Detection of diagnostic somatic copy number alterations from cerebrospinal fluid cell-free DNA in brain tumor patients

Abstract The gold standard for precise diagnostic classification of brain tumors requires tissue sampling, which carries relevant procedural risks. Brain biopsies often have limited sensitivity and fail to address tumor heterogeneity, because small tissue parts are being examined. This study aims to...

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Main Authors: Svenja Klinsing, Julia Beck, Katharina J. Weber, Kirsten Bornemann-Kolatzki, Mareike Dettki, Hans Urban, Bastian Roller, Kai U. Chow, Henning Reis, Peter J. Wild, Ekkehard Schuetz, Philipp Euskirchen, Joachim P. Steinbach, Michael W. Ronellenfitsch, Patrick N. Harter, Pia S. Zeiner
Format: Article
Language:English
Published: BMC 2024-11-01
Series:Acta Neuropathologica Communications
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Online Access:https://doi.org/10.1186/s40478-024-01887-9
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author Svenja Klinsing
Julia Beck
Katharina J. Weber
Kirsten Bornemann-Kolatzki
Mareike Dettki
Hans Urban
Bastian Roller
Kai U. Chow
Henning Reis
Peter J. Wild
Ekkehard Schuetz
Philipp Euskirchen
Joachim P. Steinbach
Michael W. Ronellenfitsch
Patrick N. Harter
Pia S. Zeiner
author_facet Svenja Klinsing
Julia Beck
Katharina J. Weber
Kirsten Bornemann-Kolatzki
Mareike Dettki
Hans Urban
Bastian Roller
Kai U. Chow
Henning Reis
Peter J. Wild
Ekkehard Schuetz
Philipp Euskirchen
Joachim P. Steinbach
Michael W. Ronellenfitsch
Patrick N. Harter
Pia S. Zeiner
author_sort Svenja Klinsing
collection DOAJ
description Abstract The gold standard for precise diagnostic classification of brain tumors requires tissue sampling, which carries relevant procedural risks. Brain biopsies often have limited sensitivity and fail to address tumor heterogeneity, because small tissue parts are being examined. This study aims to explore the detection and quantification of diagnostically relevant somatic copy number aberrations (SCNAs) in cell-free DNA (cfDNA) extracted from cerebrospinal fluid (CSF) in a real-world cohort of patients with defined brain tumor subtypes. A total of 33 CSF samples were collected from 30 patients for cfDNA extraction. Shallow whole-genome sequencing was conducted on CSF samples containing > 3ng of cfDNA and corresponding tissue DNA from nine patients. The sequencing cohort encompassed 26 samples of 23 patients, comprising 12 with confirmed CNS cancer as compared to 11 patients with either ambiguous CNS lesions (n = 5) or non-cancer CNS lesions (n = 6). After mapping and quality filtering SCNAs were called by depth-of-coverage analyses with a binning of 5.5 Mbp. SCNAs were exclusively identified in CSF cfDNA from brain tumor patients (10/12, 83%). In tumor patients, SCNAs were detectable in cfDNA from all patients with, but also in five of seven patients without tumor cells detected by CSF cytopathology. A substantial number of shared SCNAs were traceable between tissue and CSF in matched pair analyses. Additionally, some SCNAs unique to either CSF or tissue indicating spatial heterogeneity or tumor evolution. Also, diagnostically relevant genomic alterations as well as essential and desirable SCNAs as implemented in the current WHO classification of CNS tumors for certain primary brain tumor subtypes were traceable. In summary, this minimally invasive cfDNA-based LB approach employing shallow whole genome sequencing demonstrates potential for providing a molecularly informed diagnosis of CNS cancers, mapping tumor heterogeneity, tracking tumor evolution, and surveilling tumor patients. Further prospective trials are warranted.
