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.
Bingqian Xue, Mengjun Luo, Yang Wen, Yanfeng Yang, Hua Lai, Yizhou Wen, Qin Zhou
Frontiers in immunology · 2026-01
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.
MeSH terms
- Humans
- Mucocutaneous Lymph Node Syndrome
- Male
- Coronary Aneurysm
- Child, Preschool
- Latent Tuberculosis
- Immunoglobulins, Intravenous
- Blood Coagulation
- Treatment Outcome