Abstract 4370075: Leveraging Single-Nucleus Chromatin Accessibility and Transcriptome Profiling to Define the Regulatory Landscape of Dilated Cardiomyopathy

L Logan Dunkenberger (Stanford University, Stanford, California, United States) C Casey Gilles (Stanford University, Stanford, California, United States) N Nadjet Belbachir (Stanford Cardiovascular Institute, Stanford University, CA (L.R., N.B., H.Z., R.H., W.Z., X.W., M.X., J.G., H.Y.K., T.L., J.S., J.C.W.).) E Eyal Metzl Raz (Stanford University, Stanford, California, United States) L Lasemahang Limbu (Stanford University, Stanford, California, United States) S Spencer Bonham (Stanford University, Stanford, California, United States) J Jennifer Arthur (Stanford University, Stanford, California, United States) A Alex Dalal (Stanford University, Menlo Park, California, United States) M Maryam Kay (Stanford University, Stanford, California, United States) R Rachel Baum (Stanford University, Stanford, California, United States) E Evgenios Neofytou (Stanford University, Stanford, California, United States) R Rohin Ramchandani (Stanford University, Stanford, California, United States) M Mo Ameen (Stanford University, Stanford, California, United States) K Kevin Wang (Department of Neurobiology and Behavior, University of California) M Michael Fischbein (Stanford University, Menlo Park, California, United States) Y Y Joseph Woo (STANFORD UNIV SCHOOL MEDICINE, Stanford, California, United States) I Ioannis Karakikes (Stanford University, Stanford, California, United States)

Abstract

Introduction: While genetic variants linked to dilated cardiomyopathy (DCM) are increasingly well characterized, the molecular mechanisms driving pathogenesis remain poorly understood. Traditional bulk and in vitro methods lack the resolution to capture the cellular heterogeneity and complexity of DCM. Here, we use single-nucleus multiome chromatin accessibility and RNA sequencing to resolve cell-type-specific gene regulatory changes in genetic DCM. Methods: We performed 10X Genomics Multiome (simultaneous RNA and chromatin accessibility) profiling on nuclei isolated from left ventricular tissue of 13 DCM patients with diverse genetic etiologies ( LMNA , PLN , RBM20 , TNNT2 , TTN ) and 5 non-failing hearts. Integrated analysis identified 12 distinct cardiac cell types based on joint transcriptomic and epigenomic signatures. Intercellular communication networks were inferred using CellChat. We leveraged paired RNA and chromatin accessibility data to elucidate potential enhancer-gene interactions driving disease-associated cell state changes. Results: We observed disease-specific alterations in cellular composition and signaling networks across DCM genetic etiologies. Integrated chromatin accessibility and gene expression data revealed changes in the gene regulatory networks of cardiomyocytes, fibroblasts, and endothelial cells. Notably, the transcription factor PRRX1 emerged as a key differentially regulated gene in LMNA -related DCM. Functional assays in iPSC-derived cardiomyocytes demonstrated that PRRX1 knockdown significantly improved calcium handling and reduced arrythmia incidence. Conclusions: This single nucleus multiomic atlas of genetic DCM provides insights into cell-type-specific gene regulation and cell communication programs associated with disease states. Our findings highlight PRRX1 as a potential therapeutic target in LMNA -related DCM and underscore the value of multiomic profiling for uncovering the regulatory basis of cardiomyopathies.

Article Details

Journal Circulation
Volume / Issue Vol. 152, Issue Suppl_3
Published November 04, 2025
ISSN 0009-7322
Publisher Lippincott Williams & Wilkins

Journal Info

Circulation

Lippincott Williams & Wilkins

ISSN: 0009-7322 Health Sciences

Authors (17)

L

Logan Dunkenberger

Stanford University, Stanford, California, United States

C

Casey Gilles

Stanford University, Stanford, California, United States

N

Nadjet Belbachir

Stanford Cardiovascular Institute, Stanford University, CA (L.R., N.B., H.Z., R.H., W.Z., X.W., M.X., J.G., H.Y.K., T.L., J.S., J.C.W.).

E

Eyal Metzl Raz

Stanford University, Stanford, California, United States

L

Lasemahang Limbu

Stanford University, Stanford, California, United States

S

Spencer Bonham

Stanford University, Stanford, California, United States

J

Jennifer Arthur

Stanford University, Stanford, California, United States

A

Alex Dalal

Stanford University, Menlo Park, California, United States

M

Maryam Kay

Stanford University, Stanford, California, United States

R

Rachel Baum

Stanford University, Stanford, California, United States

E

Evgenios Neofytou

Stanford University, Stanford, California, United States

R

Rohin Ramchandani

Stanford University, Stanford, California, United States

M

Mo Ameen

Stanford University, Stanford, California, United States

K

Kevin Wang

Department of Neurobiology and Behavior, University of California

M

Michael Fischbein

Stanford University, Menlo Park, California, United States

Y

Y Joseph Woo

STANFORD UNIV SCHOOL MEDICINE, Stanford, California, United States

I

Ioannis Karakikes

Stanford University, Stanford, California, United States