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We report on an unusual case of a year-old Caucasian male displaying severe Kawasaki-like symptoms. The disease lasted for seven years before diffuse coronary aneurysms occurred, leading to the patient's death, despite ongoing treatment by intravenous immunoglobulins IVIGs. The patient had also been suffering from a disabling inflammation of the spine, which was reported to have started at the onset of the disorder. Whereas neither NSAIDS, nor high doses corticosteroids, or anti-TNF drugs had a clear effect, the clinical features of spinal inflammation were highly sensitive to IVIGs, and were attributed definitively to HLA-Bnegative axial spondylarthropathy after bone scan and magnetic resonance imaging disclosed typical enthesitis of both heels and bilateral sacroiliitis.
Kawasaki disease is a rare multi-systemic form of vasculitis, affecting mostly young children. It rarely occurs in adults. Indeed, less than a hundred cases have been reported [1] , [2]. Moreover, Kawasaki disease usually evolves as an acute disorder, and has not been associated thus far with clinical or imaging features suggestive of spondylarthropathy. The disease lasted for seven years before diffuse coronary aneurysms occurred, leading to the patient's death, despite ongoing treatment by intravenous IVIGs.
The clinical features of spinal inflammation were highly sensitive to IVIGs, and were attributed definitively to HLA-Bnegative axial spondylarthropathy after bone scan and magnetic resonance imaging disclosed typical enthesitis of both heels and bilateral sacroiliitis.
The patient was a farmer with no previous medical history. He began to suffer from painful migrating arthralgia in August ; this was associated with tarsal pain, heel enthesitis, and inflammatory back pain pain of the lower back, occurring mainly during the second part of the night, and associated with a long lasting morning stiffness, partly relieved by daily exercise. The disorder followed a remitting-relapsing course. The patient was treated with sulfasalazine for four months, despite the absence of the HLA-B27 gene; however, this treatment was ineffective.
