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All forms of OI also feature short stature, implying an effect on endochondral ossification. Using the\n                    <jats:italic>\n                      Aga2\n                      <jats:sup>+\/\u2013<\/jats:sup>\n                    <\/jats:italic>\n                    mouse, which has a mutation in type I collagen, we show an affected growth plate primarily due to a shortened proliferative zone. We used single-cell RNA-Seq analysis of tibial and femoral growth plate tissues to understand transcriptional consequences on growth plate cell types. We show that perichondrial cells, which express abundant type I procollagen, and growth plate chondrocytes, which were found to express low amounts of type I procollagen, had ER stress and dysregulation of the same unfolded protein response pathway as previously demonstrated in osteoblasts.\n                    <jats:italic>\n                      Aga2\n                      <jats:sup>+\/\u2013<\/jats:sup>\n                    <\/jats:italic>\n                    proliferating chondrocytes showed increased FGF and MAPK signaling, findings consistent with accelerated differentiation. There was also increased\n                    <jats:italic>Sox9<\/jats:italic>\n                    expression throughout the growth plate, which is expected to accelerate early chondrocyte differentiation but reduce late hypertrophic differentiation. These data reveal that mutant type I collagen expression in OI has an impact on the cartilage growth plate. These effects on endochondral ossification indicate that OI is a biologically complex phenotype going beyond its known impacts on bone to negatively affect linear growth.\n                  <\/jats:p>","DOI":"10.1172\/jci.insight.171984","type":"journal-article","created":{"date-parts":[[2023,10,5]],"date-time":"2023-10-05T12:00:29Z","timestamp":1696507229000},"source":"Crossref","is-referenced-by-count":11,"title":["Altered Sox9 and FGF signaling gene expression in Aga2 OI mice negatively affects linear growth"],"prefix":"10.1172","volume":"8","author":[{"given":"Jennifer","family":"Zieba","sequence":"first","affiliation":[{"name":"Department of Orthopaedic Surgery, David Geffen School of Medicine at University of California, Los Angeles, Los Angeles, California, USA."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lisette","family":"Nevarez","sequence":"additional","affiliation":[{"name":"Department of Molecular Cell and Developmental Biology, University of 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