The case of Kawasaki disease after rituximab infusion triggered by human anti-chimeric antibodies

CEN Case Rep. 2020 Nov;9(4):392-394. doi: 10.1007/s13730-020-00492-8. Epub 2020 Jun 13.

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: Human anti-chimeric antibodies; Kawasaki disease; Refractory nephrotic syndrome; Rituximab; Serum sickness.

Publication types

  • Case Reports

MeSH terms

  • Antirheumatic Agents / administration & dosage
  • Antirheumatic Agents / adverse effects
  • Antirheumatic Agents / therapeutic use
  • Autoantibodies / blood*
  • Child
  • Coronary Vessel Anomalies / diagnosis
  • Coronary Vessel Anomalies / etiology
  • Diagnosis, Differential
  • Exanthema / diagnosis
  • Female
  • Fever / diagnosis
  • Humans
  • Immunoglobulins, Intravenous / administration & dosage
  • Immunoglobulins, Intravenous / therapeutic use
  • Injection Site Reaction / complications
  • Mucocutaneous Lymph Node Syndrome / diagnosis*
  • Mucocutaneous Lymph Node Syndrome / drug therapy
  • Mucocutaneous Lymph Node Syndrome / immunology
  • Nephrotic Syndrome / diagnosis*
  • Nephrotic Syndrome / drug therapy
  • Nephrotic Syndrome / immunology
  • Rituximab / administration & dosage
  • Rituximab / adverse effects*
  • Rituximab / therapeutic use
  • Serum Sickness / chemically induced*
  • Serum Sickness / pathology
  • Treatment Outcome

Substances

  • Antirheumatic Agents
  • Autoantibodies
  • Immunoglobulins, Intravenous
  • Rituximab