Sandwell and West Birmingham Hospitals NHS Trust, City Hospital, Dudley Road, Birmingham, UK.
Clin Rheumatol. 2012 Jun;31(6):1013-8. doi: 10.1007/s10067-011-1933-9. Epub 2012 Jan 17.
Chronic progressive multisystem granulomatous disease is seen in 10-30% of patients with sarcoidosis and can result in end organ damage. Corticosteroids are the mainstay of treatment with the addition of cytotoxic agents in severe cases. Some patients are refractory to such treatment and, therefore, management is a challenge. There is currently limited evidence for biological agents such as infliximab, a monoclonal anti-tumor necrosis factor-α antibody in the treatment of multisystem sarcoidosis. We report outcomes of three patients with extensive multisystem sarcoidosis refractory to conventional treatment and treated at our center. Clinical assessment and radiographic imaging were used to assess the response to infliximab treatment. Infliximab therapy induced clinical remission in all three patients, and this clinical response correlated with radiographic evidence of the resolution of granulomatous disease. Serum ACE level was reduced in all cases, and daily steroid dosage was reduced. We propose that infliximab can be an effective treatment in patients with multisystem complex sarcoidosis refractory to conventional drug therapy and can result in sustained clinical remission. Our experience supports the urgent need for randomized controlled clinical trials of anti-TNF therapy in refractory systemic sarcoidosis.
慢性进行性多系统肉芽肿病可见于 10-30%的结节病患者，可导致终末器官损害。皮质类固醇是治疗的主要方法，在严重病例中加用细胞毒性药物。一些患者对此类治疗有抗药性，因此治疗具有挑战性。目前，生物制剂（如抗肿瘤坏死因子-α单克隆抗体英夫利昔单抗）在治疗多系统结节病方面的证据有限。我们报告了在我们中心治疗的 3 例对常规治疗有抗药性的广泛多系统结节病患者的结果。临床评估和放射影像学用于评估英夫利昔单抗治疗的反应。英夫利昔单抗治疗使所有 3 例患者的临床症状缓解，这种临床反应与肉芽肿性疾病消退的放射影像学证据相关。所有病例的血清 ACE 水平降低，每日类固醇剂量减少。我们提出，英夫利昔单抗可有效治疗对常规药物治疗有抗药性的多系统复杂结节病，并可导致持续的临床缓解。我们的经验支持迫切需要对难治性系统性结节病进行抗 TNF 治疗的随机对照临床试验。
