{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,14]],"date-time":"2026-07-14T00:25:09Z","timestamp":1783988709958,"version":"3.55.0"},"reference-count":63,"publisher":"Oxford University Press (OUP)","issue":"18","license":[{"start":{"date-parts":[[2020,9,9]],"date-time":"2020-09-09T00:00:00Z","timestamp":1599609600000},"content-version":"vor","delay-in-days":1,"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"}]},{"DOI":"10.13039\/100000071","name":"National Institute of Child Health and Human Development","doi-asserted-by":"publisher","award":["NICHD IDDRC U54"],"award-info":[{"award-number":["NICHD IDDRC U54"]}],"id":[{"id":"10.13039\/100000071","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000071","name":"National Institute of Child Health and Human Development","doi-asserted-by":"publisher","award":["HD 090257"],"award-info":[{"award-number":["HD 090257"]}],"id":[{"id":"10.13039\/100000071","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Georgetown University and Children\u2019s National Medical Center"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2020,11,4]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>We identified divergent modes of initial axon growth that prefigure disrupted differentiation of the trigeminal nerve (CN V), a cranial nerve essential for suckling, feeding and swallowing (S\/F\/S), a key innate behavior compromised in multiple genetic developmental disorders including DiGeorge\/22q11.2 Deletion Syndrome (22q11.2 DS). We combined rapid in vivo labeling of single CN V axons in LgDel+\/\u2212 mouse embryos, a genomically accurate 22q11.2DS model, and 3D imaging to identify and quantify phenotypes that could not be resolved using existing methods. We assessed these phenotypes in three 22q11.2-related genotypes to determine whether individual CN V motor and sensory axons wander, branch and sprout aberrantly in register with altered anterior\u2013posterior hindbrain patterning and gross morphological disruption of CN V seen in LgDel+\/\u2212. In the additional 22q11.2-related genotypes: Tbx1+\/\u2212, Ranbp1\u2212\/\u2212, Ranbp1+\/\u2212 and LgDel+\/\u2212:Raldh2+\/\u2212; axon phenotypes are seen when hindbrain patterning and CN V gross morphology is altered, but not when it is normal or restored toward WT. This disordered growth of CN V sensory and motor axons, whose appropriate targeting is critical for optimal S\/F\/S, may be an early, critical determinant of imprecise innervation leading to inefficient oropharyngeal function associated with 22q11.2 deletion from birth onward.<\/jats:p>","DOI":"10.1093\/hmg\/ddaa199","type":"journal-article","created":{"date-parts":[[2020,9,3]],"date-time":"2020-09-03T19:13:53Z","timestamp":1599160433000},"page":"3081-3093","source":"Crossref","is-referenced-by-count":8,"title":["Aberrant early growth of individual trigeminal sensory and motor axons in a series of mouse genetic models of 22q11.2 deletion syndrome"],"prefix":"10.1093","volume":"29","author":[{"given":"Zahra","family":"Motahari","sequence":"first","affiliation":[{"name":"Institute for Neuroscience, Washington, DC 20037, USA"},{"name":"Department of Anatomy and Cell Biology, The George Washington University School of Medicine and Health Sciences, Washington, DC 20037, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6976-3936","authenticated-orcid":false,"given":"Thomas M","family":"Maynard","sequence":"additional","affiliation":[{"name":"The Fralin Biomedical Research Institute at Virginia Tech-Carilion School of Medicine, Roanoke, VA 24016, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anastas","family":"Popratiloff","sequence":"additional","affiliation":[{"name":"Institute for Neuroscience, Washington, DC 20037, USA"},{"name":"Department of Anatomy and Cell Biology, The George Washington University School of Medicine and Health Sciences, Washington, DC 20037, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sally A","family":"Moody","sequence":"additional","affiliation":[{"name":"Institute for Neuroscience, Washington, DC 20037, USA"},{"name":"Department of Anatomy and Cell Biology, The George Washington University School of Medicine and Health Sciences, Washington, DC 20037, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anthony-S","family":"LaMantia","sequence":"additional","affiliation":[{"name":"The Fralin Biomedical Research Institute at Virginia Tech-Carilion School of Medicine, Roanoke, VA 24016, USA"},{"name":"Department of Biological Sciences, Virginia Tech, Blacksburg, VA 24060, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2020,9,8]]},"reference":[{"key":"2020110605012550400_ref1","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1016\/j.pneurobio.2015.03.004","article-title":"Modeling a model: mouse genetics, 22q11.2 deletion syndrome, and disorders of cortical circuit development","volume":"130","author":"Meechan","year":"2015","journal-title":"Prog. 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