Analysis of male reproductive parameters in a murine model of mucopolysaccharidosis type I (MPS I)

Analysis of male reproductive parameters in a murine model of mucopolysaccharidosis type I (MPS I)

Author Nascimento, Cinthia Castro do Autor UNIFESP Google Scholar
Aguiar Junior, Odair Autor UNIFESP Google Scholar
D'Almeida, Vania Autor UNIFESP Google Scholar
Institution Universidade Federal de São Paulo (UNIFESP)
Abstract Mucopolysaccharidosis (MPS) I is a lysosomal storage disorder (LSD) that is characterised by alpha-L-idu-ronidase (Idua) deficiency and continuous deposition of glycosaminoglycans (GAGs), which consequently interferes with cell signalling mechanisms and results in multisystemic and progressive symptoms. The animal model of MPS I (Idua-/-) has been widely studied to elucidate the consequences and progression of the disorder; however, studies specifically assessing the male reproductive tract are lacking. The aim of this study was to evaluate some of the reproductive characteristics of male MPS I mice in two phases of life. Reproductive organ biometry, sperm counts, sperm morphological evaluation, plasma testosterone measurements and histopathological, histomorphometrical and immunohistochemical analysis were performed in 3- and 6-month-old C57BL/6 Idua+/+ and Idua-/- mice. Seminal vesicle weights were decreased in both the 3- and 6-month-old Idua-/- mice. Decrease in sperm counts and the majority of the histopathological signs were observed in the 6-month-old Idua-/- mice. No differences were detected in the sperm morphological analysis. Immunohistochemistry revealed that seminiferous tubules from 3-month-old Idua-/- mice were more intensely stained with anti-caspase-3 than 3-month-old Idua+/+ mice, but no difference was found at 6 months. These results suggest that MPS I interferes with male reproductive parameters both in 3 and 6-month-old animals and histopathological signs are more pronounced in 6-month-old mice, indicating that the effects of the disorder may intensify with the disease progression.
Keywords Knockout mice
mucopolysaccharidosis
spermatogenesis
testis
Language English
Sponsor Coordenação de Aperfeiçoamento de Pessoal de Nível Superior (CAPES)
AFIP
Fundação de Amparo à Pesquisa do Estado de São Paulo (FAPESP)
Conselho Nacional de Desenvolvimento Científico e Tecnológico (CNPq)
Date 2014-01-01
Published in International Journal Of Clinical And Experimental Pathology. Madison: E-century Publishing Corp, v. 7, n. 6, p. 3488-3497, 2014.
ISSN 1936-2625 (Sherpa/Romeo, impact factor)
Publisher E-century Publishing Corp
Extent 3488-3497
Origin https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4097293/
Access rights Open access Open Access
Type Article
Web of Science ID WOS:000338770500089
URI http://repositorio.unifesp.br/11600/44034

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