Compensation between FOXP transcription factors maintains proper striatal function

被引：3

作者：

Ahmed, Newaz I. ^{[1
,2
]}

Khandelwal, Nitin ^{[1
,2
]}

Anderson, Ashley G. ^{[1
,3
,4
]}

Oh, Emily ^{[1
,2
]}

Vollmer, Rachael M. ^{[1
,2
]}

Kulkarni, Ashwinikumar ^{[1
,2
]}

Gibson, Jay R. ^{[1
,2
]}

Konopka, Genevieve ^{[1
,2
]}

机构：

[1] UT Southwestern Med Ctr, Dept Neurosci, Dallas, TX 75390 USA

[2] UT Southwestern Med Ctr, Peter ODonnell Jr Brain Inst, Dallas, TX 75390 USA

[3] Baylor Coll Med, Dept Mol & Human Genet, Houston, TX 77030 USA

[4] Texas Childrens Hosp, Jan & Dan Duncan Neurol Res Inst, Houston, TX 77030 USA

来源：

CELL REPORTS | 2024年 / 43卷 / 05期

关键词：

BASAL GANGLIA CIRCUITS; PROTEIN-KINASE-C; VOCAL COMMUNICATION; ADENYLYL-CYCLASE; FORKHEAD-DOMAIN; SEVERE SPEECH; GENE; EXPRESSION; LANGUAGE; IDENTIFICATION;

D O I：

10.1016/j.celrep.2024.114257

中图分类号：

Q2 [细胞生物学];

学科分类号：

071009 ; 090102 ;

摘要：

Spiny projection neurons (SPNs) of the striatum are critical in integrating neurochemical information to coordinate motor and reward-based behavior. Mutations in the regulatory transcription factors expressed in SPNs can result in neurodevelopmental disorders (NDDs). Paralogous transcription factors Foxp1 and Foxp2, , which are both expressed in the dopamine receptor 1 (D1) expressing SPNs, are known to have variants implicated in NDDs. Utilizing mice with a D1-SPN-specific loss of Foxp1, , Foxp2, , or both and a combination of behavior, electrophysiology, and cell-type-specific genomic analysis, loss of both genes results in impaired motor and social behavior as well as increased firing of the D1-SPNs. Differential gene expression analysis implicates genes involved in autism risk, electrophysiological properties, and neuronal development and function. Viral-mediated re-expression of Foxp1 into the double knockouts is sufficient to restore electrophysiological and behavioral deficits. These data indicate complementary roles between Foxp1 and Foxp2 in the D1-SPNs.

引用

页数：25

共 129 条

[21] Foxp2 controls synaptic wiring of corticostriatal circuits and vocal communication by opposing Mef2c [J].