Identification of two biological subgroups of complex regional pain syndrome type 1 by transcriptomic profiling of skin and blood in women

Abstract Background Patients with Complex Regional Pain Syndrome (CRPS) present prolonged, debilitating pain and functional impairment. Treatments are not disease-modifying due to the poorly understood underlying pathomechanisms. This study aimed to identify the molecular signatures of potential CRP...

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Main Authors: Melina Pérez Vertti Valdés, Astrid Jüngel, Pamela Bitterli, Jan Devan, Hubert Rehrauer, Lennart Opitz, Laura Sirucek, Petra Schweinhardt, Sabrina Catanzaro, Oliver Distler, Florian Brunner, Stefan Dudli
Format: Article
Language:English
Published: BMC 2025-03-01
Series:Molecular Medicine
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Online Access:https://doi.org/10.1186/s10020-025-01148-y
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author Melina Pérez Vertti Valdés
Astrid Jüngel
Pamela Bitterli
Jan Devan
Hubert Rehrauer
Lennart Opitz
Laura Sirucek
Petra Schweinhardt
Sabrina Catanzaro
Oliver Distler
Florian Brunner
Stefan Dudli
author_facet Melina Pérez Vertti Valdés
Astrid Jüngel
Pamela Bitterli
Jan Devan
Hubert Rehrauer
Lennart Opitz
Laura Sirucek
Petra Schweinhardt
Sabrina Catanzaro
Oliver Distler
Florian Brunner
Stefan Dudli
author_sort Melina Pérez Vertti Valdés
collection DOAJ
description Abstract Background Patients with Complex Regional Pain Syndrome (CRPS) present prolonged, debilitating pain and functional impairment. Treatments are not disease-modifying due to the poorly understood underlying pathomechanisms. This study aimed to identify the molecular signatures of potential CRPS type 1 subgroups. Methods Twelve women with CRPS type 1 were included. Demographics and pain questionnaires were recorded. Skin biopsies of the affected and non-affected limbs (n = 6 + 6) and peripheral blood (n = 11) were collected. RNA sequencing was performed on skin and peripheral blood mononuclear cells (PBMCs). Twenty cytokines were quantified in blood plasma (n = 12). Results Cluster analysis of the affected skin identified two CRPS subgroups (SG). SG1 exhibited increased gene expression related to epidermal development, metabolic processes, and a greater abundance of keratinocytes. SG2 showed enhanced transcriptomic changes in inflammatory, immune, and fibrotic processes, along with higher abundance of fibroblasts, macrophages, and endothelial cells. PBMCs transcriptomics revealed the same SG1/SG2 clusters and highlighted a stronger inflammatory response in the blood of SG1, suggesting distinct tissue-specific immune responses for the subgroups. Interleukin-1 receptor antagonist (IL-1RA) levels were higher in the blood plasma of SG1 (FDR = 0.01), consistent with its encoding gene IL1RN expression in PBMCs (log2 FC = 1.10, P < 0.001) and affected skin (log2 FC = 0.88, P = 0.006). Subgroups did not differ in demographic or clinical parameters but correlations among clinical factors varied between them. Conclusions This study identified two potential biological subgroups of CRPS type 1 in women through skin and blood transcriptomic profiling, advancing the understanding of this condition. This could facilitate the development of targeted treatments for CRPS type 1.
