The Object Space Task reveals increased expression of cumulative memory in a mouse model of Kleefstra syndrome
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Kleefstra syndrome is a disorder caused by a mutation in the EHMT1 gene characterized in humans by general developmental delay, mild to severe intellectual disability and autism. Here, we characterized cumulative memory in the Ehmt1+/− mouse model using the Object Space Task. We combined conventional behavioral analysis with automated analysis by deep-learning networks, a session-based computational learning model, and a trial-based classifier. Ehmt1+/− mice showed more anxiety-like features and generally explored objects less, but the difference decreased over time. Interestingly, when analyzing memory-specific exploration, Ehmt1+/− show increased expression of cumulative memory, but a deficit in a more simple, control memory condition. Using our automatic classifier to differentiate between genotypes, we found that cumulative memory features are better suited for classification than general exploration differences. Thus, detailed behavioral classification with the Object Space Task produced a more detailed behavioral phenotype of the Ehmt1+/− mouse model.
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Other monogenic syndromes like Kleefstra, KBG, WitKoS, Angelman, Coffin-Sirris, Rubinstein-Taybi, Phelan-McDermid, Smith-Magenis Syndrome and Kabuki syndrome also have a clear prefrontal component in their behavioral and cognitive phenotype.
“Other monogenic syndromes like Kleefstra, KBG, WitKoS, Angelman, Coffin-Sirris, Rubinstein-Taybi, Phelan-McDermid, Smith–Magenis Syndrome and Kabuki syndrome also have a clear prefrontal component in their behavioral and cognitive phenotype [272–280].”
From Development of prefrontal cortex (2022) · cites this paper as [275]
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- Authors
- Schut, E. H. S., Alonso, A., Smits, S., Khamassi, M., Samanta, A., Negwer, M., Kasri, N. N., Navarro Lobato, I., & Genzel, L.
- Affiliations
- Radboud University Nijmegen (NL)
- Centre National de la Recherche Scientifique (FR)
- Radboud University Medical Center (NL)
- Corresponding author
- Genzel, Lisa
- ORCID
- Venue
- Neurobiology of Learning and Memory
- Publisher
- Elsevier BV
- Published
- 2020-09
- Volume
- 173
- Pages
- 107265
- ISSN
- 1074-7427 (ISSN-L)
- OpenAlex
- W3034343597
- Cited by
- 8
- References
- 42
- Citations by year
- 2025: 2 · 2024: 1 · 2023: 2 · 2022: 1 · 2021: 2
- FWCI
- 0.73
- Type
- journal article
- Language
- en
- Open access
- hybrid
- doi.org
- Licence
- Locations
- Neurobiology of Learning and Memory (publishedVersion, cc-by)
- PubMed (publishedVersion)
- HAL (submittedVersion, other-oa)
- Radboud Repository (Radboud University) (submittedVersion)
- Radboud Repository (Radboud University) (submittedVersion, other-oa)
- Funders
- Branco Weiss Society in Science
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
- Life Sciences › Neuroscience › Cognitive Neuroscience › Memory and Neural Mechanisms
- Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Neuroscience and Neuropharmacology Research
- Keywords
- Kleefstra syndrome, EHMT1, anxiety-like behavior, object exploration, deep learning, computational models of learning, intellectual disability, autism spectrum disorder, developmental delay
- MeSH terms
- Deep Learning, Animals, Behavior, Animal, Chromosome Deletion, Chromosomes, Human, Pair 9, Disease Models, Animal, Exploratory Behavior, Heart Defects, Congenital, Male, Memory, Intellectual Disability, Histone-Lysine N-Methyltransferase, Craniofacial Abnormalities, Mice
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