川崎病合并冠状动脉病变患儿21例冠状动脉造影复查分析

doi:10.3969/j.issn.1674-8115.2023.12.008

摘要/Abstract

摘要：

目的·通过冠状动脉（冠脉）造影复查分析川崎病所致严重冠脉病变患儿的进展情况，并评价心脏超声（心超）对此类患儿的诊断价值。方法·采用回顾性分析，收集2013年1月至2023年1月上海交通大学医学院附属上海儿童医学中心的冠脉病变分级达到Ⅳ级及以上的川崎病患儿，要求纳入对象接受过至少2次的冠脉造影，收集其临床及影像学检查资料，分析病变进展情况；并将心超与冠脉造影结果进行对比分析。结果·共纳入21例患儿，其中男15例、女6例，中位发病年龄为3岁6个月，初次冠脉造影的中位年龄为7岁11个月，发病时间与初次造影中位间隔时间为4年5个月；造影复查时的中位年龄为9岁2个月，与初次造影中位间隔时间为1年3个月。初次造影发现有13例患儿存在冠脉狭窄或闭塞，其中6例行冠状动脉旁路移植术（coronary artery bypass grafting，CABG），并在1年后复查造影，结果显示桥血管通畅，未见明显狭窄。15例患儿在后续随访中，心超发现病变进展并行造影复查，其中8例患儿冠状病变较前明显进展，并有1例患儿于术中行冠脉内球囊扩张术，1例复查后行CABG。在21例患儿初次造影时，共发现16处冠脉狭窄或闭塞；而同期心超仅提示1处冠脉狭窄。21例患儿初次造影时共发现28处中大型冠脉瘤，将心超和冠脉造影对此28处冠脉瘤的测量直径进行Bland-Altman一致性分析，最大直径差值为（1.63±2.33）mm，95%CI为-2.95~6.21 mm。结论·川崎病所致严重冠脉病变可能是不断进展的；对病变严重的患儿，心超可能对冠脉狭窄或闭塞存在漏诊或误诊，对冠脉瘤直径测量存在一定误差，需定期复查冠脉造影。

关键词: 川崎病, 冠状动脉瘤, 冠状动脉狭窄, 冠状动脉造影, 心脏超声

Abstract:

Objective ·To analyze the progression of children with severe coronary artery lesions due to Kawasaki disease by coronary artery angiography, and evaluate the diagnostic value of echocardiography in these children. Methods ·A retrospective analysis was performed to enroll children with Kawasaki disease whose coronary artery lesions were graded Ⅳ or above from Shanghai Children's Medical Center, Shanghai Jiao Tong University School of Medicine, from January 2013 to January 2023. The subjects were required to have received at least 2 times of coronary angiogram, and their clinical and imaging data were collected to analyze the progression of the lesions. Echocardiography results were compared with the results of the coronary angiogram. Results ·A total of 21 children were included, including 15 males and 6 females, with a median age at onset of 3 years and 6 months, a median age at initial coronary angiography of 7 years and 11 months, a median interval of 4 years and 5 months between the time of onset and initial angiography, a median age at angiographic review of 9 years and 2 months, and a median interval of 1 year and 3 months between the time of initial angiography and review. Coronary stenosis or occlusion was detected in 13 children in the initial angiography, of whom 6 underwent coronary artery bypass grafting (CABG) and had their angiography reviews 1 year later. The review results showed that the bridging vessels were unobstructed and no obvious stenosis was observed. Fifteen children had progression of the lesions detected by echocardiography in the subsequent follow-up and had their angiogram reviews, of whom 8 had significant progression of the coronary lesions. Intracoronary balloon dilatation was performed in 1 case, and CABG was performed in another case. Sixteen lesions of coronary stenosis or occlusion were detected in the initial angiography in 21 children, while only 1 lesion of coronary stenosis was detected by echocardiography during the same period of time. Twenty-eight medium- to large-sized coronary aneurysms were detected in the initial angiography in the 21 children, and the diameters of the 28 aneurysms measured by echocardiography and coronary angiogram were subjected to the Bland-Altman analysis. The Bland-Altman analysis showed that the difference in maximum diameter between 2 methods was (1.63±2.33) mm, with 95%CI of -2.95?6.21 mm. Conclusion ·Coronary artery lesions due to Kawasaki disease may be progressive; in the children with severe lesions, coronary artery stenosis or occlusion may be missed or misdiagnosed and some errors may exist in the measurement of diameters of aneurysms by echocardiography. Regular review of coronary angiography is needed.

