Pediatr Cardiol 12:33-35, 1991
Pediatric Cardiology 9 Springer-VerlagNew York Inc. 1991
Case Reports Percutaneous Transluminal Coronary Angioplasty for Kawasaki Disease: A Case R e p o r t and Literature R e v i e w Toshihiro Ino,I Kei Nishimoto,~ Katsumi Akimoto,l Insam Park,l Shinjiro Shimazaki, Keijiro Yabuta, I and Hiroshi Yamaguchi ~ ~Department of Pediatrics and -'Division of Cardiology, Department of Internal Medicine, Juntendo University School of Medicine, Tokyo, Japan
SUMMARY. A 31-month-old boy developed right coronary artery stenosis after Kawasaki disease for which he underwent percutaneous transluminal coronary angioplasty (PTCA). The narrowing of the right coronary artery was successfully dilated by angioplasty without apparent complication. This case suggests that PTCA may have a potential advantage as a temporary method to postpone the aortocoronary bypass surgery in a child with coronary artery stenosis due to Kawasaki disease. However, strict patient selection is recommended for coronary angioplasty. KEY WORDS: Kawasaki diseasewPercutaneous transluminal coronary angioplasty--Coronary artery aneurysm--Coronary artery stenosis
Approximately 10-20% of coronary artery aneurysms resulting from Kawasaki disease usually progress to become obstructive [7, 8, 10]. In patients with these obstructive lesions, aortocoronary bypass surgery may sometimes be required to preserve ventricular function [3, 9]. However, the long-term patency rate of the bypass graft remains unsatisfactory, particularly in a young child with this disease [5]. Therefore, other therapeutic alternatives to aortocoronary bypass surgery should be considered in this age group. We report a 3 l-monthold boy in whom percutaneous transluminal coronary angioplasty (PTCA) was successfully performed for coronary artery stenosis caused by Kawasaki disease.
Case Report A 2-year-old Japanese boy was referred to our hospital for angiographic evaluation of coronary aneurysms. At 4 months of age he was first hospitalized at a local hospital, 7 days after onset because of prolonged fever (temperature to 39~ nonexudative conjunctivitis, cervical lymphadenopathy, strawberry tongue with red lips, and polymorphous examthem. The initial clinical
Address offprint requests to." Dr. Toshihiro Ino, Department of Pediatrics, Juntendo University School of Medicine, 2-1-1 Hongo, Bunkyou-ku, Tokyo 113, Japan.
laboratory data suggested the diagnosis of Kawasaki disease. Pulsed methylprednisolone therapy (30 mg/kg/day i.v.) with aspirin (30 mg/kg/day p.o.) was begun on the 8th day of hospitalization. An echocardiogram on the 13th days showed aneurysms of bilateral coronary arteries: maximally 7 mm (right), 6 mm (left) in diameter. He was discharged on aspirin (10 mg/kg/day p.o.) and dipyridamole (3 mg/kg/day p.o.) and has since remained entirely well. Initial angiography was performed at 10 months of age. Coronary angiograms revealed saccular and spindle aneurysms of the right coronary artery and mild dilatation of the left coronary artery (Fig. 1). An extensive filling defect was found in the distal aneurysm of the right coronary artery, suggesting a large thrombus. The left ventriculogram showed normal contractility and no evidence of wall motion abnormalities. The treatment with aspirin and dipyridamole was continued with an increased dosage (aspirin 10--~30 mg/kg/day). The follow-up coronary angiography was performed 12 months after the initial one. Right aneurysms had progressed to severe, localized stenosis in the middle portion. The dilatation of the left coronary artery remained unchange. Myocardial imaging with thallium-201 at that time revealed no significant perfusion defects of the left ventricle, and he has since remained symptomfree. At that time, PTCA was planned because the angiographic evidence suggested a potential risk of sudden death by acute myocardial infarction. At 31 months of age, he was admitted to our hospital for reevaluation and coronary angioplasty of the right coronary artery stenosis. Physical examination on admission revealed a well-developed boy. His weight was 11.5 kg. No heart mumur or gallop rhythm was heard. A chest roentogenogram showed no cardiomegaly. An electrocardiogram (ECG) showed incomplete
34
Fig. 1.
