Literature DB >> 21235448

Treatment of fabry disease: current and emerging strategies.

Paula Rozenfeld1, Pablo M Neumann.   

Abstract

Fabry disease is an X-linked lysosomal storage disorder (LSD) due to deficiency of the enzyme α-galactosidase A (GLA). Absent or reduced enzyme activity leads to impaired catabolism of neutral glycosphingolipids, particularly globotriaosylceramide (Gb3), resulting in intracellular deposition of such lipids. Clinical manifestations in hemizygote males include angiokeratoma, hypohydrosis, acroparesthesia, abdominal pain, proteinuria, renal insufficiency, left ventricular hypertrophy and cerebrovascular accidents. Heterozygote women may present with mild to severe signs and symptoms. Since year 2001, enzyme replacement therapy (ERT) is the only specific treatment for Fabry disease. The beneficial effect of ERT on different organs/systems has been extensively evaluated, and an improvement in renal function, cardiac mass and quality of life has been reported. Different treatment approaches are currently on development. One of them implies the use of the active-site-specific chaperone 1-deoxygalactonojirimycin that acts facilitating folding of mutant GLA in the endoplasmic reticulum and increasing its lysosomal residual activity. Reduction of Gb3 deposits has been shown in lymphoblasts from Fabry patients with missense mutations and transgenic mouse model expressing a missense mutation GLA. Gene therapy has been also developed as a potential option for treatment of Fabry disease. This review will discuss these novel therapeutic options along with their advantages and limitations.

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Year:  2011        PMID: 21235448     DOI: 10.2174/138920111795542705

Source DB:  PubMed          Journal:  Curr Pharm Biotechnol        ISSN: 1389-2010            Impact factor:   2.837


  5 in total

1.  Relative distribution of Gb3 isoforms/analogs in NOD/SCID/Fabry mice tissues determined by tandem mass spectrometry.

Authors:  Philippe Provençal; Michel Boutin; Shaalee Dworski; Bryan Au; Jeffrey A Medin; Christiane Auray-Blais
Journal:  Bioanalysis       Date:  2016-08-15       Impact factor: 2.681

2.  A high throughput glucocerebrosidase assay using the natural substrate glucosylceramide.

Authors:  Omid Motabar; Ehud Goldin; William Leister; Ke Liu; Noel Southall; Wenwei Huang; Juan J Marugan; Ellen Sidransky; Wei Zheng
Journal:  Anal Bioanal Chem       Date:  2011-10-28       Impact factor: 4.142

3.  Fabry disease and early stroke.

Authors:  U Feldt-Rasmussen
Journal:  Stroke Res Treat       Date:  2011-06-23

4.  The potential consequences of bidirectional promoter methylation on GLA and HNRNPH2 expression in Fabry disease phenotypes in a family of patients carrying a GLA deletion variant.

Authors:  Mohammed A Al-Obaide; Ibtisam I Al-Obaidi; Tetyana L Vasylyeva
Journal:  Biomed Rep       Date:  2022-06-24

Review 5.  Fabry nephropathy: a review - how can we optimize the management of Fabry nephropathy?

Authors:  Stephen Waldek; Sandro Feriozzi
Journal:  BMC Nephrol       Date:  2014-05-06       Impact factor: 2.388

  5 in total

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