Key words: Kawasaki disease, coronary artery aneurysm, coronary artery stenosis, coronary angiography, echocardiography

中图分类号:

R725.4

张锰, 崔青, 朱荻绮, 张玉奇, 钟玉敏, 沈捷. 川崎病合并冠状动脉病变患儿21例冠状动脉造影复查分析[J]. 上海交通大学学报（医学版）, 2023, 43(12): 1535-1541.

ZHANG Meng, CUI Qing, ZHU Diqi, ZHANG Yuqi, ZHONG Yumin, SHEN Jie. Coronary angiography review in 21 children with Kawasaki disease complicated with coronary artery disease[J]. Journal of Shanghai Jiao Tong University (Medical Science), 2023, 43(12): 1535-1541.

图/表 5

参考文献 10

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No.	Gender	Age of diagnosis	Age of first CAG	Echocardiography before the first CAG	Result of first CAG	Age of CAG review	Result of CAG review
1	Male	13 years	13 years and 7 months	A large coronary aneurysm in the LM; a large proximal coronary aneurysm in the proximal segment of LAD, and a coronary dilatation in the distal segment; a large coronary aneurysm in the proximal segment of RCA	A large coronary artery aneurysm in the proximal segment of LM; a beaded dilatation in the LAD; a large coronary artery aneurysm in the proximal segment of RCA, and a post-aneurysm coronary atresia with collaterals formation	17 years and 2 months	The LAD coronary artery aneurysm progressed and the length of the LM coronary artery aneurysm nearly doubled
2	Male	6 months	3 years and 6 months	Significant enlargement of the left ventricle with LVEF of 50%; a nodule in the proximal segment of LAD; 2 sites of dilatation in the RCA without clear display of the distal segment	Torsion and stenosis of the main trunk of RCA with collaterals formation; the LAD without significant lesions	7 years and 6 months (chest pain for 10 d)	The proximal segment of RCA was severely narrow or atresia with several collaterals formation, and the distal segment displayed well
3	Male	3 years	3 years and 3 months	A large aneurysm in the proximal segment of LAD; a beaded dilatation in RCA	A medium coronary aneurysm in the proximal segment of LAD and post-aneurysm coronary artery occlusion with collaterals formation; beaded dilatations in the RCA without filling defects	4 years and 3 months	The blood flow of LCX was unobstructed; three aneurysms in the RCA progressed, of which diameters were wider than before, and filling defects could be seen in the RCA
4	Male	4 months	4 years	A medium aneurysm in the proximal segment of LAD and a medium aneurysm in the proximal segment of RCA, and the coronary arteries not clear after the 2 aneurysms	Small aneurysms in the middle section of RCA with calcification and collaterals formation; stenosis in the proximal segment of LAD without coronary artery formation	6 years	The stenosis in the LAD progressed, and the diameter of the narrowest point was 1.2 mm. Intraoperative balloon dilation was performed, and then the blood flow was unobstructed
5	Female	2 years and 4 months	3 years	A large aneurysm in the LM; the opening of LCX dilated with thrombosis formation; a small aneurysm in the proximal segment of RCA with thrombosis after the aneurysm	A large aneurysm in the LM with the stenotic LAD; a small aneurysm at the beginning of RCA with post-aneurysm atresia and collaterals formation	3 years and 9 months	The aneurysm in the LM progressed with narrower LAD; proximal coronary atresia of the RCA was found with collaterals formation
6	Male	2 years	5 years	A large aneurysm in the proximal segment LAD with thrombosis and without clear display of the distal segment; the RCA dilatated in the middle segment	The blood flow was unobstructed in the LAD; small artery dilatations in the LAD and the distal segment of RCA	7 years	The diameter of the opening of RCA was 3.1 mm, while the stenosis in the proximal segment was 2.01 mm in diameter, and a beaded dilatation in RCA after the stenosis
7	Male	9 years	12 years (2 times of syncope in 3 years)	The LM dilatated; a large aneurysm in the LAD; beaded dilatations in the RCA	A medium aneurysm in the LAD with a post-aneurysm coronary atresia and calcification formation; 3 beaded coronary aneurysms in the RCA	15 years	Thrombosis at the beginning of LAD with atresia and calcification after the thrombus; the RCA supplied the LAD by collaterals in reverse direction
8	Female	2 years	3 years	A large aneurysm in the LAD without clear display of the distal segment	A dilatation at the beginning of LAD, about 6.3 mm in diameter, with filling defection and thrombosis formation after the dilatation	4 years and 5 months	A aneurysm with the diameter of about 8.5 mm at the openings of LCX and LAD (bifurcation of LM)

