tetano
Editor, Senior Moderator
J Infect Chemother
. 2022 Jan 24;S1341-321X(22)00023-X.
doi: 10.1016/j.jiac.2022.01.011. Online ahead of print.
Infliximab treatment for refractory COVID-19-associated multisystem inflammatory syndrome in a Japanese child
Yohei Yamaguchi[SUP] 1 [/SUP], Kei Takasawa[SUP] 2 [/SUP], Hitoshi Irabu[SUP] 3 [/SUP], Kanako Hiratoko[SUP] 4 [/SUP], Yosuke Ichigi[SUP] 1 [/SUP], Ko Hirata[SUP] 1 [/SUP], Yumie Tamura[SUP] 1 [/SUP], Miki Murakoshi[SUP] 1 [/SUP], Motoi Yamashita[SUP] 1 [/SUP], Hisae Nakatani[SUP] 1 [/SUP], Masuhiro Shimoda[SUP] 4 [/SUP], Taku Ishii[SUP] 1 [/SUP], Tomohiro Udagawa[SUP] 1 [/SUP], Masaki Shimizu[SUP] 5 [/SUP], Hirokazu Kanegane[SUP] 5 [/SUP], Tomohiro Morio[SUP] 1 [/SUP]
Affiliations
Abstract
Patients with multisystem inflammatory syndrome in children (MIS-C) can develop clinical features resembling Kawasaki disease (KD). A full picture of MIS-C in East Asia which has higher incidence of KD than other regions remains unclear. We report on a 15-year-old Japanese boy with refractory MIS-C who was successfully treated with infliximab. A Japanese boy who was diagnosed with coronavirus disease 2019 (COVID-19) before a month developed MIS-C with fulfilling six principal symptoms of KD. Laboratory data showed extreme hyperferritinemia (11,404 ng/mL), besides lymphopenia and thrombocytopenia. The patient was refractory to initial therapy with intravenous immunoglobulin (IVIG; 2 g/kg), aspirin, and prednisolone. He was therefore administered a second IVIG (2 g/kg) and infliximab (5 mg/kg) on days 7 and 8 from the onset of fever, respectively, which resulted in an improvement of clinical symptoms. Only four Japanese cases with MIS-C were reported and all of them were responsive to IVIG. The hyperferritinemia in this case was distinctive from previously reported MIS-C cases in Japan and other cohorts and may be associated with refractoriness to IVIG therapy. Marked elevation of circulating ferritin levels is known to be induced by tumor necrosis factor-α, which plays a key role in the pathogenesis of both KD and MIS-C. Thus, for MIS-C patients with hyperferritinemia, early intervention with adjunctive infliximab may induce a more rapid resolution of inflammation and improve outcome. Because MIS-C may be heterogeneous with respect to immunopathology, genetic background, clinical phenotypes and response to therapies, optimized treatment strategies according to immunopathogenesis are required.
Keywords: COVID-19; Hyperinflammation; Infliximab; Multisystem inflammatory syndrome in children.
. 2022 Jan 24;S1341-321X(22)00023-X.
doi: 10.1016/j.jiac.2022.01.011. Online ahead of print.
Infliximab treatment for refractory COVID-19-associated multisystem inflammatory syndrome in a Japanese child
Yohei Yamaguchi[SUP] 1 [/SUP], Kei Takasawa[SUP] 2 [/SUP], Hitoshi Irabu[SUP] 3 [/SUP], Kanako Hiratoko[SUP] 4 [/SUP], Yosuke Ichigi[SUP] 1 [/SUP], Ko Hirata[SUP] 1 [/SUP], Yumie Tamura[SUP] 1 [/SUP], Miki Murakoshi[SUP] 1 [/SUP], Motoi Yamashita[SUP] 1 [/SUP], Hisae Nakatani[SUP] 1 [/SUP], Masuhiro Shimoda[SUP] 4 [/SUP], Taku Ishii[SUP] 1 [/SUP], Tomohiro Udagawa[SUP] 1 [/SUP], Masaki Shimizu[SUP] 5 [/SUP], Hirokazu Kanegane[SUP] 5 [/SUP], Tomohiro Morio[SUP] 1 [/SUP]
Affiliations
- PMID: 35125343
- DOI: 10.1016/j.jiac.2022.01.011
Abstract
Patients with multisystem inflammatory syndrome in children (MIS-C) can develop clinical features resembling Kawasaki disease (KD). A full picture of MIS-C in East Asia which has higher incidence of KD than other regions remains unclear. We report on a 15-year-old Japanese boy with refractory MIS-C who was successfully treated with infliximab. A Japanese boy who was diagnosed with coronavirus disease 2019 (COVID-19) before a month developed MIS-C with fulfilling six principal symptoms of KD. Laboratory data showed extreme hyperferritinemia (11,404 ng/mL), besides lymphopenia and thrombocytopenia. The patient was refractory to initial therapy with intravenous immunoglobulin (IVIG; 2 g/kg), aspirin, and prednisolone. He was therefore administered a second IVIG (2 g/kg) and infliximab (5 mg/kg) on days 7 and 8 from the onset of fever, respectively, which resulted in an improvement of clinical symptoms. Only four Japanese cases with MIS-C were reported and all of them were responsive to IVIG. The hyperferritinemia in this case was distinctive from previously reported MIS-C cases in Japan and other cohorts and may be associated with refractoriness to IVIG therapy. Marked elevation of circulating ferritin levels is known to be induced by tumor necrosis factor-α, which plays a key role in the pathogenesis of both KD and MIS-C. Thus, for MIS-C patients with hyperferritinemia, early intervention with adjunctive infliximab may induce a more rapid resolution of inflammation and improve outcome. Because MIS-C may be heterogeneous with respect to immunopathology, genetic background, clinical phenotypes and response to therapies, optimized treatment strategies according to immunopathogenesis are required.
Keywords: COVID-19; Hyperinflammation; Infliximab; Multisystem inflammatory syndrome in children.