Development of a score for early identification of children with Kawasaki disease requiring second-line treatment in multi-ethnic populations in Europe: A multicentre retrospective cohort study
Summary: Background: Early identification of high-risk patients is essential to stratify treatment algorithms of Kawasaki disease (KD) and to appropriately select patients at risk for complicated disease who would benefit from intensified first-line treatment. Several scores have been developed and...
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| Format: | Article |
| Language: | English |
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Elsevier
2022-11-01
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| Series: | The Lancet Regional Health. Europe |
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| Online Access: | http://www.sciencedirect.com/science/article/pii/S2666776222001776 |
| _version_ | 1828152888909627392 |
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| author | Naim Ouldali Rosa Maria Dellepiane Sofia Torreggiani Lucia Mauri Gladys Beaujour Constance Beyler Martina Cucchetti Cécile Dumaine Adriano La Vecchia Isabelle Melki Rita Stracquadaino Caroline Vinit Rolando Cimaz Ulrich Meinzer |
| author_facet | Naim Ouldali Rosa Maria Dellepiane Sofia Torreggiani Lucia Mauri Gladys Beaujour Constance Beyler Martina Cucchetti Cécile Dumaine Adriano La Vecchia Isabelle Melki Rita Stracquadaino Caroline Vinit Rolando Cimaz Ulrich Meinzer |
| author_sort | Naim Ouldali |
| collection | DOAJ |
| description | Summary: Background: Early identification of high-risk patients is essential to stratify treatment algorithms of Kawasaki disease (KD) and to appropriately select patients at risk for complicated disease who would benefit from intensified first-line treatment. Several scores have been developed and validated in Asian populations but have shown low sensitivity in predicting intravenous immunoglobulin (IVIG) resistance in non-Asian populations. We sought methods to predict the need for secondary treatment after initial IVIG in non-Asian populations. Methods: We conducted a retrospective, multicenter study including consecutive patients with KD admitted to two tertiary pediatric hospitals in France and Italy from 2005 to 2019. We evaluated the performance of the Kawanet-score and compared it with the performances of initial echocardiography findings, and of a newly proposed score combining the Kawanet-score and initial echocardiography findings. For each score, we assessed the AUC, sensitivity and specificity for predicting the need for second-line treatment. Findings: We included 363 children with KD, 186 from France and 177 from Italy, of whom 57 (16%) required second-line therapy after the first IVIG dose. The Kawanet score, coronary artery dilation or aneurysm with maximal Z-score ≥2.0 at baseline, and abnormal initial echocardiography had a sensitivity of 43%, 55% and 65% and a specificity of 73%, 78%, 73%, respectively, for predicting the need for second-line treatment. The Kawanet-score was significantly improved by combining it with initial echocardiography findings. The best predictive performance (Sensitivity 76%, Specificity 54%) was obtained by combining the Kawanet-score with abnormal initial echocardiography, defined by the presence of either coronary artery maximal Z-score ≥2.0, pericarditis, myocarditis and/or ventricular dysfunction. This score predicted the need for second-line treatment in European, African/Afro-Caribbean and Asian ethnicity with a sensitivity of 80%, 65% and 100%, respectively, and a specificity of 56%, 51% and 61%, respectively. Interpretation: Our study proposes a score that we named the Kawanet-echo score, which allows early identification of children with KD who require a second-line treatment in multi-ethnic populations in Europe. Funding: None. |
| first_indexed | 2024-04-11T22:16:49Z |
| format | Article |
| id | doaj.art-e78447e471a74ce9864133600b849bd2 |
| institution | Directory Open Access Journal |
| issn | 2666-7762 |
| language | English |
| last_indexed | 2024-04-11T22:16:49Z |
| publishDate | 2022-11-01 |
| publisher | Elsevier |
| record_format | Article |
| series | The Lancet Regional Health. Europe |
