Journal
CEREBRAL CORTEX
Volume 26, Issue 10, Pages 3991-4003Publisher
OXFORD UNIV PRESS INC
DOI: 10.1093/cercor/bhw229
Keywords
animal model; cognition; copy number variation; 22q11; 2 deletion syndrome
Categories
Funding
- Innovative Medicine Initiative Joint Undertaking [115008]
- European Union
- Medical Research Council
- Wellcome Trust
- Medical Research Council [G1000183, G1000183B] Funding Source: researchfish
- MRC [G1000183] Funding Source: UKRI
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A chromosomal microdeletion at the 22q11.2 locus is associated with extensive cognitive impairments, schizophrenia and other psychopathology in humans. Previous reports indicate that mouse models of the 22q11.2 microdeletion syndrome (22q11.2DS) may model the genetic basis of cognitive deficits relevant for neuropsychiatric disorders such as schizophrenia. To assess the models usefulness for drug discovery, a novel mouse (Df(h22q11)/+) was assessed in an extensive battery of cognitive assays by partners within the NEWMEDS collaboration (Innovative Medicines Initiative Grant Agreement No. 115008). This battery included classic and touchscreen-based paradigms with recognized sensitivity and multiple attempts at reproducing previously published findings in 22q11.2DS mouse models. This work represents one of the most comprehensive reports of cognitive functioning in a transgenic animal model. In accordance with previous reports, there were non-significant trends or marginal impairment in some tasks. However, the Df(h22q11)/+ mouse did not show comprehensive deficits; no robust impairment was observed following more than 17 experiments and 14 behavioral paradigms. Thus - within the current protocols - the 22q11.2DS mouse model fails to mimic the cognitive alterations observed in human 22q11.2 deletion carriers. We suggest that the 22q11.2DS model may induce liability for cognitive dysfunction with additional hits being required for phenotypic expression.
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