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1.
J Pediatr ; 274: 114145, 2024 Nov.
Article in English | MEDLINE | ID: mdl-38878963

ABSTRACT

OBJECTIVES: To determine the long-term outcomes among a cohort of patients with Kawasaki disease (KD) and a history of giant coronary artery aneurysms (CAAs) at a single US center. STUDY DESIGN: Medical records for all patients with KD and giant CAAs at a pediatric academic institution were reviewed. Primary outcomes included major adverse cardiovascular events (MACE) and normalization of CA luminal diameter, using Kaplan-Meier analyses. RESULTS: There were 60 patients with KD and giant CAAs identified between 1989 and 2023. The majority of patients were male (71.7%) with a median age at diagnosis of 0.9 years (range, 0.2-13.3 years). Patients were followed for a median of 11 years, up to 34.5 years. MACE occurred in 13 patients (21.7%) at a median of 1.4 years (range, 0.04-22.6 years) after KD diagnosis. The 10-, 20-, and 30-year MACE-free rates were 75%, 75%, and 60%. Patients with maximal CA z scores of ≥20 or bilateral CAA were more likely to have MACE. During follow-up, 26.7% of CAA regressed to a normal luminal diameter at a median of 3.6 years (range, 0.6-12.0 years). The 10-, 20- and 30-year likelihood of CA regression to normal luminal diameter was 36%, 46%, and 46%. CONCLUSIONS: Over 30 years, MACE occurred in nearly 22% of patients, more often in those with bilateral CAA or CA z scores of ≥20. Despite regression to a normal luminal diameter in >25% of CAAs, patients with a history of KD-associated giant CAA require ongoing surveillance for cardiac complications, even years after the initial disease.


Subject(s)
Coronary Aneurysm , Mucocutaneous Lymph Node Syndrome , Humans , Mucocutaneous Lymph Node Syndrome/complications , Mucocutaneous Lymph Node Syndrome/epidemiology , Coronary Aneurysm/etiology , Coronary Aneurysm/epidemiology , Male , Female , Child , Child, Preschool , Adolescent , Infant , Retrospective Studies , United States/epidemiology , Follow-Up Studies , Kaplan-Meier Estimate
2.
Cytokine ; 180: 156667, 2024 Aug.
Article in English | MEDLINE | ID: mdl-38857561

ABSTRACT

BACKGROUND: Kawasaki disease (KD) is a vasculitis of unknown etiology in children aged under 5 years. Coronary arterial aneurysm (CAA) is the major complication of KD. It is no longer though to be a self-limiting disease because its cardiovascular sequelae might persist into adulthood. NLRP3 is a key protein of the NLRP3 inflammasome that participates in sterile inflammatory disease. This study investigated the serum levels of NLRP3 in patients with KD at different stages to explore the relationships between serum NLRP3 and clinical parameters. METHODS: A total of 247 children enrolled in this study. There were 123 patients in the acute stage of KD, and 93 healthy children made up the healthy control (HC) group. Among the acute KD patients, 52 had coronary arterial aneurysm (KD-CAA) and 71 did not (KD-NCAA). 36 patient samples were collected after IVIG and aspirin treatment. Additionally, 29 patients were in the cardiovascular sequelae stage. Enzyme-linked immunosorbent assay was used to measure serum NLRP3 levels in all subjects. RESULTS: Serum NLRP3 was elevated in the KD group and was even higher in the KD-CAA subgroup than in the KD-NCAA subgroup of acute-stage patients. Serum NLRP3 declined when the patients were treated with IVIG and aspirin, but during the convalescent (coronary sequelae) stage, serum NLRP3 re-increased. Serum NLRP3 was higher in the ≥ 6-mm-coronary-arterial-diameter group than that the < 6-mm-diameter group. The ROC curve of serum NLRP3 indicated its utility in the prediction of both KD and KD-CAA. CONCLUSIONS: NLRP3 may be involved in the development of KD and CAA in children with KD. Targeting NLRP3 might mitigate CAA, thereby reducing the risk of cardiovascular events in adulthood.


Subject(s)
Biomarkers , Coronary Aneurysm , Mucocutaneous Lymph Node Syndrome , NLR Family, Pyrin Domain-Containing 3 Protein , Humans , Mucocutaneous Lymph Node Syndrome/blood , Mucocutaneous Lymph Node Syndrome/complications , NLR Family, Pyrin Domain-Containing 3 Protein/blood , Male , Female , Coronary Aneurysm/blood , Coronary Aneurysm/etiology , Child, Preschool , Biomarkers/blood , Infant , Child , Aspirin/therapeutic use , Immunoglobulins, Intravenous/therapeutic use
3.
Catheter Cardiovasc Interv ; 104(2): 247-251, 2024 Aug.
Article in English | MEDLINE | ID: mdl-38860616

ABSTRACT

In the absence of standardized management guidelines, coronary artery aneurysms (CAAs) present therapeutic challenges. Percutaneous coronary intervention (PCI) is rarely explored, especially in giant aneurysms with persistent angina, where surgery might be presumed as a preferred option. We describe the technical aspects and feasibility of PCI using Gore Viabahn expanded polytetrafluoroethylene (ePTFE)-covered nitinol self-expanding stents in a 66-year-old woman with a complex medical history and an enlarging, symptomatic right coronary artery aneurysm. The case was complicated by endoleak after the first stent, but intravascular ultrasound guidance enabled the precise deployment of additional stents, resulting in the successful exclusion of the aneurysm. This case demonstrates steps to successful CAA PCI with Gore Viabahn ePTFE-covered nitinol self-expanding stents and emphasizes that in unsuitable surgical candidates, PCI might be a potential alternative for symptomatic CAAs.