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spelling doaj-art-bb49ac9722b644f68630890aba9b37852025-08-20T04:02:44ZengBMCActa Neuropathologica Communications2051-59602024-11-0112111310.1186/s40478-024-01887-9Detection of diagnostic somatic copy number alterations from cerebrospinal fluid cell-free DNA in brain tumor patientsSvenja Klinsing0Julia Beck1Katharina J. Weber2Kirsten Bornemann-Kolatzki3Mareike Dettki4Hans Urban5Bastian Roller6Kai U. Chow7Henning Reis8Peter J. Wild9Ekkehard Schuetz10Philipp Euskirchen11Joachim P. Steinbach12Michael W. Ronellenfitsch13Patrick N. Harter14Pia S. Zeiner15Dr. Senckenberg Institute of Neurooncology, University Hospital, Goethe University FrankfurtChronix Biomedical, OncocyteInstitute of Neurology (Edinger-Institute), University Hospital, Goethe University FrankfurtChronix Biomedical, OncocyteDr. Senckenberg Institute of Neurooncology, University Hospital, Goethe University FrankfurtDr. Senckenberg Institute of Neurooncology, University Hospital, Goethe University FrankfurtDr. Senckenberg Institute of Neurooncology, University Hospital, Goethe University FrankfurtAmbulantes KrebszentrumDr. Senckenberg Institute of Pathology, Goethe University Frankfurt, University HospitalDr. Senckenberg Institute of Pathology, Goethe University Frankfurt, University HospitalChronix Biomedical, OncocyteDepartment of Neuropathology, Charité-Universitätsmedizin Berlin, Corporate Member of Freie Universität Berlin Und Humboldt Universität Zu BerlinDr. Senckenberg Institute of Neurooncology, University Hospital, Goethe University FrankfurtDr. Senckenberg Institute of Neurooncology, University Hospital, Goethe University FrankfurtInstitute of Neurology (Edinger-Institute), University Hospital, Goethe University FrankfurtDr. Senckenberg Institute of Neurooncology, University Hospital, Goethe University FrankfurtAbstract The gold standard for precise diagnostic classification of brain tumors requires tissue sampling, which carries relevant procedural risks. Brain biopsies often have limited sensitivity and fail to address tumor heterogeneity, because small tissue parts are being examined. This study aims to explore the detection and quantification of diagnostically relevant somatic copy number aberrations (SCNAs) in cell-free DNA (cfDNA) extracted from cerebrospinal fluid (CSF) in a real-world cohort of patients with defined brain tumor subtypes. A total of 33 CSF samples were collected from 30 patients for cfDNA extraction. Shallow whole-genome sequencing was conducted on CSF samples containing > 3ng of cfDNA and corresponding tissue DNA from nine patients. The sequencing cohort encompassed 26 samples of 23 patients, comprising 12 with confirmed CNS cancer as compared to 11 patients with either ambiguous CNS lesions (n = 5) or non-cancer CNS lesions (n = 6). After mapping and quality filtering SCNAs were called by depth-of-coverage analyses with a binning of 5.5 Mbp. SCNAs were exclusively identified in CSF cfDNA from brain tumor patients (10/12, 83%). In tumor patients, SCNAs were detectable in cfDNA from all patients with, but also in five of seven patients without tumor cells detected by CSF cytopathology. A substantial number of shared SCNAs were traceable between tissue and CSF in matched pair analyses. Additionally, some SCNAs unique to either CSF or tissue indicating spatial heterogeneity or tumor evolution. Also, diagnostically relevant genomic alterations as well as essential and desirable SCNAs as implemented in the current WHO classification of CNS tumors for certain primary brain tumor subtypes were traceable. In summary, this minimally invasive cfDNA-based LB approach employing shallow whole genome sequencing demonstrates potential for providing a molecularly informed diagnosis of CNS cancers, mapping tumor heterogeneity, tracking tumor evolution, and surveilling tumor patients. Further prospective trials are warranted.https://doi.org/10.1186/s40478-024-01887-9Cell-free DNACerebrospinal fluidLiquid biopsyNext-generation sequencingBrain tumor
spellingShingle Svenja Klinsing
Julia Beck
Katharina J. Weber
Kirsten Bornemann-Kolatzki
Mareike Dettki
Hans Urban
Bastian Roller
Kai U. Chow
Henning Reis
Peter J. Wild
Ekkehard Schuetz
Philipp Euskirchen
Joachim P. Steinbach
Michael W. Ronellenfitsch
Patrick N. Harter
Pia S. Zeiner
Detection of diagnostic somatic copy number alterations from cerebrospinal fluid cell-free DNA in brain tumor patients
Acta Neuropathologica Communications
Cell-free DNA
Cerebrospinal fluid
Liquid biopsy
Next-generation sequencing
Brain tumor
title Detection of diagnostic somatic copy number alterations from cerebrospinal fluid cell-free DNA in brain tumor patients
title_full Detection of diagnostic somatic copy number alterations from cerebrospinal fluid cell-free DNA in brain tumor patients
title_fullStr Detection of diagnostic somatic copy number alterations from cerebrospinal fluid cell-free DNA in brain tumor patients
title_full_unstemmed Detection of diagnostic somatic copy number alterations from cerebrospinal fluid cell-free DNA in brain tumor patients
title_short Detection of diagnostic somatic copy number alterations from cerebrospinal fluid cell-free DNA in brain tumor patients
title_sort detection of diagnostic somatic copy number alterations from cerebrospinal fluid cell free dna in brain tumor patients
topic Cell-free DNA
Cerebrospinal fluid
Liquid biopsy
Next-generation sequencing
Brain tumor
url https://doi.org/10.1186/s40478-024-01887-9
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