Department of Pathology, University of Washington, Seattle, Washington, USA.
Department of Medicine, Haukeland University Hospital, Bergen, Norway; Institute of Medicine, University of Bergen, Bergen, Norway.
Kidney Int. 2011 Mar;79(6):663-670. doi: 10.1038/ki.2010.484. Epub 2010 Dec 15.
Progressive renal failure often complicates Fabry disease, the pathogenesis of which is not well understood. To further explore this we applied unbiased stereological quantitative methods to electron microscopic changes of Fabry nephropathy and the relationship between parameters of glomerular structure and renal function in 14 young Fabry patients (median age 12 years). Renal biopsies were obtained shortly before enzyme replacement therapy from these patients and from nine normal living kidney donors as controls. Podocyte globotriaosylceramide (GL-3) inclusion volume density increased progressively with age; however, there were no significant relationships between age and endothelial or mesangial inclusion volume densities. Foot process width, greater in male Fabry patients, also progressively increased with age compared with the controls, and correlated directly with proteinuria. In comparison to the biopsies of the controls, endothelial fenestration was reduced in Fabry patients. Thus, our study found relationships between quantitative parameters of glomerular structure in Fabry nephropathy and age, as well as urinary protein excretion. Hence, podocyte injury may play a pivotal role in the development and progression of Fabry nephropathy.
进行性肾衰竭常使法布瑞病复杂化,但该病的发病机制尚未完全阐明。为了进一步探究这一问题,我们采用无偏立体定量方法研究了法布瑞肾病的电镜变化,以及 14 名年轻法布瑞病患者(中位年龄 12 岁)肾小球结构和肾功能参数之间的关系。这些患者和 9 名正常活体供肾者在接受酶替代治疗前不久均接受了肾活检。足细胞糖鞘脂(GL-3)包涵体体积密度随年龄逐渐增加;然而,内皮或系膜包涵体体积密度与年龄之间无显著关系。足突宽度在男性法布瑞病患者中更大,与对照组相比也随年龄逐渐增加,并且与蛋白尿直接相关。与对照组的活检相比,法布瑞病患者的内皮窗孔减少。因此,我们的研究发现法布瑞肾病肾小球结构的定量参数与年龄以及尿蛋白排泄之间存在关系。因此,足细胞损伤可能在法布瑞肾病的发生和进展中起关键作用。