Subject(s)
Alloys , Coronary Aneurysm , Prosthesis Design , Ultrasonography, Interventional , Humans , Coronary Aneurysm/diagnostic imaging , Coronary Aneurysm/surgery , Coronary Aneurysm/therapy , Aged , Female , Treatment Outcome , Self Expandable Metallic Stents , Coronary Angiography , Polytetrafluoroethylene , Percutaneous Coronary Intervention/instrumentation , Stents , Angioplasty, Balloon, Coronary/instrumentation
4.
Pediatr Res ; 95(1): 267-274, 2024 Jan.
Article in English | MEDLINE | ID: mdl-37670043

ABSTRACT

BACKGROUND: We aimed to examine predictive measures for medium and giant coronary artery aneurysms (CAA) in Kawasaki disease (KD) patients. METHODS: Patients who were diagnosed with KD from 2015 to 2021 were retrospectively reviewed. The clinical and laboratory data were compared between medium-giant group and non-medium-giant group. RESULTS: A total of 1331 KD patients were investigated, of whom 63 patients (4.7%) developed medium-giant CAA including 27 patients (2%) with giant CAA. Sex, age, fever duration, intravenous immunoglobulin (IVIG) resistance, platelet count, and albumin level independently predicted medium or giant CAA by multivariate logistic regression analysis. Male, age, duration of fever, IVIG resistance, platelet count, hemoglobin, and erythrocyte sedimentation rate were independent predictors for giant CAA. The two new scoring systems using these factors in identifying patients with medium-giant CAA and giant CAA had respectively sensitivities of 86.89% and 92.59%, and specificities of 81.65% and 87.93%. Validation in 2021 dataset (193 KD patients) showed comparable sensitivity and specificity to development dataset. CONCLUSIONS: Male, age, fever duration, IVIG resistance, platelet count, albumin, hemoglobin, and erythrocyte sedimentation rate might be significant predictors of medium and giant CAA. The sensitivity and specificity in our risk prediction model were higher than in previous research. IMPACT: This is the first study to search for risk factors and establish a prediction model for the development of medium-giant CAA in the Chinese population using z-scores and absolute inner diameter values based on large sample sizes. The sensitivity and specificity in our model were higher than in previous studies. Our research could help clinicians better predict medium-giant CAA and choose more appropriate treatment.


Subject(s)
Coronary Aneurysm , Coronary Artery Disease , Mucocutaneous Lymph Node Syndrome , Humans , Male , Infant , Immunoglobulins, Intravenous/therapeutic use , Retrospective Studies , Mucocutaneous Lymph Node Syndrome/complications , Mucocutaneous Lymph Node Syndrome/diagnosis , Mucocutaneous Lymph Node Syndrome/drug therapy , Coronary Vessels/diagnostic imaging , Coronary Aneurysm/diagnostic imaging , Coronary Aneurysm/etiology , Risk Factors , Albumins , Hemoglobins , Coronary Artery Disease/complications
5.
BMC Cardiovasc Disord ; 24(1): 473, 2024 Sep 05.
Article in English | MEDLINE | ID: mdl-39237865

ABSTRACT

BACKGROUND: Coronary artery thrombosis and myocardial ischemia caused by giant coronary aneurysms are the main causes of death in children with Kawasaki disease. The use of thrombolytic therapy in children with Kawasaki disease who have coronary thrombosis is a controversial topic, especially with respect to the timing of treatment. CASE PRESENTATION: In this article, we report a case of a child aged two years and nine months with Kawasaki disease whose coronary arteries had no involvement in the acute phase. However, by only one week after discharge, the patient returned because we found giant coronary aneurysms complicated by thrombosis via echocardiography. Despite aggressive thrombolytic therapy, the child developed myocardial ischemia during thrombolytic therapy. Fortunately, because of timely treatment, the child's thrombus has dissolved, and the myocardial ischemia has resolved. CONCLUSIONS: This case suggests that for patients at high risk of coronary artery aneurysms, echocardiography may need to be reviewed earlier. Low-molecular-weight heparin should be added to antagonize the early procoagulant effects of warfarin when warfarin therapy is initiated. In the case of first-detected coronary thrombosis, aggressive thrombolytic therapy may be justified, particularly during the acute and subacute phases of the disease course.


