Infliximab treatment of intravenous immunoglobulin-resistant Kawasaki disease in 2 children and literature review

Abstract

Objective To investigate the clinical effect and the prospect of Infliximab in treatment of intravenous immunoglobulin(IVIG)-resistant Kawasaki disease(KD) patients. Methods Clinical features, inflammatory markers and coronary changes were observed in 2 cases of IVIG-resistant KD patients hospitalized in Peking University First Hospital, who were treated effectively by Infliximab.Relevant researches on the mechanism and progress of the Infliximab treatment for IVIG-resistant KD in the last 10 years were reviewed at the same time. Results Two KD patients hospitalized in Peking University First Hospital had been treated with 2 g/kg IVIG for 2 times and followed by methylprednisolone treatment.However, fever and other clinical manifestations occurred again after 2 days and 6 days when temperature returned normal.They both defervesced and all the symptoms were improved after 1 dose of Infliximab(5 mg/kg) by laboratory examinations.Four published literatures of the basic research and 9 retrospective or prospective clinical researches of Infliximab treatment of KD showed that Infliximab alleviated the inflammatory level in the KD patients significantly.Complete remission was up to 72.73%-92.11%.Those KD patients defervesced within 12 h, with dramatic improvement of symptoms and signs.Arthralgia also disappeared in the patients with arthritis.Only 1 case was complicated with hepatitis in the acute phase and cholecystitis in recovery time.A phase 3 randomised, double-blinded, placebo-controlled trial had been done to assess the addition of Infliximab to the standard therapy. Conclusions Infliximab is a feasible choice for IVIG-resistant KD patients.Efficacy and safety of Infliximab for KD treatment have been proved in the literature.However, Infliximab for KD treatment has not been indicated in the drug instruction, so the informed consent from the guardians and Ethics Committee is needed. Key words: Kawasaki disease; Intravenous immunoglobulin-resistance; Treatment; Infliximab; Tumor necrosis factor-α