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Postnatal Arx transcriptional activity regulates functional properties of PV interneurons

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Postnatal Arx transcriptional activity regulates functional properties of PV interneurons. / Joseph, Donald J; von Deimling, Markus; Hasegawa, Yuiko; Cristanco, Ana G; Ahrens-Nicklas, Rebecca C ; Rogers, Stephanie L; Risbud, Rashmi; McCoy, Almedia J; Marsh, Eric D.

In: ISCIENCE, Vol. 24, No. 1, 101999, 22.01.2021, p. 101999.

Research output: SCORING: Contribution to journal › SCORING: Journal article › Research › peer-review

Harvard

Joseph, DJ, von Deimling, M, Hasegawa, Y, Cristanco, AG, Ahrens-Nicklas, RC, Rogers, SL, Risbud, R, McCoy, AJ & Marsh, ED 2021, 'Postnatal Arx transcriptional activity regulates functional properties of PV interneurons', ISCIENCE, vol. 24, no. 1, 101999, pp. 101999. https://doi.org/10.1016/j.isci.2020.101999

APA

Joseph, D. J., von Deimling, M., Hasegawa, Y., Cristanco, A. G., Ahrens-Nicklas, R. C., Rogers, S. L., Risbud, R., McCoy, A. J., & Marsh, E. D. (2021). Postnatal Arx transcriptional activity regulates functional properties of PV interneurons. ISCIENCE, 24(1), 101999. [101999]. https://doi.org/10.1016/j.isci.2020.101999

Vancouver

Joseph DJ, von Deimling M, Hasegawa Y, Cristanco AG, Ahrens-Nicklas RC, Rogers SL et al. Postnatal Arx transcriptional activity regulates functional properties of PV interneurons. ISCIENCE. 2021 Jan 22;24(1):101999. 101999. https://doi.org/10.1016/j.isci.2020.101999

Bibtex

@article{208e6cc529394ec780be9a6e6134aea5,
title = "Postnatal Arx transcriptional activity regulates functional properties of PV interneurons",
abstract = "The transcription factor Aristaless-related X-linked gene (Arx) is a monogenic factor in early onset epileptic encephalopathies (EOEEs) and a fundamental regulator of early stages of brain development. However, Arx expression persists in mature GABAergic neurons with an unknown role. To address this issue, we generated a conditional knockout (CKO) mouse in which postnatal Arx was ablated in parvalbumin interneurons (PVIs). Electroencephalogram (EEG) recordings in CKO mice revealed an increase in theta oscillations and the occurrence of occasional seizures. Behavioral analysis uncovered an increase in anxiety. Genome-wide sequencing of fluorescence activated cell sorted (FACS) PVIs revealed that Arx impinged on network excitability via genes primarily associated with synaptic and extracellular matrix pathways. Whole-cell recordings revealed prominent hypoexcitability of various intrinsic and synaptic properties. These results revealed important roles for postnatal Arx expression in PVIs in the control of neural circuits and that dysfunction in those roles alone can cause EOEE-like network abnormalities.",
author = "Joseph, {Donald J} and {von Deimling}, Markus and Yuiko Hasegawa and Cristanco, {Ana G} and Ahrens-Nicklas, {Rebecca C} and Rogers, {Stephanie L} and Rashmi Risbud and McCoy, {Almedia J} and Marsh, {Eric D}",
year = "2021",
month = jan,
day = "22",
doi = "10.1016/j.isci.2020.101999",
language = "English",
volume = "24",
pages = "101999",
journal = "ISCIENCE",
issn = "2589-0042",
publisher = "Elsevier Inc.",
number = "1",

}

RIS

TY - JOUR

T1 - Postnatal Arx transcriptional activity regulates functional properties of PV interneurons

AU - Joseph, Donald J

AU - von Deimling, Markus

AU - Hasegawa, Yuiko

AU - Cristanco, Ana G

AU - Ahrens-Nicklas, Rebecca C

AU - Rogers, Stephanie L

AU - Risbud, Rashmi

AU - McCoy, Almedia J

AU - Marsh, Eric D

PY - 2021/1/22

Y1 - 2021/1/22

N2 - The transcription factor Aristaless-related X-linked gene (Arx) is a monogenic factor in early onset epileptic encephalopathies (EOEEs) and a fundamental regulator of early stages of brain development. However, Arx expression persists in mature GABAergic neurons with an unknown role. To address this issue, we generated a conditional knockout (CKO) mouse in which postnatal Arx was ablated in parvalbumin interneurons (PVIs). Electroencephalogram (EEG) recordings in CKO mice revealed an increase in theta oscillations and the occurrence of occasional seizures. Behavioral analysis uncovered an increase in anxiety. Genome-wide sequencing of fluorescence activated cell sorted (FACS) PVIs revealed that Arx impinged on network excitability via genes primarily associated with synaptic and extracellular matrix pathways. Whole-cell recordings revealed prominent hypoexcitability of various intrinsic and synaptic properties. These results revealed important roles for postnatal Arx expression in PVIs in the control of neural circuits and that dysfunction in those roles alone can cause EOEE-like network abnormalities.

AB - The transcription factor Aristaless-related X-linked gene (Arx) is a monogenic factor in early onset epileptic encephalopathies (EOEEs) and a fundamental regulator of early stages of brain development. However, Arx expression persists in mature GABAergic neurons with an unknown role. To address this issue, we generated a conditional knockout (CKO) mouse in which postnatal Arx was ablated in parvalbumin interneurons (PVIs). Electroencephalogram (EEG) recordings in CKO mice revealed an increase in theta oscillations and the occurrence of occasional seizures. Behavioral analysis uncovered an increase in anxiety. Genome-wide sequencing of fluorescence activated cell sorted (FACS) PVIs revealed that Arx impinged on network excitability via genes primarily associated with synaptic and extracellular matrix pathways. Whole-cell recordings revealed prominent hypoexcitability of various intrinsic and synaptic properties. These results revealed important roles for postnatal Arx expression in PVIs in the control of neural circuits and that dysfunction in those roles alone can cause EOEE-like network abnormalities.

U2 - 10.1016/j.isci.2020.101999

DO - 10.1016/j.isci.2020.101999

M3 - SCORING: Journal article

VL - 24

SP - 101999

JO - ISCIENCE

JF - ISCIENCE

SN - 2589-0042

IS - 1

M1 - 101999

ER -