Subject(s)
Coronary Aneurysm , Coronary Thrombosis , Mucocutaneous Lymph Node Syndrome , Myocardial Ischemia , Thrombolytic Therapy , Humans , Mucocutaneous Lymph Node Syndrome/complications , Mucocutaneous Lymph Node Syndrome/diagnosis , Mucocutaneous Lymph Node Syndrome/drug therapy , Coronary Thrombosis/diagnostic imaging , Coronary Thrombosis/etiology , Coronary Aneurysm/diagnostic imaging , Coronary Aneurysm/etiology , Treatment Outcome , Child, Preschool , Myocardial Ischemia/etiology , Myocardial Ischemia/drug therapy , Myocardial Ischemia/diagnostic imaging , Male , Anticoagulants/therapeutic use , Fibrinolytic Agents/therapeutic use , Coronary Angiography
6.
BMC Cardiovasc Disord ; 24(1): 187, 2024 Apr 01.
Article in English | MEDLINE | ID: mdl-38561678

ABSTRACT

BACKGROUND: A coronary artery aneurysm (CAA) is an abnormal dilation of a coronary artery segment often accompanied by coronary artery fistula (CAF), leading to communication between a coronary artery and a cardiac chamber or a part of the coronary venous system. Both CAAs and CAFs can present with symptoms and signs of myocardial ischemia and infarction. CASE PRESENTATION: We describe the case of a 46-year-old woman with non-ST-elevation myocardial infarction (NSTEMI) caused by a "giant" CAA. Various imaging modalities revealed a thrombus-containing aneurysm located at the right-posterior cardiac border, with established arteriovenous communication with the distal part of left circumflex artery (LCx). After initial treatment with dual antiplatelet therapy, a relapse of pain was reported along with a new increase in troponin levels, electrocardiographic abnormalities, reduced left ventricular ejection fraction (LVEF) and thrombus enlargement. Surgical excision of the aneurysm was favored, revealing its true size of 6 cm in diameter. Τhe aneurysm was excised without complications. The patient remained asymptomatic during follow-up. CONCLUSIONS: Management of rare entities such as "giant" CAAs and CAFs can be challenging. Cases such as this can serve as precedents to facilitate treatment plans and develop consistent recommendations, emphasizing the importance of personalized strategies for future patients.


Subject(s)
Arteriovenous Fistula , Coronary Aneurysm , Coronary Artery Disease , Myocardial Infarction , Thrombosis , Female , Humans , Middle Aged , Stroke Volume , Ventricular Function, Left , Myocardial Infarction/diagnostic imaging , Myocardial Infarction/etiology , Myocardial Infarction/therapy , Coronary Aneurysm/complications , Coronary Aneurysm/diagnostic imaging , Coronary Artery Disease/diagnosis , Arteriovenous Fistula/complications , Arteriovenous Fistula/diagnostic imaging , Thrombosis/complications , Coronary Angiography/methods
7.
Eur J Pediatr ; 183(10): 4229-4234, 2024 Oct.
Article in English | MEDLINE | ID: mdl-38990385

ABSTRACT

While Kawasaki disease (KD) induced coronary artery aneurysms (KD CAAs) in children are well studied, the features and prognosis of non-KD induced CAAs (non-KD CAAs) in the pediatric population are poorly documented. This case series study is to analyze the etiology and prognosis of non-KD CAAs in children and compare the characteristics of non-KD CAAs and KD CAAs. Non-KD CAA and KD CAA cases at our department from January 2022 to December 2023 were retrospectively collected. Etiologies and prognosis of non-KD CAAs were analyzed. Furthermore, demographic data, biochemical parameters and outcomes between children with Non-KD CAAs and children with KD CAAs were comparatively studied. Fifteen children with non-KD CAAs with a median age of 6 years and 117 children with KD CAAs with a median age of 2.0 years (p = 0.022) were included in this study. The causes of non-KD CAAs include: unknown etiologies (2 cases), coronary artery structural abnormalities (4), Takayasu arteritis (2), virus infection (2), cardiomyopathy (2), aplastic anemia with agranulocytosis (1), ANCA-associated vasculitis (1), and mucopolysaccharidosis (1). In the non-KD CAA group, there were a total of 19 CAAs with 3 being giant, 5 medium, and 11 small; 4 patients had complete CAA regression; an infant with a fistula between the right coronary artery and the coronary sinus complicated with cardiac enlargement died of heart failure. The KD group had significantly higher levels of CRP, white cells counts and ESR with zero mortality. Non-KD CAA cases had a significantly lower regression rate than KD-CAA cases (26.7% vs 66.7%, p = 0.004), and the probability of CAA regression in non-KD patients was 0.341 of that in KD patients (p = 0.006, OR = 0.341, 95% CI: 0.179-0.647). CONCLUSIONS: Various etiologies for Non-KD CAAs are identified. Patients with Non-KD CAAs were observed to have lower inflammatory indexes but poorer recovery than patients with KD CAAs. Therapeutic strategies different than those for KD may be needed for non-KD CAAs. WHAT IS KNOWN: • Coronary artery aneurysm (CAA) in children is most commonly induced by Kawasaki disease (KD CAA), with a 50 ~ 70% regression rate in 1 to 2 years. • CAA induced by diseases other than KD (non-KD CAA) in children is rare and its prognosis remains largely unknown. WHAT IS NEW: • Most non-KD CAA cases are caused by coronary artery structural malformations. • Non-KD CAA in children has poorer prognosis and lower regression rate compared with KD CAA. • In addition to guideline directed anti-platelet and anti-coagulant therapies, treatments targeting the causal factor are necessary for non-KD CAA.


