Significance Mutations in the FOXG1 gene cause a rare neurodevelopmental disorder called “FOXG1-syndrome”. FOXG1 is a key instructor of the developing telencephalon, and patients present with various phenotypes including microcephaly,… Click to show full abstract
Significance Mutations in the FOXG1 gene cause a rare neurodevelopmental disorder called “FOXG1-syndrome”. FOXG1 is a key instructor of the developing telencephalon, and patients present with various phenotypes including microcephaly, seizures, and cognitive dysfunctions. We explored the pleiotropy of molecular changes underlying neuronal abnormalities upon loss of FOXG1 and provide the multiomics data set exploring functions of mouse FOXG1 at the chromatin level. We report changes in the epigenetic landscape, impacting the accessibility of chromatin regions and activation of enhancers, upon reduced FOXG1 expression that alter the transcriptome in mouse hippocampal neurons. We identified cooperation of FOXG1 with the proneuronal transcription factor NEUROD1 and HDACs in controlling gene transcription, indicating complex and multimodal FOXG1 functions regarding neuronal maturation and function.
Journal Title: Proceedings of the National Academy of Sciences of the United States of America
Year Published: 2023
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