Obi Ogugua Ndili, Saketkoo Lesley Ann, Russell Anne-Marie, Baughman Robert P
Division of Pulmonary Critical Care and Sleep Medicine, Brody School of Medicine, East Carolina University, Greenville, NC, United States.
New Orleans Scleroderma and Sarcoidosis Patient Care and Research Center, New Orleans, LA, United States.
Front Med (Lausanne). 2022 Oct 12;9:991783. doi: 10.3389/fmed.2022.991783. eCollection 2022.
Sarcoidosis is a systemic granulomatous inflammatory disease of unknown etiology. It affects the lungs in over 90% of patients yet extra-pulmonary and multi-organ involvement is common. Spontaneous remission of disease occurs commonly, nonetheless, over 50% of patients will require treatment and up to 30% of patients will develop a chronic progressive non-remitting disease with marked pulmonary fibrosis leading to significant morbidity and death. Guidelines outlining an immunosuppressive treatment approach to sarcoidosis were recently published, however, the strength of evidence behind many of the guideline recommended drugs is weak. None of the drugs currently used for the treatment of sarcoidosis have been rigorously studied and prescription of these drugs is often based on off-label" indications informed by experience with other diseases. Indeed, only two medications [prednisone and repository corticotropin (RCI) injection] currently used in the treatment of sarcoidosis are approved by the United States Food and Drug Administration. This situation results in significant reimbursement challenges especially for the more advanced (and often more effective) drugs that are favored for severe and refractory forms of disease causing an over-reliance on corticosteroids known to be associated with significant dose and duration dependent toxicities. This past decade has seen a renewed interest in developing new drugs and exploring novel therapeutic pathways for the treatment of sarcoidosis. Several of these trials are active randomized controlled trials (RCTs) designed to recruit relatively large numbers of patients with a goal to determine the safety, efficacy, and tolerability of these new molecules and therapeutic approaches. While it is an exciting time, it is also necessary to exercise caution. Resources including research dollars and most importantly, patient populations available for trials are limited and thus necessitate that several of the challenges facing drug trials and drug development in sarcoidosis are addressed. This will ensure that currently available resources are judiciously utilized. Our paper reviews the ongoing and anticipated drug trials in sarcoidosis and addresses the challenges facing these and future trials. We also review several recently completed trials and draw lessons that should be applied in future.
结节病是一种病因不明的全身性肉芽肿性炎症性疾病。超过90%的患者肺部会受到影响,但肺外和多器官受累也很常见。疾病通常会自发缓解,尽管如此,超过50%的患者需要治疗,高达30%的患者会发展为慢性进行性不缓解疾病,伴有明显的肺纤维化,导致严重的发病率和死亡。最近发布了概述结节病免疫抑制治疗方法的指南,然而,许多指南推荐药物背后的证据力度较弱。目前用于治疗结节病的药物均未经过严格研究,这些药物的处方通常基于“非标签”适应症,这些适应症是根据对其他疾病的经验得出的。事实上,目前用于治疗结节病的药物中只有两种[泼尼松和长效促肾上腺皮质激素(RCI)注射剂]获得了美国食品药品监督管理局的批准。这种情况导致了重大的报销挑战,尤其是对于治疗严重和难治性疾病更常用(且通常更有效)的药物,这导致过度依赖已知与显著的剂量和疗程依赖性毒性相关的皮质类固醇。在过去十年中,人们对开发治疗结节病的新药和探索新的治疗途径重新产生了兴趣。其中一些试验是积极的随机对照试验(RCT),旨在招募相对大量的患者,目标是确定这些新分子和治疗方法的安全性、有效性和耐受性。虽然这是一个令人兴奋的时期,但也有必要谨慎行事。包括研究资金在内的资源,以及最重要的是可用于试验的患者群体有限,因此有必要解决结节病药物试验和药物开发面临的一些挑战。这将确保明智地利用现有的资源。我们的论文回顾了正在进行和预期的结节病药物试验,并讨论了这些试验以及未来试验面临的挑战。我们还回顾了一些最近完成的试验,并吸取了应在未来应用的经验教训。