Subject(s)
Coronary Aneurysm , Mucocutaneous Lymph Node Syndrome , Humans , Coronary Aneurysm/etiology , Coronary Aneurysm/diagnosis , Retrospective Studies , Male , Female , Mucocutaneous Lymph Node Syndrome/complications , Child, Preschool , Prognosis , Child , Infant
8.
Eur J Pediatr ; 183(11): 4817-4825, 2024 Nov.
Article in English | MEDLINE | ID: mdl-39240388

ABSTRACT

PURPOSE: This study is to investigate whether angiotensin type 1 receptor blockers (ARBs) or angiotensin-converting enzyme inhibitors (ACEis) can regress coronary artery aneurysm (CAA) in patients with Kawasaki disease (KD). METHODS: This multicenter, prospective, observational study was conducted at 53 institutions throughout Japan. We enrolled patients who were diagnosed with KD after January 2015 and had a medium or large CAA (maximum luminal diameter ≥ 4 mm or z score ≥ + 5) 30 days or later after KD onset. RESULTS: Of the 209 patients, 47 (22%) were taking ARBs/ ACEis. Compared with those in the non-ARB/ACEi group, the baseline CAA diameter was significantly greater (6.7 mm vs. 5.5 mm, p < 0.01), and bilateral CAA (70% vs. 59%, p = 0.01) and giant CAA (32% vs. 20%, p = 0.08) were more frequently observed in the ARB/ACEi group. Although the overall regression rates did not differ between the groups (67% vs. 65%), the regression rates of giant CAA were approximately 1.6 times greater in the ARB/ACEi group than in the non-ARB/ACEi group (36% vs. 23%). Multivariate Cox regression analysis after adjustment for other clinical variables suggested that ARBs/ACEis may be a factor in CAA regression (hazard ratio [HR]: 1.5, 95% confidence interval [CI]: 0.91-2.46). CONCLUSIONS: Although ARBs/ ACEis were used more frequently in patients with severe CAA, these patients had similar CAA regression rates to patients not taking ARBs/ACEis. ARBs/ACEis may be beneficial agents aimed at inducing CAA regression in KD patients. WHAT IS KNOWN: • Large CAAs are less likely to regress and are always at risk of life-threatening cardiac events. • Moderate CAA, age less than 1 year, and female sex have been reported to be factors that promote the regression of CAA. WHAT IS NEW: • Although ARBs/ACEis were used more frequently in patients with severe CAA, these patients had a similar rate of CAA regression to patients who did not take ARBs/ACEis. • The regression rates of giant CAA were approximately 1.6 times greater in the ARB/ACEi group than in the non-ARB/ACEi group.


Subject(s)
Angiotensin-Converting Enzyme Inhibitors , Coronary Aneurysm , Mucocutaneous Lymph Node Syndrome , Humans , Mucocutaneous Lymph Node Syndrome/drug therapy , Mucocutaneous Lymph Node Syndrome/complications , Angiotensin-Converting Enzyme Inhibitors/therapeutic use , Male , Female , Prospective Studies , Coronary Aneurysm/etiology , Coronary Aneurysm/drug therapy , Child, Preschool , Infant , Japan , Treatment Outcome , Child , Renin-Angiotensin System/drug effects , Angiotensin Receptor Antagonists/therapeutic use , Angiotensin II Type 1 Receptor Blockers/therapeutic use
9.
Eur J Pediatr ; 183(10): 4265-4274, 2024 Oct.
Article in English | MEDLINE | ID: mdl-39048743

ABSTRACT

The purpose of this study is to determine whether adding intravenous methylprednisolone pulse (IVMP) to primary adjunctive prednisolone with intravenous immunoglobulin (IVIG) improves treatment resistance and coronary artery aneurysms (CAA) in patients with Kawasaki disease (KD) with a high risk of treatment resistance. This multicenter, prospective, observational study was conducted at 28 hospitals in Japan from October 2016 to June 2020. For patients predicted to be resistant to treatment based on a Kobayashi score ≥ 5 and total bilirubin ≥ 1.0 mg/dL, each hospital independently decided to add IVMP followed by prednisolone, prednisolone alone, or nothing to the primary IVIG therapy. In total, 2856 consecutive KD patients were enrolled; of these, 399 (14.0%) were predicted to be treatment resistant. Patients who were resistant to the primary treatment and required additional treatment comprised 59%, 20%, and 26% of the IVIG-alone group, IVIG-plus-prednisolone group, and IVIG-plus-IVMP group, respectively (P < .0001). The CAA incidence (Z score ≥ 2.5) at month 1 was similar among the treatment groups (6.7%, 4.8%, and 7.3%, respectively; P = .66). CAA occurred more frequently in patients who needed third- or later-line therapy.Conclusions: Primary adjunctive corticosteroid therapy improved the treatment response and suppressed inflammation. However, the study found no benefit of adding IVMP to prednisolone therapy. Patients receiving IVIG alone achieved coronary outcomes comparable to those of patients receiving primary adjunctive corticosteroid therapy although they were more likely to require additional rescue treatment. KD inflammation should be resolved no later than the third line of additional treatment to reduce the risk of CAA.Trial registration: University Hospital Medical Information Network Clinical Trials Registry in Japan ( https://www.umin.ac.jp/ctr/index.htm ) under code UMIN000024937.