Initial coronary arteriogram at 10 months of age showing extensive aneurysm with thrombosis (arrow) in the right coronary artery (A) and mild dilatation of the left coronary artery (B).
Fig. 2. Right coronary arteriogram before PCTA showing a severe, localized stenosis (white arrow) (A). A waist was observed at the middle of inflated balloon (B). The stenosis was well dilated shortly after angioplasty (C). Follow-up angiography showing no evidence of restenosis of the right coronary artery (D).
right bundle branch block but no abnormal Q wave or ST segment change. The right coronary arteriogram before angioplasty revealed a same stenosis with an intraluminal filling defect as the second angiogram (Fig. 2A). A dilating catheter with a 2.5-mm balloon was selected because of the caliber of the distal right coronary artery. The balloon catheter was introduced by a 7-F guiding catheter with a steerable guidwire. The balloon was inflated with diluted contrast material to 4 and 6 atm pressure (Fig. 2B). The duration of inflation was approximately 60 s at each dilatation, and the procedure was repeated twice. The right coronary arteriogram was repeated immediately after the procedures. The estimated degree of the stenosis was improved from approximately 90 to 35%, without any filling defects (Fig. 2C). An ECG during the procedure revealed transient elevation of the ST segment in leads II, III and aVF. No significant complications were encountered during and after the procedure. Antiplatelet therapy of aspirin and dipyridamole was continued. No perfusion defects were found by myocardial imaging with thallium-201 at 3 months after the procedure. At 37 months of age, the follow-up angiography revealed no evidence of restenosis of the right coronary artery (Fig. 2D).
Pediatric Cardiology Vol. 12, No. 1, 1991
Discussion
PTCA has been offered as a reasonable alternative to coronary artery bypass surgery in adults with angina pectoris. To our knowledge, only two cases that had undergone PTCA for coronary artery stenosis in Kawasaki disease have been previously reported [I]. According to that report, as summarized in Table 1, one patient died of obstruction of the left coronary artery immediately after balloon rupture caused by a high inflating pressure during the procedure, and the other was successfully dilated. In our patient the stenosis was well dilated without any complications. The mechanism for relief of narrowing remains unknown in this setting. Fujiwara and colleagues [2] reported that marked intimal thickening at the site of the coronary arterial narrowing was found at autopsy specimens in the convalescent phase. It is speculated, therefore, that the mechanism is probably different from that in adults with atherosclerosis and is related to producing a tear in the intima and media by coronary angioplasty as in angioplasty for coarctation of the aorta or pulmonary artery stenosis [6]. In the early acute phase, this procedure may rupture the coronary artery because of fragility of the arterial wall by vasculitis. Therefore, this invasive intervention should be postponed until the healing of the acute inflammatory process.
Ino et al.:
Coronary Angioplasty for Kawasaki Disease
Table 1. Comparison of clinical data in our patient and a previous report
Present study Age of onset 4m Age of PTCA 2y Sex Male Symptoms -Perfusion defect -(TI-201 imaging) Location of lesion RCA for angioplasty Complication -Outcome Improved
Case 1"
Case 2"
2y II y Female + +
3m 8y Male + +
LAD
LAD
ND Improved
Balloon rupture Died
RCA, right coronary artery; LCA, left coronary artery; LAD. left anterior descending artery; m, month; y, year; ND, not described. " Data in cases 1 and 2 were obtained from the previous report (see [1]).
The correct indication for PCTA in Kawasaki heart disease remains uncertain. Although our patient had no obvious symptoms, the angiographic findings of the right coronary artery made angioplasty seem suitable as there was a single, localized stenosis. In addition, the left ventriculography revealed good systolic function. Keltz et al. [4] previously reported that nonexercise stress thallium-201 imaging using dipyridamole was sensitive for assessing myocardial ischemia, but could occasionally induce severe persistent angina. We therefore did not employ this procedure, fearing that it might cause severe angina. In summary, our case and the previous report suggest that PTCA may have a potential advantage as a temporary method of postponing aortocoronary bypass surgery in small children with se-
35
vere, localized stenosis of the coronary artery due to Kawasaki disease. However, reliable criteria of patient selection for coronary angioplasty are needed.
