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.

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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
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article

Cell-type-specific roles of FOXP1 in the excitatory neuronal lineage during early neocortical murine development

Elliot Outland, Miranda Jankovic, Geneviève Konopka, Nitin Khandelwal et al.
9 citations
Cell Reports
Single-cell and spatial transcriptomics
7.25
article

Cell-type-specific roles of FOXP1 in the excitatory neuronal lineage during early neocortical murine development

Elliot Outland, Miranda Jankovic, Geneviève Konopka, Nitin Khandelwal, Fatma Ayhan, Ana Ortiz, Matthew J. Harper
article en
9 citations

Abstract

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.

Cell ReportsVol. 44(3)
Southwestern Medical Center (US), Southwestern Medical Center (US), The University of Texas Southwestern Medical Center (US)
Openalex Percentile: Top 3%
Single-cell and spatial transcriptomics
7.25
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Cell-type-specific roles of FOXP1 in the excitatory neuronal lineage during early neocortical murine development — Elliot Outland, Miranda Jankovic, et al. · Cell Reports (2025) | TGRS Research Map | TGRS