Replicated blood-based biomarkers for myalgic encephalomyelitis not explicable by inactivity

Abstract Myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) is a common female-biased disease. ME/CFS diagnosis is hindered by the absence of biomarkers that are unaffected by patients’ low physical activity level. Our analysis used semi-parametric efficient estimators, an initial Super Lea...

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Main Authors: Sjoerd Viktor Beentjes, Artur Miralles Méharon, Julia Kaczmarczyk, Amanda Cassar, Gemma Louise Samms, Nima S Hejazi, Ava Khamseh, Chris P Ponting
Format: Article
Language:English
Published: Springer Nature 2025-06-01
Series:EMBO Molecular Medicine
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Online Access:https://doi.org/10.1038/s44321-025-00258-8
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author Sjoerd Viktor Beentjes
Artur Miralles Méharon
Julia Kaczmarczyk
Amanda Cassar
Gemma Louise Samms
Nima S Hejazi
Ava Khamseh
Chris P Ponting
author_facet Sjoerd Viktor Beentjes
Artur Miralles Méharon
Julia Kaczmarczyk
Amanda Cassar
Gemma Louise Samms
Nima S Hejazi
Ava Khamseh
Chris P Ponting
author_sort Sjoerd Viktor Beentjes
collection DOAJ
description Abstract Myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) is a common female-biased disease. ME/CFS diagnosis is hindered by the absence of biomarkers that are unaffected by patients’ low physical activity level. Our analysis used semi-parametric efficient estimators, an initial Super Learner fit followed by a one-step correction, three mediators, and natural direct and indirect estimands, to decompose the average effect of ME/CFS status on molecular and cellular traits. For this, we used UK Biobank data for up to 1455 ME/CFS cases and 131,303 controls. Hundreds of traits differed significantly between cases and controls, including 116 significant for both female and male cohorts. These were indicative of chronic inflammation, insulin resistance and liver disease. Nine of 14 traits were replicated in the smaller All-of-Us cohort. Results cannot be explained by restricted activity: via an activity mediator, ME/CFS status significantly affected only 1 of 3237 traits. Individuals with post-exertional malaise show stronger biomarker differences. Single traits could not cleanly distinguish cases from controls. Nevertheless, these results keep alive the future ambition of a blood-based biomarker panel for accurate ME/CFS diagnosis.
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spelling doaj-art-08169c5df2164cd58fadde1693f3d79b2025-08-20T03:46:25ZengSpringer NatureEMBO Molecular Medicine1757-46842025-06-011771868189110.1038/s44321-025-00258-8Replicated blood-based biomarkers for myalgic encephalomyelitis not explicable by inactivitySjoerd Viktor Beentjes0Artur Miralles Méharon1Julia Kaczmarczyk2Amanda Cassar3Gemma Louise Samms4Nima S Hejazi5Ava Khamseh6Chris P Ponting7School of Mathematics and Maxwell Institute for Mathematical Sciences, University of EdinburghSchool of Informatics, University of EdinburghSchool of Mathematics and Maxwell Institute for Mathematical Sciences, University of EdinburghSchool of Informatics, University of EdinburghMRC Human Genetics Unit, Institute of Genetics & Cancer, University of EdinburghHarvard T.H. Chan School of Public Health, Department of BiostatisticsMRC Human Genetics Unit, Institute of Genetics & Cancer, University of EdinburghMRC Human Genetics Unit, Institute of Genetics & Cancer, University of EdinburghAbstract Myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) is a common female-biased disease. ME/CFS diagnosis is hindered by the absence of biomarkers that are unaffected by patients’ low physical activity level. Our analysis used semi-parametric efficient estimators, an initial Super Learner fit followed by a one-step correction, three mediators, and natural direct and indirect estimands, to decompose the average effect of ME/CFS status on molecular and cellular traits. For this, we used UK Biobank data for up to 1455 ME/CFS cases and 131,303 controls. Hundreds of traits differed significantly between cases and controls, including 116 significant for both female and male cohorts. These were indicative of chronic inflammation, insulin resistance and liver disease. Nine of 14 traits were replicated in the smaller All-of-Us cohort. Results cannot be explained by restricted activity: via an activity mediator, ME/CFS status significantly affected only 1 of 3237 traits. Individuals with post-exertional malaise show stronger biomarker differences. Single traits could not cleanly distinguish cases from controls. Nevertheless, these results keep alive the future ambition of a blood-based biomarker panel for accurate ME/CFS diagnosis.https://doi.org/10.1038/s44321-025-00258-8Myalgic EncephalomyelitisSemi-parametric Mediation AnalysisPost-exertional MalaiseUK BiobankAll of Us Biobank
spellingShingle Sjoerd Viktor Beentjes
Artur Miralles Méharon
Julia Kaczmarczyk
Amanda Cassar
Gemma Louise Samms
Nima S Hejazi
Ava Khamseh
Chris P Ponting
Replicated blood-based biomarkers for myalgic encephalomyelitis not explicable by inactivity
EMBO Molecular Medicine
Myalgic Encephalomyelitis
Semi-parametric Mediation Analysis
Post-exertional Malaise
UK Biobank
All of Us Biobank
title Replicated blood-based biomarkers for myalgic encephalomyelitis not explicable by inactivity
title_full Replicated blood-based biomarkers for myalgic encephalomyelitis not explicable by inactivity
title_fullStr Replicated blood-based biomarkers for myalgic encephalomyelitis not explicable by inactivity
title_full_unstemmed Replicated blood-based biomarkers for myalgic encephalomyelitis not explicable by inactivity
title_short Replicated blood-based biomarkers for myalgic encephalomyelitis not explicable by inactivity
title_sort replicated blood based biomarkers for myalgic encephalomyelitis not explicable by inactivity
topic Myalgic Encephalomyelitis
Semi-parametric Mediation Analysis
Post-exertional Malaise
UK Biobank
All of Us Biobank
url https://doi.org/10.1038/s44321-025-00258-8
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