Acknowledgment. We thank Drs. Takaya, Daida, and Higashiyarea for their assistance in this report.
References 1. Echigo S (1988) PTCA. In Abstracts o f the 3rd International Kawasaki Disease Symposium. Tokyo, p 64 2. Fujiwara T, Fujiwara H, Hamashima Y (1987) Frequency and size of coronary arterial aneurysm at necropsy in Kawasaki disease. Am J Cardiol 58:808-811 3. lno T, Iwahara M, Boku H, et al. (1987) Aortocornary bypass surgery for Kawasaki disease. Pediatr Cardiol 8:195197 4. Keltz TN, Innerfield M, Gitler B, Cooper JA (1987) Dipyridamole-induced myocardial ischemia. J A M A 25:1515-1516 5. Kitamura S, Kawachi K, Oyama C, et al. (1985) Severe Kawasaki heart disease treated with an internal mammary artery graft in pediatric patients. J Thorac Cardiovasc Surg 89:860-866 6. Lock JE, Niemi T, Burke BA, et al. (1982) Transcutaneous angioplasty of experimental aortic coarctation. Circulation 66:1280-1286 7. Nakano H, Ueda K, Saito A, Nojima K (1985) Repeated quantitative angiograms in coronary arterial aneurysm in Kawasaki disease. Am J Cardiol 56:846-851 8. Suzuki A, Kamiya T, Kuwahara N, et al. (1986) Coronary arterial lesions in Kawasaki disease: Cardiac-catheterization findings of 1100 cases. Pediatr Cardiol 7:3-9 9. Suzuki A, Kitamura T, Ona Y, Takahashi N, Naito Y, Koh Y (1985) Indication of aortocoronary bypass for coronary arterial obstruction due to Kawasaki disease. Heart Vessels 1:94-100 10. Tatara K, Kusakawa S (1987) Long-term prognosis of giant coronary aneurysm in Kawasaki disease: An angiographic study. J Pediatr 111:705-10
Pediatric Cardiology 9 Springer-VerlagNew York Inc. 1991
Case Reports Percutaneous Transluminal Coronary Angioplasty for Kawasaki Disease: A Case R e p o r t and Literature R e v i e w Toshihiro Ino,I Kei Nishimoto,~ Katsumi Akimoto,l Insam Park,l Shinjiro Shimazaki, Keijiro Yabuta, I and Hiroshi Yamaguchi ~ ~Department of Pediatrics and -'Division of Cardiology, Department of Internal Medicine, Juntendo University School of Medicine, Tokyo, Japan
SUMMARY. A 31-month-old boy developed right coronary artery stenosis after Kawasaki disease for which he underwent percutaneous transluminal coronary angioplasty (PTCA). The narrowing of the right coronary artery was successfully dilated by angioplasty without apparent complication. This case suggests that PTCA may have a potential advantage as a temporary method to postpone the aortocoronary bypass surgery in a child with coronary artery stenosis due to Kawasaki disease. However, strict patient selection is recommended for coronary angioplasty. KEY WORDS: Kawasaki diseasewPercutaneous transluminal coronary angioplasty--Coronary artery aneurysm--Coronary artery stenosis
Approximately 10-20% of coronary artery aneurysms resulting from Kawasaki disease usually progress to become obstructive [7, 8, 10]. In patients with these obstructive lesions, aortocoronary bypass surgery may sometimes be required to preserve ventricular function [3, 9]. However, the long-term patency rate of the bypass graft remains unsatisfactory, particularly in a young child with this disease [5]. Therefore, other therapeutic alternatives to aortocoronary bypass surgery should be considered in this age group. We report a 3 l-monthold boy in whom percutaneous transluminal coronary angioplasty (PTCA) was successfully performed for coronary artery stenosis caused by Kawasaki disease.
