A Case Diagnosed with Takayasu Arteritis during the Course of Treatment with Adalimumab for Chronic Recurrent Multifocal Osteomyelitis

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  • TAKEUCHI Shio
    Department of Pediatrics, Iida Municipal Hospital Department of Pediatrics, Shinshu University School of Medicine
  • MARUYAMA Yuta
    Department of Pediatrics, Shinshu University School of Medicine
  • Nakazawa Yozo
    Department of Pediatrics, Shinshu University School of Medicine

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Other Title
  • 先行した慢性再発性多発性骨髄炎に対するアダリムマブ治療経過中に高安動脈炎の診断に至った１例
  • センコウ シタ マンセイ サイハツセイ タハツセイ コツズイエン ニ タイスル アダリムマブ チリョウ ケイカ チュウ ニ コウアンドウミャクエン ノ シンダン ニ イタッタ 1レイ

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A 15-year-old female was referred to our hospital because of fever and fatigue for 1 month. C-reactive protein elevation was observed (8.2mg/dL), and imaging studies including 18F-fluoro-D-glucose-positron emission tomography/computed tomography (FDG-PET/CT) showed no abnormal findings. Prednisolone (PSL) was started, and her fever went down subsequently. When PSL was tapered, fever and headache recurred ; so tocilizumab (TCZ) was initiated as a treatment for systemic juvenile idiopathic arthritis, and fever was resolved. Six months later she experienced pain around both knees. Multifocal abnormal bone marrow signals were observed through magnetic resonance imaging. Lesion bone biopsy revealed nonspecific inflammation without malignancy or infection. Our diagnosis was chronic recurrent multifocal osteomyelitis (CRMO), naproxen was started, and TCZ was switched to adalimumab (ADA). However, her pain around both knees and headache persisted. A year after CRMO diagnosis, 18F-FDG-PET/CT re-examination revealed FDG accumulation in the common carotid artery, subclavian artery, aortic arch, and femoral artery to the popliteal artery. Contrast-enhanced CT showed vessel wall thickening and arterial stenosis. Accordingly, the patient was diagnosed with Takayasu arteritis. PSL was increased, and azathioprine was initiated, and ADA was switched to subcutaneous TCZ. At the 10-month follow-up, headache and leg pain had decreased, and the steroid dose was tapered.<br>CRMO is a noninfectious, autoinflammatory disease causing recurrent sterile inflammatory bone lesions. CRMO has been described in association with other inflammatory conditions such as Takayasu arteritis. Physicians should consider that in a case of CRMO with persistent inflammation after treatment, other inflammatory diseases such as Takayasu arteritis may coexist.

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