Ouldali Naim, Dellepiane Rosa Maria, Torreggiani Sofia, Mauri Lucia, Beaujour Gladys, Beyler Constance, Cucchetti Martina, Dumaine Cécile, La Vecchia Adriano, Melki Isabelle, Stracquadaino Rita, Vinit Caroline, Cimaz Rolando, Meinzer Ulrich
Department 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.
Lancet Reg Health Eur. 2022 Aug 6;22:100481. doi: 10.1016/j.lanepe.2022.100481. eCollection 2022 Nov.
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.
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.
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.
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.
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早期识别高危患者对于分层川崎病（KD）的治疗方案以及恰当选择可能从强化一线治疗中获益的复杂疾病高危患者至关重要。亚洲人群已开发并验证了多种评分系统，但这些评分系统在预测非亚洲人群静脉注射免疫球蛋白（IVIG）抵抗方面敏感性较低。我们试图寻找预测非亚洲人群初始IVIG治疗后是否需要二线治疗的方法。
我们开展了一项回顾性多中心研究，纳入了2005年至2019年期间在法国和意大利两家三级儿科医院连续收治的KD患者。我们评估了川网评分（Kawanet-score）的性能，并将其与初始超声心动图检查结果以及新提出的将川网评分与初始超声心动图检查结果相结合的评分的性能进行比较。对于每个评分，我们评估了预测二线治疗需求的曲线下面积（AUC）、敏感性和特异性。
我们纳入了363例KD患儿，其中186例来自法国，177例来自意大利，其中57例（16%）在首次IVIG剂量后需要二线治疗。川网评分、基线时最大Z值≥2.0的冠状动脉扩张或动脉瘤以及初始超声心动图异常预测二线治疗需求的敏感性分别为43%、55%和65%，特异性分别为73%、78%、73%。将川网评分与初始超声心动图检查结果相结合可显著提高其性能。将川网评分与初始超声心动图异常（定义为冠状动脉最大Z值≥2.0、心包炎、心肌炎和/或心室功能障碍中的任何一种）相结合可获得最佳预测性能（敏感性76%，特异性54%）。该评分预测欧洲、非洲/非裔加勒比和亚洲种族二线治疗需求的敏感性分别为80%、65%和100%，特异性分别为56%、51%和61%。
我们的研究提出了一个我们命名为川网-超声评分（Kawanet-echo score）的评分系统，该系统能够早期识别欧洲多民族人群中需要二线治疗的KD患儿。
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