Genomic instability in blood cells from murine model of mucopolysaccharidosis type I

Genomic instability in blood cells from murine model of mucopolysaccharidosis type I

Author Noguti, Juliana Autor UNIFESP Google Scholar
Pereira, Vanessa Goncalves Autor UNIFESP Google Scholar
Martins, Ana Maria Autor UNIFESP Google Scholar
D'Almeida, Vania Autor UNIFESP Google Scholar
Ribeiro, Daniel Araki Autor UNIFESP Google Scholar
Institution Universidade Federal de São Paulo (UNIFESP)
Abstract Mucopolysaccharidosis type I (MPS I) is caused by a deficiency of alfa-iduronidase (IDUA), which leads to intralysosomal accumulation of glycosaminoglycans. Some studies have revealed that oxidative stress plays an important role in MPS I. However, the mechanisms by which these alterations occur are still not fully understood. the aim of this study was to analyze genomic instability in blood cells from murine model of MPS I by single cell gel (comet) assay and micronucleus test. the results pointed out genetic damage in blood cells as depicted by the single cell gel (comet) assay results. By contrast, no increase of micronucleated cells were found in mouse blood cells when compared to negative control. Taken together, our results suggest that IDUA deficiency induces genomic damage in blood cells. Certainly, this finding offers new insights into the mechanisms underlying the relation between IDUA deficiency and clinical manifestations that can occur in MPS I patients.
Keywords Mucopolysaccharidosis
Blood cells
Genomic damage
Language English
Sponsor Coordenação de Aperfeiçoamento de Pessoal de Nível Superior (CAPES)
Fundação de Amparo à Pesquisa do Estado de São Paulo (FAPESP)
AFIP
Conselho Nacional de Desenvolvimento Científico e Tecnológico (CNPq)
Grant number CNPq: 501343/2010-5
Date 2011-12-01
Published in Journal of Molecular Histology. Dordrecht: Springer, v. 42, n. 6, p. 575-578, 2011.
ISSN 1567-2379 (Sherpa/Romeo, impact factor)
Publisher Springer
Extent 575-578
Origin http://dx.doi.org/10.1007/s10735-011-9361-3
Access rights Closed access
Type Article
Web of Science ID WOS:000300371400010
URI http://repositorio.unifesp.br/handle/11600/34277

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