Case Report A 2-year-old Japanese boy was referred to our hospital for angiographic evaluation of coronary aneurysms. At 4 months of age he was first hospitalized at a local hospital, 7 days after onset because of prolonged fever (temperature to 39~ nonexudative conjunctivitis, cervical lymphadenopathy, strawberry tongue with red lips, and polymorphous examthem. The initial clinical
Address offprint requests to." Dr. Toshihiro Ino, Department of Pediatrics, Juntendo University School of Medicine, 2-1-1 Hongo, Bunkyou-ku, Tokyo 113, Japan.
laboratory data suggested the diagnosis of Kawasaki disease. Pulsed methylprednisolone therapy (30 mg/kg/day i.v.) with aspirin (30 mg/kg/day p.o.) was begun on the 8th day of hospitalization. An echocardiogram on the 13th days showed aneurysms of bilateral coronary arteries: maximally 7 mm (right), 6 mm (left) in diameter. He was discharged on aspirin (10 mg/kg/day p.o.) and dipyridamole (3 mg/kg/day p.o.) and has since remained entirely well. Initial angiography was performed at 10 months of age. Coronary angiograms revealed saccular and spindle aneurysms of the right coronary artery and mild dilatation of the left coronary artery (Fig. 1). An extensive filling defect was found in the distal aneurysm of the right coronary artery, suggesting a large thrombus. The left ventriculogram showed normal contractility and no evidence of wall motion abnormalities. The treatment with aspirin and dipyridamole was continued with an increased dosage (aspirin 10--~30 mg/kg/day). The follow-up coronary angiography was performed 12 months after the initial one. Right aneurysms had progressed to severe, localized stenosis in the middle portion. The dilatation of the left coronary artery remained unchange. Myocardial imaging with thallium-201 at that time revealed no significant perfusion defects of the left ventricle, and he has since remained symptomfree. At that time, PTCA was planned because the angiographic evidence suggested a potential risk of sudden death by acute myocardial infarction. At 31 months of age, he was admitted to our hospital for reevaluation and coronary angioplasty of the right coronary artery stenosis. Physical examination on admission revealed a well-developed boy. His weight was 11.5 kg. No heart mumur or gallop rhythm was heard. A chest roentogenogram showed no cardiomegaly. An electrocardiogram (ECG) showed incomplete
34
Fig. 1.
Initial coronary arteriogram at 10 months of age showing extensive aneurysm with thrombosis (arrow) in the right coronary artery (A) and mild dilatation of the left coronary artery (B).
Fig. 2. Right coronary arteriogram before PCTA showing a severe, localized stenosis (white arrow) (A). A waist was observed at the middle of inflated balloon (B). The stenosis was well dilated shortly after angioplasty (C). Follow-up angiography showing no evidence of restenosis of the right coronary artery (D).
right bundle branch block but no abnormal Q wave or ST segment change. The right coronary arteriogram before angioplasty revealed a same stenosis with an intraluminal filling defect as the second angiogram (Fig. 2A). A dilating catheter with a 2.5-mm balloon was selected because of the caliber of the distal right coronary artery. The balloon catheter was introduced by a 7-F guiding catheter with a steerable guidwire. The balloon was inflated with diluted contrast material to 4 and 6 atm pressure (Fig. 2B). The duration of inflation was approximately 60 s at each dilatation, and the procedure was repeated twice. The right coronary arteriogram was repeated immediately after the procedures. The estimated degree of the stenosis was improved from approximately 90 to 35%, without any filling defects (Fig. 2C). An ECG during the procedure revealed transient elevation of the ST segment in leads II, III and aVF. No significant complications were encountered during and after the procedure. Antiplatelet therapy of aspirin and dipyridamole was continued. No perfusion defects were found by myocardial imaging with thallium-201 at 3 months after the procedure. At 37 months of age, the follow-up angiography revealed no evidence of restenosis of the right coronary artery (Fig. 2D).
