@article{oai:hirosaki.repo.nii.ac.jp:00005783, author = {藤田, 円 and 湯澤, 健太郎 and 三浦, 文武 and 相澤, 知美 and 嶋田, 淳 and 北川, 陽介 and 渡邊, 祥二郎 and 津川, 浩二 and 敦賀, 和志 and 丹代, 諭 and 大谷, 勝記 and 田中, 完 and 高橋, 徹 and 伊藤, 悦朗}, issue = {1}, journal = {弘前医学}, month = {Nov}, note = {小児期発症の高安動脈炎(TA)に対し IL-6 受容体抗体であるtocilizumab (TCZ)を使用した報告は少なく,加えてTA 症例に対して冠動脈バイパス術(CABG)を施行した症例も稀である.症例は11歳女子.反復する胸痛,心臓超音波検査で大動脈弁閉鎖不全,心電図で ST 低下,血液検査でCRP 上昇と赤沈亢進,トロポニンT陽性,CT で左冠動脈開口部の狭窄を認めTA と診断した.ステロイド薬,各種免疫抑制剤薬,抗TNFα 抗体のインフリキシマブを併用しても寛解が得られなかった.倫理委員会承認後にTCZ の点滴投与を行い,速やかに症状と検査所見の改善を認め,ステロイド薬を減量・中止した.TCZ 使用に伴う有害事象は認められなかった.炎症が鎮静化後,左冠動脈開口部 99%狭窄に対してCABG が施行され術後の経過は良好である.本邦でも成人領域において難治性のTA に対するTCZ 皮下注射製剤が適応となった.今後,小児例においても適応の拡大が期待される. Although Takayasu arteritis (TA) is rarely seen in children, difficult cases of childhood-onset TA have sometimes been reported. Recently, clinical efficacy of anti-proinflammatory cytokines treatment, including antiinterleukin- 6 receptor antibody, tocilizumab( TCZ) has been reported in adult patients with refractory TA. However, only a few papers describing successful TCZ treatment in childhood onset TA has been reported to date. A 11-yearold girl complained of recurrent chest pain visited to a regional hospital. An echocardiography revealed aortic regurgitation and an electro-cardiogram revealed ST segment depression. Blood examinations revealed elevated serum C-reactive protein and troponin T. Various image inspection showed extended ascending aorta and stenosis of left coronary artery ostium, suggesting that she had probable TA. Initially, treatment with prednisolone (PSL), various immunosuppressants and anti-tumor necrosis factor α monoclonal antibody, infliximab was started. However, the disease activity could not be controlled. Then, after obtaining the approval of the ethic committee, an initiation of intravenous TCZ resulted in a rapid clinical remission in this patient. PSL was successfully tapered. Since her coronary angiogram showed 99% stenosis of left coronary artery ostium, coronary artery bypass grafting was successfully conducted thereafter. In our patient, treatment with TCZ was effective and safe including perioperative period. In the case of TA with high risk of organ damage, we believe that early initiation of TCZ would be necessary even in pediatric cases.}, pages = {56--61}, title = {トシリズマブが奏功した難治性小児高安動脈炎の女子例}, volume = {70}, year = {2019} }