{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,28]],"date-time":"2026-09-28T22:56:48Z","timestamp":1790636208848,"version":"4.1.0"},"reference-count":47,"publisher":"eLife Sciences Publications, Ltd","license":[{"start":{"date-parts":[[2021,5,26]],"date-time":"2021-05-26T00:00:00Z","timestamp":1621987200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2021,5,26]],"date-time":"2021-05-26T00:00:00Z","timestamp":1621987200000},"content-version":"am","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2021,5,26]],"date-time":"2021-05-26T00:00:00Z","timestamp":1621987200000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100009633","name":"Eunice Kennedy Shriver National Institute of Child Health and Human Development","doi-asserted-by":"publisher","award":["HD024064"],"award-info":[{"award-number":["HD024064"]}],"id":[{"id":"10.13039\/100009633","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100007856","name":"Baylor College of Medicine","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100007856","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000069","name":"National Institute of Arthritis and Musculoskeletal and Skin Diseases","doi-asserted-by":"publisher","award":["AR373318"],"award-info":[{"award-number":["AR373318"]}],"id":[{"id":"10.13039\/100000069","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000071","name":"National Institute of Child Health and Human Development","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000071","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["elifesciences.org"],"crossmark-restriction":false},"short-container-title":[],"abstract":"<jats:p>\n                    Osteogenesis imperfecta (OI) is characterized by short stature, skeletal deformities, low bone mass, and motor deficits. A subset of OI patients also present with joint hypermobility; however, the role of tendon dysfunction in OI pathogenesis is largely unknown. Using the\n                    <jats:italic>\n                      Crtap\n                      <jats:sup>-\/-<\/jats:sup>\n                    <\/jats:italic>\n                    mouse model of severe, recessive OI, we found that mutant Achilles and patellar tendons were thinner and weaker with increased collagen cross-links and reduced collagen fibril size at 1- and 4-months compared to wildtype. Patellar tendons from\n                    <jats:italic>\n                      Crtap\n                      <jats:sup>-\/-<\/jats:sup>\n                    <\/jats:italic>\n                    mice also had altered numbers of CD146\n                    <jats:sup>+<\/jats:sup>\n                    CD200\n                    <jats:sup>+<\/jats:sup>\n                    and CD146\n                    <jats:sup>-<\/jats:sup>\n                    CD200\n                    <jats:sup>+<\/jats:sup>\n                    progenitor-like cells at skeletal maturity. RNA-seq analysis of Achilles and patellar tendons from 1-month\n                    <jats:italic>\n                      Crtap\n                      <jats:sup>-\/-<\/jats:sup>\n                    <\/jats:italic>\n                    mice revealed dysregulation in matrix and tendon marker gene expression concomitant with predicted alterations in TGF-\u03b2, inflammatory, and metabolic signaling. At 4-months,\n                    <jats:italic>\n                      Crtap\n                      <jats:sup>-\/-<\/jats:sup>\n                    <\/jats:italic>\n                    mice showed increased \u03b1SMA, MMP2, and phospho-NF\u03baB staining in the patellar tendon consistent with excess matrix remodeling and tissue inflammation. Finally, a series of behavioral tests showed severe motor impairments and reduced grip strength in 4-month\n                    <jats:italic>\n                      Crtap\n                      <jats:sup>-\/-<\/jats:sup>\n                    <\/jats:italic>\n                    mice \u2013 a phenotype that correlates with the tendon pathology.\n                  <\/jats:p>","DOI":"10.7554\/elife.63488","type":"journal-article","created":{"date-parts":[[2021,5,26]],"date-time":"2021-05-26T08:00:29Z","timestamp":1622016029000},"update-policy":"https:\/\/doi.org\/10.7554\/elife.63488","source":"Crossref","is-referenced-by-count":19,"title":["Tendon and motor phenotypes in the Crtap-\/- mouse model of recessive osteogenesis imperfecta"],"prefix":"10.7554","volume":"10","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6514-9066","authenticated-orcid":true,"given":"Matthew William","family":"Grol","sequence":"first","affiliation":[{"name":"Department of Molecular and Human Genetics, Baylor 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