{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,25]],"date-time":"2026-08-25T19:05:24Z","timestamp":1787684724919,"version":"build-2784847793"},"reference-count":64,"publisher":"American Society for Clinical Investigation","issue":"1","license":[{"start":{"date-parts":[[2025,1,2]],"date-time":"2025-01-02T00:00:00Z","timestamp":1735776000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,1,2]]},"abstract":"<jats:p>\n                    ATP-dependent chromatin remodeling protein ATRX is an essential regulator involved in maintenance of DNA structure and chromatin state and regulation of gene expression during development.\n                    <jats:italic>ATRX<\/jats:italic>\n                    was originally identified as the monogenic cause of X-linked \u03b1-thalassemia mental retardation (ATR-X) syndrome. Affected individuals display a variety of developmental abnormalities and skeletal deformities. Studies from others investigated the role of ATRX in skeletal development by tissue-specific\n                    <jats:italic>Atrx<\/jats:italic>\n                    knockout. However, the impact of ATRX during early skeletal development has not been examined. Using preosteoblast-specific\n                    <jats:italic>Atrx<\/jats:italic>\n                    conditional knockout mice, we observed increased trabecular bone mass and decreased osteoclast number in bone. In vitro coculture of\n                    <jats:italic>Atrx<\/jats:italic>\n                    conditional knockout bone marrow stromal cells (BMSCs) with WT splenocytes showed impaired osteoclast differentiation. Additionally,\n                    <jats:italic>Atrx<\/jats:italic>\n                    deletion was associated with decreased receptor activator of nuclear factor \u03ba-B ligand (\n                    <jats:italic>Rankl<\/jats:italic>\n                    )\/ osteoprotegerin (\n                    <jats:italic>Opg<\/jats:italic>\n                    ) expression ratio in BMSCs. Notably,\n                    <jats:italic>Atrx<\/jats:italic>\n                    -deficient osteolineage cells expressed high levels of the neuropeptide cocaine- and amphetamine-regulated transcript prepropeptide (\n                    <jats:italic>Cartpt<\/jats:italic>\n                    ). Mechanistically, ATRX suppresses\n                    <jats:italic>Cartpt<\/jats:italic>\n                    transcription by binding to the promoter, which is otherwise poised for\n                    <jats:italic>Cartpt<\/jats:italic>\n                    expression by RUNX2 binding to the distal enhancer. Finally,\n                    <jats:italic>Cartpt<\/jats:italic>\n                    silencing in\n                    <jats:italic>Atrx<\/jats:italic>\n                    conditional knockout BMSCs rescued the molecular phenotype by increasing the\n                    <jats:italic>Rankl<\/jats:italic>\n                    \/\n                    <jats:italic>Opg<\/jats:italic>\n                    expression ratio. Together, our data show a potent repressor function of ATRX in restricting\n                    <jats:italic>Cartpt<\/jats:italic>\n                    expression during skeletal development.\n                  <\/jats:p>","DOI":"10.1172\/jci163587","type":"journal-article","created":{"date-parts":[[2025,1,1]],"date-time":"2025-01-01T14:00:42Z","timestamp":1735740042000},"source":"Crossref","is-referenced-by-count":3,"title":["ATRX silences Cartpt expression in osteoblastic cells during skeletal development"],"prefix":"10.1172","volume":"135","author":[{"given":"Yi-Ting","family":"Chen","sequence":"first","affiliation":[{"name":"Integrative Molecular and Biomedical Sciences Program and"},{"name":"Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ming-Ming","family":"Jiang","sequence":"additional","affiliation":[{"name":"Department of Molecular and Human Genetics, Baylor College 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