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Our report details an atypical presentation of Kawasaki disease (KD) in an infant with a late initial presentation. The patient presented with large coronary artery aneurysms and was refractory to traditional KD treatment, intravenous immunoglobulin and aspirin. The patient was treated with anakinra, an interleukin-1 receptor antagonist, which improved inflammatory markers and prevented the progression of the patient’s coronary artery aneurysms. There are no clear guidelines for refractory KD and limited reports of anakinra use in KD. Emerging treatment options for patient at high risk for refractory KD, such as anakinra, may offer significant benefits and further research is needed to create standard of care guidelines for this patient population.