LeBleu Valerie, Sugimoto Hikaru, Mundel Thomas M, Gerami-Naini Behzad, Finan Elizabeth, Miller Caroline A, Gattone Vincent H, Lu Lingge, Shield Charles F, Folkman Judah, Kalluri Raghu
Division of Matrix Biology, Beth Israel Deaconess Medical Center and Harvard Medical School, Boston, MA 02215, USA.
J Am Soc Nephrol. 2009 Nov;20(11):2359-70. doi: 10.1681/ASN.2009010123. Epub 2009 Oct 15.
Patients with Alport syndrome progressively lose renal function as a result of defective type IV collagen in their glomerular basement membrane. In mice lacking the alpha3 chain of type IV collagen (Col4A3 knockout mice), a model for Alport syndrome, transplantation of wild-type bone marrow repairs the renal disease. It is unknown whether cell-based therapies that do not require transplantation have similar potential. Here, infusion of wild-type bone marrow-derived cells into unconditioned, nonirradiated Col4A3 knockout mice during the late stage of disease significantly improved renal histology and function. Furthermore, transfusion of unfractionated wild-type blood into unconditioned, nonirradiated Col4A3 knockout mice improved the renal phenotype and significantly improved survival. Injection of mouse and human embryonic stem cells into Col4A3 knockout mice produced similar results. Regardless of treatment modality, the improvement in the architecture of the glomerular basement membrane is associated with de novo expression of the alpha3(IV) chain. These data provide further support for testing cell-based therapies for Alport syndrome.
由于肾小球基底膜中IV型胶原蛋白存在缺陷,奥尔波特综合征患者的肾功能会逐渐丧失。在缺乏IV型胶原蛋白α3链的小鼠(Col4A3基因敲除小鼠)中,这是一种奥尔波特综合征模型,野生型骨髓移植可修复肾脏疾病。尚不清楚不需要移植的细胞疗法是否具有类似的潜力。在此,在疾病晚期将野生型骨髓来源的细胞注入未经预处理、未接受辐射的Col4A3基因敲除小鼠体内,显著改善了肾脏组织学和功能。此外,将未分级的野生型血液输给未经预处理、未接受辐射的Col4A3基因敲除小鼠,改善了肾脏表型并显著提高了存活率。将小鼠和人类胚胎干细胞注射到Col4A3基因敲除小鼠体内也产生了类似的结果。无论治疗方式如何,肾小球基底膜结构的改善都与α3(IV)链的从头表达有关。这些数据为测试针对奥尔波特综合征的细胞疗法提供了进一步的支持。