Subject(s)
Glucocorticoids , Immunoglobulins, Intravenous , Methylprednisolone , Mucocutaneous Lymph Node Syndrome , Prednisolone , Child, Preschool , Female , Humans , Infant , Male , Coronary Aneurysm/etiology , Coronary Aneurysm/prevention & control , Drug Resistance , Drug Therapy, Combination , Glucocorticoids/administration & dosage , Glucocorticoids/therapeutic use , Immunoglobulins, Intravenous/therapeutic use , Immunoglobulins, Intravenous/administration & dosage , Immunologic Factors/therapeutic use , Immunologic Factors/administration & dosage , Japan , Methylprednisolone/administration & dosage , Methylprednisolone/therapeutic use , Mucocutaneous Lymph Node Syndrome/drug therapy , Mucocutaneous Lymph Node Syndrome/complications , Prednisolone/therapeutic use , Prednisolone/administration & dosage , Prospective Studies , Pulse Therapy, Drug , Treatment Outcome
10.
Pathol Int ; 74(7): 408-414, 2024 Jul.
Article in English | MEDLINE | ID: mdl-38751008

ABSTRACT

We conducted an autopsy on a 3-month-old boy in whom Kawasaki disease (KD) was strongly suspected based on the autopsy findings. The infant had a fever and was brought to a nearby clinic, where he was prescribed antipyretics and kept under observation. However, 15 days after onset of the fever, he suddenly died in bed. He exhibited no obvious redness of the lips, tongue, or conjunctiva. Membranous desquamation was present on his distal fingers. Vasculitis was observed in the coronary arteries, renal artery, splenic artery, and pulmonary vein. In addition, coronary artery aneurysms were present in the right coronary artery and left anterior descending artery. Thrombotic occlusion was observed in one aneurysm in the right coronary artery, resulting in acute myocardial infarction. The coronary artery wall showed infiltration of numerous macrophages and neutrophils. This case was classified as incomplete KD because the coronary artery aneurysm could not be demonstrated before death and was only recognized at autopsy. Pathologists and forensic scientists need to be aware that there are cases in which KD goes undiagnosed and untreated, leading to coronary artery aneurysm formation and sudden death.


Subject(s)
Autopsy , Mucocutaneous Lymph Node Syndrome , Sudden Infant Death , Humans , Mucocutaneous Lymph Node Syndrome/pathology , Mucocutaneous Lymph Node Syndrome/complications , Mucocutaneous Lymph Node Syndrome/diagnosis , Male , Infant , Sudden Infant Death/pathology , Sudden Infant Death/etiology , Sudden Infant Death/diagnosis , Coronary Aneurysm/pathology , Coronary Aneurysm/diagnosis , Coronary Vessels/pathology
11.
BMC Pediatr ; 24(1): 139, 2024 Feb 23.
Article in English | MEDLINE | ID: mdl-38395821

ABSTRACT

BACKGROUND: Japanese Kawasaki disease (KD) risk scores cannot be adopted in non-Japanese patients. In North American populations a baseline coronary artery Z-score > 2 and the Son score are associated with coronary artery aneurysms (CAAs) at 4 and 8 weeks from disease onset. In European populations, the Kawanet and Kawanet-echo scores are associated with intravenous immunoglobulin resistance. This study aims to evaluate the association between KD risk scores and baseline coronary artery Z-scores with CAAs at one, two, and six months in a European population. METHODS: Historical cohort study of all the children diagnosed with KD in a tertiary care hospital in Milan, Italy, between 1st January 2015 and 31st May 2021. Univariate and multivariate (adjusting for age and corticosteroid therapy) logistic regression analyses were used to study the association between the risk scores, a baseline Z-score ≥ 2 and ≥ 2.5 with CAAs. RESULTS: Eighty-nine patients were diagnosed with KD at our Centre, and 12 were excluded based on the exclusion criteria. We included 77 patients, 51 (66%) males, and 26 (34%) females, with a median age at presentation of 27 months (IQR 13-46). A baseline Z-score ≥ 2 was correlated with CAAs at one and two-month follow-ups (odds ratio (OR) 10, 95% confidence interval (CI) 2-72, and OR 18, CI 3-357) but not at six-month follow-up. The Son score showed an association with one and two-month follow-up CAAs (OR 3, CI 1.3-7, and OR 3, CI 1.3-8) but not with a six-month follow-up. CONCLUSIONS: Patients with a baseline Z-score ≥ 2 are at higher risk for CAAs in the long term. The Son score should be tested in larger European samples. Further studies should keep the observational periods longer than 8 weeks from KD onset.


