Sheikh Virginia, Porter Brian O, DerSimonian Rebecca, Kovacs Stephen B, Thompson William L, Perez-Diez Ainhoa, Freeman Alexandra F, Roby Gregg, Mican JoAnn, Pau Alice, Rupert Adam, Adelsberger Joseph, Higgins Jeanette, Bourgeois Jeffrey S, Jensen Stig M R, Morcock David R, Burbelo Peter D, Osnos Leah, Maric Irina, Natarajan Ven, Croughs Therese, Yao Michael D, Estes Jacob D, Sereti Irini
National Institute of Allergy and Infectious Diseases, National Institutes of Health (NIH), Bethesda, MD;
Leidos Biomedical Research, Inc., Frederick National Laboratory for Cancer Research, Frederick, MD;
Blood. 2016 Feb 25;127(8):977-88. doi: 10.1182/blood-2015-05-645077. Epub 2015 Dec 16.
Idiopathic CD4 lymphopenia (ICL) is a rare syndrome defined by low CD4 T-cell counts (<300/µL) without evidence of HIV infection or other known cause of immunodeficiency. ICL confers an increased risk of opportunistic infections and has no established treatment. Interleukin-7 (IL-7) is fundamental for thymopoiesis, T-cell homeostasis, and survival of mature T cells, which provides a rationale for its potential use as an immunotherapeutic agent for ICL. We performed an open-label phase 1/2A dose-escalation trial of 3 subcutaneous doses of recombinant human IL-7 (rhIL-7) per week in patients with ICL who were at risk of disease progression. The primary objectives of the study were to assess safety and the immunomodulatory effects of rhIL-7 in ICL patients. Injection site reactions were the most frequently reported adverse events. One patient experienced a hypersensitivity reaction and developed non-neutralizing anti-IL-7 antibodies. Patients with autoimmune diseases that required systemic therapy at screening were excluded from the study; however, 1 participant developed systemic lupus erythematosus while on study and was excluded from further rhIL-7 dosing. Quantitatively, rhIL-7 led to an increase in the number of circulating CD4 and CD8 T cells and tissue-resident CD3 T cells in the gut mucosa and bone marrow. Functionally, these T cells were capable of producing cytokines after mitogenic stimulation. rhIL-7 was well tolerated at biologically active doses and may represent a promising therapeutic intervention in ICL. This trial was registered at www.clinicaltrials.gov as #NCT00839436.
特发性CD4淋巴细胞减少症(ICL)是一种罕见综合征,其定义为CD4 T细胞计数低(<300/µL),且无HIV感染证据或其他已知免疫缺陷病因。ICL会增加机会性感染风险,且尚无既定治疗方法。白细胞介素-7(IL-7)对胸腺生成、T细胞稳态及成熟T细胞存活至关重要,这为其作为ICL免疫治疗药物的潜在用途提供了理论依据。我们对有疾病进展风险的ICL患者进行了一项开放标签的1/2A期剂量递增试验,每周皮下注射3次重组人IL-7(rhIL-7)。该研究的主要目的是评估rhIL-7在ICL患者中的安全性和免疫调节作用。注射部位反应是最常报告的不良事件。一名患者发生超敏反应并产生非中和性抗IL-7抗体。筛查时需要全身治疗的自身免疫性疾病患者被排除在研究之外;然而,1名参与者在研究期间发生系统性红斑狼疮,被排除在进一步的rhIL-7给药之外。从数量上看,rhIL-7导致循环CD4和CD8 T细胞以及肠道黏膜和骨髓中组织驻留CD3 T细胞数量增加。从功能上看,这些T细胞在有丝分裂原刺激后能够产生细胞因子。rhIL-7在生物活性剂量下耐受性良好,可能是ICL中有前景的治疗干预措施。该试验已在www.clinicaltrials.gov上注册,编号为#NCT00839436。