Journal of Pediatric Cardiology and Cardiac Surgery

Online ISSN: 2433-1783 Print ISSN: 2433-2720
Japanese Society of Pediatric Cardiology and Cardiac Surgery
Japanese Society of Pediatric Cardiology and Cardiac Surgery Academy Center, 358-5 Yamabuki-cho, Shinju-ku, Tokyo 162-0801, Japan
Journal of Pediatric Cardiology and Cardiac Surgery 5(1): 20-23 (2021)

Case ReportCase Report

Successful Treatment with Infliximab in a Child with Kawasaki Disease Refractory to Additional Combination Therapy with Intravenous Immunoglobulin and Oral Ciclosporin

Department of Pediatric Cardiology, National Cerebral and Cardiovascular Center ◇ Osaka, Japan

受付日:2020年3月29日Received: March 29, 2020
受理日:2020年9月16日Accepted: September 16, 2020
発行日:2021年3月1日Published: March 1, 2021

It remains unclear how to treat patients with Kawasaki disease (KD) refractory to conventional intravenous gammaglobulin (IVIg) therapy. We report successful early aggressive combination treatment with ciclosporin and infliximab in an IVIg-resistant case. A 2-year 6-month-old boy with 5 principle symptoms was transferred to our hospital on day 3 of illness. Body temperature was 40.8°C, and appeared ill. Laboratory testing on admission showed: aspartate aminotransferase (AST), 2,439 IU/L; alanine aminotransferase (ALT), 1,142 IU/L; total bilirubin, 2.1 mg/dL; and C-reactive protein, 11.8 mg/dL. White blood cell count was 8.8×109/L (85% neutrophils). Kobayashi, Egami, and Sano scores, each of which predict resistance to IVIg therapy, were 8, 5, and 3, respectively. Aspirin was not used as an anti-inflammatory agent, because of the elevated AST and ALT levels. IVIg and oral ciclosporin were administered on days 3 and 6, respectively, but fever persisted. After infliximab was administered on day 9, fever was immediately alleviated. He had no coronary aneurysms. This case suggested that third-line therapy should be performed by day 10 of illness to prevent coronary aneurysms. As additional treatment for IVIg-resistant KD, ciclosporin, infliximab and plasmapheresis should be considered, in that order. Further, aspirin may not be needed with aggressive therapies such as ciclosporin.

Key words: Kawasaki disease; ciclosporin; infliximab; intravenous immunoglobulin resistance; liver dysfunction

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