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)
Please use this url to cite or link to this publication:
https://lup.lub.lu.se/record/2519539
- author
- Bartolini, Barbara
LU
; Thelin, Martin
LU
; Rauch, Uwe
LU
; Feinstein, Ricardo
; Oldberg, Åke
LU
; Malmström, Anders
LU
and Maccarana, Marco
LU
- organization
- publishing date
- 2012
- 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
- pmid:22496542
- 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
- 2016-04-04 09:04:02
- date last changed
- 2022-04-23 18:47:39
@article{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 = {{Oxford University Press}},
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}},
doi = {{10.1093/glycob/cws065}},
volume = {{22}},
year = {{2012}},
}