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The Specific Role of Dermatan Sulfate as an Instructive Glycosaminoglycan in Tissue Development
The crucial roles of dermatan sulfate (DS) have been demonstrated in tissue development of the cutis, blood vessels, and bone through construction of the extracellular matrix and cell signaling. Although DS classically exerts physiological functions via interaction with collagens, growth factors, an...
Autores principales: | , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2022
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9267682/ https://www.ncbi.nlm.nih.gov/pubmed/35806490 http://dx.doi.org/10.3390/ijms23137485 |
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author | Mizumoto, Shuji Yamada, Shuhei |
author_facet | Mizumoto, Shuji Yamada, Shuhei |
author_sort | Mizumoto, Shuji |
collection | PubMed |
description | The crucial roles of dermatan sulfate (DS) have been demonstrated in tissue development of the cutis, blood vessels, and bone through construction of the extracellular matrix and cell signaling. Although DS classically exerts physiological functions via interaction with collagens, growth factors, and heparin cofactor-II, new functions have been revealed through analyses of human genetic disorders as well as of knockout mice with loss of DS-synthesizing enzymes. Mutations in human genes encoding the epimerase and sulfotransferase responsible for the biosynthesis of DS chains cause connective tissue disorders including spondylodysplastic type Ehlers–Danlos syndrome, characterized by skin hyperextensibility, joint hypermobility, and tissue fragility. DS-deficient mice show perinatal lethality, skin fragility, vascular abnormalities, thoracic kyphosis, myopathy-related phenotypes, acceleration of nerve regeneration, and impairments in self-renewal and proliferation of neural stem cells. These findings suggest that DS is essential for tissue development in addition to the assembly of collagen fibrils in the skin, and that DS-deficient knockout mice can be utilized as models of human genetic disorders that involve impairment of DS biosynthesis. This review highlights a novel role of DS in tissue development studies from the past decade. |
format | Online Article Text |
id | pubmed-9267682 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
spelling | pubmed-92676822022-07-09 The Specific Role of Dermatan Sulfate as an Instructive Glycosaminoglycan in Tissue Development Mizumoto, Shuji Yamada, Shuhei Int J Mol Sci Review The crucial roles of dermatan sulfate (DS) have been demonstrated in tissue development of the cutis, blood vessels, and bone through construction of the extracellular matrix and cell signaling. Although DS classically exerts physiological functions via interaction with collagens, growth factors, and heparin cofactor-II, new functions have been revealed through analyses of human genetic disorders as well as of knockout mice with loss of DS-synthesizing enzymes. Mutations in human genes encoding the epimerase and sulfotransferase responsible for the biosynthesis of DS chains cause connective tissue disorders including spondylodysplastic type Ehlers–Danlos syndrome, characterized by skin hyperextensibility, joint hypermobility, and tissue fragility. DS-deficient mice show perinatal lethality, skin fragility, vascular abnormalities, thoracic kyphosis, myopathy-related phenotypes, acceleration of nerve regeneration, and impairments in self-renewal and proliferation of neural stem cells. These findings suggest that DS is essential for tissue development in addition to the assembly of collagen fibrils in the skin, and that DS-deficient knockout mice can be utilized as models of human genetic disorders that involve impairment of DS biosynthesis. This review highlights a novel role of DS in tissue development studies from the past decade. MDPI 2022-07-05 /pmc/articles/PMC9267682/ /pubmed/35806490 http://dx.doi.org/10.3390/ijms23137485 Text en © 2022 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/). |
spellingShingle | Review Mizumoto, Shuji Yamada, Shuhei The Specific Role of Dermatan Sulfate as an Instructive Glycosaminoglycan in Tissue Development |
title | The Specific Role of Dermatan Sulfate as an Instructive Glycosaminoglycan in Tissue Development |
title_full | The Specific Role of Dermatan Sulfate as an Instructive Glycosaminoglycan in Tissue Development |
title_fullStr | The Specific Role of Dermatan Sulfate as an Instructive Glycosaminoglycan in Tissue Development |
title_full_unstemmed | The Specific Role of Dermatan Sulfate as an Instructive Glycosaminoglycan in Tissue Development |
title_short | The Specific Role of Dermatan Sulfate as an Instructive Glycosaminoglycan in Tissue Development |
title_sort | specific role of dermatan sulfate as an instructive glycosaminoglycan in tissue development |
topic | Review |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9267682/ https://www.ncbi.nlm.nih.gov/pubmed/35806490 http://dx.doi.org/10.3390/ijms23137485 |
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