title: Behavioural characteristics of the Prader-Willi syndrome related biallelic Snord116 mouse model.
abstract: Prader-Willi syndrome (PWS) is the predominant genetic cause of obesity in humans and is associated with several behavioural phenotypes such as altered motoric function, reduced activity, and learning disabilities. It can include mood instability and, in some cases, psychotic episodes. Recently, the Snord116 gene has been associated with the development of PWS, however, it's contribution to the behavioural aspects of the disease are unknown. Here we show that male and female mice lacking Snord116 on both alleles exhibit normal motor behaviours and exploration but do display task-dependent alterations to locomotion and anxiety-related behaviours. Sociability is well developed in Snord116 deficient mice as are social recognition memory, spatial working memory, and fear-associated behaviours. No sex-specific effects were found. In conclusion, the biallelic Snord116 deficiency mouse model exhibits particular endophenotypes with some relevance to PWS, suggesting partial face validity for the syndrome.
authors.affiliation: Neuroscience Research Australia, Randwick, NSW 2031, Australia; Schizophrenia Research Institute, Darlinghurst, NSW 2010, Australia.
authors.forename: Jerzy
authors.initials: J
authors.lastname: Zieba
authors.affiliation: Neuroscience Research Australia, Randwick, NSW 2031, Australia.
authors.forename: Jac Kee
authors.initials: JK
authors.lastname: Low
authors.affiliation: Neuroscience Division, Garvan Institute of Medical Research, Darlinghurst, NSW 2010, Australia.
authors.forename: Louise
authors.initials: L
authors.lastname: Purtell
authors.affiliation: Neuroscience Division, Garvan Institute of Medical Research, Darlinghurst, NSW 2010, Australia.
authors.forename: Yue
authors.initials: Y
authors.lastname: Qi
authors.affiliation: Neuroscience Division, Garvan Institute of Medical Research, Darlinghurst, NSW 2010, Australia.
authors.forename: Lesley
authors.initials: L
authors.lastname: Campbell
authors.affiliation: Neuroscience Division, Garvan Institute of Medical Research, Darlinghurst, NSW 2010, Australia.
authors.forename: Herbert
authors.initials: H
authors.lastname: Herzog
authors.affiliation: Neuroscience Research Australia, Randwick, NSW 2031, Australia; Schizophrenia Research Institute, Darlinghurst, NSW 2010, Australia; School of Medical Sciences, University of New South Wales, NSW 2052, Australia. Electronic address: t.karl@neura.edu.au.
authors.forename: Tim
authors.initials: T
authors.lastname: Karl
mesh_terms: D000483:Alleles; D000818:Animals; D001007:Anxiety / Q000523:psychology; D001522:Behavior, Animal; D003071:Cognition; D005106:Exploratory Behavior; D005260:Female; D008297:Male; D051379:Mice; D018345:Mice, Knockout; D009043:Motor Activity / Q000235:genetics; D011218:Prader-Willi Syndrome / Q000235:genetics* / Q000523:psychology*; D020537:RNA, Small Nucleolar / Q000235:genetics*; D021641:Recognition, Psychology; D012919:Social Behavior
chemical_list: D020537:RNA, Small Nucleolar; C000609194:SNORD116 RNA, mouse
keywords: Anxiety; Behaviour; Cognition; HBII-85; MBII-85; Mouse model; Prader–Willi syndrome; SNORD116; Social preference