Altered Behavior and Neuronal Activity with Paternal Snord116 Deletion

Daniel S Scott1, Violeta Zaric2, Carol A Tamminga1,3

  • 1Department of Psychiatry, UT Southwestern Medical Center, Dallas, TX 75390, USA.

Genes
|August 28, 2025
PubMed

Insights

Deletion of the Snord116 gene in Prader-Willi Syndrome (PWS) mice increases neural activity in key brain regions. This genetic change is linked to psychosis-like behaviors, particularly under stress.

Area of Science:

  • Neuroscience
  • Genetics
  • Behavioral Science

Background:

  • Prader-Willi Syndrome (PWS) is a neurodevelopmental disorder characterized by behavioral issues, including psychosis.
  • PWS is linked to the deletion or silencing of the paternal copy of chromosome 15q11-13.
  • The Snord116 gene within this region plays a critical role in PWS manifestation.

Purpose of the Study:

  • To investigate how the microdeletion of the paternal Snord116 gene affects neural activity in psychosis-associated brain regions.
  • To evaluate the impact of Snord116 deletion on psychosis-like behaviors in a mouse model.

Main Methods:

  • Assessed regional neural activity using c-Fos expression in specific brain areas of mice.
  • Evaluated behavioral phenotypes related to psychosis in mice with Snord116 microdeletion.
  • Utilized stressful paradigms to identify behavioral deficits and altered fear conditioning.

Main Results:

  • Snord116 deletion led to increased c-Fos expression in the hippocampus and anterior cingulate cortex.
  • Mice lacking Snord116 exhibited psychosis-consistent behaviors, including sensorimotor gating deficits.
  • Augmented contextual and cued fear conditioning was observed in Snord116-deleted mice.

Conclusions:

  • The study implicates Snord116 targets in the development of psychosis-like states.
  • The observed effects of Snord116 deletion on neural activity and behavior show regional specificity.
Abstract

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