Institute of Translational Immunology, Research Center for Immunotherapy, University Medical Center, Johannes Gutenberg University, Mainz, Germany.
Department of Molecular Biology, Central Institute of Mental Health, Medical Faculty Mannheim/Heidelberg University, Mannheim, Germany.
Matrix Biol. 2018 Mar;66:34-49. doi: 10.1016/j.matbio.2017.11.002. Epub 2017 Nov 6.
Organ fibrosis is characterized by a chronic wound-healing response, with excess deposition of extracellular matrix components. Here, collagen type I represents the most abundant scar component and a primary target for antifibrotic therapies. Liver fibrosis can progress to cirrhosis and primary liver cancer, which are the major causes of liver related morbidity and mortality. However, a (pro-)collagen type I specific therapy remains difficult and its therapeutic abrogation may incur unwanted side effects. We therefore designed tetracycline-regulated procollagen alpha1(I) short hairpin (sh)RNA expressing mice that permit a highly efficient inducible knockdown of the procollagen alpha1(I) gene in activated (myo-)fibroblasts, to study the effect of induced procollagen type I deficiency. Transgenic mice were generated using recombinase-mediated integration in embryonic stem cells or zinc-finger nuclease-aided genomic targeting combined with miR30-shRNA technology. Liver fibrosis was induced in transgenic mice by carbon tetrachloride, either without or with doxycycline supplementation. Doxycycline treated mice showed an 80-90% suppression of procollagen alpha1(I) transcription and a 40-50% reduction in hepatic collagen accumulation. Procollagen alpha1(I) knockdown also downregulated procollagens type III, IV and VI and other fibrosis related parameters. Moreover, this was associated with an attenuation of chronic inflammation, suggesting that collagen type I serves not only as major scar component, but also as modulator of other collagens and promoter of chronic inflammation.
器官纤维化的特征是慢性伤口愈合反应,细胞外基质成分过度沉积。在这里,I 型胶原代表最丰富的疤痕成分,也是抗纤维化治疗的主要靶点。肝纤维化可进展为肝硬化和原发性肝癌,这是导致肝相关发病率和死亡率的主要原因。然而,(前)I 型胶原特异性治疗仍然很困难,其治疗阻断可能会产生不必要的副作用。因此,我们设计了四环素调控的前胶原 alpha1(I)短发夹(sh)RNA 表达小鼠,允许在激活的(肌成)纤维细胞中高效诱导前胶原 alpha1(I)基因的敲低,以研究诱导的 I 型胶原缺乏的效果。通过重组酶介导的整合在胚胎干细胞或锌指核酸酶辅助的基因组靶向与 miR30-shRNA 技术的组合,生成了转基因小鼠。在转基因小鼠中,通过四氯化碳诱导肝纤维化,要么不补充,要么补充强力霉素。强力霉素处理的小鼠显示前胶原 alpha1(I)转录的 80-90%抑制和肝胶原积累的 40-50%减少。前胶原 alpha1(I)敲低也下调了 I 型、III 型、IV 型和 VI 型前胶原和其他纤维化相关参数。此外,这与慢性炎症的减轻有关,表明胶原 I 不仅作为主要的疤痕成分,而且作为其他胶原的调节剂和慢性炎症的促进剂。