Fragmentation signatures in cancer patients resemble those of patients with vascular or autoimmune diseases

S Samuel D. Curtis (Department of Pharmacology and Molecular Sciences, Johns Hopkins University School of Medicine) T Tingshan Liu (Department of Biomedical Engineering, Johns Hopkins University) Y Yuxin Bai (Department of Biomedical Engineering, Johns Hopkins University) Y Yuxuan Wang S Sambit Panda (Department of Biomedical Engineering, Johns Hopkins University) A Adam Li (Department of Computer Science, Columbia University) H Haoyin Xu (Department of Biomedical Engineering, Johns Hopkins University) E Eliza O’Reilly (Department of Applied Mathematics and Statistic, Johns Hopkins University) L Lisa Dobbyn (Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine) M Maria Popoli (Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine) J Janine Ptak (Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine) N Natalie Silliman (Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine) C Chris Thoburn (Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine) J Jeanne Tie (Division of Personalized Oncology, Walter and Eliza Hall Institute of Medical Research) P Peter Gibbs (Division of Personalized Oncology, Walter and Eliza Hall Institute of Medical Research) L Lan T. Ho-Pham (BioMedical Research Center, Pham Ngoc Thach University of Medicine) B Bich N. H. Tran (Saigon Precision Medicine Research Center) T Thach S. Tran (Saigon Precision Medicine Research Center) T Tuan V. Nguyen (Saigon Precision Medicine Research Center) M Maximilian F. Konig (The Ludwig Center for Cancer Genetics and Therapeutics, Johns Hopkins University School of Medicine) M Michelle Petri (Division of Rheumatology, Department of Medicine, The Johns Hopkins University School of Medicine) A Antony Rosen (Division of Rheumatology, Department of Medicine, The Johns Hopkins University School of Medicine) C Christopher A. Mecoli (Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine) A Ami A. Shah (Division of Rheumatology, Department of Medicine, The Johns Hopkins University School of Medicine) C Chetan Bettegowda K Kenneth W. Kinzler (Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine) N Nickolas Papadopoulos (Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine) J Joshua T. Vogelstein (Department of Biomedical Engineering, Johns Hopkins University) B Bert Vogelstein (Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine) C Christopher Douville (Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine) F Frederikus A. Klok J Jan Beyer-Westendorf L Luis Jara-Palomares W Walter Ageno M Mike J.L. Peters A Aurélien Delluc M Myron Best N Noori Guman H Hans-Martin Otten F Frits Mulder (Department of Vascular Medicine, Amsterdam Cardiovascular Sciences, Amsterdam University Medical Centers, University of Amsterdam) N Nick van Es (Department of Vascular Medicine, Amsterdam Cardiovascular Sciences, Amsterdam University Medical Centers, University of Amsterdam)

Abstract

Multiple case-controlled studies have shown that analyzing fragmentation patterns in plasma cell-free DNA (cfDNA) can distinguish individuals with cancer from healthy controls. However, there have been few studies that investigate various types of cfDNA fragmentomics patterns in individuals with other diseases. We therefore developed a comprehensive statistic, called fragmentation signatures, that integrates the distributions of fragment positioning, fragment length, and fragment end-motifs in cfDNA. We found that individuals with venous thromboembolism, systemic lupus erythematosus, dermatomyositis, or scleroderma have cfDNA fragmentation signatures that closely resemble those found in individuals with advanced cancers. Furthermore, these signatures were highly correlated with increases in inflammatory markers in the blood. We demonstrate that these similarities in fragmentation signatures lead to high rates of false positives in individuals with autoimmune or vascular disease when evaluated using conventional binary classification approaches for multicancer earlier detection (MCED). To address this issue, we introduced a multiclass approach for MCED that integrates fragmentation signatures with protein biomarkers and achieves improved specificity in individuals with autoimmune or vascular disease while maintaining high sensitivity. Though these data put substantial limitations on the specificity of fragmentomics-based tests for cancer diagnostics, they also offer ways to improve the interpretability of such tests. Moreover, we expect these results will lead to a better understanding of the process—most likely inflammatory—from which abnormal fragmentation signatures are derived.

Article Details

Volume / Issue Vol. 122, Issue 34
Published August 26, 2025
ISSN 0027-8424
Publisher National Academy of Sciences

Authors (41)

S

Samuel D. Curtis

Department of Pharmacology and Molecular Sciences, Johns Hopkins University School of Medicine

T

Tingshan Liu

Department of Biomedical Engineering, Johns Hopkins University

Y

Yuxin Bai

Department of Biomedical Engineering, Johns Hopkins University

Y

Yuxuan Wang

S

Sambit Panda

Department of Biomedical Engineering, Johns Hopkins University

A

Adam Li

Department of Computer Science, Columbia University

H

Haoyin Xu

Department of Biomedical Engineering, Johns Hopkins University

E

Eliza O’Reilly

Department of Applied Mathematics and Statistic, Johns Hopkins University

L

Lisa Dobbyn

Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine

M

Maria Popoli

Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine

J

Janine Ptak

Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine

N

Natalie Silliman

Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine

C

Chris Thoburn

Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine

J

Jeanne Tie

Division of Personalized Oncology, Walter and Eliza Hall Institute of Medical Research

P

Peter Gibbs

Division of Personalized Oncology, Walter and Eliza Hall Institute of Medical Research

L

Lan T. Ho-Pham

BioMedical Research Center, Pham Ngoc Thach University of Medicine

B

Bich N. H. Tran

Saigon Precision Medicine Research Center

T

Thach S. Tran

Saigon Precision Medicine Research Center

T

Tuan V. Nguyen

Saigon Precision Medicine Research Center

M

Maximilian F. Konig

The Ludwig Center for Cancer Genetics and Therapeutics, Johns Hopkins University School of Medicine

M

Michelle Petri

Division of Rheumatology, Department of Medicine, The Johns Hopkins University School of Medicine

A

Antony Rosen

Division of Rheumatology, Department of Medicine, The Johns Hopkins University School of Medicine

C

Christopher A. Mecoli

Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine

A

Ami A. Shah

Division of Rheumatology, Department of Medicine, The Johns Hopkins University School of Medicine

C

Chetan Bettegowda

K

Kenneth W. Kinzler

Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine

N

Nickolas Papadopoulos

Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine

J

Joshua T. Vogelstein

Department of Biomedical Engineering, Johns Hopkins University

B

Bert Vogelstein

Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine

C

Christopher Douville

Department of Oncology, the Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine

F

Frederikus A. Klok

J

Jan Beyer-Westendorf

L

Luis Jara-Palomares

W

Walter Ageno

M

Mike J.L. Peters

A

Aurélien Delluc

M

Myron Best

N

Noori Guman

H

Hans-Martin Otten

F

Frits Mulder

Department of Vascular Medicine, Amsterdam Cardiovascular Sciences, Amsterdam University Medical Centers, University of Amsterdam

N

Nick van Es

Department of Vascular Medicine, Amsterdam Cardiovascular Sciences, Amsterdam University Medical Centers, University of Amsterdam