ArticleAutism research : official journal of the International Society for Autism Research2020
Generation of a Novel Rat Model of Angelman Syndrome with a Complete Ube3a Gene Deletion.
Article in Autism research : official journal of the International Society for Autism Research, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 25 papers.
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25 citing papers in PubMed, 38 citations in OpenAlex.
- Operant behavior is reliably impaired in a mouse model of Angelman syndrome.Research square · 2026Article
- Crossing the finish line towards a disease-modifying treatment for Angelman syndrome.Journal of neurodevelopmental disorders · 2026Review
- A preclinical pig model of Angelman syndrome mirrors the early developmental trajectory of the human condition.Proceedings of the National Academy of Sciences of the United States of America · 2025Article
- Acute administration of lovastatin had no pronounced effect on motor abilities, motor coordination, gait nor simple cognition in a mouse model of Angelman syndrome.Journal of neurodevelopmental disorders · 2025Article
- Perinatal exposure to pesticides alters synaptic plasticity signaling and induces behavioral deficits associated with neurodevelopmental disorders.Cell biology and toxicology · 2023Article
- Vector enabled CRISPR gene editing - A revolutionary strategy for targeting the diversity of brain pathologies.Coordination chemistry reviews · 2023Article
- Modernising autism spectrum disorder model engineering and treatmentWorld journal of clinical cases · 2023Review
- Unique Features of the Gut Microbiome Characterized in Animal Models of Angelman Syndrome.mSystems · 2023Article
- Stem cell models of Angelman syndrome.Frontiers in cell and developmental biology · 2023Review
- Multidimensional analysis of behavior predicts genotype with high accuracy in a mouse model of Angelman syndrome.Translational psychiatry · 2022Article
- Touchscreen cognitive deficits, hyperexcitability and hyperactivity in males and females using two models of Cdkl5 deficiency.Human molecular genetics · 2022Article
- Generation and Characterization of a Novel Angelman Syndrome Mouse Model with a Full Deletion of theCells · 2022Article
- Improving Gene Therapy for Angelman Syndrome with Secreted Human UBE3A.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2022Article
- Gait as a quantitative translational outcome measure in Angelman syndrome.Autism research : official journal of the International Society for Autism Research · 2022Article
- Recent Advances in the Production of Genome-Edited Rats.International journal of molecular sciences · 2022Review
- Circadian Rhythms and Sleep Are Dependent Upon Expression Levels of Key Ubiquitin LigaseFrontiers in behavioral neuroscience · 2022Article
- Excessive Laughter-like Vocalizations, Microcephaly, and Translational Outcomes in theThe Journal of neuroscience : the official journal of the Society for Neuroscience · 2021Article
- Insulin-like growth factor-2 does not improve behavioral deficits in mouse and rat models of Angelman Syndrome.Molecular autism · 2021Article
- Emerging Gene and Small Molecule Therapies for the Neurodevelopmental Disorder Angelman Syndrome.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2021Review
- Functional rescue in an Angelman syndrome model following treatment with lentivector transduced hematopoietic stem cells.Human molecular genetics · 2021Article
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Authors and funding
20 authors at 5 institutions in 2 countries.
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Abstract
Angelman syndrome (AS) is a rare genetic disorder characterized by severe intellectual disability, seizures, lack of speech, and ataxia. The gene responsible for AS was identified as Ube3a and it encodes for E6AP, an E3 ubiquitin ligase. Currently, there is very little known about E6AP's mechanism of action in vivo or how the lack of this protein in neurons may contribute to the AS phenotype. Elucidating the mechanistic action of E6AP would enhance our understanding of AS and drive current research into new avenues that could lead to novel therapeutic approaches that target E6AP's various functions. To facilitate the study of AS, we have generated a novel rat model in which we deleted the rat Ube3a gene using CRISPR. The AS rat phenotypically mirrors human AS with loss of Ube3a expression in the brain and deficits in motor coordination as well as learning and memory. This model offers a new avenue for the study of AS. Autism Res 2020, 13: 397-409. © 2020 International Society for Autism Research,Wiley Periodicals, Inc. LAY SUMMARY: Angelman syndrome (AS) is a rare genetic disorder characterized by severe intellectual disability, seizures, difficulty speaking, and ataxia. The gene responsible for AS was identified as UBE3A, yet very little is known about its function in vivo or how the lack of this protein in neurons may contribute to the AS phenotype. To facilitate the study of AS, we have generated a novel rat model in which we deleted the rat Ube3a gene using CRISPR. The AS rat mirrors human AS with loss of Ube3a expression in the brain and deficits in motor coordination as well as learning and memory. This model offers a new avenue for the study of AS.
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