The case of Kawasaki disease after rituximab infusion triggered by human anti-chimeric antibodies
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CASE REPORT
The case of Kawasaki disease after rituximab infusion triggered by human anti‑chimeric antibodies Masayuki Sato1 · Masayoshi Yamada1 · Mika Nakajima1 · Yudai Miyama1 · Hirotsugu Kitayama1 Received: 10 March 2020 / Accepted: 4 June 2020 © Japanese Society of Nephrology 2020
Abstract Rituximab (RTX) is an effective treatment for refractory nephrotic syndrome (NS), but may produce human anti-chimeric antibodies (HACA) which can cause severe infusion reaction or rituximab-induced serum sickness (RISS). RISS presents with a fever, rash, and arthralgia, which typically occurs 7–21 days after RTX infusion. On the other hand, Kawasaki disease (KD) also presents with fever and rash. There have been no reports of KD developed after RTX infusion. A 6-year-old girl with frequently relapsing NS was admitted to our hospital for fever and rash on day 7 after receiving RTX. Although it was suggestive of RISS at first, she also had conjunctival hyperemia, swelling, and erythema of the hands and feet, and a right coronary artery abnormality on echocardiography. Her symptoms met the diagnostic criteria of KD. We administered intravenous immunoglobulin (IVIg) (2 g/kg), and her symptoms resolved within a few days. The HACA titer determined using the serum collected at admission was very high. This is the first report of KD with a clinical course similar to RISS. It should be noted that a careful follow-up of coronary arteries should be performed in patients suspected of RISS. Keywords Refractory nephrotic syndrome · Rituximab · Human anti-chimeric antibodies · Serum sickness · Kawasaki disease
Introduction Rituximab (RTX), a chimeric anti-CD20 monoclonal antibody, has been shown to be an effective treatment for refractory nephrotic syndromes (NS), such as frequently relapsing (FR) NS and steroid-dependent (SD) NS [1, 2]. However, RTX treatment may cause several adverse events. RTX may produce human anti-chimeric antibodies (HACA) against the infused RTX. HACA can cause severe infusion reaction or rituximab-induced serum sickness (RISS). RISS is a rare type-III hypersensitivity reaction. It presents with a fever, rash, and arthralgia, which typically occurs 7–21 days after RTX infusion [3–5]. A 6-year-old girl with refractory FRNS was admitted to our hospital for a high fever and skin rash on day 7 after RTX infusion. Although it was suggestive of RISS at first, her symptoms and a right coronary artery (RCA) abnormality * Masayuki Sato [email protected] 1
Department of Pediatric Nephrology, Shizuoka Children’s Hospital, Urushiyama 860, Aoi‑ku, Shizuoka 420‑8660, Japan
on echocardiography met the diagnostic criteria of Kawasaki disease (KD). We administered intravenous immunoglobulin (IVIg), then her symptoms resolved. We confirmed that her serum HACA level on admission was very high. This is the first report of KD triggered by HACA with a clinical course similar to RISS.
Case report A 6-year-old girl with refractory FRNS was admitted to our pediatric nephrology department for a high fever and skin rash on day
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