{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T15:24:14Z","timestamp":1791300254778,"version":"4.1.0"},"reference-count":75,"publisher":"Oxford University Press (OUP)","issue":"6","license":[{"start":{"date-parts":[[2025,6,25]],"date-time":"2025-06-25T00:00:00Z","timestamp":1750809600000},"content-version":"vor","delay-in-days":24,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000893","name":"Simons Foundation","doi-asserted-by":"publisher","award":["SFARI 1013720"],"award-info":[{"award-number":["SFARI 1013720"]}],"id":[{"id":"10.13039\/100000893","id-type":"DOI","asserted-by":"publisher"}]},{"name":"FOXG1 Research Foundation"},{"name":"NINDS\/NIH","award":["NS118748"],"award-info":[{"award-number":["NS118748"]}]},{"name":"NINDS\/NIH","award":["NS100471"],"award-info":[{"award-number":["NS100471"]}]},{"name":"NINDS\/NIH","award":["NS111760"],"award-info":[{"award-number":["NS111760"]}]},{"DOI":"10.13039\/100002046","name":"Hearing Health Foundation","doi-asserted-by":"publisher","award":["114344"],"award-info":[{"award-number":["114344"]}],"id":[{"id":"10.13039\/100002046","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,6,4]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>FOXG1 syndrome (FS) is a rare and devastating neurodevelopmental disorder affected by FOXG1 gene mutations and reduced sound tolerance has been reported in children with FS. Effects of single missense mutation of Foxg1 gene on auditory function and behavior were studied using the G216S mouse model. G216S mice showed significantly reduced gap-induced prepulse inhibition, suggesting poor temporal processing without hearing loss. Increased running and freezing behaviors under loud sounds were also found in G216 mice, suggesting aversive sound behaviors. Electrophysiological assessment of the auditory cortex of G216 mice revealed a slightly reduced amplitude and enlarged poststimulus responses to the sound stimulus. The layer function analysis using current source density revealed reduced layer\u2013specific response in the G216S mice. Immunocytochemistry found Foxg1 gene mutation affects cortical layer differentiations and reduced cortical neurons, which are consistent with the physiological results. Our study suggests that the Foxg1 mutation impaired cortical development. The results are consistent with other models of autism spectrum disorders (ASDs), suggesting that the G216S mouse model may represent a hyperacusis model of ASD. Our results provide direct evidence that a single-nucleotide mutation of the Foxg1 gene can affect cortical layer development and auditory processing and reduce sound tolerance.<\/jats:p>","DOI":"10.1093\/cercor\/bhaf166","type":"journal-article","created":{"date-parts":[[2025,6,25]],"date-time":"2025-06-25T19:43:08Z","timestamp":1750880588000},"source":"Crossref","is-referenced-by-count":5,"title":["Foxg1 gene mutation impairs auditory cortex response and reduces sound tolerance"],"prefix":"10.1093","volume":"35","author":[{"given":"Fei","family":"Xu","sequence":"first","affiliation":[{"name":"Department of Communicative Disorders and Sciences, State University of New York at Buffalo , 3435 Main Street, Buffalo, NY 14214 ,","place":["United States"]},{"name":"Department of Audiology and Speech Pathology, Zhejiang Chinese Medical University , 48 Binwen Road, Binjiang District, Hangzhou City, Zhejiang Province 310053 ,","place":["P.R. China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Guangdi","family":"Chen","sequence":"additional","affiliation":[{"name":"Department of Communicative Disorders and Sciences, State University of New York at Buffalo , 3435 Main Street, Buffalo, NY 14214 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shin","family":"Jeon","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences, FOXG1 Research Center, State University of New York at Buffalo , C533 Cooke Hall, 211 Mary Talbert Way, Buffalo, NY 14260 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lynne","family":"Ling","sequence":"additional","affiliation":[{"name":"Department of Pharmacology, School of Medicine, Southern Illinois University , 801 N. Rutledge St., Springfield, IL 62702 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Senthilvelan","family":"Manohar","sequence":"additional","affiliation":[{"name":"Department of Communicative Disorders and Sciences, State University of New York at Buffalo , 3435 Main Street, Buffalo, NY 14214 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dalian","family":"Ding","sequence":"additional","affiliation":[{"name":"Department of Communicative Disorders and Sciences, State University of New York at Buffalo , 3435 Main Street, Buffalo, NY 14214 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Benjamin D","family":"Auerbach","sequence":"additional","affiliation":[{"name":"Beckman Institute, Department of Molecular & Integrative Physiology, University of Illinois Urbana-Champaign , 405 N Mathews Ave, Urbana, IL 61801 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jae","family":"Lee","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences, FOXG1 Research Center, State University of New York at Buffalo , C533 Cooke Hall, 211 Mary Talbert Way, Buffalo, NY 14260 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Soo-Kyung","family":"Lee","sequence":"additional","affiliation":[{"name":"Department of Biological Sciences, FOXG1 Research Center, State University of New York at Buffalo , C533 Cooke Hall, 211 Mary Talbert Way, Buffalo, NY 14260 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wei","family":"Sun","sequence":"additional","affiliation":[{"name":"Department of Communicative Disorders and Sciences, State University of New York at Buffalo , 3435 Main Street, Buffalo, NY 14214 ,","place":["United 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