Molecular diagnostics enables detection of actionable targets: the Pediatric Targeted Therapy 2.0 registry.

Brain tumour Cancer predisposition Constitutional DNA variant Molecular diagnostics Molecular targeted therapy Paediatric oncology Precision oncology Sarcoma

Journal

European journal of cancer (Oxford, England : 1990)
ISSN: 1879-0852
Titre abrégé: Eur J Cancer
Pays: England
ID NLM: 9005373

Informations de publication

Date de publication:
02 2023
Historique:
received: 18 10 2022
revised: 11 11 2022
accepted: 11 11 2022
pubmed: 22 12 2022
medline: 28 1 2023
entrez: 21 12 2022
Statut: ppublish

Résumé

Precision oncology requires diagnostic accuracy and robust detection of actionable alterations. The Pediatric Targeted Therapy (PTT) 2.0 program aims at improving diagnostic accuracy by addition of molecular analyses to the existing histological diagnosis and detection of actionable alterations for relapsed paediatric oncology patients, in cases with limited availability of tumour material. Paediatric patients diagnosed with relapse or progression of a central nervous system tumour (n = 178), a sarcoma (n = 41) or another solid tumour (n = 44) were included. DNA methylation array, targeted gene panel sequencing on tumour and blood (130 genes), RNA sequencing in selected cases and a pathway-specific immunohistochemistry (IHC) panel were performed using limited formalin-fixed paraffin embedded tissue from any disease episode available. The clinical impact of reported findings was assessed by a serial questionnaire-based follow-up. Integrated molecular diagnostics resulted in refined or changed diagnosis in 117/263 (44%) tumours. Actionable targets were detected in 155/263 (59%) cases. Constitutional DNA variants with clinical relevance were identified in 16/240 (7%) of patients, half of which were previously unknown. Clinical follow-up showed that 26/263 (10%) of patients received mechanism-of-action based treatment matched to the molecular findings. Next-generation diagnostics adds robust and relevant information on diagnosis, actionable alterations and cancer predisposition syndromes even when tissue from the current disease episode is limited.

Sections du résumé

BACKGROUND
Precision oncology requires diagnostic accuracy and robust detection of actionable alterations. The Pediatric Targeted Therapy (PTT) 2.0 program aims at improving diagnostic accuracy by addition of molecular analyses to the existing histological diagnosis and detection of actionable alterations for relapsed paediatric oncology patients, in cases with limited availability of tumour material.
METHODS
Paediatric patients diagnosed with relapse or progression of a central nervous system tumour (n = 178), a sarcoma (n = 41) or another solid tumour (n = 44) were included. DNA methylation array, targeted gene panel sequencing on tumour and blood (130 genes), RNA sequencing in selected cases and a pathway-specific immunohistochemistry (IHC) panel were performed using limited formalin-fixed paraffin embedded tissue from any disease episode available. The clinical impact of reported findings was assessed by a serial questionnaire-based follow-up.
RESULTS
Integrated molecular diagnostics resulted in refined or changed diagnosis in 117/263 (44%) tumours. Actionable targets were detected in 155/263 (59%) cases. Constitutional DNA variants with clinical relevance were identified in 16/240 (7%) of patients, half of which were previously unknown. Clinical follow-up showed that 26/263 (10%) of patients received mechanism-of-action based treatment matched to the molecular findings.
CONCLUSION
Next-generation diagnostics adds robust and relevant information on diagnosis, actionable alterations and cancer predisposition syndromes even when tissue from the current disease episode is limited.

Identifiants

pubmed: 36542877
pii: S0959-8049(22)01759-2
doi: 10.1016/j.ejca.2022.11.015
pii:
doi:

Substances chimiques

DNA 9007-49-2

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

71-84

Commentaires et corrections

Type : ErratumIn

Informations de copyright

Copyright © 2022 Elsevier Ltd. All rights reserved.

Déclaration de conflit d'intérêts

Conflict of interest statement The authors declare the following financial interests/personal relationships which may be considered as potential competing interests: Cornelis M. van Tilburg participated in advisory boards of Novartis and Bayer. Olaf Witt participated in advisory boards of Astra Zeneca, BMS, Novartis, Bayer, Roche and Janssen. Stefan M. Pfister coordinates an IMI-2 project (www.itccp4.eu) with funding from the EU, Eli-Lilly, Roche, Pfizer, Bayer, PharmaMar, Astra Zeneca, Johnson & Johnson, Sanofi, Servier and Amgen. Stefan M, Pfister is involved in a patent on DNA methylation-based tumor classification. Olaf Witt and Till Milde received research funding from Biomed Valley and Day One Therapeutics. All other authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

Auteurs

Jonas Ecker (J)

Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany; Clinical Cooperation Unit Pediatric Oncology, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany; Department of Pediatric Hematology, Oncology, Immunology and Pulmonology, Heidelberg University Hospital, Heidelberg, Germany.

