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Mouse development is not obviously affected by the absence of dermatan sulfate epimerase 2 in spite of a modified brain dermatan sulfate composition.

Bartolini, Barbara LU ; Thelin, Martin LU ; Rauch, Uwe LU ; Feinstein, Ricardo; Oldberg, Åke LU ; Malmström, Anders LU and Maccarana, Marco LU (2012) In Glycobiology 22(7). p.1007-1016
Abstract
Dermatan sulfate epimerase 2 (DS-epi2), together with its homologue DS-epi1, transform glucuronic acid into iduronic acid in dermatan sulfate polysaccharide chains. Iduronic acid gives dermatan sulfate increased chain flexibility and promotes protein binding. DS-epi2 is ubiquitously expressed and is the predominant epimerase in brain. Here we report the generation and initial characterization of DS-epi2 null mice. DS-epi2 deficient mice showed no anatomical, histological or morphological abnormalities. The body weights and lengths of mutated and wild-type littermates were indistinguishable. They were fertile and had a normal lifespan. Chondroitin/dermatan sulfate (CS/DS) isolated from newborn mutated mouse brains had a 38% reduction in... (More)
Dermatan sulfate epimerase 2 (DS-epi2), together with its homologue DS-epi1, transform glucuronic acid into iduronic acid in dermatan sulfate polysaccharide chains. Iduronic acid gives dermatan sulfate increased chain flexibility and promotes protein binding. DS-epi2 is ubiquitously expressed and is the predominant epimerase in brain. Here we report the generation and initial characterization of DS-epi2 null mice. DS-epi2 deficient mice showed no anatomical, histological or morphological abnormalities. The body weights and lengths of mutated and wild-type littermates were indistinguishable. They were fertile and had a normal lifespan. Chondroitin/dermatan sulfate (CS/DS) isolated from newborn mutated mouse brains had a 38% reduction in iduronic acid compared to wild type littermates and compositional analysis revealed a decrease of 4-O-sulfate and an increase of 6-O-sulfate containing structures. Despite the reduction in iduronic acid, adult DS-epi2-/- brain showed normal extracellular matrix features by immunohistological stainings. We conclude that DS-epi1 compensates in vivo for the loss of DS-epi2.. These results extend previous findings of functional redundancy of brain extracellular matrix components. (Less)
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author
organization
publishing date
type
Contribution to journal
publication status
published
subject
in
Glycobiology
volume
22
issue
7
pages
1007 - 1016
publisher
Oxford University Press
external identifiers
  • WOS:000304195500012
  • PMID:22496542
  • Scopus:84861416926
ISSN
1460-2423
DOI
10.1093/glycob/cws065
language
English
LU publication?
yes
id
96afb899-0c7c-4980-889c-fee49a2679b6 (old id 2519539)
alternative location
http://www.ncbi.nlm.nih.gov/pubmed/22496542?dopt=Abstract
date added to LUP
2012-05-06 16:48:36
date last changed
2016-10-13 04:23:17
@misc{96afb899-0c7c-4980-889c-fee49a2679b6,
  abstract     = {Dermatan sulfate epimerase 2 (DS-epi2), together with its homologue DS-epi1, transform glucuronic acid into iduronic acid in dermatan sulfate polysaccharide chains. Iduronic acid gives dermatan sulfate increased chain flexibility and promotes protein binding. DS-epi2 is ubiquitously expressed and is the predominant epimerase in brain. Here we report the generation and initial characterization of DS-epi2 null mice. DS-epi2 deficient mice showed no anatomical, histological or morphological abnormalities. The body weights and lengths of mutated and wild-type littermates were indistinguishable. They were fertile and had a normal lifespan. Chondroitin/dermatan sulfate (CS/DS) isolated from newborn mutated mouse brains had a 38% reduction in iduronic acid compared to wild type littermates and compositional analysis revealed a decrease of 4-O-sulfate and an increase of 6-O-sulfate containing structures. Despite the reduction in iduronic acid, adult DS-epi2-/- brain showed normal extracellular matrix features by immunohistological stainings. We conclude that DS-epi1 compensates in vivo for the loss of DS-epi2.. These results extend previous findings of functional redundancy of brain extracellular matrix components.},
  author       = {Bartolini, Barbara and Thelin, Martin and Rauch, Uwe and Feinstein, Ricardo and Oldberg, Åke and Malmström, Anders and Maccarana, Marco},
  issn         = {1460-2423},
  language     = {eng},
  number       = {7},
  pages        = {1007--1016},
  publisher    = {ARRAY(0x79d3950)},
  series       = {Glycobiology},
  title        = {Mouse development is not obviously affected by the absence of dermatan sulfate epimerase 2 in spite of a modified brain dermatan sulfate composition.},
  url          = {http://dx.doi.org/10.1093/glycob/cws065},
  volume       = {22},
  year         = {2012},
}