No.	Gender	Age of diagnosis	Age of first CAG	Echocardiography before the first CAG	Result of first CAG	Age of CAG review	Result of CAG review
1	Male	13 years	13 years and 7 months	A large coronary aneurysm in the LM; a large proximal coronary aneurysm in the proximal segment of LAD, and a coronary dilatation in the distal segment; a large coronary aneurysm in the proximal segment of RCA	A large coronary artery aneurysm in the proximal segment of LM; a beaded dilatation in the LAD; a large coronary artery aneurysm in the proximal segment of RCA, and a post-aneurysm coronary atresia with collaterals formation	17 years and 2 months	The LAD coronary artery aneurysm progressed and the length of the LM coronary artery aneurysm nearly doubled
2	Male	6 months	3 years and 6 months	Significant enlargement of the left ventricle with LVEF of 50%; a nodule in the proximal segment of LAD; 2 sites of dilatation in the RCA without clear display of the distal segment	Torsion and stenosis of the main trunk of RCA with collaterals formation; the LAD without significant lesions	7 years and 6 months (chest pain for 10 d)	The proximal segment of RCA was severely narrow or atresia with several collaterals formation, and the distal segment displayed well
3	Male	3 years	3 years and 3 months	A large aneurysm in the proximal segment of LAD; a beaded dilatation in RCA	A medium coronary aneurysm in the proximal segment of LAD and post-aneurysm coronary artery occlusion with collaterals formation; beaded dilatations in the RCA without filling defects	4 years and 3 months	The blood flow of LCX was unobstructed; three aneurysms in the RCA progressed, of which diameters were wider than before, and filling defects could be seen in the RCA
4	Male	4 months	4 years	A medium aneurysm in the proximal segment of LAD and a medium aneurysm in the proximal segment of RCA, and the coronary arteries not clear after the 2 aneurysms	Small aneurysms in the middle section of RCA with calcification and collaterals formation; stenosis in the proximal segment of LAD without coronary artery formation	6 years	The stenosis in the LAD progressed, and the diameter of the narrowest point was 1.2 mm. Intraoperative balloon dilation was performed, and then the blood flow was unobstructed
5	Female	2 years and 4 months	3 years	A large aneurysm in the LM; the opening of LCX dilated with thrombosis formation; a small aneurysm in the proximal segment of RCA with thrombosis after the aneurysm	A large aneurysm in the LM with the stenotic LAD; a small aneurysm at the beginning of RCA with post-aneurysm atresia and collaterals formation	3 years and 9 months	The aneurysm in the LM progressed with narrower LAD; proximal coronary atresia of the RCA was found with collaterals formation
6	Male	2 years	5 years	A large aneurysm in the proximal segment LAD with thrombosis and without clear display of the distal segment; the RCA dilatated in the middle segment	The blood flow was unobstructed in the LAD; small artery dilatations in the LAD and the distal segment of RCA	7 years	The diameter of the opening of RCA was 3.1 mm, while the stenosis in the proximal segment was 2.01 mm in diameter, and a beaded dilatation in RCA after the stenosis
7	Male	9 years	12 years (2 times of syncope in 3 years)	The LM dilatated; a large aneurysm in the LAD; beaded dilatations in the RCA	A medium aneurysm in the LAD with a post-aneurysm coronary atresia and calcification formation; 3 beaded coronary aneurysms in the RCA	15 years	Thrombosis at the beginning of LAD with atresia and calcification after the thrombus; the RCA supplied the LAD by collaterals in reverse direction
8	Female	2 years	3 years	A large aneurysm in the LAD without clear display of the distal segment	A dilatation at the beginning of LAD, about 6.3 mm in diameter, with filling defection and thrombosis formation after the dilatation	4 years and 5 months	A aneurysm with the diameter of about 8.5 mm at the openings of LCX and LAD (bifurcation of LM)

Detection method	Coronary artery aneurysm				Coronary artery stenosis and atresia
Detection method	LM	LAD	RCA	LCX	LM	LAD	RCA
CAG	6	15	23	4	1	8	7
Echocardiography	8	15	25	3	1	0	0

Detection method	Coronary artery aneurysm				Coronary artery stenosis and atresia
Detection method	LM	LAD	RCA	LCX	LM	LAD	RCA
CAG	6	15	23	4	1	8	7
Echocardiography	8	15	25	3	1	0	0

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