| spelling | doaj.art-e78447e471a74ce9864133600b849bd22022-12-22T04:00:21ZengElsevierThe Lancet Regional Health. Europe2666-77622022-11-0122100481Development of a score for early identification of children with Kawasaki disease requiring second-line treatment in multi-ethnic populations in Europe: A multicentre retrospective cohort studyNaim Ouldali0Rosa Maria Dellepiane1Sofia Torreggiani2Lucia Mauri3Gladys Beaujour4Constance Beyler5Martina Cucchetti6Cécile Dumaine7Adriano La Vecchia8Isabelle Melki9Rita Stracquadaino10Caroline Vinit11Rolando Cimaz12Ulrich Meinzer13Department of General Paediatrics, Paediatric Internal Medicine, Rheumatology and Infectious Diseases, National Reference Centre for Rare Paediatric Inflammatory Rheumatisms and Systemic Autoimmune diseases RAISE, Robert Debré University Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France; Clinical Epidemiology Unit, ECEVE INSERM UMR 1123, Robert Debré University Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France; ACTIV, Association Clinique et Thérapeutique Infantile du Val-de-Marne, Créteil, France; Université de Paris, UFR de Médecine, Paris, FrancePediatric Intermediate Care Unit, Fondazione IRCCS Ca’ Granda Ospedale Maggiore Policlinico, Milan, ItalyPediatric Intermediate Care Unit, Fondazione IRCCS Ca’ Granda Ospedale Maggiore Policlinico, Milan, ItalyDepartment of Paediatric Cardiology, Fondazione IRCCS Ca’ Granda Ospedale Maggiore Policlinico, Milan, ItalyDepartment of General Paediatrics, Paediatric Internal Medicine, Rheumatology and Infectious Diseases, National Reference Centre for Rare Paediatric Inflammatory Rheumatisms and Systemic Autoimmune diseases RAISE, Robert Debré University Hospital, Assistance Publique-Hôpitaux de Paris, Paris, FranceDepartment of Paediatric Cardiology, Robert Debré University Hospital, Assistance Publique-Hôpitaux de Paris, Paris, FranceUniversity of Milan, Milan, ItalyDepartment of General Paediatrics, Paediatric Internal Medicine, Rheumatology and Infectious Diseases, National Reference Centre for Rare Paediatric Inflammatory Rheumatisms and Systemic Autoimmune diseases RAISE, Robert Debré University Hospital, Assistance Publique-Hôpitaux de Paris, Paris, FranceUniversity of Milan, Milan, ItalyDepartment of General Paediatrics, Paediatric Internal Medicine, Rheumatology and Infectious Diseases, National Reference Centre for Rare Paediatric Inflammatory Rheumatisms and Systemic Autoimmune diseases RAISE, Robert Debré University Hospital, Assistance Publique-Hôpitaux de Paris, Paris, FranceUniversity of Milan, Milan, ItalyDepartment of General Paediatrics, Paediatric Internal Medicine, Rheumatology and Infectious Diseases, National Reference Centre for Rare Paediatric Inflammatory Rheumatisms and Systemic Autoimmune diseases RAISE, Robert Debré University Hospital, Assistance Publique-Hôpitaux de Paris, Paris, FranceDepartment of Clinical Sciences and Community Health, Research Center for Adult and Pediatric Rheumatic Diseases, University of Milan, Milan, ItalyDepartment of General Paediatrics, Paediatric Internal Medicine, Rheumatology and Infectious Diseases, National Reference Centre for Rare Paediatric Inflammatory Rheumatisms and Systemic Autoimmune diseases RAISE, Robert Debré University Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France; ACTIV, Association Clinique et Thérapeutique Infantile du Val-de-Marne, Créteil, France; Université de Paris, UFR de Médecine, Paris, France; Université de Paris, INSERM, UMR1149, Paris, France; Biology and Genetics of Bacterial Cell Wall Unit, Pasteur Institute, University de Paris, Paris, France; Corresponding author at: Department of General Pediatrics, Pediatric Internal Medicine, Rheumatology and Infectious Diseases, Reference Centre for Rare Pediatric Inflammatory Rheumatisms and Systemic Autoimmune diseases RAISE, Robert Debré University Hospital, Assistance Publique-Hôpitaux de Paris, F-75019 Paris, France.Summary: Background: Early identification of high-risk patients is essential to stratify treatment algorithms of Kawasaki disease (KD) and to appropriately select patients at risk for complicated disease who would benefit from intensified first-line treatment. Several scores have been developed and validated in