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Infliximab in IVIG-resistant Kawasaki disease with cholestatic hepatitis: case reportCan a new drug stop heart damage in Kawasaki disease when standard treatments fail?

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Key Takeaway
Consider dissociation between systemic and coronary effects in infliximab use for IVIG-resistant KD.

This case report involves a 2-year-old Han Chinese male with IVIG-resistant Kawasaki disease and cholestatic hepatitis. The patient received infliximab (5 mg/kg) after failing initial high-dose IVIG (2 g/kg) combined with clopidogrel and a second dose of IVIG (1 g/kg) combined with high-dose methylprednisolone pulse therapy. The primary outcome was prevention of coronary artery aneurysm (CAA) progression, with secondary outcomes including fever subsidence, inflammatory indicators (CRP, ESR), and liver function indicators (ALT, AST, bilirubin, total bile acids).

Main results showed fever subsided rapidly within 24 h, and inflammatory and liver function indicators improved significantly. However, coronary artery aneurysms progressed, eventually forming giant CAAs. Echocardiographic follow-up was conducted, but specific durations or numerical improvements in markers were not reported. Safety and tolerability data were not provided.

Key limitations include dissociation between systemic inflammation control and coronary protection, and the critical importance of optimizing the timing of infliximab administration. Practice relevance suggests further randomized controlled trials are needed to clarify optimal timing, dose, and patient selection criteria for infliximab in IVIG-resistant Kawasaki disease. This case underscores the need for careful monitoring in such complex presentations.

Imagine a two-year-old boy fighting a fierce battle against Kawasaki disease. His body was on fire with fever, and his liver was struggling. Doctors tried the standard heavy-hitting treatments, including high doses of IVIG and steroids, but the inflammation kept raging. The heart, specifically the coronary arteries, was in danger of developing aneurysms, which are dangerous bulges that can lead to heart attacks later in life. In this specific case, the doctors added a biologic drug called infliximab to the mix. Within just 24 hours, the fever vanished, and the markers of inflammation in his blood improved significantly. His liver function also bounced back to normal levels quickly.

However, there was a heartbreaking disconnect in the results. Even though the drug successfully calmed the body's inflammatory storm, the damage to the heart continued. The coronary artery aneurysms did not stop growing; instead, they progressed until they became giant aneurysms. This case highlights a critical and confusing reality: controlling the fever and inflammation does not automatically mean the heart is safe. The boy received infliximab at a specific dose, but the outcome was not the protection doctors had hoped for in this instance.

This story is a warning and a call to action. It shows that we cannot assume a treatment works for everyone or in every situation. The timing of giving this powerful drug might be the key factor, but we do not know it yet. Because this is just one person, we cannot say this drug is a miracle cure or a failure. We simply know that more research is needed to figure out the right timing, dose, and patient selection criteria before we can recommend this for everyone.

What this means for you:
In one case, infliximab lowered fever but did not stop heart artery growth in Kawasaki disease.

Study Details

Study typeRct
EvidenceLevel 2
PublishedApr 2026
View Original Abstract ↓
BackgroundKawasaki disease (KD) is the leading cause of acquired heart disease in children under 5 years of age worldwide. Approximately 15%–20% of KD patients are resistant to intravenous immunoglobulin (IVIG), and these patients have a significantly higher risk of developing coronary artery aneurysms (CAAs), a severe complication leading to long-term cardiovascular morbidity. Cholestatic hepatitis is a rare manifestation of KD, which further increases the difficulty of clinical treatment. This study reports a case of IVIG-resistant KD complicated with cholestatic hepatitis treated with infliximab, and explores the clinical challenges in preventing CAA progression.Case reportA 2-year-old Han Chinese male presented with persistent fever as the initial symptom, followed by typical clinical manifestations of KD and cholestatic hepatitis. Initial treatment with high-dose IVIG (2 g/kg) combined with clopidogrel (1 mg/kg/day) was ineffective. A second dose of IVIG (1 g/kg) combined with high-dose methylprednisolone pulse therapy also failed to control the disease. On the ninth day of illness, salvage therapy with the tumor necrosis factor-alpha (TNF-α) inhibitor infliximab (5 mg/kg) was administered, and the child's fever subsided rapidly within 24 h. Subsequent laboratory examinations showed that inflammatory indicators [C-reactive protein (CRP), erythrocyte sedimentation rate (ESR)] and liver function indicators [alanine aminotransferase (ALT), aspartate aminotransferase (AST), bilirubin, total bile acids (TBA)] improved significantly. However, despite effective inflammation control, echocardiographic follow-up revealed progressive development of CAAs, eventually forming giant CAAs.ConclusionInfliximab can effectively suppress systemic inflammation and improve liver function in patients with IVIG-resistant KD complicated with cholestatic hepatitis. However, the development of giant CAAs in this case underscores the dissociation between systemic inflammation control and coronary protection, highlighting the critical importance of optimizing the timing of infliximab administration. Further randomized controlled trials are needed to clarify the optimal timing, dose, and patient selection criteria for infliximab in the treatment of IVIG-resistant KD.
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