Literature DB >> 29274327

Investigation of correlation of urinary globotriaosylceramide (Gb3) levels with markers of renal function in patients with Fabry disease.

Alana Pimentel Moura1, Tatiane Hammerschmidt2, Marion Deon2, Roberto Giugliani3, Carmen Regla Vargas4.   

Abstract

Fabry disease (FD) is a disorder that results from mutations of hydrolase α-galactosidase A. The enzymatic defect leads to accumulation of globotriaosylceramide (Gb3) in the kidney. Substrate deposition is related to tissue damage in FD, but the relation of urinary Gb3 levels in patients and the renal function markers remain not completely understood. Once nephropathy is one of the main features of FD and is marked by an insidious development, we investigated a possible correlation of Gb3 with biochemical markers of nephropathy including albuminuria, estimated glomerular filtration rate (eGFR), serum creatinine and urea, and proteinuria in male and female patients under or not enzyme replacement therapy (ERT).Gb3, proteinuria and albuminuria were increased in male and female FD patients. We found no correlation between urinary Gb3 levels and all renal function parameters evaluated in Fabry patients (in both sexes and using or not ERT). On the other hand, albuminuria showed negative correlation with eGFR only in male under or not ERT, demonstrating that albuminuria seems to be an early marker of renal function alteration. In conclusion, the results suggest that urinary Gb3 level does not reflect the renal function and that albuminuria is an important biomarker in male FD patients.
Copyright © 2017 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Albuminuria; Fabry disease; Globotriaosylceramide; Renal function

Mesh:

Substances:

Year:  2017        PMID: 29274327     DOI: 10.1016/j.cca.2017.12.033

Source DB:  PubMed          Journal:  Clin Chim Acta        ISSN: 0009-8981            Impact factor:   3.786


  4 in total

1.  Brazilian consensus recommendations for the diagnosis, screening, and treatment of individuals with fabry disease: Committee for Rare Diseases - Brazilian Society of Nephrology/2021.

Authors:  Cassiano Augusto Braga Silva; Luis Gustavo Modelli de Andrade; Maria Helena Vaisbich; Fellype de Carvalho Barreto
Journal:  J Bras Nefrol       Date:  2022 Apr-Jun

Review 2.  Renal Manifestations of Fabry Disease: A Narrative Review.

Authors:  Cassiano Augusto Braga Silva; José A Moura-Neto; Marlene Antônia Dos Reis; Osvaldo Merege Vieira Neto; Fellype Carvalho Barreto
Journal:  Can J Kidney Health Dis       Date:  2021-01-19

3.  Human kidney organoids reveal the role of glutathione in Fabry disease.

Authors:  Jin Won Kim; Hyung Wook Kim; Sun Ah Nam; Jong Young Lee; Hae Jin Cho; Tae-Min Kim; Yong Kyun Kim
Journal:  Exp Mol Med       Date:  2021-10-15       Impact factor: 8.718

4.  Reduced α-galactosidase A activity in zebrafish (Danio rerio) mirrors distinct features of Fabry nephropathy phenotype.

Authors:  Hassan O A Elsaid; Jessica Furriol; Maria Blomqvist; Mette Diswall; Sabine Leh; Naouel Gharbi; Jan Haug Anonsen; Janka Babickova; Camilla Tøndel; Einar Svarstad; Hans-Peter Marti; Maximilian Krause
Journal:  Mol Genet Metab Rep       Date:  2022-02-17
  4 in total

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