Kurbegovic Almira, Trudel Marie
Molecular Genetics and Development, Institut de Recherches Cliniques de Montréal, Faculté de Médecine, Université de Montréal, Montréal, Québec, Canada.
Molecular Genetics and Development, Institut de Recherches Cliniques de Montréal, Faculté de Médecine, Université de Montréal, Montréal, Québec, Canada
Am J Physiol Renal Physiol. 2016 Oct 1;311(4):F740-F751. doi: 10.1152/ajprenal.00167.2016. Epub 2016 Aug 3.
Acute kidney injury (AKI) and autosomal dominant polycystic kidney disease (ADPKD) are considered separate entities that both frequently cause renal failure. Since ADPKD appears to depend on a polycystin-1 (Pc1) or Pc2 dosage mechanism, we investigated whether slow progression of cystogenesis in two Pkd1 transgenic mouse models can be accelerated with moderate ischemia-reperfusion injury (IRI). Transient unilateral left ischemic kidneys in both nontransgenic and transgenic mice reproducibly develop tubular dilatations, cysts, and typical PKD cellular defects within 3 mo post-IRI. Similar onset and severity of IRI induced-cystogenesis independently of genotype revealed that IRI is sufficient to promote renal cyst formation; however, this response was not further amplified by the transgene in Pkd1 mouse models. The IRI nontransgenic and transgenic kidneys showed from 16 days post-IRI strikingly increased and sustained Pkd1/Pc1 (>3-fold) and Pc2 (>8-fold) expression that can individually be cystogenic in mice. In parallel, long-term and important stimulation of hypoxia-inducible factor 1α expression was induced as in polycystic kidney disease. While mammalian target of rapamycin signaling is activated, stimulation of the Wnt pathway, with markedly increased active β-catenin and c-Myc expression in IRI renal epithelium, uncovered a similar regulatory cystogenic response shared by IRI and ADPKD. Our study demonstrates that long-term AKI induces cystogenesis and cross talk with ADPKD Pc1/Pc2 pathogenic signaling.
急性肾损伤(AKI)和常染色体显性多囊肾病(ADPKD)被认为是两种不同的疾病实体,它们都经常导致肾衰竭。由于ADPKD似乎依赖于多囊蛋白-1(Pc1)或Pc2的剂量机制,我们研究了在两种Pkd1转基因小鼠模型中,适度的缺血再灌注损伤(IRI)是否能加速囊肿形成的缓慢进程。在非转基因和转基因小鼠中,短暂性单侧左肾缺血均可在IRI后3个月内重现性地出现肾小管扩张、囊肿以及典型的PKD细胞缺陷。IRI诱导的囊肿形成的起始和严重程度与基因型无关,这表明IRI足以促进肾囊肿的形成;然而,在Pkd1小鼠模型中,转基因并未进一步放大这种反应。IRI后的非转基因和转基因肾脏从第16天起显示出Pkd1/Pc1(>3倍)和Pc2(>8倍)表达显著增加且持续存在,这两种蛋白在小鼠中均可单独导致囊肿形成。同时,与多囊肾病一样,诱导了缺氧诱导因子1α表达的长期且重要的上调。虽然雷帕霉素靶蛋白信号被激活,但在IRI肾上皮细胞中,Wnt通路受到刺激,活性β-连环蛋白和c-Myc表达显著增加,揭示了IRI和ADPKD共有的类似调节性囊肿形成反应。我们的研究表明,长期的AKI可诱导囊肿形成,并与ADPKD的Pc1/Pc2致病信号发生相互作用。