{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,2]],"date-time":"2026-06-02T15:04:11Z","timestamp":1780412651419,"version":"3.54.1"},"reference-count":48,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2023,3,29]],"date-time":"2023-03-29T00:00:00Z","timestamp":1680048000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["#82071103,#81570958"],"award-info":[{"award-number":["#82071103,#81570958"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100004826","name":"Natural Science Foundation of Beijing Municipality","doi-asserted-by":"publisher","award":["#7162075"],"award-info":[{"award-number":["#7162075"]}],"id":[{"id":"10.13039\/501100004826","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Oral Diseases"],"published-print":{"date-parts":[[2024,4]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Objectives<\/jats:title>\n                    <jats:p>\n                      Gnathodiaphyseal dysplasia (GDD; OMIM#166260) is a rare skeletal genetic disorder characterized by sclerosis of tubular bones and cemento\u2010osseous lesions in mandibles.\n                      <jats:italic>TMEM16E\/ANO5<\/jats:italic>\n                      gene mutations have been identified in patients with GDD. Here,\n                      <jats:italic>Ano5<\/jats:italic>\n                      knockout (\n                      <jats:italic>Ano5<\/jats:italic>\n                      <jats:sup>\n                        <jats:italic>\u2212\/\u2212<\/jats:italic>\n                      <\/jats:sup>\n                      ) mice with enhanced osteoblastogenesis were used to investigate whether\n                      <jats:italic>Ano5<\/jats:italic>\n                      disruption affects osteoclastogenesis.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Subjects and Methods<\/jats:title>\n                    <jats:p>The maturation of osteoclasts, formation of F\u2010actin ring and bone resorption were detected by immunohistochemistry, TRAP, phalloidin staining and Coming Osteo assays. The expression of osteoclast\u2010related factors was measured by qRT\u2010PCR. Early signaling pathways were verified by western blot.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      <jats:italic>Ano5<\/jats:italic>\n                      <jats:sup>\n                        <jats:italic>\u2212\/\u2212<\/jats:italic>\n                      <\/jats:sup>\n                      mice exhibited inhibitory formation of multinucleated osteoclasts with a reduction of TRAP activity. The expression of\n                      <jats:italic>Nfatc1<\/jats:italic>\n                      ,\n                      <jats:italic>c\u2010Fos<\/jats:italic>\n                      ,\n                      <jats:italic>Trap<\/jats:italic>\n                      ,\n                      <jats:italic>Ctsk<\/jats:italic>\n                      ,\n                      <jats:italic>Mmp9<\/jats:italic>\n                      ,\n                      <jats:italic>Rank<\/jats:italic>\n                      and\n                      <jats:italic>Dc\u2010stamp<\/jats:italic>\n                      was significantly decreased in bone tissues and bone marrow\u2010derived macrophages (BMMs) of\n                      <jats:italic>Ano5<\/jats:italic>\n                      <jats:sup>\n                        <jats:italic>\u2212\/\u2212<\/jats:italic>\n                      <\/jats:sup>\n                      mice.\n                      <jats:italic>Ano5<\/jats:italic>\n                      <jats:sup>\n                        <jats:italic>\u2212\/\u2212<\/jats:italic>\n                      <\/jats:sup>\n                      osteoclasts manifested disrupted actin ring and less mineral resorption. RANKL\u2010induced early signaling pathways were suppressed in\n                      <jats:italic>Ano5<\/jats:italic>\n                      <jats:sup>\n                        <jats:italic>\u2212\/\u2212<\/jats:italic>\n                      <\/jats:sup>\n                      osteoclasts and\n                      <jats:italic>Ano5<\/jats:italic>\n                      knockdown RAW264.7 cells. Moreover, the inhibitory effects of NF\u2010\u03baB signalling pathway on osteoclastogenesis were partially attenuated with NF\u2010\u03baB signalling activator.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      <jats:italic>Ano5<\/jats:italic>\n                      deficiency impairs osteoclastogenesis, which leads to enhanced osteogenic phenotypes mediated by bone homeostasis dysregulation.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/odi.14562","type":"journal-article","created":{"date-parts":[[2023,3,17]],"date-time":"2023-03-17T22:18:37Z","timestamp":1679091517000},"page":"1403-1415","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Genetic disruption of\n                    <i>Ano5<\/i>\n                    leads to impaired osteoclastogenesis for gnathodiaphyseal dysplasia"],"prefix":"10.1111","volume":"30","author":[{"given":"Xiu","family":"Liu","sequence":"first","affiliation":[{"name":"Beijing Institute of Dental Research, Beijing Stomatological Hospital Capital Medical University  Beijing 100050 China"},{"name":"Department of Oral Medicine, Beijing Stomatological Hospital Capital Medical University  Beijing 100050 China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaoyu","family":"Wang","sequence":"additional","affiliation":[{"name":"Beijing Institute of Dental Research, Beijing Stomatological Hospital Capital Medical University  Beijing 100050 China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xinrong","family":"Ma","sequence":"additional","affiliation":[{"name":"Beijing Institute of Dental Research, Beijing Stomatological Hospital Capital Medical University  Beijing 100050 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