{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,11]],"date-time":"2026-04-11T14:11:46Z","timestamp":1775916706020,"version":"3.50.1"},"reference-count":24,"publisher":"Public Library of Science (PLoS)","issue":"10","license":[{"start":{"date-parts":[[2022,10,12]],"date-time":"2022-10-12T00:00:00Z","timestamp":1665532800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["R01HD083181"],"award-info":[{"award-number":["R01HD083181"]}],"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":["U54HD083211"],"award-info":[{"award-number":["U54HD083211"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Neuroscience Core and Neuroimaging Core","award":["1P50HD103537"],"award-info":[{"award-number":["1P50HD103537"]}]},{"name":"Mouse Neurobehavioral Phenotyping Core and the Translational Neuroscience Core","award":["T32MH065215"],"award-info":[{"award-number":["T32MH065215"]}]},{"name":"Mouse Neurobehavioral Phenotyping Core and the Translational Neuroscience Core","award":["T32 MH064913"],"award-info":[{"award-number":["T32 MH064913"]}]},{"name":"Mouse Neurobehavioral Phenotyping Core and the Translational Neuroscience Core","award":["T32 GM07628"],"award-info":[{"award-number":["T32 GM07628"]}]},{"name":"International FOXG1 Foundation"}],"content-domain":{"domain":["www.plosone.org"],"crossmark-restriction":false},"short-container-title":["PLoS ONE"],"abstract":"<jats:p>FOXG1 Syndrome (FS) is a devastating neurodevelopmental disorder that is caused by a heterozygous loss-of-function (LOF) mutation of the <jats:italic>FOXG1<\/jats:italic> gene, which encodes a transcriptional regulator important for telencephalic brain development. People with FS have marked developmental delays, impaired ambulation, movement disorders, seizures, and behavior abnormalities including autistic features. Current therapeutic approaches are entirely symptomatic, however the ability to rescue phenotypes in mouse models of other genetic neurodevelopmental disorders such as Rett syndrome, Angelman syndrome, and Phelan-McDermid syndrome by postnatal expression of gene products has led to hope that similar approaches could help modify the disease course in other neurodevelopmental disorders such as FS. While FoxG1 protein function plays a critical role in embryonic brain development, the ongoing adult expression of FoxG1 and behavioral phenotypes that present when FoxG1 function is removed postnatally provides support for opportunity for improvement with postnatal treatment. Here we generated a new mouse allele of <jats:italic>Foxg1<\/jats:italic> that disrupts protein expression and characterized the behavioral and structural brain phenotypes in heterozygous mutant animals. These mutant animals display changes in locomotor behavior, gait, anxiety, social interaction, aggression, and learning and memory compared to littermate controls. Additionally, they have structural brain abnormalities reminiscent of people with FS. This information provides a framework for future studies to evaluate the potential for post-natal expression of FoxG1 to modify the disease course in this severe neurodevelopmental disorder.<\/jats:p>","DOI":"10.1371\/journal.pone.0266861","type":"journal-article","created":{"date-parts":[[2022,10,12]],"date-time":"2022-10-12T17:36:06Z","timestamp":1665596166000},"page":"e0266861","update-policy":"https:\/\/doi.org\/10.1371\/journal.pone.corrections_policy","source":"Crossref","is-referenced-by-count":9,"title":["Behavioral and brain anatomical analysis of Foxg1 heterozygous mice"],"prefix":"10.1371","volume":"17","author":[{"given":"Kirsty R.","family":"Erickson","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Rebekah","family":"Farmer","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jonathan 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