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spelling doaj-art-89bc814634ad4536a6716dd8eaa1408b2025-08-20T03:02:21ZengBMCMolecular Medicine1528-36582025-03-0131111710.1186/s10020-025-01148-yIdentification of two biological subgroups of complex regional pain syndrome type 1 by transcriptomic profiling of skin and blood in womenMelina Pérez Vertti Valdés0Astrid Jüngel1Pamela Bitterli2Jan Devan3Hubert Rehrauer4Lennart Opitz5Laura Sirucek6Petra Schweinhardt7Sabrina Catanzaro8Oliver Distler9Florian Brunner10Stefan Dudli11Center of Experimental Rheumatology, Balgrist Campus, University Hospital Zurich, University of ZurichCenter of Experimental Rheumatology, Balgrist Campus, University Hospital Zurich, University of ZurichCenter of Experimental Rheumatology, Balgrist Campus, University Hospital Zurich, University of ZurichCenter of Experimental Rheumatology, Balgrist Campus, University Hospital Zurich, University of ZurichFunctional Genomics Center Zurich, ETH Zurich and University of ZurichFunctional Genomics Center Zurich, ETH Zurich and University of ZurichDepartment of Chiropractic Medicine, Integrative Spinal Research Group, Balgrist University Hospital, University of ZurichDepartment of Chiropractic Medicine, Integrative Spinal Research Group, Balgrist University Hospital, University of ZurichUnit of Clinical and Applied Research, Balgrist University Hospital, University of Zurich, SwitzerlandCenter of Experimental Rheumatology, Balgrist Campus, University Hospital Zurich, University of ZurichDepartment of Physical Medicine and Rheumatology, Balgrist University Hospital, University of ZurichCenter of Experimental Rheumatology, Balgrist Campus, University Hospital Zurich, University of ZurichAbstract Background Patients with Complex Regional Pain Syndrome (CRPS) present prolonged, debilitating pain and functional impairment. Treatments are not disease-modifying due to the poorly understood underlying pathomechanisms. This study aimed to identify the molecular signatures of potential CRPS type 1 subgroups. Methods Twelve women with CRPS type 1 were included. Demographics and pain questionnaires were recorded. Skin biopsies of the affected and non-affected limbs (n = 6 + 6) and peripheral blood (n = 11) were collected. RNA sequencing was performed on skin and peripheral blood mononuclear cells (PBMCs). Twenty cytokines were quantified in blood plasma (n = 12). Results Cluster analysis of the affected skin identified two CRPS subgroups (SG). SG1 exhibited increased gene expression related to epidermal development, metabolic processes, and a greater abundance of keratinocytes. SG2 showed enhanced transcriptomic changes in inflammatory, immune, and fibrotic processes, along with higher abundance of fibroblasts, macrophages, and endothelial cells. PBMCs transcriptomics revealed the same SG1/SG2 clusters and highlighted a stronger inflammatory response in the blood of SG1, suggesting distinct tissue-specific immune responses for the subgroups. Interleukin-1 receptor antagonist (IL-1RA) levels were higher in the blood plasma of SG1 (FDR = 0.01), consistent with its encoding gene IL1RN expression in PBMCs (log2 FC = 1.10, P < 0.001) and affected skin (log2 FC = 0.88, P = 0.006). Subgroups did not differ in demographic or clinical parameters but correlations among clinical factors varied between them. Conclusions This study identified two potential biological subgroups of CRPS type 1 in women through skin and blood transcriptomic profiling, advancing the understanding of this condition. This could facilitate the development of targeted treatments for CRPS type 1.https://doi.org/10.1186/s10020-025-01148-yComplex regional pain syndrome (CRPS)RNA-seqPeripheral blood mononuclear cells (PBMCs)Cytokine quantificationChronic pain
spellingShingle Melina Pérez Vertti Valdés
Astrid Jüngel
Pamela Bitterli
Jan Devan
Hubert Rehrauer
Lennart Opitz
Laura Sirucek
Petra Schweinhardt
Sabrina Catanzaro
Oliver Distler
Florian Brunner
Stefan Dudli
Identification of two biological subgroups of complex regional pain syndrome type 1 by transcriptomic profiling of skin and blood in women
Molecular Medicine
Complex regional pain syndrome (CRPS)
RNA-seq
Peripheral blood mononuclear cells (PBMCs)
Cytokine quantification
Chronic pain
title Identification of two biological subgroups of complex regional pain syndrome type 1 by transcriptomic profiling of skin and blood in women
title_full Identification of two biological subgroups of complex regional pain syndrome type 1 by transcriptomic profiling of skin and blood in women
title_fullStr Identification of two biological subgroups of complex regional pain syndrome type 1 by transcriptomic profiling of skin and blood in women
title_full_unstemmed Identification of two biological subgroups of complex regional pain syndrome type 1 by transcriptomic profiling of skin and blood in women
title_short Identification of two biological subgroups of complex regional pain syndrome type 1 by transcriptomic profiling of skin and blood in women
title_sort identification of two biological subgroups of complex regional pain syndrome type 1 by transcriptomic profiling of skin and blood in women
topic Complex regional pain syndrome (CRPS)
RNA-seq
Peripheral blood mononuclear cells (PBMCs)
Cytokine quantification
Chronic pain
url https://doi.org/10.1186/s10020-025-01148-y
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