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dc.contributor.author
Rozenfeld, Paula Adriana  
dc.contributor.author
Neumann, Pablo M.  
dc.date.available
2023-05-04T12:23:29Z  
dc.date.issued
2011-01  
dc.identifier.citation
Rozenfeld, Paula Adriana; Neumann, Pablo M.; Treatment of fabry disease: Current and emerging strategies; Bentham Science Publishers; Current Pharmaceutical Biotechnology; 12; 6; 1-2011; 916-922  
dc.identifier.issn
1389-2010  
dc.identifier.uri
http://hdl.handle.net/11336/196240  
dc.description.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.  
dc.format
application/pdf  
dc.language.iso
eng  
dc.publisher
Bentham Science Publishers  
dc.rights
info:eu-repo/semantics/openAccess  
dc.rights.uri
https://creativecommons.org/licenses/by-nc-sa/2.5/ar/  
dc.subject
AGALSIDASE ALFA  
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ALPHA-GALACTOSIDASE A  
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CHAPERONE TREATMENT  
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ENZYME REPLACEMENT THERAPY  
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FABRY DISEASE  
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GENE THERAPY  
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Genética Humana  
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Medicina Básica  
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CIENCIAS MÉDICAS Y DE LA SALUD  
dc.title
Treatment of fabry disease: Current and emerging strategies  
dc.type
info:eu-repo/semantics/article  
dc.type
info:ar-repo/semantics/artículo  
dc.type
info:eu-repo/semantics/publishedVersion  
dc.date.updated
2023-04-10T10:16:17Z  
dc.journal.volume
12  
dc.journal.number
6  
dc.journal.pagination
916-922  
dc.journal.pais
Estados Unidos  
dc.description.fil
Fil: Rozenfeld, Paula Adriana. Universidad Nacional de La Plata. Facultad de Ciencias Exactas. Departamento de Ciencias Biológicas. Cátedra de Inmunología; Argentina. Consejo Nacional de Investigaciones Científicas y Técnicas. Centro Científico Tecnológico Conicet - La Plata; Argentina  
dc.description.fil
Fil: Neumann, Pablo M.. Instituto Universitario del Hospital Italiano de Buenos Aires; Argentina  
dc.journal.title
Current Pharmaceutical Biotechnology  
dc.relation.alternativeid
info:eu-repo/semantics/altIdentifier/url/https://www.eurekaselect.com/article/18967  
dc.relation.alternativeid
info:eu-repo/semantics/altIdentifier/doi/http://dx.doi.org/10.2174/138920111795542705