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病例报告:不完全川崎病患者冠状动脉血管壁的结构变化

Case Report: Structural Changes in the Coronary Vessel Wall in a Patient With Incomplete Kawasaki Disease.

作者信息

Ishikawa Takamichi, Uchiyama Hiroki, Mogi Satoshi, Ohtani Hayato

机构信息

Department of Pediatrics, Hamamatsu University School of Medicine, Hamamatsu, Japan.

Division of Cardiology, Internal Medicine 3, Hamamatsu University School of Medicine, Hamamatsu, Japan.

出版信息

Front Pediatr. 2022 Mar 3;10:845723. doi: 10.3389/fped.2022.845723. eCollection 2022.

Abstract

BACKGROUND

Kawasaki disease (KD) is an acute systemic vasculitis of infants and young children that affects medium-sized vessels. Conventional cardiac imaging techniques, such as cardiac catheterization, are useful for characterizing the coronary arterial lesion (CAL) size and luminal diameter of the diseased coronary artery segment in patients with KD, but there are limitations to the visualization of the detailed vascular anatomy. Optical coherence tomography (OCT) is a high-resolution intracoronary arterial imaging modality that can distinguish the three layers of the coronary arterial wall. Several studies have reported coronary artery wall abnormalities in KD patients with coronary arterial aneurysm or regressed aneurysm. However, there have been no reports on changes in the coronary artery wall in cases of incomplete KD without CAL.

CASE PRESENTATION

We herein report an 11-year-old girl with a history of incomplete KD without coronary arterial aneurysms. She had been diagnosed with perimembranous ventricular septal defect (VSD) after birth and had experienced incomplete KD at 1 year old. During her hospitalization for KD, she did not receive intravenous immunoglobulin (IVIG), because she did not meet the Harada score or criteria for treatment in patients with incomplete KD established by the American Heart Association. No dilatation or coronary artery aneurysm were observed on transthoracic echocardiography in the acute or follow-up period. At 11 years old, she received cardiac catheterization and coronary angiography (CAG) for the evaluation of a VSD and follow-up of KD. CAG demonstrated no aneurysm, dilatation, or significant stenosis of the coronary arteries. We performed an OCT study, which revealed the presence of intimal thickening, disruption of the media, and neovascularization in the left anterior descending artery.

CONCLUSION

OCT demonstrates the structural changes of CA even in the patient with incomplete KD who have not been treated with IVIG.

摘要

背景

川崎病(KD)是一种影响婴幼儿的急性全身性血管炎,累及中等大小血管。传统的心脏成像技术,如心导管检查,对于确定KD患者病变冠状动脉节段的冠状动脉病变(CAL)大小和管腔直径很有用,但在详细血管解剖结构可视化方面存在局限性。光学相干断层扫描(OCT)是一种高分辨率的冠状动脉内成像方式,可区分冠状动脉壁的三层结构。几项研究报告了患有冠状动脉瘤或消退性动脉瘤的KD患者的冠状动脉壁异常情况。然而,对于无CAL的不完全KD病例的冠状动脉壁变化尚无报道。

病例报告

我们在此报告一名11岁女孩,有不完全KD病史且无冠状动脉瘤。她出生后被诊断为膜周部室间隔缺损(VSD),1岁时患不完全KD。在她因KD住院期间,由于不符合原田评分或美国心脏协会制定的不完全KD患者治疗标准,未接受静脉注射免疫球蛋白(IVIG)治疗。在急性期或随访期经胸超声心动图未观察到冠状动脉扩张或动脉瘤。11岁时,她接受了心导管检查和冠状动脉造影(CAG)以评估VSD和随访KD。CAG显示冠状动脉无动脉瘤、扩张或明显狭窄。我们进行了OCT研究,结果显示左前降支存在内膜增厚、中层破坏和新生血管形成。

结论

即使在未接受IVIG治疗的不完全KD患者中,OCT也能显示冠状动脉的结构变化。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9ca8/8927284/2eae0fd71303/fped-10-845723-g0001.jpg

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