Behavioral Phenotypes of Foxg1 Heterozygous Mice

Skyler Younger, Sydney Boutros, Francesca Cargnin, Shin Jeon, Jae Lee, Soo Kyung Lee, Jacob Raber

Research output: Contribution to journalArticlepeer-review

Abstract

FOXG1 syndrome (FS, aka a congenital variant of Rett syndrome) is a recently defined rare and devastating neurodevelopmental disorder characterized by various symptoms, including severe intellectual disability, autistic features, involuntary, and continuous jerky movements, feeding problems, sleep disturbances, seizures, irritability, and excessive crying. FS results from mutations in a single allele of the FOXG1 gene, leading to impaired FOXG1 function. Therefore, in establishing mouse models for FS, it is important to test if heterozygous (HET) mutation in the Foxg1 gene, mimicking genotypes of the human FS individuals, also manifests phenotypes similar to their symptoms. We analyzed HET mice with a null mutation allele in a single copy of Foxg1, and found that they show various phenotypes resembling the symptoms of the human FS individuals. These include increased anxiety in the open field as well as impairment in object recognition, motor coordination, and fear learning and contextual and cued fear memory. Our results suggest that Foxg1 HET mice recapitulate at least some symptoms of the human FS individuals.

Original languageEnglish (US)
Article number927296
JournalFrontiers in Pharmacology
Volume13
DOIs
StatePublished - Jun 8 2022

Keywords

  • contextual fear memory
  • cued fear memory
  • object recognition
  • open field
  • wire hang

ASJC Scopus subject areas

  • Pharmacology
  • Pharmacology (medical)

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