Division of Rheumatology, Department of Internal Medicine, Keio University School of Medicine, Tokyo, Japan.
Division of Gastroenterology and Hepatology, Department of Internal Medicine, Keio University School of Medicine, Tokyo, Japan.
Mod Rheumatol Case Rep. 2023 Dec 29;8(1):33-36. doi: 10.1093/mrcr/rxad031.
An 18 year-old man with autoimmune hepatitis-primary sclerosing cholangitis-overlap syndrome and ulcerative colitis was admitted due to relapsed enteritis and polyarthritis after cessation of infliximab. Colonoscopy and articular ultrasonography revealed large ulcers in the colon with crypt abscess in the specimens and active enthesitis and synovitis, respectively. His intestinitis was improved with golimumab but arthritis was persistent. Golimumab was switched to secukinumab, which was effective for arthritis. However, colitis was flared resulting in total colorectal resection. One month after colectomy, polyarthritis was relapsed. Tocilizumab ameliorated arthritis but enteritis emerged again, and switching tocilizumab to adalimumab improved enteritis but arthritis exacerbated. Finally, we restarted tocilizumab for arthritis with continued adalimumab for enteritis. The dual cytokine blocking strategy, tumour necrosis factor-α and interleukin-6 inhibition, subsided both of his refractory enteritis and arthritis and maintained remission for more than 3 years without any serious adverse event. Our case suggests that enteritis and arthritis in inflammatory bowel disease may be different in pathophysiology and raises the possible usefulness of simultaneous inhibition of two inflammatory cytokines in such cases.
一位 18 岁男性，患有自身免疫性肝炎-原发性硬化性胆管炎重叠综合征和溃疡性结肠炎，因停用英夫利昔单抗后肠炎和多发性关节炎复发而入院。结肠镜检查和关节超声检查显示结肠有大溃疡，标本中有隐窝脓肿，分别存在活动性附着点炎和滑膜炎。他的肠炎用戈利木单抗改善，但关节炎仍持续存在。将戈利木单抗转换为司库珠单抗后，关节炎得到有效缓解。然而，结肠炎发作，导致全结肠切除。结肠切除术后 1 个月，多发性关节炎复发。托珠单抗改善了关节炎，但肠炎再次出现，将托珠单抗转换为阿达木单抗改善了肠炎，但关节炎加重。最后，我们重新开始使用托珠单抗治疗关节炎，并继续使用阿达木单抗治疗肠炎。双重细胞因子阻断策略，即肿瘤坏死因子-α和白细胞介素-6 抑制，缓解了他的难治性肠炎和关节炎，并且在 3 年多的时间里没有任何严重的不良事件，病情一直处于缓解状态。我们的病例提示炎症性肠病中的肠炎和关节炎在发病机制上可能不同，并提示在这种情况下同时抑制两种炎症细胞因子可能有用。