Subject(s)
Coronary Aneurysm , Coronary Artery Disease , Mucocutaneous Lymph Node Syndrome , Child , Male , Female , Humans , Infant , Child, Preschool , Mucocutaneous Lymph Node Syndrome/complications , Mucocutaneous Lymph Node Syndrome/diagnosis , Cohort Studies , Coronary Vessels , Retrospective Studies , Risk Factors , Coronary Aneurysm/diagnostic imaging , Coronary Aneurysm/etiology , Immunoglobulins, Intravenous/therapeutic use
12.
Pediatr Cardiol ; 45(3): 552-559, 2024 Mar.
Article in English | MEDLINE | ID: mdl-38261062

ABSTRACT

Multisystem inflammatory syndrome in children (MIS-C) is a rare condition following COVID-19 infection. Cardiac involvement is common and includes left ventricular systolic dysfunction, cardiac marker elevation, electrocardiogram (ECG) changes, and coronary artery dilation. This single-center retrospective cohort study compares cardiovascular disease between three major SARS-CoV-2 variants and describes the evolution of findings in medium-term follow-up. Of 69 total children (mean age 9.2 years, 58% male), 60 (87%) had cardiovascular involvement with the most common features being troponin elevation in 33 (47%) and left ventricular dysfunction in 22 (32%). Based on presumed infection timing, 61 patients were sorted into variant cohorts of Alpha, Delta, and Omicron. Hospitalization was longer for the Delta group (7.7 days) vs Alpha (5.1 days, p = 0.0065) and Omicron (4.9 days, p = 0.012). Troponin elevation was more common in Delta compared to Alpha (13/20 vs 7/25, p = 0.18), and cumulative evidence of cardiac injury (echocardiographic abnormality and/or troponin elevation) was more common in Delta (17/20) compared with Alpha (12/25, p = 0.013) or Omicron (8/16, p = 0.034). Forty-nine (77%) of the original cohort (n = 69) had no cardiac symptoms or findings beyond 3 months post-hospitalization. Cardiac MRI was performed in 28 patients (between 3 and 6 months post-hospitalization) and was normal in 25 patients (89%). The differences in the variant cohorts may be due to alteration of the immune landscape with higher severity of COVID-19 infection. Despite overall reassuring cardiac outcomes, it is important to note the variability of presentation and remain vigilant with future variants.


Subject(s)
COVID-19/complications , Coronary Aneurysm , Child , Humans , Male , Female , SARS-CoV-2 , Retrospective Studies , Coronary Vessels , Troponin , Systemic Inflammatory Response Syndrome/diagnosis
13.
Cardiol Young ; 34(4): 793-802, 2024 Apr.
Article in English | MEDLINE | ID: mdl-37830370

ABSTRACT

BACKGROUND: Children who develop coronary artery aneurysms after Kawasaki disease are at risk for cardiovascular morbidity, requiring health care transition and lifelong follow-up with an adult specialist. Follow-up losses after health care transition have been reported but without outcome and patient experience evaluation. OBJECTIVE: The Theoretical Domains Framework underpinned our aim to explore the required self-care behaviours and experiences of young adults' post-health care transition. METHODS: A qualitative description approach was used for virtual, 1:1 interviews with 11 participants, recruited after health care transition from a regional cardiac centre in Ontario. Directed content analysis was employed. RESULTS: Health, psychosocial, and lifestyle challenges were compounded by a sense of loss. Six themes emerged within the Theoretical Domains Framework categories. Participants offered novel health care transition programme recommendations. CONCLUSIONS: The realities of health care transition involve multiple, overlapping stressors for young adults with Kawasaki disease and coronary artery aneurysms. Our findings will inform a renewed health care transition programme and will include outcome evaluation.


Subject(s)
Coronary Aneurysm , Mucocutaneous Lymph Node Syndrome , Transition to Adult Care , Child , Young Adult , Humans , Mucocutaneous Lymph Node Syndrome/therapy , Patient Transfer , Coronary Aneurysm/etiology , Coronary Aneurysm/therapy , Ontario
14.
Cardiol Young ; 34(6): 1375-1377, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38618865

ABSTRACT

Giant coronary aneurysm with ventricular fistula is rare. Due to the limited data from randomised clinical trials, therapeutic strategies for coronary aneurysms predominantly rely on on case series and anecdotal evidences. Reporting cases that provide practical experience in managing these aneurysms is therefore crucial. In this article, we report a rare case of a successful surgical management for a thrombosed giant left coronary aneurysm with right ventricular fistula, which is larger than any previously reported cases.


