Practice Guideline

. 2024 Aug 15;30(16):3395-3406.

doi: 10.1158/1078-0432.CCR-24-0101.

Biological Sample Collection to Advance Research and Treatment: A Fight Osteosarcoma Through European Research and Euro Ewing Consortium Statement

Darrell Green¹, Roelof van Ewijk², Elisa Tirtei^{3

4}, Dimosthenis Andreou⁵, Fredrik Baecklund⁶, Daniel Baumhoer⁷, Stefan S Bielack⁸, Rajesh Botchu⁹, Kjetil Boye¹⁰, Bernadette Brennan¹¹, Michael Capra¹², Lucia Cottone¹³, Uta Dirksen¹⁴, Franca Fagioli^{3

4}, Natalia Fernandez¹⁵, Adrienne M Flanagan^{13

16}, Marco Gambarotti¹⁷, Nathalie Gaspar^{18

19}, Hans Gelderblom²⁰, Craig Gerrand²¹, Anne Gomez-Mascard^{22

23}, Jendrik Hardes²⁴, Stefanie Hecker-Nolting⁸, Edita Kabickova²⁵, Leo Kager^{26

27}, Jukka Kanerva²⁸, Lennart A Kester², Marieke L Kuijjer^{29

30

31}, Valérie Laurence³², Cyril Lervat³³, Antonin Marchais¹⁸, Perrine Marec-Berard³⁴, Cristina Mendes³⁵, Johannes H M Merks^{2

36}, Benjamin Ory³⁷, Emanuela Palmerini³⁸, Pan Pantziarka^{15

39

40}, Evgenia Papakonstantinou⁴¹, Sophie Piperno-Neumann³³, Anna Raciborska⁴², Elizabeth A Roundhill⁴³, Vilma Rutkauskaite⁴⁴, Akmal Safwat⁴⁵, Katia Scotlandi⁴⁶, Eric L Staals⁴⁷, Sandra J Strauss⁴⁸, Didier Surdez⁴⁹, Gwen M L Sys⁵⁰, Marie-Dominique Tabone⁵¹, Maud Toulmonde⁵², Claudia Valverde⁵³, Michiel A J van de Sande⁵⁴, Klaus Wörtler⁵⁵, Quentin Campbell-Hewson⁵⁶, Martin G McCabe^{57

58}, Michaela Nathrath^{59

60}

Affiliations

¹ Biomedical Research Centre, Norwich Medical School, University of East Anglia, Norwich, United Kingdom.
² Princess Maxima Center for Pediatric Oncology, Utrecht, the Netherlands.
³ Pediatric Oncology, Regina Margherita Children's Hospital, Turin, Italy.
⁴ Department of Public Health and Pediatrics, University of Turin, Turin, Italy.
⁵ Department of Orthopaedics and Trauma, Medical University of Graz, Graz, Austria.
⁶ Pediatric Oncology Unit, Karolinska University Hospital, Stockholm, Sweden.
⁷ Institute of Medical Genetics and Pathology, University Hospital Basel, Basel, Switzerland.
⁸ Center for Pediatric, Adolescent and Women's Medicine, Klinikum Stuttgart-Olgahospital, Stuttgart Cancer Centre, Stuttgart, Germany.
⁹ Department of Musculoskeletal Radiology, Royal Orthopaedic Hospital NHS Foundation Trust, Birmingham, United Kingdom.
¹⁰ Department of Oncology, Oslo University Hospital, Oslo, Norway.
¹¹ Paediatric Oncology, Royal Manchester Children's Hospital, Central Manchester University Hospital NHS Foundation Trust, Manchester, United Kingdom.
¹² Haematology/Oncology, Children's Health Ireland at Crumlin, Dublin, Ireland.
¹³ Department of Pathology, UCL Cancer Institute, University College London, London, United Kingdom.
¹⁴ Pediatrics III, West German Cancer Center, University Hospital Essen, German Cancer Consortium (DKTK) Site Essen, Cancer Research Center (NCT) Cologne-Essen, University of Duisburg-Essen, Essen, Germany.
¹⁵ Patient and Parent Advocacy Group, FOSTER, Washington, District of Columbia.
¹⁶ Histopathology, The Royal National Orthopaedic Hospital NHS Trust, Stanmore, United Kingdom.
¹⁷ Department of Pathology, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy.
¹⁸ Department of Oncology for Child and Adolescent, Gustave Roussy Cancer Campus, Université Paris-Saclay, Villejuif, France.
¹⁹ U1015, Université Paris-Saclay, Villejuif, France.
²⁰ Medical Oncology, Leiden University Medical Center, Leiden, the Netherlands.
²¹ Orthopaedic Oncology, The Royal National Orthopaedic Hospital NHS Trust, Stanmore, United Kingdom.
²² Department of Pathology, Institut Universitaire du Cancer de Toulouse-Oncopole, Toulouse, France.
²³ EQ ONCOSARC, CRCT Inserm/UT3, ERL CNRS, Toulouse, France.
²⁴ Tumour Orthopaedics, University Hospital Essen, German Cancer Consortium (DKTK) Site Essen, Cancer Research Center (NCT) Cologne-Essen, University of Duisburg-Essen, Essen, Germany.
²⁵ Paediatric Haematology and Oncology, University Hospital Motol, Prague, Czech Republic.
²⁶ Pediatrics, St Anna Children's Hospital, Medical University Vienna, Vienna, