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CPT2 Deficiency Modeled in Zebrafish: Abnormal Neural Development, Electrical Activity, Behavior, and Schizophrenia-Related Gene Expression.

التفاصيل البيبلوغرافية
العنوان: CPT2 Deficiency Modeled in Zebrafish: Abnormal Neural Development, Electrical Activity, Behavior, and Schizophrenia-Related Gene Expression.
المؤلفون: Baker CE; Department of Biomedical Sciences, Creighton University, Omaha, NE 68178, USA., Marta AG; Department of Biology, Creighton University, Omaha, NE 68178, USA., Zimmerman ND; Department of Biology, Creighton University, Omaha, NE 68178, USA., Korade Z; Department of Pediatrics, Department of Biochemistry & Molecular Biology, College of Medicine, University of Nebraska Medical Center, Omaha, NE 68178, USA., Mathy NW; Department of Biology, Creighton University, Omaha, NE 68178, USA., Wilton D; Department of Biology, Creighton University, Omaha, NE 68178, USA., Simeone T; Department of Pharmacology and Neuroscience, Creighton University, Omaha, NE 68178, USA., Kochvar A; Department of Biology, Creighton University, Omaha, NE 68178, USA., Kramer KL; Department of Biomedical Sciences, Creighton University, Omaha, NE 68178, USA., Stessman HAF; Department of Pharmacology and Neuroscience, Creighton University, Omaha, NE 68178, USA., Shibata A; Department of Biology, Creighton University, Omaha, NE 68178, USA.
المصدر: Biomolecules [Biomolecules] 2024 Jul 26; Vol. 14 (8). Date of Electronic Publication: 2024 Jul 26.
نوع المنشور: Journal Article
اللغة: English
بيانات الدورية: Publisher: MDPI Country of Publication: Switzerland NLM ID: 101596414 Publication Model: Electronic Cited Medium: Internet ISSN: 2218-273X (Electronic) Linking ISSN: 2218273X NLM ISO Abbreviation: Biomolecules Subsets: MEDLINE
أسماء مطبوعة: Original Publication: Basel, Switzerland : MDPI, 2011-
مواضيع طبية MeSH: Zebrafish*/genetics , Zebrafish*/embryology , Carnitine O-Palmitoyltransferase*/genetics , Carnitine O-Palmitoyltransferase*/deficiency , Carnitine O-Palmitoyltransferase*/metabolism , Schizophrenia*/genetics , Schizophrenia*/metabolism , Brain*/metabolism , Brain*/growth & development, Animals ; Zebrafish Proteins/genetics ; Zebrafish Proteins/metabolism ; Zebrafish Proteins/deficiency ; Disease Models, Animal ; Gene Knockdown Techniques ; Behavior, Animal ; Gene Expression Regulation, Developmental
مستخلص: Carnitine palmitoyltransferase 2 (CPT2) is an inner mitochondrial membrane protein of the carnitine shuttle and is involved in the beta-oxidation of long chain fatty acids. Beta-oxidation provides an alternative pathway of energy production during early development and starvation. CPT2 deficiency is a genetic disorder that we recently showed can be associated with schizophrenia. We hypothesize that CPT2 deficiency during early brain development causes transcriptional, structural, and functional abnormalities that may contribute to a CNS environment that is susceptible to the emergence of schizophrenia. To investigate the effect of CPT2 deficiency on early vertebrate development and brain function, CPT2 was knocked down in a zebrafish model system. CPT2 knockdown resulted in abnormal lipid utilization and deposition, reduction in body size, and abnormal brain development. Axonal projections, neurotransmitter synthesis, electrical hyperactivity, and swimming behavior were disrupted in CPT2 knockdown zebrafish. RT-qPCR analyses showed significant increases in the expression of schizophrenia-associated genes in CPT2 knockdown compared to control zebrafish. Taken together, these data demonstrate that zebrafish are a useful model for studying the importance of beta-oxidation for early vertebrate development and brain function. This study also presents novel findings linking CPT2 deficiency to the regulation of schizophrenia and neurodegenerative disease-associated genes.
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معلومات مُعتمدة: P20 GM103427 United States GM NIGMS NIH HHS; R15 AI156879 United States AI NIAID NIH HHS; 5P20GM103427-25A1 United States NH NIH HHS; 1 R15 AI156879 United States AI NIAID NIH HHS
فهرسة مساهمة: Keywords: CPT2 deficiency; brain development; carnitine palmitoyltransferase; neurodegenerative disease; schizophrenia-related gene expression; seizure-like activity; zebrafish; β-oxidation
المشرفين على المادة: EC 2.3.1.21 (Carnitine O-Palmitoyltransferase)
0 (Zebrafish Proteins)
تواريخ الأحداث: Date Created: 20240829 Date Completed: 20240829 Latest Revision: 20240904
رمز التحديث: 20240904
مُعرف محوري في PubMed: PMC11353230
DOI: 10.3390/biom14080914
PMID: 39199302
قاعدة البيانات: MEDLINE
الوصف
تدمد:2218-273X
DOI:10.3390/biom14080914