Lee Eun Ji, Park Eun Young, Mun HyoWon, Chang EunSun, Ko Je Yeong, Kim Do Yeon, Park Jong Hoon
Department of Biological Science, Sookmyung Women's University, Seoul, Republic of Korea.
Department of Biological Science, Sookmyung Women's University, Seoul, Republic of Korea
FASEB J. 2015 Aug;29(8):3506-14. doi: 10.1096/fj.15-272302. Epub 2015 May 1.
Autosomal polycystic kidney disease (ADPKD) is a highly prevalent genetic renal disorder in which epithelial-lining fluid-filled cysts appear in kidneys. It is accompanied by hyperactivation of cell proliferation, interstitial inflammation, and fibrosis around the cyst lining cells, finally reaching end-stage renal disease. Previously, we found high expression of ligands stimulating the receptor for advanced glycation end products (RAGE) in ADPKD mice. Furthermore, gene silencing of RAGE was revealed to cause reduction of cystogenesis via down-regulation of cell proliferation in vitro, and intravenous administration of anti-RAGE adenovirus in vivo also displayed alleviation of the disease. Here, we attempted to identify the role of soluble RAGE (sRAGE) in inhibiting the progression of ADPKD using 2 different ADPKD mouse models. sRAGE is an endogenously expressed form of RAGE that has no membrane-anchoring domain, thereby giving it the ability to neutralize the ligands that stimulate RAGE signals. Both overexpression of sRAGE and sRAGE treatment blocked RAGE-mediated cell proliferation in vitro. In addition, sRAGE-injected ADPKD mice showed reduced cysts accompanied by enhanced renal function, inhibition of cell proliferation, inflammation, and fibrosis. These positive therapeutic effects of sRAGE displayed little liver toxicity, suggesting it as a new potential therapeutic target of ADPKD with low side effects.
常染色体显性多囊肾病(ADPKD)是一种高度流行的遗传性肾脏疾病,其特征是肾脏中出现内衬上皮细胞的充满液体的囊肿。该病伴有细胞增殖过度激活、间质炎症以及囊肿内衬细胞周围的纤维化,最终发展为终末期肾病。此前,我们发现ADPKD小鼠中刺激晚期糖基化终产物受体(RAGE)的配体表达较高。此外,RAGE基因沉默显示可通过体外下调细胞增殖来减少囊肿形成,并且在体内静脉注射抗RAGE腺病毒也显示出病情缓解。在此,我们试图使用2种不同的ADPKD小鼠模型来确定可溶性RAGE(sRAGE)在抑制ADPKD进展中的作用。sRAGE是RAGE的一种内源性表达形式,没有膜锚定结构域,因此具有中和刺激RAGE信号的配体的能力。sRAGE的过表达和sRAGE处理均在体外阻断了RAGE介导的细胞增殖。此外,注射sRAGE的ADPKD小鼠囊肿减少,同时肾功能增强,细胞增殖、炎症和纤维化受到抑制。sRAGE的这些积极治疗效果几乎没有肝毒性,表明它是一种具有低副作用的ADPKD新潜在治疗靶点。