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Psychotic illness is a challenging problem for individuals with PWS and has different rates of prevalence in distinct PWS genotypes. Previously, we demonstrated behavioural and cognitive endophenotypes of relevance to psychiatric illness in a mouse model for one of the associated PWS genotypes, namely PWS-IC, in which deletion of the imprinting centre leads to loss of paternally imprinted gene expression and over-expression of\n                    <jats:italic>Ube3a<\/jats:italic>\n                    . Here we examine the broader gene expression changes that are specific to the psychiatric endophenotypes seen in this model. To do this we compared the brain transcriptomic profile of the PWS-IC mouse to the PWS-cr model that carries a deletion of the PWS minimal critical interval spanning the snoRNA\n                    <jats:italic>Snord116<\/jats:italic>\n                    and\n                    <jats:italic>Ipw<\/jats:italic>\n                    . Firstly, we examined the same behavioural and cognitive endophenotypes of relevance to psychiatric illness in the PWS-cr mice. Unlike the PWS-IC mice, PWS-cr exhibit no differences in locomotor activity, sensory-motor gating, and attention. RNA-seq analysis of neonatal whole brain tissue revealed a greater number of transcriptional changes between PWS-IC and wild-type littermates than between PWS-cr and wild-type littermates. Moreover, the differentially expressed genes in the PWS-IC brain were enriched for GWAS variants of episodes of psychotic illness but, interestingly, not schizophrenia. These data illustrate the molecular pathways that may underpin psychotic illness in PWS and have implications for potential therapeutic interventions.\n                  <\/jats:p>","DOI":"10.1038\/s41398-021-01561-x","type":"journal-article","created":{"date-parts":[[2021,8,20]],"date-time":"2021-08-20T03:04:32Z","timestamp":1629428672000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":11,"title":["Comparison of mouse models reveals a molecular distinction between psychotic illness in PWS and schizophrenia"],"prefix":"10.1038","volume":"11","author":[{"given":"Simona K.","family":"Zahova","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Trevor","family":"Humby","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jennifer R.","family":"Davies","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joanne E.","family":"Morgan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7587-5712","authenticated-orcid":false,"given":"Anthony R.","family":"Isles","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,8,20]]},"reference":[{"key":"1561_CR1","doi-asserted-by":"publisher","first-page":"721","DOI":"10.1042\/BST20130008","volume":"41","author":"GI McNamara","year":"2013","unstructured":"McNamara GI, Isles AR. 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