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A Ruptured Coronary Artery Aneurysm Secondary to Kawasaki Disease.

ABSTRACT: Coronary artery aneurysm occurs in 0.3%-0.8% of patients with Kawasaki disease, and cases of rupture are extremely rare. Only 2 cases have been reported in which the patients survived. We report a case of ruptured coronary artery aneurysm that was treated with coronary artery bypass grafting and extracorporeal membrane oxygenation. (Level of Difficulty: Advanced.).

SUBMITTER: Suzuki H

PROVIDER: S-EPMC9270623 | biostudies-literature | 2022 Jul

REPOSITORIES: biostudies-literature

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A Ruptured Coronary Artery Aneurysm Secondary to Kawasaki Disease.

Suzuki Hiroyuki H Kuroko Yosuke Y Kotani Yasuhiro Y Sakoda Naoya N Kasahara Shingo S

JACC. Case reports 20220706 13

Coronary artery aneurysm occurs in 0.3%-0.8% of patients with Kawasaki disease, and cases of rupture are extremely rare. Only 2 cases have been reported in which the patients survived. We report a case of ruptured coronary artery aneurysm that was treated with coronary artery bypass grafting and extracorporeal membrane oxygenation. (<b>Level of Difficulty: Advanced.</b>). ...[more]

PMID: 35818603

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High risk of coronary artery aneurysm in Kawasaki disease.

Project description:ObjectiveKawasaki disease (KD) is a vasculitis of unknown aetiology with a high risk of coronary aneurysms if untreated. Timely treatment with intravenous immunoglobulin decreases the risk for coronary artery aneurysms (CAA). In this study, we set out to elucidate the factors associated with the risk of developing CAA.MethodsRecords of all KD-diagnosed children in Skåne between 2004 and 2014 were collected and clinical and demographic data were compiled. KD is defined according to the revised American Heart Association diagnostic criteria and classified as either complete KD (cKD) or incomplete KD (iKD).ResultsKD was diagnosed in 77 children and CAA was found in 31% (n = 24). Children with CAA were younger compared with children without (median; 20 vs 34 months) and intravenous immunoglobulin treatment within 10 days was less likely to be received (75% vs 91%). In children presenting with iKD, 47% developed CAA compared with 21% in cKD patients. Using multivariate analysis, an association between the risk of CAA with low age in children with iKD was observed.ConclusionThe risk of CAA development is disturbingly high in young children with iKD. This highlights the importance of rapid intense treatment and vigilance in infants, who are the most difficult to diagnose, in order to reduce the frequency of CAA.

| S-EPMC8023999 | biostudies-literature

Coronary Artery Aneurysm Rupture in Kawasaki Disease and SARS-CoV-2 Infection.

Project description: Not available

| S-EPMC10899712 | biostudies-literature

Matrix metalloproteinase haplotypes associated with coronary artery aneurysm formation in patients with Kawasaki disease.

Project description:Aneurysms of the vascular wall represent a final common pathway for a number of inflammatory processes, including atherosclerosis and idiopathic vasculitis syndromes. Kawasaki disease (KD) is an acute, self-limited vasculitis in children and the leading cause of acquired coronary artery aneurysms. We sought to identify shared molecular mechanisms of aneurysm formation by genotyping eight polymorphisms in matrix metalloproteinase (MMP)-1, 3, 7, 12 and 13 in the gene cluster on Chr.11q22, whose gene products have been implicated in aneurysm formation or are known to have elastase activity. We genotyped 482 US-UK KD patients (aneurysm+: n=111, aneurysm-: n=371) and tested our findings in an independent cohort of 200 Japanese KD patients (aneurysm+: n=58, aneurysm-: n=142). Analysis of the five MMP genes identified modest trends in allele and genotype frequencies for MMP-3 rs3025058 (-/T) and haplotypes containing MMP-3 rs3025058 (-/T) and MMP-12 rs2276109 (A/G) (nominal P=2 to 4 × 10(-5)) that conferred increased risk of aneurysm formation in US-UK subjects. This finding was validated in Japanese subjects and suggests the importance of this locus in aneurysm formation in children with KD. The region encompassing these risk haplotypes is a prime candidate for resequencing to look for rare genetic variation that may influence aneurysm formation.

| S-EPMC3005773 | biostudies-literature

Sorting nexin 24 genetic variation associates with coronary artery aneurysm severity in Kawasaki disease patients.