Asian populations but have shown low sensitivity in predicting intravenous immunoglobulin (IVIG) resistance in non-Asian populations. We sought methods to predict the need for secondary treatment after initial IVIG in non-Asian populations. Methods: We conducted a retrospective, multicenter study including consecutive patients with KD admitted to two tertiary pediatric hospitals in France and Italy from 2005 to 2019. We evaluated the performance of the Kawanet-score and compared it with the performances of initial echocardiography findings, and of a newly proposed score combining the Kawanet-score and initial echocardiography findings. For each score, we assessed the AUC, sensitivity and specificity for predicting the need for second-line treatment. Findings: We included 363 children with KD, 186 from France and 177 from Italy, of whom 57 (16%) required second-line therapy after the first IVIG dose. The Kawanet score, coronary artery dilation or aneurysm with maximal Z-score ≥2.0 at baseline, and abnormal initial echocardiography had a sensitivity of 43%, 55% and 65% and a specificity of 73%, 78%, 73%, respectively, for predicting the need for second-line treatment. The Kawanet-score was significantly improved by combining it with initial echocardiography findings. The best predictive performance (Sensitivity 76%, Specificity 54%) was obtained by combining the Kawanet-score with abnormal initial echocardiography, defined by the presence of either coronary artery maximal Z-score ≥2.0, pericarditis, myocarditis and/or ventricular dysfunction. This score predicted the need for second-line treatment in European, African/Afro-Caribbean and Asian ethnicity with a sensitivity of 80%, 65% and 100%, respectively, and a specificity of 56%, 51% and 61%, respectively. Interpretation: Our study proposes a score that we named the Kawanet-echo score, which allows early identification of children with KD who require a second-line treatment in multi-ethnic populations in Europe. Funding: None.http://www.sciencedirect.com/science/article/pii/S2666776222001776Kawasaki diseaseVasculitisPediatric rheumatologyCoronary arteryEchocardiographySeverity score |
| spellingShingle | Naim Ouldali Rosa Maria Dellepiane Sofia Torreggiani Lucia Mauri Gladys Beaujour Constance Beyler Martina Cucchetti Cécile Dumaine Adriano La Vecchia Isabelle Melki Rita Stracquadaino Caroline Vinit Rolando Cimaz Ulrich Meinzer Development of a score for early identification of children with Kawasaki disease requiring second-line treatment in multi-ethnic populations in Europe: A multicentre retrospective cohort study The Lancet Regional Health. Europe Kawasaki disease Vasculitis Pediatric rheumatology Coronary artery Echocardiography Severity score |
| title | Development of a score for early identification of children with Kawasaki disease requiring second-line treatment in multi-ethnic populations in Europe: A multicentre retrospective cohort study |
| title_full | Development of a score for early identification of children with Kawasaki disease requiring second-line treatment in multi-ethnic populations in Europe: A multicentre retrospective cohort study |
| title_fullStr | Development of a score for early identification of children with Kawasaki disease requiring second-line treatment in multi-ethnic populations in Europe: A multicentre retrospective cohort study |
| title_full_unstemmed | Development of a score for early identification of children with Kawasaki disease requiring second-line treatment in multi-ethnic populations in Europe: A multicentre retrospective cohort study |
| title_short | Development of a score for early identification of children with Kawasaki disease requiring second-line treatment in multi-ethnic populations in Europe: A multicentre retrospective cohort study |
| title_sort | development of a score for early identification of children with kawasaki disease requiring second line treatment in multi ethnic populations in europe a multicentre retrospective cohort study |
| topic | Kawasaki disease Vasculitis Pediatric rheumatology Coronary artery Echocardiography Severity score |
| url | http://www.sciencedirect.com/science/article/pii/S2666776222001776 |
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