Subject(s)
Coronary Aneurysm , Coronary Angiography , Heart Ventricles , Humans , Coronary Aneurysm/surgery , Coronary Aneurysm/complications , Coronary Aneurysm/diagnostic imaging , Heart Ventricles/surgery , Male , Fistula/surgery , Cardiac Surgical Procedures/methods , Echocardiography
15.
Cardiol Young ; 34(4): 809-814, 2024 Apr.
Article in English | MEDLINE | ID: mdl-37850457

ABSTRACT

OBJECTIVE: I encountered three adult patients with major coronary artery occlusion after Kawasaki disease in childhood, who had developed again acute coronary syndrome of adults in the peripheral branches, such as the 4th segments, the atrioventricular node artery, and the posterior descending artery, of the right coronary artery. METHODS: I reviewed their clinical course and coronary angiograms. RESULTS: Their age at onset of acute coronary syndrome ranged from 29 to 33 years. The male patient with a previous anteroseptal myocardial infarction in children had a symptomatic occlusion of the branch of the 4th posterior descending artery at 32 years of age. Acute coronary syndrome occurred in the area of 4th atrioventricular node artery in two female patients. The collateral arteries from the circumflex artery to the 4th atrioventricular node arteries were not clearly injected. It was suspected that they had developed bilateral giant aneurysms after acute Kawasaki disease. Two patients had an acute myocardial infarction due to thrombotic occlusion in a giant aneurysm of the right coronary artery or the left anterior descending artery, and one patient had an asymptomatic coronary occlusion of the right coronary artery and left anterior descending artery in children. CONCLUSION: Occlusion of peripheral coronary arteries in adulthood can occur in patients with multi-vessel disease caused by Kawasaki disease. Recurrent events of acute coronary syndrome can occur in adults, although its prevalence may be low. Careful follow-up in adults is also needed in this population.


Subject(s)
Acute Coronary Syndrome , Aneurysm , Coronary Aneurysm , Coronary Occlusion , Mucocutaneous Lymph Node Syndrome , Myocardial Infarction , Child , Humans , Male , Female , Young Adult , Adult , Acute Coronary Syndrome/diagnostic imaging , Acute Coronary Syndrome/etiology , Mucocutaneous Lymph Node Syndrome/complications , Myocardial Infarction/etiology , Coronary Angiography , Coronary Vessels/diagnostic imaging , Aneurysm/complications , Coronary Aneurysm/diagnostic imaging , Coronary Aneurysm/etiology
16.
Cardiol Young ; 34(2): 348-355, 2024 Feb.
Article in English | MEDLINE | ID: mdl-37424509

ABSTRACT

BACKGROUND: Coronary artery aneurysms have been considered the most serious complication of Kawasaki disease. However, some coronary artery aneurysms do regress. Therefore, the ability to predict the expected time of coronary artery aneurysm regression is critical. Herein, we have created a nomogram prediction system to determine the early regression (<1 month) among patients with small to medium coronary artery aneurysms. METHODS: Seventy-six Kawasaki disease patients identified with coronary artery aneurysms during the acute or subacute phase were included. All the patients who met inclusion criteria demonstrated regression of coronary artery aneurysms within the first-year post Kawasaki disease diagnosis. The clinical and laboratory parameters were compared between the groups of coronary artery aneurysms regression duration within and beyond 1 month. Multivariate logistic regression analysis was used to identify the independent parameters for early regression based on the results from the univariable analysis. Then nomogram prediction systems were established with associated receiver operating characteristic curves. RESULTS: Among the 76 included patients, 40 cases recovered within 1 month. Haemoglobin, globulin, activated partial thromboplastin time, the number of lesions, location of the aneurysm, and coronary artery aneurysm size were identified as independent factors for early regression of coronary artery aneurysms in Kawasaki disease patients. The predictive nomogram models revealed a high efficacy in predicting early regression of coronary artery aneurysms. CONCLUSION: The size of coronary artery aneurysms, the number of lesions, and the location of aneurysms presented better predictive value for predicting coronary artery aneurysms regression. The nomogram system created from the identified risk factors successfully predicted early coronary artery aneurysm regression.


Subject(s)
Coronary Aneurysm , Coronary Artery Disease , Mucocutaneous Lymph Node Syndrome , Humans , Coronary Vessels/diagnostic imaging , Coronary Vessels/pathology , Nomograms , Mucocutaneous Lymph Node Syndrome/complications , Mucocutaneous Lymph Node Syndrome/diagnosis , Mucocutaneous Lymph Node Syndrome/pathology , Coronary Aneurysm/diagnosis , Coronary Aneurysm/etiology , ROC Curve , Retrospective Studies , Coronary Artery Disease/etiology , Coronary Artery Disease/complications
17.
Cardiol Young ; 34(1): 224-227, 2024 Jan.
Article in English | MEDLINE | ID: mdl-38038187

ABSTRACT

Although Kawasaki disease is often self-limiting, significant cardiovascular sequelae may occur in the acute or late stage. The most common late complication is persistent coronary artery aneurysm, which can lead to myocardial ischaemia and even myocardial infarction. We report a case of coronary artery bypass grafting in a 16-year-old boy with a history of undiagnosed Kawasaki disease. Increased awareness of Kawasaki disease, especially among children between the ages of 6 months and 5 years, can increase early treatment and prevent serious complications that may occur in the future.


