José Tiburcio do Monte Neto, Gianna Mastroianni Kirsztajn
{"title":"荚膜损伤在法布里病肾病发病机制中的作用。","authors":"José Tiburcio do Monte Neto, Gianna Mastroianni Kirsztajn","doi":"10.1590/2175-8239-JBN-2024-0035en","DOIUrl":null,"url":null,"abstract":"<p><p>Renal involvement is one of the most severe morbidities of Fabry disease (FD), a multisystemic lysosomal storage disease with an X-linked inheritance pattern. It results from pathogenic variants in the GLA gene (Xq22.2), which encodes the production of alpha-galactosidase A (α-Gal), responsible for glycosphingolipid metabolism. Insufficient activity of this lysosomal enzyme generates deposits of unprocessed intermediate substrates, especially globotriaosylceramide (Gb3) and derivatives, triggering cellular injury and subsequently, multiple organ dysfunction, including chronic nephropathy. Kidney injury in FD is classically attributed to Gb3 deposits in renal cells, with podocytes being the main target of the pathological process, in which structural and functional alterations are established early and severely. This configures a typical hereditary metabolic podocytopathy, whose clinical manifestations are proteinuria and progressive renal failure. Although late clinical outcomes and morphological changes are well established in this nephropathy, the molecular mechanisms that trigger and accelerate podocyte injury have not yet been fully elucidated. Podocytes are highly specialized and differentiated cells that cover the outer surface of glomerular capillaries, playing a crucial role in preserving the structure and function of the glomerular filtration barrier. They are frequent targets of injury in many nephropathies. Furthermore, dysfunction and depletion of glomerular podocytes are essential events implicated in the pathogenesis of chronic kidney disease progression. We will review the biology of podocytes and their crucial role in regulating the glomerular filtration barrier, analyzing the main pathogenic pathways involved in podocyte injury, especially related to FD nephropathy.</p>","PeriodicalId":14724,"journal":{"name":"Jornal brasileiro de nefrologia : 'orgao oficial de Sociedades Brasileira e Latino-Americana de Nefrologia","volume":"46 3","pages":"e20240035"},"PeriodicalIF":1.3000,"publicationDate":"2024-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11287863/pdf/","citationCount":"0","resultStr":"{\"title\":\"The role of podocyte injury in the pathogenesis of Fabry disease nephropathy.\",\"authors\":\"José Tiburcio do Monte Neto, Gianna Mastroianni Kirsztajn\",\"doi\":\"10.1590/2175-8239-JBN-2024-0035en\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Renal involvement is one of the most severe morbidities of Fabry disease (FD), a multisystemic lysosomal storage disease with an X-linked inheritance pattern. It results from pathogenic variants in the GLA gene (Xq22.2), which encodes the production of alpha-galactosidase A (α-Gal), responsible for glycosphingolipid metabolism. Insufficient activity of this lysosomal enzyme generates deposits of unprocessed intermediate substrates, especially globotriaosylceramide (Gb3) and derivatives, triggering cellular injury and subsequently, multiple organ dysfunction, including chronic nephropathy. Kidney injury in FD is classically attributed to Gb3 deposits in renal cells, with podocytes being the main target of the pathological process, in which structural and functional alterations are established early and severely. This configures a typical hereditary metabolic podocytopathy, whose clinical manifestations are proteinuria and progressive renal failure. Although late clinical outcomes and morphological changes are well established in this nephropathy, the molecular mechanisms that trigger and accelerate podocyte injury have not yet been fully elucidated. Podocytes are highly specialized and differentiated cells that cover the outer surface of glomerular capillaries, playing a crucial role in preserving the structure and function of the glomerular filtration barrier. They are frequent targets of injury in many nephropathies. Furthermore, dysfunction and depletion of glomerular podocytes are essential events implicated in the pathogenesis of chronic kidney disease progression. 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引用次数: 0
摘要
肾脏受累是法布里病(FD)最严重的病症之一,法布里病是一种多系统溶酶体贮积病,具有 X 连锁遗传模式。该病是由 GLA 基因(Xq22.2)的致病变体引起的,该基因编码产生α-半乳糖苷酶 A(α-Gal),负责糖磷脂代谢。这种溶酶体酶的活性不足会产生未加工的中间底物沉积,尤其是球糖基甘油三酯(Gb3)及其衍生物,引发细胞损伤,进而导致多器官功能障碍,包括慢性肾病。FD的肾损伤通常归因于肾细胞中的Gb3沉积,荚膜细胞是病理过程的主要目标,其结构和功能的改变发生得早且严重。这就是典型的遗传代谢性荚膜细胞病,其临床表现为蛋白尿和进行性肾衰竭。虽然这种肾病的晚期临床结果和形态学变化已得到证实,但引发和加速荚膜细胞损伤的分子机制尚未完全阐明。荚膜细胞是高度特化和分化的细胞,覆盖在肾小球毛细血管的外表面,在维护肾小球滤过屏障的结构和功能方面起着至关重要的作用。在许多肾病中,绒毛膜细胞经常成为损伤的目标。此外,肾小球荚膜细胞的功能障碍和耗竭也是慢性肾病进展的重要发病机制。我们将回顾荚膜细胞的生物学特性及其在调节肾小球滤过屏障中的关键作用,分析荚膜细胞损伤所涉及的主要致病途径,尤其是与 FD 肾病相关的途径。
The role of podocyte injury in the pathogenesis of Fabry disease nephropathy.
Renal involvement is one of the most severe morbidities of Fabry disease (FD), a multisystemic lysosomal storage disease with an X-linked inheritance pattern. It results from pathogenic variants in the GLA gene (Xq22.2), which encodes the production of alpha-galactosidase A (α-Gal), responsible for glycosphingolipid metabolism. Insufficient activity of this lysosomal enzyme generates deposits of unprocessed intermediate substrates, especially globotriaosylceramide (Gb3) and derivatives, triggering cellular injury and subsequently, multiple organ dysfunction, including chronic nephropathy. Kidney injury in FD is classically attributed to Gb3 deposits in renal cells, with podocytes being the main target of the pathological process, in which structural and functional alterations are established early and severely. This configures a typical hereditary metabolic podocytopathy, whose clinical manifestations are proteinuria and progressive renal failure. Although late clinical outcomes and morphological changes are well established in this nephropathy, the molecular mechanisms that trigger and accelerate podocyte injury have not yet been fully elucidated. Podocytes are highly specialized and differentiated cells that cover the outer surface of glomerular capillaries, playing a crucial role in preserving the structure and function of the glomerular filtration barrier. They are frequent targets of injury in many nephropathies. Furthermore, dysfunction and depletion of glomerular podocytes are essential events implicated in the pathogenesis of chronic kidney disease progression. We will review the biology of podocytes and their crucial role in regulating the glomerular filtration barrier, analyzing the main pathogenic pathways involved in podocyte injury, especially related to FD nephropathy.