Pediatric Cardiology Vol. 12, No. 1, 1991
Discussion
PTCA has been offered as a reasonable alternative to coronary artery bypass surgery in adults with angina pectoris. To our knowledge, only two cases that had undergone PTCA for coronary artery stenosis in Kawasaki disease have been previously reported [I]. According to that report, as summarized in Table 1, one patient died of obstruction of the left coronary artery immediately after balloon rupture caused by a high inflating pressure during the procedure, and the other was successfully dilated. In our patient the stenosis was well dilated without any complications. The mechanism for relief of narrowing remains unknown in this setting. Fujiwara and colleagues [2] reported that marked intimal thickening at the site of the coronary arterial narrowing was found at autopsy specimens in the convalescent phase. It is speculated, therefore, that the mechanism is probably different from that in adults with atherosclerosis and is related to producing a tear in the intima and media by coronary angioplasty as in angioplasty for coarctation of the aorta or pulmonary artery stenosis [6]. In the early acute phase, this procedure may rupture the coronary artery because of fragility of the arterial wall by vasculitis. Therefore, this invasive intervention should be postponed until the healing of the acute inflammatory process.
Ino et al.:
Coronary Angioplasty for Kawasaki Disease
Table 1. Comparison of clinical data in our patient and a previous report
Present study Age of onset 4m Age of PTCA 2y Sex Male Symptoms -Perfusion defect -(TI-201 imaging) Location of lesion RCA for angioplasty Complication -Outcome Improved
Case 1"
Case 2"
2y II y Female + +
3m 8y Male + +
LAD
LAD
ND Improved
Balloon rupture Died
RCA, right coronary artery; LCA, left coronary artery; LAD. left anterior descending artery; m, month; y, year; ND, not described. " Data in cases 1 and 2 were obtained from the previous report (see [1]).
The correct indication for PCTA in Kawasaki heart disease remains uncertain. Although our patient had no obvious symptoms, the angiographic findings of the right coronary artery made angioplasty seem suitable as there was a single, localized stenosis. In addition, the left ventriculography revealed good systolic function. Keltz et al. [4] previously reported that nonexercise stress thallium-201 imaging using dipyridamole was sensitive for assessing myocardial ischemia, but could occasionally induce severe persistent angina. We therefore did not employ this procedure, fearing that it might cause severe angina. In summary, our case and the previous report suggest that PTCA may have a potential advantage as a temporary method of postponing aortocoronary bypass surgery in small children with se-
35
vere, localized stenosis of the coronary artery due to Kawasaki disease. However, reliable criteria of patient selection for coronary angioplasty are needed.
Acknowledgment. We thank Drs. Takaya, Daida, and Higashiyarea for their assistance in this report.
References 1. Echigo S (1988) PTCA. In Abstracts o f the 3rd International Kawasaki Disease Symposium. Tokyo, p 64 2. Fujiwara T, Fujiwara H, Hamashima Y (1987) Frequency and size of coronary arterial aneurysm at necropsy in Kawasaki disease. Am J Cardiol 58:808-811 3. lno T, Iwahara M, Boku H, et al. (1987) Aortocornary bypass surgery for Kawasaki disease. Pediatr Cardiol 8:195197 4. Keltz TN, Innerfield M, Gitler B, Cooper JA (1987) Dipyridamole-induced myocardial ischemia. J A M A 25:1515-1516 5. Kitamura S, Kawachi K, Oyama C, et al. (1985) Severe Kawasaki heart disease treated with an internal mammary artery graft in pediatric patients. J Thorac Cardiovasc Surg 89:860-866 6. Lock JE, Niemi T, Burke BA, et al. (1982) Transcutaneous angioplasty of experimental aortic coarctation. Circulation 66:1280-1286 7. Nakano H, Ueda K, Saito A, Nojima K (1985) Repeated quantitative angiograms in coronary arterial aneurysm in Kawasaki disease. Am J Cardiol 56:846-851 8. Suzuki A, Kamiya T, Kuwahara N, et al. (1986) Coronary arterial lesions in Kawasaki disease: Cardiac-catheterization findings of 1100 cases. Pediatr Cardiol 7:3-9 9. Suzuki A, Kitamura T, Ona Y, Takahashi N, Naito Y, Koh Y (1985) Indication of aortocoronary bypass for coronary arterial obstruction due to Kawasaki disease. Heart Vessels 1:94-100 10. Tatara K, Kusakawa S (1987) Long-term prognosis of giant coronary aneurysm in Kawasaki disease: An angiographic study. J Pediatr 111:705-10
