Characterizing and controlling CRISPR repair outcomes in nondividing human cells

G Gokul N. Ramadoss S Samali J. Namaganda M Manasi M. Kumar J Jennifer R. Hamilton R Rohit Sharma K Karena G. Chow L Luke A. Workley B Bria L. Macklin M Mengyuan Sun A Alvin S. Ha J Jia-Cheng Liu C Christof Fellmann H Hannah L. Watry P Philip H. Dierks R Rudra S. Bose J Julianne Jin B Barbara S. Perez C Cindy R. Sandoval Espinoza M Madeline P. Matia S Serena H. Lu L Luke M. Judge B Brian R. Shy A André Nussenzweig B Britt Adamson N Niren Murthy J Jennifer A. Doudna M Martin Kampmann B Bruce R. Conklin

Abstract

Abstract Genome editing is poised to revolutionize treatment of genetic diseases, but poor understanding and control of DNA repair outcomes hinders its therapeutic potential. DNA repair is especially understudied in nondividing cells like neurons, limiting the efficiency and precision of genome editing in many clinically relevant tissues. Here, we address this barrier by using induced pluripotent stem cells (iPSCs) and iPSC-derived neurons to examine how postmitotic human neurons repair Cas9-induced DNA damage. CRISPR editing outcomes differ dramatically in neurons compared to genetically identical dividing cells: neurons take longer to fully resolve this damage, and upregulate non-canonical DNA repair factors in the process. Manipulating this response with chemical or genetic perturbations allows us to direct DNA repair toward desired editing outcomes in nondividing human neurons, cardiomyocytes, and primary T cells. By studying DNA repair in clinically relevant cells, we reveal unforeseen challenges and opportunities for precise therapeutic editing.

Article Details

Volume / Issue Vol. 16, Issue 1
Published November 17, 2025
ISSN 2041-1723
Publisher Nature Portfolio

Journal Info

Nature Communications

Nature Portfolio

ISSN: 2041-1723 Open Access Life Sciences

Authors (28)

G

Gokul N. Ramadoss

S

Samali J. Namaganda

M

Manasi M. Kumar

J

Jennifer R. Hamilton

R

Rohit Sharma

K

Karena G. Chow

L

Luke A. Workley

B

Bria L. Macklin

M

Mengyuan Sun

A

Alvin S. Ha

J

Jia-Cheng Liu

C

Christof Fellmann

H

Hannah L. Watry

P

Philip H. Dierks

R

Rudra S. Bose

J

Julianne Jin

B

Barbara S. Perez

C

Cindy R. Sandoval Espinoza

M

Madeline P. Matia

S

Serena H. Lu

L

Luke M. Judge

B

Brian R. Shy

A

André Nussenzweig

B

Britt Adamson

N

Niren Murthy

J

Jennifer A. Doudna

M

Martin Kampmann

B

Bruce R. Conklin