{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,7,30]],"date-time":"2025-07-30T09:47:18Z","timestamp":1753868838026,"version":"3.41.2"},"reference-count":48,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2019,9,30]],"date-time":"2019-09-30T00:00:00Z","timestamp":1569801600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["R01DE020843","R01DE025897","R01DC012308"],"award-info":[{"award-number":["R01DE020843","R01DE025897","R01DC012308"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Genesis"],"published-print":{"date-parts":[[2020,1]]},"abstract":"<jats:title>Summary<\/jats:title><jats:p>Proper development of taste organs including the tongue and taste papillae requires interactions with the underlying mesenchyme through multiple molecular signaling pathways. The effects of bone morphogenetic proteins (BMPs) and antagonists are profound, however, the tissue\u2010specific roles of distinct receptors are largely unknown. Here, we report that constitutive activation (ca) of ALK2\u2010BMP signaling in the tongue mesenchyme (marked by <jats:italic>Wnt1\u2010Cre<\/jats:italic>) caused microglossia\u2014a dramatically smaller and misshapen tongue with a progressively severe reduction in size along the anteroposterior axis and absence of a pharyngeal region. At E10.5, the tongue primordia (branchial arches 1\u20134) formed in <jats:italic>Wnt1\u2010Cre\/caAlk2<\/jats:italic> mutants while each branchial arch responded to elevated BMP signaling distinctly in gene expression of BMP targets (<jats:italic>Id1<\/jats:italic>, <jats:italic>Snai1<\/jats:italic>, <jats:italic>Snai2<\/jats:italic>, and <jats:italic>Runx2<\/jats:italic>), proliferation (<jats:italic>Cyclin\u2010D1<\/jats:italic>) and apoptosis (<jats:italic>p53<\/jats:italic>). Moreover, elevated ALK2\u2010BMP signaling in the mesenchyme resulted in apparent defects of lingual epithelium, muscles, and nerves. In <jats:italic>Wnt1\u2010Cre\/caAlk2<\/jats:italic> mutants, a circumvallate papilla was missing and further development of formed fungiform papillae was arrested in late embryos. Our data collectively demonstrate that ALK2\u2010BMP signaling in the mesenchyme plays essential roles in orchestrating various tissues for proper development of the tongue and its appendages in a region\u2010specific manner.<\/jats:p>","DOI":"10.1002\/dvg.23337","type":"journal-article","created":{"date-parts":[[2019,10,1]],"date-time":"2019-10-01T06:03:01Z","timestamp":1569909781000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Increased activity of mesenchymal ALK2\u2010BMP signaling causes posteriorly truncated microglossia and disorganization of lingual tissues"],"prefix":"10.1002","volume":"58","author":[{"given":"Mohamed","family":"Ishan","sequence":"first","affiliation":[{"name":"Department of Animal and Dairy Science Regenerative Bioscience Center, College of Agricultural and Environmental Sciences, University of Georgia  Athens Georgia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Guiqian","family":"Chen","sequence":"additional","affiliation":[{"name":"Department of Animal and Dairy Science Regenerative Bioscience Center, College of Agricultural and Environmental Sciences, University of Georgia  Athens Georgia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Chenming","family":"Sun","sequence":"additional","affiliation":[{"name":"Department of Physiology and Pharmacology College of Veterinary Medicine, University of Georgia  Athens Georgia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shi\u2010You","family":"Chen","sequence":"additional","affiliation":[{"name":"Department of Physiology and Pharmacology College of Veterinary Medicine, University of Georgia  Athens Georgia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yoshihiro","family":"Komatsu","sequence":"additional","affiliation":[{"name":"Department of Pediatrics The University of Texas Medical School at Houston  Houston Texas"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6268-4204","authenticated-orcid":false,"given":"Yuji","family":"Mishina","sequence":"additional","affiliation":[{"name":"Department of Biologic and Materials Sciences School of Dentistry, University of Michigan  Ann Arbor Michigan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5515-9869","authenticated-orcid":false,"given":"Hong\u2010Xiang","family":"Liu","sequence":"additional","affiliation":[{"name":"Department of Animal and Dairy Science Regenerative Bioscience Center, College of Agricultural and Environmental Sciences, University of Georgia  Athens 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