Florian Selt (F)

Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany; Clinical Cooperation Unit Pediatric Oncology, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany; Department of Pediatric Hematology, Oncology, Immunology and Pulmonology, Heidelberg University Hospital, Heidelberg, Germany.

Dominik Sturm (D)

Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany; Department of Pediatric Hematology, Oncology, Immunology and Pulmonology, Heidelberg University Hospital, Heidelberg, Germany; Division of Pediatric Glioma Research, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany.

Martin Sill (M)

Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany; Department of Pediatric Hematology, Oncology, Immunology and Pulmonology, Heidelberg University Hospital, Heidelberg, Germany; Division of Pediatric Neurooncology, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany.

Andrey Korshunov (A)

Department of Neuropathology, Heidelberg University Hospital, Heidelberg, Germany; Clinical Cooperation Unit Neuropathology, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany.

Steffen Hirsch (S)

Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany; Department of Human Genetics, Institute of Human Genetics, Heidelberg University Hospital, Heidelberg, Germany.

David Capper (D)

Department of Neuropathology, Charité - Universitätsmedizin Berlin, Campus Charité Mitte, And German Consortium for Translational Cancer Research (DKTK), Berlin, Germany.

Nicola Dikow (N)

Department of Human Genetics, Institute of Human Genetics, Heidelberg University Hospital, Heidelberg, Germany.

Christian Sutter (C)

Department of Human Genetics, Institute of Human Genetics, Heidelberg University Hospital, Heidelberg, Germany.

Carina Müller (C)

Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany; Clinical Cooperation Unit Pediatric Oncology, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany.

Romain Sigaud (R)

Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany; Clinical Cooperation Unit Pediatric Oncology, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany.

Angelika Eggert (A)

Department of Pediatric Oncology and Hematology, Charité - Universitätsmedizin Berlin, Campus Virchow-Klinikum, Berlin, Germany.

Thorsten Simon (T)

Department of Pediatric Oncology and Hematology, University Hospital Cologne, Cologne, Germany.

Tim Niehues (T)

Children's Cancer Center, Helios Hospital Krefeld, Krefeld, Germany.

Andreas von Deimling (A)

Department of Neuropathology, Heidelberg University Hospital, Heidelberg, Germany; Clinical Cooperation Unit Neuropathology, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany.

Kristian W Pajtler (KW)

Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany; Department of Pediatric Hematology, Oncology, Immunology and Pulmonology, Heidelberg University Hospital, Heidelberg, Germany; Division of Pediatric Neurooncology, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany.

Cornelis M van Tilburg (CM)

Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany; Clinical Cooperation Unit Pediatric Oncology, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany; Department of Pediatric Hematology, Oncology, Immunology and Pulmonology, Heidelberg University Hospital, Heidelberg, Germany.

David T W Jones (DTW)

Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany; Division of Pediatric Glioma Research, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany.

Felix Sahm (F)

Department of Neuropathology, Heidelberg University Hospital, Heidelberg, Germany; Clinical Cooperation Unit Neuropathology, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany.

Stefan M Pfister (SM)

Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany; Department of Pediatric Hematology, Oncology, Immunology and Pulmonology, Heidelberg University Hospital, Heidelberg, Germany; Division of Pediatric Neurooncology, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany.

Olaf Witt (O)

Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany; Clinical Cooperation Unit Pediatric Oncology, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany; Department of Pediatric Hematology, Oncology, Immunology and Pulmonology, Heidelberg University Hospital, Heidelberg, Germany.

Till Milde (T)

Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany; Clinical Cooperation Unit Pediatric Oncology, German Cancer Research Center (DKFZ) and German Consortium for Translational Cancer Research (DKTK), Heidelberg, Germany; Department of Pediatric Hematology, Oncology, Immunology and Pulmonology, Heidelberg University Hospital, Heidelberg, Germany. Electronic address: t.milde@kitz-heidelberg.de.

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