Project description:BACKGROUND:The sorting nexin (SNX) family is involved in endocytosis and protein trafficking and plays multiple roles in various diseases. The role of SNX proteins in Kawasaki disease (KD) is not known. We attempted to test whether genetic SNX variation associates with the risk of coronary artery aneurysm (CAA) formation in KD. METHODS AND RESULTS:Chi-square tests were used to identify SNX24 genetic variants associated with KD susceptibility and CAA formation in KD; models were adjusted for fever duration and time of first administration of intravenous immunoglobulin. We obtained clinical characteristics and genotypes from KD patients (76 with CAA and 186 without CAA) in a population-based retrospective KD cohort study (n?=?262). Clinical and genetic factors were associated with CAA formation in KD. In addition, endothelial cell inflammation was evaluated. Significant correlation was observed between KD with CAA complications and the rs28891 single-nucleotide polymorphism in SNX24. Patients with CC?+?CT genotypes had lesser CAA complications. In lipopolysaccharide-treated human umbilical vein endothelial cells, siRNA knockdown of SNX24 significantly decreased gene expression of the proinflammatory cytokines IL-1 beta, IL-6, and IL-8. CONCLUSIONS:Polymorphisms in SNX24 may be used as genetic markers for the diagnosis and prognosis of CAA formation in KD.

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Preventing coronary artery lesions in Kawasaki disease.

Project description:A form of systemic vasculitis that affects mostly small and medium-sized vessels, Kawasaki disease (KD) is most commonly found in children under the age of 5 years old. Though its etiology is unknown, KD has been the most frequent acquired heart disease in developing countries. Its incidence has increased over recent decades in many centuries, including Japan, Korea, and China. The most severe complications of KD are coronary artery lesions (CAL), including dilation, fistula, aneurysm, arterial remodeling, stenosis, and occlusion. Aneurysm formation has been observed in 20-25% of KD patients that do not receive intravenous immunoglobulin (IVIG) treatment, and in 3-5% that do receive it. Coronary artery dilation has been found in about 30% of KD patients in the acute stage, although mostly in the transient form. Diminishing the occurrence and regression of CAL is a vital part of treating KD. In this review article, I demonstrate the clinical method to prevent CAL formation used at the Kawasaki Disease Center in Taiwan.

| S-EPMC6136281 | biostudies-other

Phase I/IIa Trial of Atorvastatin in Patients with Acute Kawasaki Disease with Coronary Artery Aneurysm.

Project description:ObjectivesTo determine the safety, tolerability, pharmacokinetics, and immunomodulatory effects of a 6-week course of atorvastatin in patients with acute Kawasaki disease with coronary artery (CA) aneurysm (CAA).Study designThis was a Phase I/IIa 2-center dose-escalation study of atorvastatin (0.125-0.75 mg/kg/day) in 34 patients with Kawasaki disease (aged 2-17 years) with echocardiographic evidence of CAA. We measured levels of the brain metabolite 24(S)-hydroxycholesterol (24-OHC), serum lipids, acute-phase reactants, liver enzymes, and creatine phosphokinase; peripheral blood mononuclear cell populations; and CA internal diameter normalized for body surface area before atorvastatin treatment and at 2 and 6 weeks after initiation of atorvastatin treatment.ResultsA 6-week course of up to 0.75 mg/kg/day of atorvastatin was well tolerated by the 34 subjects (median age, 5.3 years; IQR, 2.6-6.4 years), with no serious adverse events attributable to the study drug. The areas under the curve for atorvastatin and its metabolite were larger in the study subjects compared with those reported in adults, suggesting a slower rate of metabolism in children. The 24-OHC levels were similar between the atorvastatin-treated subjects and matched controls.ConclusionsAtorvastatin was safe and well tolerated in our cohort of children with acute Kawasaki disease and CAA. A Phase III efficacy trial is warranted in this patient population, which may benefit from the known anti-inflammatory and immunomodulatory effects of this drug.

| S-EPMC6878161 | biostudies-literature

The Relationship Between Lipoprotein-Associated Phospholipase-A2 and Coronary Artery Aneurysm in Children With Kawasaki Disease.

