Responsiveness to intravenous immunoglobulins and occurrence of coronary artery abnormalities in a single-center cohort of Italian patients with Kawasaki syndrome

Rigante, Donato; Valentini, Piero; Rizzo, Daniela; Leo, Andrea; Rosa, Gabriella De; Onesimo, Roberta; Nisco, Alessia De; Angelone, Donatella Francesca; Compagnone, Adele; Delogu, Angelica Bibiana

doi:10.1007/s00296-009-1337-1

Cited by 56 publications

(35 citation statements)

References 27 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…However, because of the small cohort this observation could only be a coincidence. In comparison to an Italian study that shows how higher values of CRP may play a crucial role in determining the development of coronary complications [32], our analysis revealed no laboratory value or any distinct clinical symptom that can be correlated with the development of coronary aneurysms. An echocardiography is therefore necessary whenever KD is suspected.…”

Section: Discussioncontrasting

confidence: 84%

Kawasaki disease in children and adolescents: clinical data of Kawasaki patients in a western region (Tyrol) of Austria from 2003–2012

et al. 2014

View full text Add to dashboard Cite

BackgroundKawasaki disease (KD) is a rare vasculitis seen predominantly in children. In developing countries, it is the leading cause of childhood-acquired heart disease. Besides a case report from 1981 there have been no data published dealing with the epidemiology and clinical aspects of KD in Austria.MethodsThe purpose of the present study was to investigate the clinical spectrum of KD in a geographically determined cohort of infants, children, and adolescents that were diagnosed and treated at the University Hospital of Innsbruck from 2003–2012.ResultsThirty-two patients were included in the study with a median age of 32.96 months (2–192). 59.4% of the patients were aged between six months and four years. The male-to-female ratio was 1:1.13. Clinical examination revealed non-purulent conjunctivitis and exanthema as the most common symptoms (84.4%). 75% showed oropharyngeal changes, 21.9% had gastrointestinal complaints such as diarrhoe, stomachache or vomiting prior to diagnosis. One third of the patients were admitted with a preliminary diagnosis, whereas 78.1% were pre-treated with antibiotics. The median fever duration at the time of presentation was estimated with 4.96 days (1–14), at time of diagnosis 6.76 days (3–15).75% were diagnosed with complete KD, and 25% with an incomplete form of the disease. There was no significant difference in the duration of fever neither between complete and incomplete KD, nor between the different age groups. Typical laboratory findings included increased C-reactive protein (CRP) (80.6%) and erythrocyte sedimentation rate (ESR) (96%),leukocytosis (48.4%) and thrombocytosis (40.6%) without any significant quantitative difference between complete and incomplete KD. Coronary complications could be observed in six patients: one with a coronary aneurysm and five with tubular dilatation of the coronary arteries. Our patient cohort represents the age distribution as described in literature and emphasizes that KD could affect persons of any age. The frequency of occurrence of the clinical symptoms differs from previous reports – in our study, we predominantly observed non-purulent conjunctivitis and exanthema.ConclusionKD should always be considered as a differential diagnosis in a child with fever of unknown origin, as treatment can significantly decrease the frequency of coronary complications.Electronic supplementary materialThe online version of this article (doi:10.1186/1546-0096-12-37) contains supplementary material, which is available to authorized users.

show abstract

Section: Discussioncontrasting

confidence: 84%

Kawasaki disease in children and adolescents: clinical data of Kawasaki patients in a western region (Tyrol) of Austria from 2003–2012

et al. 2014

View full text Add to dashboard Cite

show abstract

“…However, ~30% of cases have been reported to be unresponsive to IVIG, and additional therapy is required (9). If patients who are unresponsive to IVIG therapy are not identified in a timely manner, CAAs may still develop (10). Therefore, the early diagnosis of KD and the prediction of the therapeutic outcomes of IVIG are important issues.…”

Section: Introductionmentioning

confidence: 99%

Serum exosomal miR‑328, miR‑575, miR‑134 and miR‑671‑5p as potential biomarkers for the diagnosis of Kawasaki disease and the prediction of therapeutic outcomes of intravenous immunoglobulin therapy

