{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,19]],"date-time":"2026-08-19T20:02:42Z","timestamp":1787169762563,"version":"build-2736575974"},"reference-count":52,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2023,1,19]],"date-time":"2023-01-19T00:00:00Z","timestamp":1674086400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["anatomypubs.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Developmental Dynamics"],"published-print":{"date-parts":[[2023,5]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>\n                      The gene encoding the transcription factor, Grainyhead\u2010like 3 (\n                      <jats:italic>Grhl3<\/jats:italic>\n                      ), plays critical roles in mammalian development and homeostasis.\n                      <jats:italic>Grhl3<\/jats:italic>\n                      \u2010null embryos exhibit thoraco\u2010lumbo\u2010sacral spina bifida and soft\u2010tissue syndactyly. Additional studies reveal that these embryos also exhibit an epidermal proliferation\/differentiation imbalance. This manifests as skin barrier defects resulting in peri\u2010natal lethality and defective wound repair. Despite these extensive analyses of\n                      <jats:italic>Grhl3<\/jats:italic>\n                      loss\u2010of\u2010function models, the consequences of gain\u2010of\u2010function of this gene have been difficult to achieve.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      In this study, we generated a novel mouse model that expresses\n                      <jats:italic>Grhl3<\/jats:italic>\n                      from a transgene integrated in the\n                      <jats:italic>Rosa26<\/jats:italic>\n                      locus on an endogenous\n                      <jats:italic>Grhl3<\/jats:italic>\n                      \u2010null background. Expression of the transgene rescues both the neurulation and skin barrier defects of the knockout mice, allowing survival into adulthood. Despite this, the mice are not normal, exhibiting a range of phenotypes attributable to dysregulated\n                      <jats:italic>Grhl3<\/jats:italic>\n                      expression. In mice homozygous for the transgene, we observe a severe Shaker\u2010Waltzer phenotype associated with hearing impairment. Micro\u2010CT scanning of the inner ear revealed profound structural alterations underlying these phenotypes. In addition, these mice exhibit other developmental anomalies including hair loss, digit defects, and epidermal dysmorphogenesis.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>\n                      Taken together, these findings indicate that diverse developmental processes display low tolerance to dysregulation of\n                      <jats:italic>Grhl3<\/jats:italic>\n                      .\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/dvdy.565","type":"journal-article","created":{"date-parts":[[2023,1,6]],"date-time":"2023-01-06T04:19:10Z","timestamp":1672978750000},"page":"647-667","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Dysregulation of Grainyhead\u2010like 3 expression causes widespread developmental 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