دورية أكاديمية

Postnatal expression profiles of atypical cadherin FAT1 suggest its role in autism.

التفاصيل البيبلوغرافية
العنوان: Postnatal expression profiles of atypical cadherin FAT1 suggest its role in autism.
المؤلفون: Frei JA; Program in Neuroscience, Hussman Institute for Autism, Baltimore, MD 21201, USA., Brandenburg C; Program in Neuroscience, Hussman Institute for Autism, Baltimore, MD 21201, USA.; Graduate Program in Neuroscience, University of Maryland, School of Medicine, Baltimore, MD 21201, USA., Nestor JE; Program in Neuroscience, Hussman Institute for Autism, Baltimore, MD 21201, USA., Hodzic DM; Department of Developmental Biology, Washington University School of Medicine, St Louis, MO 63110, USA., Plachez C; Program in Neuroscience, Hussman Institute for Autism, Baltimore, MD 21201, USA.; Department of Anatomy and Neurobiology, University of Maryland, School of Medicine, Baltimore, MD 21201, USA., McNeill H; Department of Developmental Biology, Washington University School of Medicine, St Louis, MO 63110, USA., Dykxhoorn DM; Hussman Institute for Human Genomics and John T. Macdonald Foundation Department of Human Genetics, University of Miami, Miller School of Medicine, Miami, FL 33136, USA., Nestor MW; Program in Neuroscience, Hussman Institute for Autism, Baltimore, MD 21201, USA., Blatt GJ; Program in Neuroscience, Hussman Institute for Autism, Baltimore, MD 21201, USA., Lin YC; Program in Neuroscience, Hussman Institute for Autism, Baltimore, MD 21201, USA.
المصدر: Biology open [Biol Open] 2021 Jun 15; Vol. 10 (6). Date of Electronic Publication: 2021 Jun 08.
نوع المنشور: Journal Article; Research Support, Non-U.S. Gov't
اللغة: English
بيانات الدورية: Publisher: Company of Biologists Ltd Country of Publication: England NLM ID: 101578018 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 2046-6390 (Electronic) Linking ISSN: 20466390 NLM ISO Abbreviation: Biol Open Subsets: MEDLINE
أسماء مطبوعة: Original Publication: Cambridge : Company of Biologists Ltd.
مواضيع طبية MeSH: Disease Susceptibility*, Autistic Disorder/*etiology , Cadherins/*genetics, Animals ; Autistic Disorder/metabolism ; Biomarkers ; Cadherins/metabolism ; Cerebellum/metabolism ; Disease Models, Animal ; Gene Expression Profiling ; Gene Expression Regulation ; Hippocampus/metabolism ; Induced Pluripotent Stem Cells/metabolism ; Interneurons/metabolism ; Mice ; Neurites/metabolism ; Protein Transport ; Transcriptome
مستخلص: Genetic studies have linked FAT1 (FAT atypical cadherin 1) with autism spectrum disorder (ASD); however, the role that FAT1 plays in ASD remains unknown. In mice, the function of Fat1 has been primarily implicated in embryonic nervous system development with less known about its role in postnatal development. We show for the first time that FAT1 protein is expressed in mouse postnatal brains and is enriched in the cerebellum, where it localizes to granule neurons and Golgi cells in the granule layer, as well as inhibitory neurons in the molecular layer. Furthermore, subcellular characterization revealed FAT1 localization in neurites and soma of granule neurons, as well as being present in the synaptic plasma membrane and postsynaptic densities. Interestingly, FAT1 expression was decreased in induced pluripotent stem cell (iPSC)-derived neural precursor cells (NPCs) from individuals with ASD. These findings suggest a novel role for FAT1 in postnatal development and may be particularly important for cerebellum function. As the cerebellum is one of the vulnerable brain regions in ASD, our study warrants further investigation of FAT1 in the disease etiology.
Competing Interests: Competing interests The authors declare no competing or financial interests.
(© 2021. Published by The Company of Biologists Ltd.)
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فهرسة مساهمة: Keywords: Autism; Cadherin; Cerebellum; FAT1; Granule cells; Neural precursor cells
المشرفين على المادة: 0 (Biomarkers)
0 (Cadherins)
0 (fat1 protein, mouse)
تواريخ الأحداث: Date Created: 20210608 Date Completed: 20220118 Latest Revision: 20240402
رمز التحديث: 20240402
مُعرف محوري في PubMed: PMC8214424
DOI: 10.1242/bio.056457
PMID: 34100899
قاعدة البيانات: MEDLINE
الوصف
تدمد:2046-6390
DOI:10.1242/bio.056457