (dynamic ST-T changes in leads I, aVL, and V3–V6). This occlusion led to his symptomatic AMI.
The management of coronary artery stenoses in KD is principally surgical, although percutaneous coronary inter-vention has been used in patients with isolated, discrete stenoses [13–15]. The surgical options consist of either CABG or cardiac transplantation. In the case of coronary insufficiency related to KD, coronary revascularization using the saphenous vein, internal mammary arteries, and a combination of both types of grafts has been performed with considerable success [16,17]. In our case, due to se-vere three-vessel CAD and depressed LV function, CABG seemed the best option and was performed successfully.
We believe that our patient’s AMI, coronary artery aneurysms, and ectasia were a consequence of KD, which he had had since childhood. Since KD may cause CAD in young adults, cardiologists should keep in mind and include the sequelae of KD in the differential diagnosis of early-onset ischemic heart disease.
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EFERENCES1. Daoud AS, Pankin D, Tulgan H, et al. Aneurysms of the coro-nary artery: report of ten cases and review of the literature. Am J Cardiol 1963;11:228–37.
2. Eshchar Y, Yahini JH, Deutsch V, et al. Arteriosclerotic aneu-rysm of the coronary artery. Chest 1977;72:374–5.
3. Kato H, Koike S, Yamamoto M, et al. Coronary aneurysms in infants and young children with acute febrile mucocutaneous lymph node syndrome. J Pediatr 1975;86:892–8.
4. Swaye PS, Fisher LD, Litwin P, et al. Aneurysmal coronary artery disease. Circulation 1983;67:134–8.
5. Takahashi M, Mason W, Lewis AB. Regression of coronary aneurysms in patients with Kawasaki syndrome. Circulation 1987;75:387–94.
6. Swanton RH, Thomas ML, Coltart DJ, et al. Coronary artery ectasia – a variant of occlusive coronary arteriosclerosis. Br Heart J 1978;40:393–400.
7. Kitamura S, Kawachi K, Harima R, et al. Surgery for coronary heart disease due to mucocutaneous lymph node syndrome (Kawasaki’s disease). Am J Cardiol 1983;51:444–8.
8. Yoshikawa J, Yanagihara K, Owaki T, et al. Cross-sectional echocardiographic diagnosis of coronary artery aneurysms in patients with the mucocutaneous lymph node syndrome. Circulation 1979;59:133–9.
9. Kawasaki T. Acute febrile mucocutaneous syndrome with
lymphoid involvement and specific desquamation of the fingers and toes in children. Clinical observation in 50 cases. Jpn J Allergy 1967;16:178–222.
10. Dajani AS, Taubert KA, Gerber MA, et al. Diagnosis and therapy of Kawasaki disease in children. Circulation 1993;87: 1776–80.
11. Leung DY. Clinical and immunologic aspects of Kawasaki’s disease. Immunodefic Rev 1989;1:261–7.
12. Gersony WM. Diagnosis and management of Kawasaki di-sease. JAMA 1991;265:2699–703.
13. Negoro N, Nariyama J, Nakagawa A, et al. Successful catheter interventional therapy for acute coronary syndrome secondary to Kawasaki disease in young adults. Circ J 2003;67:362–5. 14. Rivera DA, Aggarwal N, Parrillo JE, et al. Probable Kawasaki
disease in a 52-year-old man presenting with acute myocardial infarction treated with percutaneous revascularization. Heart Dis 2001;3:302–5.
15. Shiraishi J, Sawada T, Tatsumi T, et al. Acute myocardial infarction due to a regressed giant coronary aneurysm as possible sequela of Kawasaki disease. J Invasive Cardiol 2001;13:569–72. 16. Suzuki A, Kamiya T, Ono Y, et al. Aortocoronary bypass
sur-gery for coronary arterial lesions resulting from Kawasaki di-sease. J Pediatr 1990;116:567–73.
17. Yamagishi I, Abe T, Kuribayashi R, et al. Coronary artery by-pass grafting in a patient with severe coronary disease due to Kawasaki disease: a case report. Kyobu Geka 1996;49:151–4.