2018

View full text Add to dashboard Cite

The present study was conducted to screen serum exosomal microRNAs (miRNAs) for the early diagnosis of Kawasaki disease (KD) and to investigate their underlying mechanisms by analyzing microarray data under accession numbers GSE60965 [exosomal miRNA, including three pooled serum samples from 5 healthy children, 5 patients with KD and 5 patients with KD following intravenous immunoglobulin (IVIG) therapy] and GSE73577 (mRNA, including peripheral blood mononuclear cell samples from 19 patients with KD prior to and following IVIG treatment) from the Gene Expression Omnibus database. Differentially expressed miRNAs (DE-miRNAs) and genes (DEGs) were identified using the Linear Models for Microarray data method, and the mRNA targets of DE-miRNAs were predicted using the miRWalk 2.0 database. The functions of the target genes were analyzed using the Database for Annotation, Visualization and Integrated Discovery (DAVID). As a result, 65 DE-miRNAs were identified with different expression patterns between the healthy children and patients with KD and between patients with KD and patients with KD following IVIG therapy. The target genes of 15 common DE-miRNAs were predicted. Following overlapping the target genes of DE-miRNAs with 355 DEGs, 28 common genes were identified and further screened to construct a network containing 30 miRNA-mRNA regulatory associations. Of these associations, only miR-328-spectrin α, erythrocytic 1, miR-575-cyclic AMP-responsive element-binding protein 5/b-1,4-galactosyltransferase 5/WD repeat and FYVE domain-containing 3/cystatin-A/C-X-C motif chemokine receptor 1/protein phosphatase 1 regulatory subunit 3B, miR-134-acyl-CoA synthetase long chain family member 1/C-type lectin domain family 1 member A and miR-671-5p-tripartite motif containing 25/leucine rich repeat kinase 2/kinesin family member 1B/leucine rich repeat neuronal 1 were involved in the negative regulation of gene expression. Functional analysis indicated that the identified target genes may be associated with inflammation. Accordingly, serum exosomal miR-328, miR-575, miR-134 and miR-671-5p may act as potential biomarkers for the diagnosis of KD and the prediction of outcomes of the IVIG therapy by influencing the expression of inflammatory genes.

show abstract

“…In previous reports [1, 20, 21], patients who were IVIG resistant were found to have a higher risk of coronary artery abnormalities. In the present study, 92% (58/63) of patients responded to initial IVIG therapy and 8% (5/63) of patients did not.…”

Section: Discussionmentioning

confidence: 98%

Plasma Clusterin Concentrations May Predict Resistance to Intravenous Immunoglobulin in Patients with Kawasaki Disease

Ou‐Yang

Kuo

Lin

et al. 2013

The Scientific World Journal

View full text Add to dashboard Cite

Kawasaki disease (KD) is an acute febrile vasculitic syndrome of early childhood often complicated by coronary artery lesion that drastically reduces the quality of life. The study aimed to identify a reliable marker for predicting nonresponsiveness to the first course of intravenous immunoglobulin (IVIG) in KD patients. A total of 63 patients with KD were enrolled in the study (IVIG response, 58; IVIG resistance, 5). Plasma samples were collected before and after IVIG infusion for measurement of biomarkers. Patients' clinical characteristics and laboratory data were also analyzed. A receiver operating characteristic curve was generated to identify a cut-off value for predicting IVIG resistance. Among the biomarkers, the difference in plasma clusterin concentrations before and after IVIG infusion (CLUSTER 12) was significantly related to IVIG resistance (P = 0.040; 95% confidence interval (CI): −25.8% to −6.0%). Using a CLUSTER 12 cut-off value of <8.52 mg/L, the odds ratio for IVIG resistance was 11.467 (95% CI: 1.186 to 110.853). Patients with plasma CLUSTER 12 concentrations >8.52 mg/L had a much higher risk of IVIG resistance than those with CLUSTER 12 concentrations <8.52 mg/L. Plasma clusterin concentration shows promise as a candidate biomarker for predicting IVIG resistance in patients with KD.

show abstract

Responsiveness to intravenous immunoglobulins and occurrence of coronary artery abnormalities in a single-center cohort of Italian patients with Kawasaki syndrome

Cited by 56 publications

References 27 publications

Kawasaki disease in children and adolescents: clinical data of Kawasaki patients in a western region (Tyrol) of Austria from 2003–2012

Kawasaki disease in children and adolescents: clinical data of Kawasaki patients in a western region (Tyrol) of Austria from 2003–2012

Serum exosomal miR‑328, miR‑575, miR‑134 and miR‑671‑5p as potential biomarkers for the diagnosis of Kawasaki disease and the prediction of therapeutic outcomes of intravenous immunoglobulin therapy

Plasma Clusterin Concentrations May Predict Resistance to Intravenous Immunoglobulin in Patients with Kawasaki Disease

Contact Info

Product

Resources

About