Cell-type-specific roles of FOXP1 in the excitatory neuronal lineage during early neocortical murine development
Forkhead box protein P1 (FOXP1), a transcription factor enriched in the neocortex, is associated with autism spectrum disorders (ASDs) and FOXP1 syndrome. Emx1<sup>Cre/+</sup>;Foxp1<sup>fl/fl</sup> conditional deletion (Foxp1 conditional knockout [cKO]) in the mouse cortex leads to overall reduced cortex thickness, alterations in cortical lamination, and changes in the relative thickness of cortical layers. However, the developmental and cell-type-specific mechanisms underlying these changes remained unclear. We find that Foxp1 deletion results in accelerated pseudo-age during early neurogenesis, increased cell cycle exit during late neurogenesis, altered gene expression and chromatin accessibility, and selective migration deficits in a subset of upper-layer neurons. These data explain the postnatal differences observed in cortical layers and relative cortical thickness. We also highlight genes regulated by FOXP1 and their enrichment with high-confidence ASD or synaptic genes. Together, these results underscore a network of neurodevelopmental-disorder-related genes that may serve as potential modulatory targets for postnatal modification relevant to ASDs and FOXP1 syndrome.
Authors
- Elliot Outland (ORCID: https://orcid.org/0000-0002-3869-2992)
- Miranda Jankovic
- Geneviève Konopka (ORCID: https://orcid.org/0000-0002-3363-7302)
- Nitin Khandelwal (ORCID: https://orcid.org/0000-0001-7229-4413)
- Fatma Ayhan (ORCID: https://orcid.org/0000-0001-9717-1727)
- Ana Ortiz (ORCID: https://orcid.org/0000-0001-9047-3634)
- Matthew J. Harper (ORCID: https://orcid.org/0000-0002-0748-5392)
Institutions
- Southwestern Medical Center (US)
- Southwestern Medical Center (US)
- The University of Texas Southwestern Medical Center (US)
Publication Details
- Journal
- Cell Reports
- Published
- 2025-03-01
- DOI
- https://doi.org/10.1016/j.celrep.2025.115384
- Citations
- 9
- Primary Topic
- Single-cell and spatial transcriptomics
- Type
- article
- Field-Weighted Citation Impact
- 7.25