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March 14, 2026Frontiers in Immunology0 citationsOpen Access

Multidisciplinary management of refractory Kawasaki disease with bilateral giant coronary artery aneurysms and latent tuberculosis infection: a case report focusing on immune–coagulation–infection crosstalk

BXBingqian XueMLMengjun LuoYWYang Jing Wen

Key Result

The multidisciplinary management strategy led to normalization of luminal diameters of bilateral giant coronary artery aneurysms after 9 months of treatment.

Key Points

  • The aim is to outline a successful multidisciplinary management strategy for refractory Kawasaki disease complicated by latent tuberculosis infection.
  • Case report of a 3.5-year-old boy with refractory Kawasaki disease and bilateral giant coronary artery aneurysms.
  • Used systemic inflammation control via methylprednisolone and IVIG while ensuring safety for latent tuberculosis.
  • Executed a precision anticoagulation strategy to manage warfarin resistance due to rifampicin.
  • Successful control of systemic inflammation and resolution of giant coronary artery aneurysms at 9-month follow-up.
  • Stable therapeutic INR achieved with significant warfarin dose escalation.
  • Established a new framework for managing complex cases involving immune-coagulation-infection interactions.

Study Design

Type

Case Report (n=1)

Multicenter

No

Structured PICO

Does a multidisciplinary management strategy including alternative immunomodulation and precision anticoagulation improve outcomes in a pediatric patient with refractory Kawasaki disease, giant coronary artery aneurysms, and latent tuberculosis infection?

P
Population
3.5-year-old boy (n=1) with refractory Kawasaki disease, rapid-onset bilateral giant coronary artery aneurysms (CAAs), and latent tuberculosis infection (LTBI).
I
Intervention
Multidisciplinary management including sequential immunomodulation (methylprednisolone and a second IVIG dose), LTBI prophylaxis (isoniazid/rifampicin), and precision anticoagulation (pharmacokinetic-guided warfarin titration with a prolonged low-molecular-weight heparin bridge).
O
Outcome
Regression of coronary artery aneurysms and achievement of stable therapeutic INR without adverse events at 9-month follow-upsurrogate

A multidisciplinary approach integrating alternative immunomodulation, strategic prophylaxis, and precision anticoagulation safely and effectively managed a pediatric patient with refractory Kawasaki disease, giant CAAs, and LTBI.

Limitations

  • Single-center report limits generalizability of the protocol.
  • Lack of direct pharmacokinetic quantification of rifampicin-warfarin interaction.
  • Single-center report requiring multi-center validation
  • Rifampicin-warfarin interaction inferred pharmacodynamically without direct pharmacokinetic quantification
  • Lacked individualized, direct immune profiling (e.g., flow cytometry or scRNA-seq)

Abstract

Background The management of refractory Kawasaki disease (KD) with giant coronary artery aneurysms (CAAs) complicated by latent tuberculosis infection (LTBI) represents a formidable “triple challenge”: it requires balancing aggressive inflammation control against the risk of tuberculosis reactivation, managing the extreme cardiovascular risk of giant CAAs driven by immune-coagulation crosstalk, and navigating the pharmacological hurdles of LTBI prophylaxis, namely rifampicin-induced warfarin resistance. Developing a safe, integrated strategy to concurrently address these inflammatory, cardiovascular, and infectious risks remains an urgent and unmet clinical need. Case presentation We report a 3.5-year-old boy with refractory KD and rapid-onset bilateral giant CAAs. Pre-biologic screening identified LTBI, strictly contraindicating guideline-recommended tumor necrosis factor-alpha (TNF-α) inhibitors. A multidisciplinary team (MDT) executed a sequential, pathophysiology-driven protocol: 1) Immunomodulation Prophylaxis: Systemic inflammation was controlled using methylprednisolone and a second IVIG dose, circumventing TNF-α blockade to ensure LTBI safety. Upon vasculitis remission and hepatic recovery, LTBI prophylaxis (isoniazid/rifampicin) was initiated. 2) Precision Anticoagulation: To overcome rifampicin-accelerated warfarin metabolism, we executed a rapid, percentage-based titration alongside a “prolonged low-molecular-weight heparin (LMWH) bridge”. A stable, conservative therapeutic INR (1.5–2.5) was achieved, requiring an exact 2.5-fold warfarin dose escalation. At the 9-month follow-up, the CAAs demonstrated luminal normalization, though necessitating lifelong surveillance for vascular pseudonormalization. Conclusion This case illustrates how an MDT-led, pharmacology-guided approach effectively resolves therapeutic conflicts in high-risk KD with biologic contraindications. By integrating alternative immunomodulation, strategic prophylaxis, and precision anticoagulation, this framework provides a practical clinical paradigm for managing intricate immune-coagulation-infection crosstalk.

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Cite This Study

Xue et al. (2026) conducted a case report in Refractory Kawasaki disease with giant coronary artery aneurysms and latent tuberculosis infection (n=1). Methylprednisolone and intravenous immunoglobulin (IVIG) followed by isoniazid/rifampicin prophylaxis and warfarin was evaluated on Normalization of luminal diameters of bilateral giant coronary artery aneurysms. The multidisciplinary management strategy led to normalization of luminal diameters of bilateral giant coronary artery aneurysms after 9 months of treatment.

synapsesocial.com/papers/69b4fb8db39f7826a300bc03https://doi.org/10.3389/fimmu.2026.1796288
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