Single-cell multiomics of neuron activation reveals context-specific genetics of brain disorders

L Lifan Liang (Department of Human Genetics, The University of Chicago, Chicago, IL, USA.) S Siwei Zhang Z Zicheng Wang (Department of Human Genetics, The University of Chicago, Chicago, IL, USA.) H Hanwen Zhang C Chuxuan Li (Center for Psychiatric Genetics, Endeavor Health Research Institute, Evanston, IL, USA.) C Christina Thapa E Emily K. Oh (Center for Psychiatric Genetics, Endeavor Health Research Institute, Evanston, IL, USA.) D David Sirkin (Center for Psychiatric Genetics, Endeavor Health Research Institute, Evanston, IL, USA.) X Xiaotong Sun A Alexandra Barishman (Center for Psychiatric Genetics, Endeavor Health Research Institute, Evanston, IL, USA.) A Ada McCarroll (Center for Psychiatric Genetics, Endeavor Health Research Institute, Evanston, IL, USA.) A Alexandra C. Duhe (Center for Psychiatric Genetics, Endeavor Health Research Institute, Evanston, IL, USA.) S Sheng Qian (Department of Human Genetics, The University of Chicago, Chicago, IL, USA.) X Xiaoyuan Zhong (Department of Human Genetics, The University of Chicago, Chicago, IL, USA.) B Brendan Jamison W Whitney Wood (Center for Psychiatric Genetics, Endeavor Health Research Institute, Evanston, IL, USA.) A Alena Kozlova Z Zhiping P. Pang A Alan R. Sanders X Xin He J Jubao Duan

Abstract

Most causal variants for neuropsychiatric disorders (NPD) remain unknown. A major hurdle is that disease variants may act in specific contexts, such as during neuronal activation, which is difficult to study in vivo at the population level. We profiled single-nucleus neuron-activation multiomics in human induced pluripotent stem cell–derived neurons from 100 donors, revealing the NPD-relevant transcriptomic and epigenomic landscape of neuronal activation. We identified abundant genetic variants associated with activity-dependent gene expression and chromatin accessibility, the latter explaining larger proportions of NPD heritability. Integrating multiomics data with genome-wide association studies further revealed NPD risk variants and genes with effects detected only upon stimulation, such as activity-dependent cholesterol metabolism. Our work highlights the power of cell stimulation to reveal context-specific “hidden” genetic effects.

Article Details

Journal Science
Volume / Issue Vol. 392, Issue 6805
Published June 25, 2026
ISSN 0036-8075
Publisher American Association for the Advancement of Science

Journal Info

Science

American Association for the Advancement of Science

ISSN: 0036-8075 Social Sciences

Authors (21)

L

Lifan Liang

Department of Human Genetics, The University of Chicago, Chicago, IL, USA.

S

Siwei Zhang

Z

Zicheng Wang

Department of Human Genetics, The University of Chicago, Chicago, IL, USA.

H

Hanwen Zhang

C

Chuxuan Li

Center for Psychiatric Genetics, Endeavor Health Research Institute, Evanston, IL, USA.

C

Christina Thapa

E

Emily K. Oh

Center for Psychiatric Genetics, Endeavor Health Research Institute, Evanston, IL, USA.

D

David Sirkin

Center for Psychiatric Genetics, Endeavor Health Research Institute, Evanston, IL, USA.

X

Xiaotong Sun

A

Alexandra Barishman

Center for Psychiatric Genetics, Endeavor Health Research Institute, Evanston, IL, USA.

A

Ada McCarroll

Center for Psychiatric Genetics, Endeavor Health Research Institute, Evanston, IL, USA.

A

Alexandra C. Duhe

Center for Psychiatric Genetics, Endeavor Health Research Institute, Evanston, IL, USA.

S

Sheng Qian

Department of Human Genetics, The University of Chicago, Chicago, IL, USA.

X

Xiaoyuan Zhong

Department of Human Genetics, The University of Chicago, Chicago, IL, USA.

B

Brendan Jamison

W

Whitney Wood

Center for Psychiatric Genetics, Endeavor Health Research Institute, Evanston, IL, USA.

A

Alena Kozlova

Z

Zhiping P. Pang

A

Alan R. Sanders

X

Xin He

J

Jubao Duan