{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,1]],"date-time":"2026-10-01T12:13:30Z","timestamp":1790856810129,"version":"4.1.0"},"update-to":[{"DOI":"10.1371\/journal.pgen.1009579","type":"new_version","label":"New version","source":"publisher","updated":{"date-parts":[[2021,6,7]],"date-time":"2021-06-07T00:00:00Z","timestamp":1623024000000}}],"reference-count":67,"publisher":"Public Library of Science (PLoS)","issue":"5","license":[{"start":{"date-parts":[[2021,5,25]],"date-time":"2021-05-25T00:00:00Z","timestamp":1621900800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","award":["RO1 DE020884"],"award-info":[{"award-number":["RO1 DE020884"]}],"id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","award":["R35 DE029086"],"award-info":[{"award-number":["R35 DE029086"]}],"id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000027","name":"National Institute on Alcohol Abuse and Alcoholism","doi-asserted-by":"publisher","award":["F32AA021320"],"award-info":[{"award-number":["F32AA021320"]}],"id":[{"id":"10.13039\/100000027","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000027","name":"National Institute on Alcohol Abuse and Alcoholism","doi-asserted-by":"publisher","award":["K99AA023560"],"award-info":[{"award-number":["K99AA023560"]}],"id":[{"id":"10.13039\/100000027","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["www.plosgenetics.org"],"crossmark-restriction":false},"short-container-title":["PLoS Genet"],"abstract":"<jats:p>We sought to understand how perturbation of signaling pathways and their targets generates variable phenotypes. In humans,<jats:italic>GATA3<\/jats:italic>associates with highly variable defects, such as HDR syndrome, microsomia and choanal atresia. We previously characterized a zebrafish point mutation in<jats:italic>gata3<\/jats:italic>with highly variable craniofacial defects to the posterior palate. This variability could be due to residual Gata3 function, however, we observe the same phenotypic variability in<jats:italic>gata3<\/jats:italic>null mutants. Using<jats:italic>hsp<\/jats:italic>:<jats:italic>GATA3-GFP<\/jats:italic>transgenics, we demonstrate that Gata3 function is required between 24 and 30 hpf. At this time maxillary neural crest cells fated to generate the palate express<jats:italic>gata3<\/jats:italic>. Transplantation experiments show that neural crest cells require Gata3 function for palatal development. Via a candidate approach, we determined if Bmp signaling was upstream of<jats:italic>gata3<\/jats:italic>and if this pathway explained the mutant\u2019s phenotypic variation. Using<jats:italic>BRE<\/jats:italic>:<jats:italic>d2EGFP<\/jats:italic>transgenics, we demonstrate that maxillary neural crest cells are Bmp responsive by 24 hpf. We find that<jats:italic>gata3<\/jats:italic>expression in maxillary neural crest requires Bmp signaling and that blocking Bmp signaling, in<jats:italic>hsp<\/jats:italic>:<jats:italic>DN-Bmpr1a-GFP<\/jats:italic>embryos, can phenocopy<jats:italic>gata3<\/jats:italic>mutants. Palatal defects are rescued in<jats:italic>hsp<\/jats:italic>:<jats:italic>DN-Bmpr1a-GFP;hsp<\/jats:italic>:<jats:italic>GATA3-GFP<\/jats:italic>double transgenic embryos, collectively demonstrating that<jats:italic>gata3<\/jats:italic>is downstream of Bmp signaling. However, Bmp attenuation does not alter phenotypic variability in<jats:italic>gata3<\/jats:italic>loss-of-function embryos, implicating a different pathway. Due to phenotypes observed in hypomorphic<jats:italic>shha<\/jats:italic>mutants, the Sonic Hedgehog (Shh) pathway was a promising candidate for this pathway. Small molecule activators and inhibitors of the Shh pathway lessen and exacerbate, respectively, the phenotypic severity of<jats:italic>gata3<\/jats:italic>mutants. Importantly, inhibition of Shh can cause<jats:italic>gata3<\/jats:italic>haploinsufficiency, as observed in humans. We find that<jats:italic>gata3<\/jats:italic>mutants in a less expressive genetic background have a compensatory upregulation of Shh signaling. These results demonstrate that the level of Shh signaling can modulate the phenotypes observed in<jats:italic>gata3<\/jats:italic>mutants.<\/jats:p>","DOI":"10.1371\/journal.pgen.1009579","type":"journal-article","created":{"date-parts":[[2021,5,25]],"date-time":"2021-05-25T17:31:58Z","timestamp":1621963918000},"page":"e1009579","update-policy":"https:\/\/doi.org\/10.1371\/journal.pgen.corrections_policy","source":"Crossref","is-referenced-by-count":14,"title":["Variation in phenotypes from a Bmp-Gata3 genetic pathway is modulated by Shh signaling"],"prefix":"10.1371","volume":"17","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7601-0147","authenticated-orcid":true,"given":"Mary E.","family":"Swartz","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7838-5823","authenticated-orcid":true,"given":"C. Ben","family":"Lovely","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5258-2477","authenticated-orcid":true,"given":"Johann K.","family":"Eberhart","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"340","published-online":{"date-parts":[[2021,5,25]]},"reference":[{"issue":"3","key":"pgen.1009579.ref001","doi-asserted-by":"crossref","first-page":"167","DOI":"10.1038\/nrg2933","article-title":"Cleft lip and palate: understanding genetic and environmental influences","volume":"12","author":"MJ Dixon","year":"2011","journal-title":"Nat Rev Genet"},{"issue":"6","key":"pgen.1009579.ref002","doi-asserted-by":"crossref","first-page":"1069","DOI":"10.1242\/dev.02281","article-title":"Early Hedgehog signaling from neural to oral epithelium organizes anterior craniofacial development","volume":"133","author":"JK 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