Literature DB >> 21160462

Progressive podocyte injury and globotriaosylceramide (GL-3) accumulation in young patients with Fabry disease.

Behzad Najafian1, Einar Svarstad2, Leif Bostad3, Marie-Claire Gubler4, Camilla Tøndel5, Chester Whitley6, Michael Mauer7.   

Abstract

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.

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Year:  2010        PMID: 21160462      PMCID: PMC3640823          DOI: 10.1038/ki.2010.484

Source DB:  PubMed          Journal:  Kidney Int        ISSN: 0085-2538            Impact factor:   10.612


  35 in total

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Journal:  Ann Pediatr (Paris)       Date:  1977-09

Review 2.  The podocyte's response to injury: role in proteinuria and glomerulosclerosis.

Authors:  S J Shankland
Journal:  Kidney Int       Date:  2006-05-10       Impact factor: 10.612

3.  Long-term therapy with agalsidase alfa for Fabry disease: safety and effects on renal function in a home infusion setting.

Authors:  Raphael Schiffmann; Markus Ries; Margaret Timmons; John T Flaherty; Roscoe O Brady
Journal:  Nephrol Dial Transplant       Date:  2005-10-04       Impact factor: 5.992

4.  Early prenatal diagnosis of inborn error of metabolism: a case report of a fetus affected with Fabry's disease.

Authors:  O Tsutsumi; M Sato; K Sato; K Sato; M Mizuno; S Sakamoto
Journal:  Asia Oceania J Obstet Gynaecol       Date:  1985-03

5.  Agalsidase-beta therapy for advanced Fabry disease: a randomized trial.

Authors:  Maryam Banikazemi; Jan Bultas; Stephen Waldek; William R Wilcox; Chester B Whitley; Marie McDonald; Richard Finkel; Seymour Packman; Daniel G Bichet; David G Warnock; Robert J Desnick
Journal:  Ann Intern Med       Date:  2006-12-18       Impact factor: 25.391

6.  The efficiency of systematic sampling in stereology and its prediction.

Authors:  H J Gundersen; E B Jensen
Journal:  J Microsc       Date:  1987-09       Impact factor: 1.758

7.  High incidence of later-onset fabry disease revealed by newborn screening.

Authors:  Marco Spada; Severo Pagliardini; Makiko Yasuda; Turgut Tukel; Geetha Thiagarajan; Hitoshi Sakuraba; Alberto Ponzone; Robert J Desnick
Journal:  Am J Hum Genet       Date:  2006-04-28       Impact factor: 11.025

Review 8.  Progression of glomerular diseases: is the podocyte the culprit?

Authors:  W Kriz; N Gretz; K V Lemley
Journal:  Kidney Int       Date:  1998-09       Impact factor: 10.612

9.  Weekly enzyme replacement therapy may slow decline of renal function in patients with Fabry disease who are on long-term biweekly dosing.

Authors:  Raphael Schiffmann; Hasan Askari; Margaret Timmons; Chevalia Robinson; William Benko; Roscoe O Brady; Markus Ries
Journal:  J Am Soc Nephrol       Date:  2007-04-04       Impact factor: 10.121

10.  Widening of foot processes in normoalbuminuric adolescents with type 1 diabetes.

Authors:  Torun Birgitta Torbjörnsdotter; Nina Elisabeth Staffansdotter Sohlman Perrin; Georg Alexander Jaremko; Ulla Birgitta Berg
Journal:  Pediatr Nephrol       Date:  2005-04-13       Impact factor: 3.714

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  58 in total

1.  Electron microscopy illuminates the pathology of Fabry nephropathy.

Authors:  Steven E Bradshaw
Journal:  Nat Rev Nephrol       Date:  2011-03       Impact factor: 28.314

2.  Fabry disease: dose matters.

Authors:  David G Warnock; Michael Mauer
Journal:  J Am Soc Nephrol       Date:  2014-02-20       Impact factor: 10.121

3.  Is it or is it not a pathogenic mutation? Is it or is it not the podocyte?

Authors:  Maria Dolores Sanchez-Niño; Alberto Ortiz
Journal:  J Nephropathol       Date:  2012-10-01

Review 4.  Lipid biology of the podocyte--new perspectives offer new opportunities.

Authors:  Alessia Fornoni; Sandra Merscher; Jeffrey B Kopp
Journal:  Nat Rev Nephrol       Date:  2014-05-27       Impact factor: 28.314

5.  Long-Term Dose-Dependent Agalsidase Effects on Kidney Histology in Fabry Disease.

Authors:  Rannveig Skrunes; Camilla Tøndel; Sabine Leh; Kristin Kampevold Larsen; Gunnar Houge; Einar Skulstad Davidsen; Carla Hollak; André B P van Kuilenburg; Frédéric M Vaz; Einar Svarstad
Journal:  Clin J Am Soc Nephrol       Date:  2017-06-16       Impact factor: 8.237

6.  Sphingolipids and Redox Signaling in Renal Regulation and Chronic Kidney Diseases.

Authors:  Owais M Bhat; Xinxu Yuan; Guangbi Li; RaMi Lee; Pin-Lan Li
Journal:  Antioxid Redox Signal       Date:  2018-01-09       Impact factor: 8.401

7.  Globotriaosylsphingosine (Lyso-Gb3) as a biomarker for cardiac variant (N215S) Fabry disease.

Authors:  Fahad J Alharbi; Shanat Baig; Christiane Auray-Blais; Michel Boutin; Douglas G Ward; Nigel Wheeldon; Rick Steed; Charlotte Dawson; Derralynn Hughes; Tarekegn Geberhiwot
Journal:  J Inherit Metab Dis       Date:  2018-01-02       Impact factor: 4.982

8.  Patients with Fabry disease after enzyme replacement therapy dose reduction versus treatment switch.

Authors:  Frank Weidemann; Johannes Krämer; Thomas Duning; Malte Lenders; Sima Canaan-Kühl; Alice Krebs; Hans Guerrero González; Claudia Sommer; Nurcan Üçeyler; Markus Niemann; Stefan Störk; Michael Schelleckes; Stefanie Reiermann; Jörg Stypmann; Stefan-Martin Brand; Christoph Wanner; Eva Brand
Journal:  J Am Soc Nephrol       Date:  2014-02-20       Impact factor: 10.121

9.  A case of female Fabry disease revealed by renal biopsy.

Authors:  Sae Aratani; Hiroyuki Yamakawa; Shinya Suzuki; Tomoyuki Otsuka; Yukinao Sakai; Akira Shimizu; Shuichi Tsuruoka
Journal:  CEN Case Rep       Date:  2019-09-21

Review 10.  Strategic biomarkers for drug development in treating rare diseases and diseases in neonates and infants.

Authors:  Jane P F Bai; Jeffrey S Barrett; Gibert J Burckart; Bernd Meibohm; Hari Cheryl Sachs; Lynne Yao
Journal:  AAPS J       Date:  2013-01-19       Impact factor: 4.009

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