{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,17]],"date-time":"2026-07-17T00:09:38Z","timestamp":1784246978707,"version":"3.55.0"},"reference-count":70,"publisher":"The Company of Biologists","issue":"17","license":[{"start":{"date-parts":[[2021,9,1]],"date-time":"2021-09-01T00:00:00Z","timestamp":1630454400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/www.biologists.com\/user-licence-1-1\/"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["DE022065"],"award-info":[{"award-number":["DE022065"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["DE026434"],"award-info":[{"award-number":["DE026434"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,9,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Branchio-oto-renal syndrome (BOR) is a disorder characterized by hearing loss, and craniofacial and\/or renal defects. Variants in the transcription factor Six1 and its co-factor Eya1, both of which are required for otic development, are linked to BOR. We previously identified Sobp as a potential Six1 co-factor, and SOBP variants in mouse and humans cause otic phenotypes; therefore, we asked whether Sobp interacts with Six1 and thereby may contribute to BOR. Co-immunoprecipitation and immunofluorescence experiments demonstrate that Sobp binds to and colocalizes with Six1 in the cell nucleus. Luciferase assays show that Sobp interferes with the transcriptional activation of Six1+Eya1 target genes. Experiments in Xenopus embryos that either knock down or increase expression of Sobp show that it is required for formation of ectodermal domains at neural plate stages. In addition, altering Sobp levels disrupts otic vesicle development and causes craniofacial cartilage defects. Expression of Xenopus Sobp containing the human variant disrupts the pre-placodal ectoderm similar to full-length Sobp, but other changes are distinct. These results indicate that Sobp modifies Six1 function and is required for vertebrate craniofacial development, and identify Sobp as a potential candidate gene for BOR.<\/jats:p>","DOI":"10.1242\/dev.199684","type":"journal-article","created":{"date-parts":[[2021,8,20]],"date-time":"2021-08-20T03:45:13Z","timestamp":1629431113000},"source":"Crossref","is-referenced-by-count":20,"title":["Sobp modulates the transcriptional activation of Six1 target genes and is required during craniofacial development"],"prefix":"10.1242","volume":"148","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-9893-2305","authenticated-orcid":false,"given":"Andre L. P.","family":"Tavares","sequence":"first","affiliation":[{"name":"Department of Anatomy and Cell Biology, George Washington University School of Medicine and Health Sciences, Washington DC, DC 20037, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6987-0560","authenticated-orcid":false,"given":"Karyn","family":"Jourdeuil","sequence":"additional","affiliation":[{"name":"Department of Anatomy and Cell Biology, George Washington University School of Medicine and Health Sciences, Washington DC, DC 20037, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8085-9242","authenticated-orcid":false,"given":"Karen M.","family":"Neilson","sequence":"additional","affiliation":[{"name":"Department of Anatomy and Cell Biology, George Washington University School of Medicine and Health Sciences, Washington DC, DC 20037, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Himani D.","family":"Majumdar","sequence":"additional","affiliation":[{"name":"Department of Anatomy and Cell Biology, George Washington University School of Medicine and Health Sciences, Washington DC, DC 20037, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4192-1087","authenticated-orcid":false,"given":"Sally A.","family":"Moody","sequence":"additional","affiliation":[{"name":"Department of Anatomy and Cell Biology, George Washington University School of Medicine and Health Sciences, Washington DC, DC 20037, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2021,9,6]]},"reference":[{"key":"2021090609554008900_DEV199684C1","doi-asserted-by":"publisher","first-page":"377","DOI":"10.1016\/j.devcel.2011.12.006","article-title":"Eya1-Six1 interaction is sufficient to induce hair cell fate in the cochlea by activating Atoh1 expression in cooperation with Sox2","volume":"22","author":"Ahmed","year":"2012","journal-title":"Dev. 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