Romaker Daniel, Puetz Michael, Teschner Sven, Donauer Johannes, Geyer Marcel, Gerke Peter, Rumberger Brigitta, Dworniczak Bernd, Pennekamp Petra, Buchholz Björn, Neumann H P H, Kumar Rajiv, Gloy Joachim, Eckardt Kai-Uwe, Walz Gerd
Renal Division, University Hospital Freiburg, Hugstetter Strasse 55, D-79106 Freiburg, Germany.
J Am Soc Nephrol. 2009 Jan;20(1):48-56. doi: 10.1681/ASN.2008040345. Epub 2008 Oct 22.
Autosomal dominant polycystic kidney disease (ADPKD) is a common hereditary disease associated with progressive renal failure. Although cyst growth and compression of surrounding tissue may account for some loss of renal tissue, the other factors contributing to the progressive renal failure in patients with ADPKD are incompletely understood. Here, we report that secreted frizzled-related protein 4 (sFRP4) is upregulated in human ADPKD and in four different animal models of PKD, suggesting that sFRP4 expression is triggered by a common mechanism that underlies cyst formation. Cyst fluid from ADPKD kidneys activated the sFRP4 promoter and induced production of sFRP4 protein in renal tubular epithelial cell lines. Antagonism of the vasopressin 2 receptor blocked both promoter activity and tubular sFRP4 expression. In addition, sFRP4 selectively influenced members of the canonical Wnt signaling cascade and promoted cystogenesis of the zebrafish pronephros. sFRP4 was detected in the urine of both patients and animals with PKD, suggesting that sFRP4 may be a potential biomarker for monitoring the progression of ADPKD. Taken together, these observations suggest a potential role for SFRP4 in the pathogenesis of ADPKD.
常染色体显性多囊肾病(ADPKD)是一种与进行性肾衰竭相关的常见遗传性疾病。尽管囊肿生长及对周围组织的压迫可能导致部分肾组织丢失,但ADPKD患者发生进行性肾衰竭的其他因素仍未完全明确。在此,我们报道分泌型卷曲相关蛋白4(sFRP4)在人类ADPKD及四种不同的PKD动物模型中表达上调,提示sFRP4的表达由囊肿形成所共有的一种机制触发。ADPKD肾囊肿液可激活sFRP4启动子并诱导肾小管上皮细胞系中sFRP4蛋白的产生。血管加压素2受体拮抗剂可阻断启动子活性及肾小管sFRP4表达。此外,sFRP4可选择性影响经典Wnt信号级联反应的成员,并促进斑马鱼前肾的囊肿形成。在PKD患者及动物的尿液中均检测到sFRP4,提示sFRP4可能是监测ADPKD进展的潜在生物标志物。综上所述,这些观察结果提示SFRP4在ADPKD发病机制中可能发挥作用。