DC Field | Value | Language |
---|---|---|
dc.contributor.author | Kang, Ryeonghwa | ko |
dc.contributor.author | Kim, Kyungdeok | ko |
dc.contributor.author | Jung, Yewon | ko |
dc.contributor.author | Choi, Sang-Han | ko |
dc.contributor.author | Lee, Chanhee | ko |
dc.contributor.author | Im, Geun Ho | ko |
dc.contributor.author | Shin, Miram | ko |
dc.contributor.author | Ryu, Kwangmin | ko |
dc.contributor.author | Choi, Subin | ko |
dc.contributor.author | Yang, Esther | ko |
dc.contributor.author | Shin, Wangyong | ko |
dc.contributor.author | Lee, Seungjoon | ko |
dc.contributor.author | Lee, Suho | ko |
dc.contributor.author | Papadopoulos, Zachary | ko |
dc.contributor.author | Ahn, Ji Hoon | ko |
dc.contributor.author | Koh, Gou Young | ko |
dc.contributor.author | Kipnis, Jonathan | ko |
dc.contributor.author | Kang, Hyojin | ko |
dc.contributor.author | Kim, Hyun | ko |
dc.contributor.author | Cho, Won-Ki | ko |
dc.contributor.author | Park, Soochul | ko |
dc.contributor.author | Kim, Seong-Gi | ko |
dc.contributor.author | Kim, Eunjoon | ko |
dc.date.accessioned | 2024-06-11T09:00:22Z | - |
dc.date.available | 2024-06-11T09:00:22Z | - |
dc.date.created | 2024-06-10 | - |
dc.date.issued | 2024-05 | - |
dc.identifier.citation | PLOS BIOLOGY, v.22, no.5 | - |
dc.identifier.issn | 1544-9173 | - |
dc.identifier.uri | http://hdl.handle.net/10203/319738 | - |
dc.description.abstract | Autism spectrum disorders (ASD) frequently accompany macrocephaly, which often involves hydrocephalic enlargement of brain ventricles. Katnal2 is a microtubule-regulatory protein strongly linked to ASD, but it remains unclear whether Katnal2 knockout (KO) in mice leads to microtubule- and ASD-related molecular, synaptic, brain, and behavioral phenotypes. We found that Katnal2-KO mice display ASD-like social communication deficits and age-dependent progressive ventricular enlargements. The latter involves increased length and beating frequency of motile cilia on ependymal cells lining ventricles. Katnal2-KO hippocampal neurons surrounded by enlarged lateral ventricles show progressive synaptic deficits that correlate with ASD-like transcriptomic changes involving synaptic gene down-regulation. Importantly, early postnatal Katnal2 re-expression prevents ciliary, ventricular, and behavioral phenotypes in Katnal2-KO adults, suggesting a causal relationship and a potential treatment. Therefore, Katnal2 negatively regulates ependymal ciliary function and its deletion in mice leads to ependymal ciliary hyperfunction and hydrocephalus accompanying ASD-related behavioral, synaptic, and transcriptomic changes. | - |
dc.language | English | - |
dc.publisher | PUBLIC LIBRARY SCIENCE | - |
dc.title | Loss of Katnal2 leads to ependymal ciliary hyperfunction and autism-related phenotypes in mice | - |
dc.type | Article | - |
dc.identifier.wosid | 001218610500001 | - |
dc.identifier.scopusid | 2-s2.0-85193010732 | - |
dc.type.rims | ART | - |
dc.citation.volume | 22 | - |
dc.citation.issue | 5 | - |
dc.citation.publicationname | PLOS BIOLOGY | - |
dc.identifier.doi | 10.1371/journal.pbio.3002596 | - |
dc.contributor.localauthor | Koh, Gou Young | - |
dc.contributor.localauthor | Cho, Won-Ki | - |
dc.contributor.localauthor | Kim, Eunjoon | - |
dc.contributor.nonIdAuthor | Kim, Kyungdeok | - |
dc.contributor.nonIdAuthor | Choi, Sang-Han | - |
dc.contributor.nonIdAuthor | Lee, Chanhee | - |
dc.contributor.nonIdAuthor | Im, Geun Ho | - |
dc.contributor.nonIdAuthor | Shin, Miram | - |
dc.contributor.nonIdAuthor | Choi, Subin | - |
dc.contributor.nonIdAuthor | Yang, Esther | - |
dc.contributor.nonIdAuthor | Shin, Wangyong | - |
dc.contributor.nonIdAuthor | Lee, Seungjoon | - |
dc.contributor.nonIdAuthor | Lee, Suho | - |
dc.contributor.nonIdAuthor | Papadopoulos, Zachary | - |
dc.contributor.nonIdAuthor | Kipnis, Jonathan | - |
dc.contributor.nonIdAuthor | Kang, Hyojin | - |
dc.contributor.nonIdAuthor | Kim, Hyun | - |
dc.contributor.nonIdAuthor | Park, Soochul | - |
dc.contributor.nonIdAuthor | Kim, Seong-Gi | - |
dc.description.isOpenAccess | N | - |
dc.type.journalArticle | Article | - |
dc.subject.keywordPlus | SET ENRICHMENT ANALYSIS | - |
dc.subject.keywordPlus | ULTRASONIC VOCALIZATIONS | - |
dc.subject.keywordPlus | MOUSE MODELS | - |
dc.subject.keywordPlus | BRAIN | - |
dc.subject.keywordPlus | HYDROCEPHALUS | - |
dc.subject.keywordPlus | PNEUMOLYSIN | - |
dc.subject.keywordPlus | GENES | - |
dc.subject.keywordPlus | MECHANISMS | - |
dc.subject.keywordPlus | DISABILITY | - |
dc.subject.keywordPlus | POLARITY | - |
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