Carlos D Grasa1,2,3, Elisa Fernández-Cooke4,5,6, Sara Domínguez-Rodríguez7,8, Javier Aracil-Santos1, Ana Barrios Tascon9, Judith Sánchez-Manubens10,11, Beatriz Mercader12, Jordi Antón10, Esmeralda Nuñez13, Enrique Villalobos14, Matilde Bustillo15, Marisol Camacho16, Manuel Oltra Benavent17, Gemma Giralt18, Ana Maria Bello Naranjo19, Beatriz Rocandio20, Cristina Calvo1,2,3,21. 1. Department of Pediatric Infectious Diseases, La Paz Children's Hospital (IdiPaz Foundation), Madrid, Spain. 2. IdiPaz, Institute for Health Research from La Paz Hospital, Madrid, Spain. 3. CIBERINFEC, CIBER of Infectious Diseases in Spain (Instituto de Salud Carlos III - ISCIII), Seville, Spain. 4. Pediatric Infectious Diseases Unit, Pediatric Research and Clinical Trial Unit (UPIC), Department of Pediatrics, Hospital Universitario 12 Octubre, Avda. Córdoba s/n., 28041, Madrid, Spain. elisafcooke@gmail.com. 5. imas12, Instituto de Investigación Sanitaria Hospital 12 de Octubre, Madrid, Spain. elisafcooke@gmail.com. 6. RITIP, Spanish Network for the Research in Pediatric Infectious Diseases, Madrid, Spain. elisafcooke@gmail.com. 7. Pediatric Infectious Diseases Unit, Pediatric Research and Clinical Trial Unit (UPIC), Department of Pediatrics, Hospital Universitario 12 Octubre, Avda. Córdoba s/n., 28041, Madrid, Spain. 8. imas12, Instituto de Investigación Sanitaria Hospital 12 de Octubre, Madrid, Spain. 9. Department of Pediatrics, Hospital Universitario Infanta Sofia, San Sebastian de los Reyes, Madrid, Spain. 10. Department of Pediatric Rheumatology, Hospital Sant Joan de Deu, Universitat de Barcelona, Barcelona, Spain. 11. Department of Pediatric Rheumatology, Hospital Parc Tauli, Sabadell, Spain. 12. Department of Pediatrics, Hospital Virgen de la Arrixaca, Murcia, Spain. 13. Pediatric Rheumatology Unit, Department of Pediatrics, Hospital Regional Universitario, Malaga, Spain. 14. Department of Pediatrics, Hospital Niño Jesús, Madrid, Spain. 15. Department of Pediatric Infectious Diseases, Hospital Miguel Servet, Zaragoza, Spain. 16. Pediatric Infectious Diseases, Rheumatology and Immunology Unit, Department of Pediatrics, Hospital Virgen del Rocio, Sevilla, Spain. 17. Department of Pediatrics, Hospital Universitario y Politécnico La Fe, Valencia, Spain. 18. Department of Pediatric Cardiology, Hospital Vall d'Hebron, Barcelona, Spain. 19. Department of Pediatrics, Hospital Universitario Materno-Infantil de Las Palmas de Gran Canaria, Canarias, Spain. 20. Department of Pediatrics, Hospital Universitario Donostia, San Sebastian, Spain. 21. RITIP, Spanish Network for the Research in Pediatric Infectious Diseases, Madrid, Spain.
Abstract
INTRODUCTION/ OBJECTIVES: Asian scores developed to predict unresponsiveness to intravenous immunoglobulin (IVIG) or development of coronary artery aneurysms (CAA) in patients with Kawasaki disease (KD) are not appropriate in Western populations. The purpose of this study is to develop 2 scores, to predict unresponsiveness to IVIG and development of CAA, appropriate for Spanish population. METHOD: Data of 625 Spanish children with KD collected retrospectively (2011-2016) were used to identify variables to develop the 2 scores of interest: unresponsiveness to IVIG and development of CAA. A statistical model selected best variables to create the scores, and scores were validated with data from 98 patients collected prospectively. RESULTS: From 625 patients of the retrospective cohort, final analysis was performed in 439 subjects: 37 developed CAA, and 212 were unresponsive to IVIG. For the score to predict CAA, a cutoff ≥ 8 was considered for high risk, considering a score system with a different weight for each of the eight variables. External validation showed a sensitivity of 22% and a specificity of 75%. The score to predict unresponsiveness to IVIG established a cutoff ≥ 8 for high risk, considering a score system with a different weight for each of the nine variables. External validation showed a sensitivity of 78% and a specificity of 50%. CONCLUSIONS: Two risk scores for KD were developed from Spanish population, to predict development of CAA and unresponsiveness to IVIG; validation in other cohorts could help to implement these tools in the management of KD in other Western populations.
INTRODUCTION/ OBJECTIVES: Asian scores developed to predict unresponsiveness to intravenous immunoglobulin (IVIG) or development of coronary artery aneurysms (CAA) in patients with Kawasaki disease (KD) are not appropriate in Western populations. The purpose of this study is to develop 2 scores, to predict unresponsiveness to IVIG and development of CAA, appropriate for Spanish population. METHOD: Data of 625 Spanish children with KD collected retrospectively (2011-2016) were used to identify variables to develop the 2 scores of interest: unresponsiveness to IVIG and development of CAA. A statistical model selected best variables to create the scores, and scores were validated with data from 98 patients collected prospectively. RESULTS: From 625 patients of the retrospective cohort, final analysis was performed in 439 subjects: 37 developed CAA, and 212 were unresponsive to IVIG. For the score to predict CAA, a cutoff ≥ 8 was considered for high risk, considering a score system with a different weight for each of the eight variables. External validation showed a sensitivity of 22% and a specificity of 75%. The score to predict unresponsiveness to IVIG established a cutoff ≥ 8 for high risk, considering a score system with a different weight for each of the nine variables. External validation showed a sensitivity of 78% and a specificity of 50%. CONCLUSIONS: Two risk scores for KD were developed from Spanish population, to predict development of CAA and unresponsiveness to IVIG; validation in other cohorts could help to implement these tools in the management of KD in other Western populations.
Authors: Robert M R Tulloh; Richard Mayon-White; Anthony Harnden; Athimalaipet V Ramanan; E Jane Tizard; Delane Shingadia; Colin A Michie; Richard M Lynn; Michael Levin; Orla D Franklin; Pippa Craggs; Sue Davidson; Rebecca Stirzaker; Mike Danson; Paul A Brogan Journal: Arch Dis Child Date: 2018-08-13 Impact factor: 3.791
Authors: J W Newburger; M Takahashi; J C Burns; A S Beiser; K J Chung; C E Duffy; M P Glode; W H Mason; V Reddy; S P Sanders Journal: N Engl J Med Date: 1986-08-07 Impact factor: 91.245
Authors: Lynn A Sleeper; L Luann Minich; Brian M McCrindle; Jennifer S Li; Wilbert Mason; Steven D Colan; Andrew M Atz; Beth F Printz; Annette Baker; Victoria L Vetter; Jane W Newburger Journal: J Pediatr Date: 2010-12-18 Impact factor: 4.406
Authors: Brian W McCrindle; Anne H Rowley; Jane W Newburger; Jane C Burns; Anne F Bolger; Michael Gewitz; Annette L Baker; Mary Anne Jackson; Masato Takahashi; Pinak B Shah; Tohru Kobayashi; Mei-Hwan Wu; Tsutomu T Saji; Elfriede Pahl Journal: Circulation Date: 2017-03-29 Impact factor: 29.690