Subject(s)
Coronary Aneurysm , Coronary Artery Disease , Mucocutaneous Lymph Node Syndrome , Myocardial Infarction , Male , Child , Humans , Adolescent , Infant , Mucocutaneous Lymph Node Syndrome/complications , Mucocutaneous Lymph Node Syndrome/diagnosis , Myocardial Infarction/complications , Myocardial Infarction/diagnosis , Coronary Artery Bypass , Coronary Artery Disease/complications , Coronary Aneurysm/diagnostic imaging , Coronary Aneurysm/etiology
18.
Cardiol Young ; 34(4): 854-858, 2024 Apr.
Article in English | MEDLINE | ID: mdl-37905350

ABSTRACT

INTRODUCTION: Infection with Sars-CoV-2 is known to cause cardiac injury and coronary artery changes in moderate to severe acute COVID-19 and post-acute multisystem inflammatory syndrome in children (MIS-C). However, little is known about the potential for cardiac involvement, in particular coronary artery dilation, in asymptomatic or mild cases of COVID-19. METHODS: A retrospective review of children ≤ 18 years of age with a history of asymptomatic or mild COVID-19 disease who underwent echocardiography after Sars-CoV-2 infection is conducted. Patients were excluded if they had been hospitalised for COVID-19/MIS-C or had a history of cardiac disease that could affect coronary artery dimension. Coronary artery dilation was defined as the Boston Z-score greater than 2.0. RESULTS: One hundred and fifty-seven patients met inclusion criteria with a mean age of 9.4 years (+/- 5.4 years). Eighty-four (54%) patients were identified as having COVID-19 through positive antibody testing. All patients underwent electrocardiogram and echocardiogram as part of their cardiology evaluation. One hundred and thirty-five (86%) patients had a normal evaluation or only a minor variant on electrocardiogram, while 22 patients had abnormalities on echocardiogram, 4 of which demonstrated coronary artery dilation based on the Boston Z-score. CONCLUSIONS: Much of the literature for post-infectious screening and follow-up focuses on patients with a history of moderate to severe COVID-19 disease, emphasising the need for surveillance for the potential development of myocarditis. In this study, 4 out of 157 (2.5%) children with a history of asymptomatic or mild COVID-19 disease without MIS-C were found to have some degree of coronary artery dilation. The significance of this finding currently remains unknown.


Subject(s)
COVID-19/complications , Coronary Aneurysm , Systemic Inflammatory Response Syndrome , Child , Humans , Coronary Vessels/diagnostic imaging , Dilatation , SARS-CoV-2
19.
Kyobu Geka ; 77(6): 415-421, 2024 Jun.
Article in Japanese | MEDLINE | ID: mdl-39009533

ABSTRACT

The case is a 76-year-old woman. She was admitted to the hospital because of chest and back pain. Coronary angiography revealed a 62-mm giant coronary artery aneurysm originating from the left main trunk( LMT), and urgent surgery was performed. Coronary artery-pulmonary artery fistula along with coronary artery aneurysm was completely removed by surgery. In this case, the reconstruction strategy for the LMT was crucial. The aneurysm wall was completely resected, allowing the coronary artery to return to its original course, and the length of the LMT defect was <2 cm. We determined that anatomical reconstruction of the LMT was optimal and succeeded in replacing a short great saphenous vein corresponding to the length of the defect. The patient was discharged without any complications.


Subject(s)
Coronary Aneurysm , Humans , Aged , Female , Coronary Aneurysm/surgery , Coronary Aneurysm/diagnostic imaging , Coronary Vessels/surgery , Coronary Vessels/diagnostic imaging , Coronary Angiography
20.
Kyobu Geka ; 77(9): 697-701, 2024 Sep.
Article in Japanese | MEDLINE | ID: mdl-39370287

ABSTRACT

The patient was an 86-year-old woman. She was brought to the emergency department because of chest pain and respiratory distress symptoms that did not improve. 12-lead electrocardiogram, echocardiography, and blood tests were negative for acute coronary syndrome, but a chest contrast computed tomography( CT) -scan showed pericardial effusion and a coronary artery aneurysm 20 mm in diameter, and punctured pericardial effusion revealed bloody fluid. A cardiac catheterization was performed. He underwent emergent surgery after being diagnosed as having a ruptured coronary aneurysm associated with a coronary pulmonary artery fistula. The aneurysm was dissected using cardiopulmonary bypass, and the coronary artery-pulmonary artery fistula was closed. The patient had a good postoperative course and was discharged in good condition. There have been few reports of rupture when the aneurysm diameter is less than 30 mm. Regardless of the size of the aneurysm, it has a risk of rupture and it is important to consider surgery.


Subject(s)
Aneurysm, Ruptured , Arterio-Arterial Fistula , Coronary Aneurysm , Pulmonary Artery , Humans , Aged, 80 and over , Female , Pulmonary Artery/surgery , Pulmonary Artery/diagnostic imaging , Pulmonary Artery/abnormalities , Coronary Aneurysm/surgery , Coronary Aneurysm/diagnostic imaging , Coronary Aneurysm/complications , Aneurysm, Ruptured/surgery , Aneurysm, Ruptured/diagnostic imaging , Aneurysm, Ruptured/complications , Arterio-Arterial Fistula/surgery , Arterio-Arterial Fistula/diagnostic imaging , Arterio-Arterial Fistula/complications , Tomography, X-Ray Computed , Treatment Outcome
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