Project description:BackgroundCoronary artery lesions including aneurysm, as the most severe complications of Kawasaki disease (KD), remain of great concern. Lipoprotein-associated phospholipase A2 (Lp-PLA2) is implicated in the regulation of inflammatory response and lipid metabolism. Since excessive inflammatory response and aberrant lipid metabolism have involved in the development of KD, we in this study sought to investigate the relationship between coronary artery aneurysm (CAA) and Lp-PLA2 and other blood parameters in children with KD.MethodsThe participants included 71 KD patients, 63 healthy controls (HCs) and 51 febrile controls (FCs). KD patients were divided into KD-CAA (KD with CAA) group and KD-NCAA (KD without CAA) group. Serum Lp-PLA2 levels were measured using enzyme-linked immunosorbent assays. Other routine clinical parameters were also detected.ResultsSerum Lp-PLA2 levels in KD group [4.83 μg/mL (3.95-6.77)] were significantly higher than those in HC [1.29 μg/mL (0.95-2.05)] and FC [1.74 μg/mL (1.18-2.74)] groups. KD-CAA group [5.56 μg/mL (4.55-22.01)] presented substantially higher serum Lp-PLA2 levels as compared with KD-NCAA group [4.64 μg/mL (2.60-5.55)]. In KD group, serum Lp-PLA2 level was positively related with erythrocyte sedimentation rate, the levels of leukocytes, platelets, albumin, creatine kinase-MB, and D-dimer, and the Z-scores of left main CA, right CA, left anterior descending CA, and left circumflex CA; and negatively related with mean corpuscular hemoglobin concentration and mean platelet volume. Moreover, receiver operating characteristic curves showed that Lp-PLA2 exhibited superior and moderate diagnostic performance for distinguishing KD patients from HC and FC ones, respectively, and possessed the potential ability to predict the occurrence of CAAs in KD.ConclusionLp-PLA2 may be related to KD and the formation of CAAs, and thus may serve as a potential diagnostic biomarker for KD.

| S-EPMC9008257 | biostudies-literature

Ruptured mycotic coronary artery aneurysm hybrid treatment with covered stent and surgical debridement☆

Project description: Not available

| S-EPMC10562108 | biostudies-literature

Case report: Multimodality imaging revealing ruptured giant coronary artery aneurysm presenting with hemoptysis

Project description:Giant coronary artery aneurysm (CAA) is a relatively uncommon disease that is defined by a focal dilation of at least 20 mm and characterized by various clinical symptoms. However, cases presenting primarily with hemoptysis have not been reported. A man in his late 20 s suffering from persistent chest pain for over 2 months was transferred to our emergency department for intermittent hemoptysis lasting for 12 h. Bronchoscopy detected fresh blood in the left upper lobe bronchus without a definite bleeding source. Magnetic resonance imaging (MRI) demonstrated a heterogeneous mass and the high-intensity signals suggested active bleeding. coronary computed tomography (CT) angiography demonstrated a giant ruptured CAA wrapped in a large mediastinal mass Coronary angiography confirmed the CAA originating from the left anterior descending artery. The patient underwent an emergency sternotomy and an enormous hematoma arising from a ruptured CAA densely adhering to the left lung was identified. The patient recovered uneventfully and was discharged on the 7th day later. The ruptured CAA masquerading as hemoptysis highlights the indispensability of multimodality imaging for accurate diagnosis. Urgent surgical intervention is desirable in such life-threatening conditions.

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Coil Embolization of a Ruptured Mycotic Coronary Artery Aneurysm in a Pediatric Patient.

Project description:Mycotic coronary artery aneurysms are rare but have a high risk of mortality. Traditional management is surgical, but percutaneous intervention can be performed in patients with high surgical risk. In this report, we describe a case of mycotic coronary aneurysm in a pediatric patient successfully managed with percutaneous coil embolization. (Level of Difficulty: Advanced.).

| S-EPMC10500331 | biostudies-literature

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