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Musculocontractural Ehlers\u2013Danlos syndrome (mcEDS) caused by pathogenic variants in the gene that encodes carbohydrate sulfotransferase 14 (CHST14) (mcEDS-CHST14) is a representative disorder of DS deficiency. Skeletal lesions, such as progressive spinal deformity and osteoporosis\/osteopenia, are significant manifestations of mcEDS-CHST14, which can lead to deterioration in quality of life. To characterize skeletal alterations associated with DS deficiency, we performed comprehensive analyses of the skeletal phenotypes in Chst14 gene-deleted (Chst14\u2212\/\u2212) mice. Chst14\u2212\/\u2212 mice exhibited progressive kyphosis, proximal femoral deformities, and decreased bone strength, as well as reduced trabecular bone mass and structural parameters, from a young age. Qualitative ultrastructural alterations of collagen fibrils in cortical bone were observed in middle-aged Chst14\u2212\/\u2212 mice by transmission electron microscopy. In addition, cortical bone showed increased expression of receptor activator of NF-\u03baB (Rank), a gene involved in osteoclast differentiation, in middle age. Histomorphometric analyses of cancellous bone demonstrated reduced trabecular structural parameters in Chst14\u2212\/\u2212 mice, including decreases in bone volume and trabecular number. These findings indicate age-dependent alterations in both cortical and cancellous bone compartments that may contribute to progressive skeletal deformation. The present study provides insight into skeletal alterations associated with mcEDS-CHST14 and highlights a broader role of DS in maintaining bone structure and strength.<\/jats:p>","DOI":"10.1093\/glycob\/cwag037","type":"journal-article","created":{"date-parts":[[2026,5,13]],"date-time":"2026-05-13T12:02:39Z","timestamp":1778673759000},"source":"Crossref","is-referenced-by-count":0,"title":["Comprehensive analysis of the skeletal phenotype in\n                    <i>Chst14<\/i>\n                    <i>\u2212\/\u2212<\/i>\n                    mice: implications for dermatan sulfate in bone structure and strength"],"prefix":"10.1093","volume":"36","author":[{"ORCID":"https:\/\/orcid.org\/0009-0009-0180-4979","authenticated-orcid":false,"given":"Yuki","family":"Takahashi","sequence":"first","affiliation":[{"name":"Department of Medical Genetics, Shinshu University School of Medicine , 3-1-1 Asahi, Matsumoto, Nagano 390-8621 ,","place":["Japan"]},{"name":"Division of Clinical Sequencing, Shinshu University School of Medicine , 3-1-1 Asahi, Matsumoto, Nagano 390-8621 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9938-6569","authenticated-orcid":false,"given":"Takahiro","family":"Yoshizawa","sequence":"additional","affiliation":[{"name":"Division of Animal Research, Research Center for Advanced Science and Technology, Shinshu University , 3-1-1 Asahi, Matsumoto, Nagano 390-8621 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4641-1505","authenticated-orcid":false,"given":"Shuji","family":"Mizumoto","sequence":"additional","affiliation":[{"name":"Department of Pathobiochemistry, Faculty of Pharmacy, Meijo University , 150 Yagotoyama, Tempaku-ku, Nagoya, Aichi 468-8503 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Medicine, Rakuno Gakuen University , 582, Bunkyodai-Midorimachi, Ebetsu, Hokkaido 069-8501 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2251-9383","authenticated-orcid":false,"given":"Shin","family":"Shimada","sequence":"additional","affiliation":[{"name":"Division of Animal Research, Research Center for Advanced Science and Technology, Shinshu University , 3-1-1 Asahi, Matsumoto, Nagano 390-8621 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7119-5894","authenticated-orcid":false,"given":"Yuko","family":"Nitahara-Kasahara","sequence":"additional","affiliation":[{"name":"Division of Molecular and Medical Genetics, Center for Gene and Cell Therapy, The Institute of Medical Science, The University of Tokyo , 4-6-1 Shirokanedai, Minato-city, Tokyo 108-8639 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Center for Gene and Cell Therapy, The Institute of Medical Science, The University of Tokyo , 4-6-1 Shirokanedai, Minato-city, Tokyo 108-8639 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8344-7507","authenticated-orcid":false,"given":"Tomoki","family":"Kosho","sequence":"additional","affiliation":[{"name":"Department of Medical Genetics, Shinshu University School of Medicine , 3-1-1 Asahi, Matsumoto, Nagano 390-8621 ,","place":["Japan"]},{"name":"Division of Clinical Sequencing, Shinshu University School of Medicine , 3-1-1 Asahi, Matsumoto, Nagano 390-8621 ,","place":["Japan"]},{"name":"Center for Medical Genetics, Shinshu University Hospital , 3-1-1 Asahi, Matsumoto, Nagano 390-8621 ,","place":["Japan"]},{"name":"Research Center for Advanced Science and Technology, 3-1-1 Asahi, Shinshu University , Matsumoto, Nagano 390-8621 ,","place":["Japan"]},{"name":"BioBank Shinshu, Shinshu University Hospital , 3-1-1 Asahi, 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