Tocilizumab for uncontrollable systemic inflammatory response syndrome complicating adult-onset Still disease: Case report and review of literature.
Tocilizumab for uncontrollable systemic inflammatory response syndrome complicating adult-onset Still disease: Case report and review of literature.
复制标题
毒珠单抗无法控制的全身性炎症反应综合征使成人发作疾病复杂化：病例报告和文献综述。
DOI：
10.1097/md.0000000000007596
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发表时间：
2017-07
期刊：
影响因子：
1.6
通讯作者：
Ito M
中科院分区：
文献类型：
--
作者：
Masui-Ito A;Okamoto R;Ikejiri K;Fujimoto M;Tanimura M;Nakamori S;Murata T;Ishikawa E;Yamada N;Imai H;Ito M
成人起病斯蒂尔病(AOSD)是一种罕见的原因不明的全身性炎症性疾病，其特征是消退的橙红色皮疹、发热刺痛、关节痛和淋巴结病。AOSD通常预后良好，但有时可能是致命的，特别是当它合并全身炎症反应综合征(SIRS)和多器官衰竭时。一位先前健康的26岁女性因持续高烧和轻度全身性浮肿被转诊至本院。五天后，患者出现呼吸困难、低血压和无尿。Anasarca发展为大量胸腔积液、腹水和全身性水肿，导致体重增加47 kg。患者在排除感染、恶性肿瘤、血液系统疾病和其他自身免疫性疾病后被诊断为AOSD。我们在环孢素、强的松龙、血浆置换和持续血液透析滤过的基础上，静脉注射IL-6受体抑制剂托奇珠单抗。患者的全身情况有所改善。在所有药物稳定下来后，患者得到控制，并单独使用tocilizumab进行治疗。据我们所知，这是第一例严重SIRS合并AOSD，成功地用抗IL-6受体抗体治疗。对于类固醇耐药的AOSD和水肿症患者，不应忽视SIRS。IL-6受体抑制剂可安全有效地控制AOSD合并严重SIRS。
Adult-onset Still disease (AOSD) is a rare systemic inflammatory disease of unknown etiology characterized by evanescent salmon-pink rash, fever spikes, arthralgia, and lymphadenopathy. AOSD usually has a good prognosis, but it can sometimes be fatal, especially when it is complicated by systemic inflammatory response syndrome (SIRS) and multiple organ failure. A previously healthy 26-year-old woman was referred to our hospital for persistent high fever and mild systemic edema. Five days later, the patient presented with dyspnea, hypotension, and anuria. Anasarca developed with massive pleural effusion, ascites, and systemic edema, resulting in an increase of 47 kg in body weight. The patient was diagnosed as AOSD after infection, malignancy, hematologic disorders, and other autoimmune diseases were excluded. We administered tocilizumab, an IL-6 receptor inhibitor, intravenously in addition to cyclosporine, prednisolone, plasma exchange, and continuous hemodiafiltration. The patient's systemic condition improved. After stabilization by all medications, the patient was managed and responded to tocilizumab alone. To the best of our knowledge, this was the first case of severe SIRS complicating AOSD that was successfully treated with an anti- IL-6 receptor antibody. SIRS should not be overlooked in a patient with steroid-resistant AOSD and edema. Inhibitors of the IL-6 receptor can be used safely and effectively to control AOSD complicated with severe SIRS.
影响因子：
1
作者：
Assimakopoulos SF;Karamouzos V;Papakonstantinou C;Zolota V;Labropoulou-Karatza C;Gogos C
通讯作者：
Gogos C
影响因子：
--
作者：
Mora Alfonso SA;Rodríguez DM;Londoño JD;Valle-Oñate R;Quintana G
通讯作者：
Quintana G