Department of Immunopathology, Sanquin Research, and Landsteiner Laboratory, Amsterdam UMC, University of Amsterdam, Amsterdam, The Netherlands.
Department of Pediatric Hematology, Immunology and Infectious Diseases, Emma Children's Hospital, Amsterdam UMC, Amsterdam, The Netherlands.
Blood Adv. 2019 Feb 26;3(4):621-632. doi: 10.1182/bloodadvances.2018025692.
Mutations in the gene encoding for complement regulator factor H (FH) severely disrupt its normal function to protect human cells from unwanted complement activation, resulting in diseases such as atypical hemolytic uremic syndrome (aHUS). aHUS presents with severe hemolytic anemia, thrombocytopenia, and renal disease, leading to end-stage renal failure. Treatment of severe complement-mediated disease, such as aHUS, by inhibiting the terminal complement pathway, has proven to be successful but at the same time fails to preserve the protective role of complement against pathogens. To improve complement regulation on human cells without interfering with antimicrobial activity, we identified an anti-FH monoclonal antibody (mAb) that induced increased FH-mediated protection of primary human endothelial cells from complement, while preserving the complement-mediated killing of bacteria. Moreover, this FH-activating mAb restored complement regulation in sera from aHUS patients carrying various heterozygous mutations in FH known to impair FH function and dysregulate complement activation. Our data suggest that FH normally circulates in a less active conformation and can become more active, allowing enhanced complement regulation on human cells. Antibody-mediated potentiation of FH may serve as a highly effective approach to inhibit unwanted complement activation on human cells in a wide range of hematological diseases while preserving the protective role of complement against pathogens.
基因编码补体调节因子 H(FH)的突变严重破坏了其正常功能,无法保护人体细胞免受不受控制的补体激活,从而导致非典型溶血性尿毒症综合征(aHUS)等疾病。aHUS 表现为严重的溶血性贫血、血小板减少和肾脏疾病,导致终末期肾衰竭。通过抑制末端补体途径治疗严重的补体介导疾病(如 aHUS)已被证明是有效的,但同时也无法保留补体对病原体的保护作用。为了在不干扰抗菌活性的情况下改善对人体细胞的补体调节,我们鉴定了一种抗 FH 单克隆抗体(mAb),它可诱导 FH 介导的原发性人内皮细胞对补体的保护作用增加,同时保留补体介导的细菌杀伤作用。此外,这种 FH 激活 mAb 恢复了携带已知会损害 FH 功能和失调补体激活的各种 FH 杂合突变的 aHUS 患者血清中的补体调节。我们的数据表明,FH 通常以一种不太活跃的构象循环,并且可以变得更加活跃,从而增强对人体细胞的补体调节。抗体介导的 FH 增强作用可能是一种非常有效的方法,可以抑制广泛的血液系统疾病中人体细胞不受控制的补体激活,同时保留补体对病原体的保护作用。