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They constitute about 95% of adult bone cells and play important functions including in the regulation of bone remodeling, phosphate homeostasis, and mechanical stimuli sensing and response. However, the role of osteocytes in the pathogenesis of congenital diseases of abnormal bone matrix is poorly understood. This study characterized in vivo transcriptional changes in osteocytes from <jats:italic>Crtap<\/jats:italic>KO and <jats:italic>oim\/oim<\/jats:italic> mouse models of osteogenesis imperfecta (OI) compared with wild\u2010type (WT) control mice. To do this, RNA was extracted from osteocyte\u2010enriched cortical femurs and tibias, sequenced and subsequently analyzed to identify differentially expressed transcripts. These models were chosen because they mimic two types of OI with different genetic mutations that result in distinct type I collagen defects. A large number of transcripts were dysregulated in either model of OI, but 281 of them were similarly up\u2010 or downregulated in both compared with WT controls. Conversely, very few transcripts were differentially expressed between the <jats:italic>Crtap<\/jats:italic>KO and <jats:italic>oim\/oim<\/jats:italic> mice, indicating that distinct alterations in type I collagen can lead to shared pathogenic processes and similar phenotypic outcomes. Bioinformatics analyses identified several critical hubs of dysregulation that were enriched in annotation terms such as development and differentiation, ECM and collagen fibril organization, cell adhesion, signaling, regulatory processes, pattern binding, chemotaxis, and cell projections. The data further indicated alterations in important signaling pathways such as WNT and TGF\u2010\u03b2 but also highlighted new candidate genes to pursue in future studies. Overall, our study suggested that the osteocyte transcriptome is broadly dysregulated in OI with potential long\u2010term consequences at the cellular level, which deserve further investigations. \u00a9 2019 The Authors. <jats:italic>JBMR Plus<\/jats:italic> published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.<\/jats:p><\/jats:sec>","DOI":"10.1002\/jbm4.10171","type":"journal-article","created":{"date-parts":[[2019,1,29]],"date-time":"2019-01-29T03:37:12Z","timestamp":1548733032000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":36,"title":["The Osteocyte Transcriptome Is Extensively Dysregulated in Mouse Models of Osteogenesis Imperfecta"],"prefix":"10.1093","volume":"3","author":[{"given":"Sarah M","family":"Zimmerman","sequence":"first","affiliation":[{"name":"Department of Physiology and Biophysics University of Arkansas for Medical Sciences Little Rock AR 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