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一例托珠单抗治疗透析相关淀粉样变性所致关节病有效的病例。

A case of effective tocilizumab for arthropathy due to dialysis-related amyloidosis.

作者信息

Tsunoda Susumu, Yoshimura Yusuke, Oba Yuki, Ikuma Daisuke, Mizuno Hiroki, Yamanouchi Masayuki, Suwabe Tatsuya, Kitajima Izuru, Kono Kei, Ohashi Kenichi, Ubara Yoshifumi, Sawa Naoki

机构信息

Department of Nephrology, Nephrology Center, Toranomon Hospital, 2-2-2, Toranomon, Minato-ku, Tokyo, 105-8470, Japan.

Department of Orthopaedic Surgery, Toranomon Hospital, Minato-ku, Tokyo, 105-8470, Japan.

出版信息

CEN Case Rep. 2025 Jun 26. doi: 10.1007/s13730-025-01009-x.

Abstract

A 79-year-old male patient on hemodialysis for 29 years was admitted to the hospital with unexplained fever (C-reactive protein, 16.1 mg/dL), anemia (hemoglobin 7.6 g/dL), and multiple joint swelling that began 7 months earlier. Infection was ruled out, both rheumatoid factor and anti-cyclic citrullinated peptide antibodies were negative. Positron emission tomography-computed tomography showed positive findings in bilateral shoulder joints, palmar region, femoral head area, and cervical spine. Magnetic resonance imaging showed low intensity on T1 and low-normal intensity on T2, especially in the shoulder joint. Musculoskeletal ultrasound revealed hypoechoic material with surrounding fluid collection and Doppler signals indicating blood flow in the shoulder joint, resembling synovitis associated with rheumatoid arthritis. Biopsy of the shoulder joint mass showed positive Congo-red and direct fast scarlet staining, positive β2-microglobulin, and infiltration of CD68-positive macrophages. Given the history of previous carpal tunnel release and cervical destructive spondyloarthropathy, dialysis-related amyloidosis, and amyloid-related arthritis were diagnosed. However, as the case had the feature of rheumatoid arthritis-like synovitis, tocilizumab was initiated, resulting in improved anemia and Clinical Disease Activity Index. We speculate that dialysis-related amyloidosis can present with arthritis involving cytokines, similar to rheumatoid arthritis, and tocilizumab may be effective for the resulting synovitis.

摘要

一名接受血液透析29年的79岁男性患者因不明原因发热(C反应蛋白,16.1mg/dL)、贫血(血红蛋白7.6g/dL)以及7个月前开始出现的多个关节肿胀入院。排除感染,类风湿因子和抗环瓜氨酸肽抗体均为阴性。正电子发射断层扫描-计算机断层扫描显示双侧肩关节、手掌区域、股骨头区域和颈椎有阳性发现。磁共振成像显示T1加权像呈低信号,T2加权像呈低-正常信号,尤其是在肩关节。肌肉骨骼超声显示低回声物质伴周围液体积聚,多普勒信号提示肩关节有血流,类似类风湿关节炎相关的滑膜炎。肩关节肿块活检刚果红和直接耐晒猩红染色阳性,β2微球蛋白阳性,CD68阳性巨噬细胞浸润。鉴于既往有腕管松解术和颈椎破坏性脊椎关节病病史,诊断为透析相关淀粉样变性和淀粉样变性相关关节炎。然而,由于该病例具有类风湿关节炎样滑膜炎的特征,因此开始使用托珠单抗治疗,贫血和临床疾病活动指数得到改善。我们推测,透析相关淀粉样变性可表现为涉及细胞因子的关节炎,类似于类风湿关节炎,托珠单抗可能对由此产生的滑膜炎有效。

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