{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,9,23]],"date-time":"2025-09-23T12:14:15Z","timestamp":1758629655560,"version":"3.37.3"},"reference-count":67,"publisher":"Oxford University Press (OUP)","issue":"6","license":[{"start":{"date-parts":[[2020,2,12]],"date-time":"2020-02-12T00:00:00Z","timestamp":1581465600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000071","name":"National Institute of Child Health and Human Development","doi-asserted-by":"publisher","award":["P01 HD083157"],"award-info":[{"award-number":["P01 HD083157"]}],"id":[{"id":"10.13039\/100000071","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2020,4,15]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>LgDel mice, which model the heterozygous deletion of genes at human chromosome 22q11.2 associated with DiGeorge\/22q11.2 deletion syndrome (22q11DS), have cranial nerve and craniofacial dysfunction as well as disrupted suckling, feeding and swallowing, similar to key 22q11DS phenotypes. Divergent trigeminal nerve (CN V) differentiation and altered trigeminal ganglion (CNgV) cellular composition prefigure these disruptions in LgDel embryos. We therefore asked whether a distinct transcriptional state in a specific population of early differentiating LgDel cranial sensory neurons, those in CNgV, a major source of innervation for appropriate oropharyngeal function, underlies this departure from typical development. LgDel versus wild-type (WT) CNgV transcriptomes differ significantly at E10.5 just after the ganglion has coalesced. Some changes parallel altered proportions of cranial placode versus cranial neural crest-derived CNgV cells. Others are consistent with a shift in anterior\u2013posterior patterning associated with divergent LgDel cranial nerve differentiation. The most robust quantitative distinction, however, is statistically verifiable increased variability of expression levels for most of the over 17\u2009000 genes expressed in common in LgDel versus WT CNgV. Thus, quantitative expression changes of functionally relevant genes and increased stochastic variation across the entire CNgV transcriptome at the onset of CN V differentiation prefigure subsequent disruption of cranial nerve differentiation and oropharyngeal function in LgDel mice.<\/jats:p>","DOI":"10.1093\/hmg\/ddaa024","type":"journal-article","created":{"date-parts":[[2020,2,4]],"date-time":"2020-02-04T20:09:38Z","timestamp":1580846978000},"page":"1002-1017","source":"Crossref","is-referenced-by-count":6,"title":["Transcriptional dysregulation in developing trigeminal sensory neurons in the LgDel mouse model of DiGeorge 22q11.2 deletion syndrome"],"prefix":"10.1093","volume":"29","author":[{"given":"Thomas M","family":"Maynard","sequence":"first","affiliation":[{"name":"Fralin Biomedical Research Institute, Virginia Tech-Carilion School of Medicine, Roanoke, VA, 24016 USA"},{"name":"Institute for Neuroscience, The George Washington University, Washington, DC 20037, USA"},{"name":"Department of Anatomy and Cell Biology, School of Medicine and Health Sciences, The George Washington University, Washington, DC 20037, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anelia","family":"Horvath","sequence":"first","affiliation":[{"name":"Institute for Neuroscience, The George Washington University, Washington, DC 20037, USA"},{"name":"Department of Pharmacology and Physiology, School of Medicine and Health Sciences, The George Washington University, Washington, DC 20037, USA"},{"name":"McCormick Genomics and Proteomics Center, School of Medicine and Health Sciences, The George Washington University, Washington, DC 20037, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1769-8631","authenticated-orcid":false,"given":"James","family":"P Bernot","sequence":"first","affiliation":[{"name":"Department of Pharmacology and Physiology, School of Medicine and Health Sciences, The George Washington University, Washington, DC 20037, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Beverly A","family":"Karpinski","sequence":"first","affiliation":[{"name":"Institute for Neuroscience, The George Washington University, Washington, DC 20037, USA"},{"name":"Department of Anatomy and Cell Biology, School of Medicine and Health Sciences, The George Washington University, Washington, DC 20037, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Andre L P","family":"Tavares","sequence":"first","affiliation":[{"name":"Department of Anatomy and Cell Biology, School of Medicine and Health Sciences, The George Washington University, Washington, DC 20037, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ankita","family":"Shah","sequence":"first","affiliation":[{"name":"Institute for Neuroscience, The George Washington University, Washington, DC 20037, USA"},{"name":"Department of Anatomy and Cell Biology, School of Medicine and Health Sciences, The George Washington University, Washington, DC 20037, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Qianqian","family":"Zheng","sequence":"first","affiliation":[{"name":"Department of Anatomy and Cell Biology, School of Medicine and Health Sciences, The George Washington University, Washington, DC 20037, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Liam","family":"Spurr","sequence":"first","affiliation":[{"name":"Department of Pharmacology and Physiology, School of Medicine and Health Sciences, The George Washington University, Washington, DC 20037, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jacqueline","family":"Olender","sequence":"first","affiliation":[{"name":"Institute for Neuroscience, The George Washington University, Washington, DC 20037, USA"},{"name":"Department of Pharmacology and Physiology, School of Medicine and Health Sciences, The George Washington University, Washington, DC 20037, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sally A","family":"Moody","sequence":"first","affiliation":[{"name":"Institute for Neuroscience, The George Washington University, Washington, DC 20037, USA"},{"name":"Department of Anatomy and Cell Biology, School of Medicine and Health Sciences, The George Washington University, Washington, DC 20037, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Claire M","family":"Fraser","sequence":"first","affiliation":[{"name":"Institute for Genome Sciences, University of Maryland, Baltimore, Baltimore, MD, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anthony-S","family":"LaMantia","sequence":"first","affiliation":[{"name":"Fralin Biomedical Research Institute, Virginia Tech-Carilion School of Medicine, Roanoke, VA, 24016 USA"},{"name":"Institute for Neuroscience, The George Washington University, Washington, DC 20037, USA"},{"name":"Department of Anatomy and Cell Biology, School of Medicine and Health Sciences, The George Washington University, Washington, DC 20037, USA"},{"name":"Department of Biological Sciences, College of Science, Virginia Tech, Blacksburg VA, 24061, USA"},{"name":"Department of Pediatrics, Virginia Tech Carilion School of Medicine, Roanoke, VA, 24016, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Norman H","family":"Lee","sequence":"first","affiliation":[{"name":"Institute for Neuroscience, The George Washington University, Washington, DC 20037, USA"},{"name":"Department of Pharmacology and Physiology, School of Medicine and Health Sciences, The George Washington University, Washington, DC 20037, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2020,2,12]]},"reference":[{"key":"2020041507553039200_ref1","doi-asserted-by":"crossref","first-page":"162","DOI":"10.1016\/j.cell.2019.01.015","article-title":"Defining the genetic, genomic, cellular, and 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