Treatment of intestinal Behcet's syndrome with chimeric tumour necrosis factor α antibody
Treatment of intestinal Behcet's syndrome with chimeric tumour necrosis factor α antibody
复制标题
DOI：
10.1136/gut.49.5.725
复制
发表时间：
2001-11-01
期刊：
影响因子：
24.5
通讯作者：
Bell, AL
中科院分区：
文献类型：
--
作者：
Travis, SPL;Czajkowski, M;Bell, AL
少数白塞综合征患者有胃肠道溃疡。这类患者难以治疗，死亡率较高。面对两名肠道白塞病患者的难治性症状，我们使用肿瘤坏死因子α（TNF-α）单克隆抗体英夫利昔单抗诱导缓解。两名女性（一名27岁，另一名30岁）均表现为口生殖器溃疡、脓疱性皮疹、腹痛、结肠溃疡引起的血性腹泻、体重减轻和滑膜炎。一个患有血栓性静脉炎、指血管炎、肛周瘘和结肠旁脓肿;另一个患有结膜炎和纳塔尔溃疡。其中一位患者接受泼尼松龙、甲基泼尼松龙和沙利度胺治疗，另一位患者接受泼尼松龙、秋水仙碱和环孢菌素治疗，均无效。经过充分讨论后，给予英夫利西单抗（3 mg/kg，1例因近期脓毒症减少剂量，另1例5 mg/kg）。10天内溃疡愈合，血性腹泻和所有肠外表现消退。1例患者在8周后需要第二次输注英夫利西单抗，随后使用低剂量沙利度胺持续缓解（> 15个月）。另一组患者最初缓解持续了12个月，但由于不耐受而不得不停用沙利度胺，在第三次输注前，在没有免疫抑制的情况下，对再治疗的良好反应仅持续了12周。白塞氏综合征的病因尚不清楚，但当用佛波醇肉豆蔻酸酯乙酸酯和CD 3刺激时，白塞氏综合征患者的外周血CD 45 γ δ T细胞产生的TNF-α比对照组多50倍。英夫利西单抗可能在诱导白塞综合征缓解中发挥作用。
Few patients with Behcet's syndrome have gastrointestinal ulceration. Such patients are difficult to treat and have a higher mortality. Faced with refractory symptoms in two patients with intestinal Behcet's, we used the tumour necrosis factor alpha (TNF-alpha) monoclonal antibody infliximab to induce remission. Both women (one aged 27 years, the other 30 years) presented with orogenital ulceration, pustular rash, abdominal pain, bloody diarrhoea due to colonic ulceration, weight loss, and synovitis. One had thrombophlebitis, digital vasculitis, perianal fistula, and paracolic abscess; the other had conjunctivitis and an ulcer in the natal cleft. Treatment with prednisolone, methyl prednisolone, and thalidomide in one and prednisolone, colchicine, and cyclosporin in the other was ineffective. After full discussion, infliximab (3 mg/kg, dose reduced because of recent sepsis in one, and 5 mg/kg in the other) was administered. Within 10 days the ulcers healed, with resolution of bloody diarrhoea and all extraintestinal manifestations. A second infusion of infliximab was necessary eight weeks later in one case, followed by sustained (> 15 months) remission on low dose thalidomide. Remission was initially sustained for 12 months in the other but thalidomide had to be stopped due to intolerance, and a good response to retreatment lasted only 12 weeks without immunosuppression, before a third infusion. The cause of Behcet's syndrome is unknown but peripheral blood CD45 gamma delta T cells in Behcet's produce > 50-fold more TNF-alpha than controls when stimulated with phorbol myristate acetate and and-CD3. Infliximab could have a role for inducing remission in Behcet's syndrome.