ArticleHuman molecular genetics2016
Behavioural abnormalities in a novel mouse model for Silver Russell Syndrome.
Article in Human molecular genetics, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 16 papers.
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Who cites it
16 citing papers in PubMed, 27 citations in OpenAlex.
- How Imprinted Genes Shape Nurturing Behaviours and Neural Circuits.Brain, behavior and evolution · 2026Review
- Mom genes and dad genes: genomic imprinting in the regulation of social behaviors.Epigenomics · 2025Review
- Imprinting disorders as a window to understand pediatric feeding disorders.Orphanet journal of rare diseases · 2025Article
- Protein restriction during pregnancy alters Cdkn1c silencing, dopamine circuitry and offspring behaviour without changing expression of key neuronal marker genes.Scientific reports · 2024Article
- Epigenetic Mechanisms of ART-Related Imprinting Disorders: Lessons From iPSC and Mouse Models.Genes · 2021Review
- Case Report: Liraglutide for Weight Management in Beckwith-Wiedemann Syndromic Obesity.Frontiers in endocrinology · 2021Article
- Modeling human epigenetic disorders in mice: Beckwith-Wiedemann syndrome and Silver-Russell syndrome.Disease models & mechanisms · 2020Review
- Role of the imprinted allele of the Cdkn1c gene in mouse neocortical development.Scientific reports · 2020Article
- Wistar Kyoto Rats Display Anhedonia In Consumption but Retain Some Sensitivity to the Anticipation of Palatable Solutions.Frontiers in behavioral neuroscience · 2020Article
- Loss of Imprinting ofInternational journal of molecular sciences · 2018Article
- Handling method alters the hedonic value of reward in laboratory mice.Scientific reports · 2018Article
- Dopaminergic and behavioural changes in a loss-of-imprinting model of Cdkn1c.Genes, brain, and behavior · 2018Article
- Loss of offspring Peg3 reduces neonatal ultrasonic vocalizations and increases maternal anxiety in wild-type mothers.Human molecular genetics · 2018Article
- Territorial Behavior and Social Stability in the Mouse Require Correct Expression of ImprintedFrontiers in behavioral neuroscience · 2018Article
- Visualizing Changes in Cdkn1c Expression Links Early-Life Adversity to Imprint Mis-regulation in Adults.Cell reports · 2017Article
- Impairments in sensory-motor gating and information processing in a mouse model ofBrain and neuroscience advancesArticle
Corrections and comments
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Authors and funding
5 authors at 1 institution in 1 country.
Funding
Abstract
Silver Russell Syndrome (SRS) syndrome is an imprinting disorder involving low birth weight with complex genetics and diagnostics. Some rare SRS patients carry maternally inherited microduplications spanning the imprinted genes CDKN1C, PHLDA2, SLC22A18 and KCNQ1, suggesting that overexpression of one of more of these genes contributes to the SRS phenotype. While this molecular alteration is very rare, feeding difficulties are a very common feature of this condition. Given that SRS children also have very low body mass index, understanding the underpinning biology of the eating disorder is important, as well as potential co-occurring behavioural alterations. Here, we report that a mouse model of this microduplication exhibits a number of behavioural deficits. The mice had a blunted perception of the palatability of a given foodstuff. This perception may underpin the fussiness with food. We additionally report hypoactivity, unrelated to anxiety or motoric function, and a deficit in the appropriate integration of incoming sensory information. Importantly, using a second genetic model, we were able to attribute all altered behaviours to elevated expression of a single gene, Cdkn1c. This is the first report linking elevated Cdkn1c to altered behaviour in mice. Importantly, the findings from our study may have relevance for SRS and highlight a potentially underreported aspect of this disorder.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.