An Overview of in vivo Functions of Chondroitin Sulfate and Dermatan Sulfate Revealed by Their Deficient Mice

Chondroitin sulfate (CS), dermatan sulfate (DS) and heparan sulfate (HS) are covalently attached to specific core proteins to form proteoglycans in their biosynthetic pathways. They are constructed through the stepwise addition of respective monosaccharides by various glycosyltransferases and matura...

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Autores principales:	Shuji Mizumoto, Shuhei Yamada
Formato:	article
Lenguaje:	EN
Publicado:	Frontiers Media S.A. 2021
Materias:	chondroitin sulfate dermatan sulfate epimerase glycosyltransferase knockout mouse proteoglycan Biology (General) QH301-705.5
Acceso en línea:	https://doaj.org/article/ba9a7e4359fc4dcba51146d3466f6ef8
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spelling	oai:doaj.org-article:ba9a7e4359fc4dcba51146d3466f6ef82021-11-30T18:28:46ZAn Overview of in vivo Functions of Chondroitin Sulfate and Dermatan Sulfate Revealed by Their Deficient Mice2296-634X10.3389/fcell.2021.764781https://doaj.org/article/ba9a7e4359fc4dcba51146d3466f6ef82021-11-01T00:00:00Zhttps://www.frontiersin.org/articles/10.3389/fcell.2021.764781/fullhttps://doaj.org/toc/2296-634XChondroitin sulfate (CS), dermatan sulfate (DS) and heparan sulfate (HS) are covalently attached to specific core proteins to form proteoglycans in their biosynthetic pathways. They are constructed through the stepwise addition of respective monosaccharides by various glycosyltransferases and maturated by epimerases as well as sulfotransferases. Structural diversities of CS/DS and HS are essential for their various biological activities including cell signaling, cell proliferation, tissue morphogenesis, and interactions with a variety of growth factors as well as cytokines. Studies using mice deficient in enzymes responsible for the biosynthesis of the CS/DS and HS chains of proteoglycans have demonstrated their essential functions. Chondroitin synthase 1-deficient mice are viable, but exhibit chondrodysplasia, progression of the bifurcation of digits, delayed endochondral ossification, and reduced bone density. DS-epimerase 1-deficient mice show thicker collagen fibrils in the dermis and hypodermis, and spina bifida. These observations suggest that CS/DS are essential for skeletal development as well as the assembly of collagen fibrils in the skin, and that their respective knockout mice can be utilized as models for human genetic disorders with mutations in chondroitin synthase 1 and DS-epimerase 1. This review provides a comprehensive overview of mice deficient in CS/DS biosyntheses.Shuji MizumotoShuhei YamadaFrontiers Media S.A.articlechondroitin sulfatedermatan sulfateepimeraseglycosyltransferaseknockout mouseproteoglycanBiology (General)QH301-705.5ENFrontiers in Cell and Developmental Biology, Vol 9 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	chondroitin sulfate dermatan sulfate epimerase glycosyltransferase knockout mouse proteoglycan Biology (General) QH301-705.5
spellingShingle	chondroitin sulfate dermatan sulfate epimerase glycosyltransferase knockout mouse proteoglycan Biology (General) QH301-705.5 Shuji Mizumoto Shuhei Yamada An Overview of in vivo Functions of Chondroitin Sulfate and Dermatan Sulfate Revealed by Their Deficient Mice
description	Chondroitin sulfate (CS), dermatan sulfate (DS) and heparan sulfate (HS) are covalently attached to specific core proteins to form proteoglycans in their biosynthetic pathways. They are constructed through the stepwise addition of respective monosaccharides by various glycosyltransferases and maturated by epimerases as well as sulfotransferases. Structural diversities of CS/DS and HS are essential for their various biological activities including cell signaling, cell proliferation, tissue morphogenesis, and interactions with a variety of growth factors as well as cytokines. Studies using mice deficient in enzymes responsible for the biosynthesis of the CS/DS and HS chains of proteoglycans have demonstrated their essential functions. Chondroitin synthase 1-deficient mice are viable, but exhibit chondrodysplasia, progression of the bifurcation of digits, delayed endochondral ossification, and reduced bone density. DS-epimerase 1-deficient mice show thicker collagen fibrils in the dermis and hypodermis, and spina bifida. These observations suggest that CS/DS are essential for skeletal development as well as the assembly of collagen fibrils in the skin, and that their respective knockout mice can be utilized as models for human genetic disorders with mutations in chondroitin synthase 1 and DS-epimerase 1. This review provides a comprehensive overview of mice deficient in CS/DS biosyntheses.
format	article
author	Shuji Mizumoto Shuhei Yamada
author_facet	Shuji Mizumoto Shuhei Yamada
author_sort	Shuji Mizumoto
title	An Overview of in vivo Functions of Chondroitin Sulfate and Dermatan Sulfate Revealed by Their Deficient Mice
title_short	An Overview of in vivo Functions of Chondroitin Sulfate and Dermatan Sulfate Revealed by Their Deficient Mice
title_full	An Overview of in vivo Functions of Chondroitin Sulfate and Dermatan Sulfate Revealed by Their Deficient Mice
title_fullStr	An Overview of in vivo Functions of Chondroitin Sulfate and Dermatan Sulfate Revealed by Their Deficient Mice
title_full_unstemmed	An Overview of in vivo Functions of Chondroitin Sulfate and Dermatan Sulfate Revealed by Their Deficient Mice
title_sort	overview of in vivo functions of chondroitin sulfate and dermatan sulfate revealed by their deficient mice
publisher	Frontiers Media S.A.
publishDate	2021
url	https://doaj.org/article/ba9a7e4359fc4dcba51146d3466f6ef8
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An Overview of in vivo Functions of Chondroitin Sulfate and Dermatan Sulfate Revealed by Their Deficient Mice

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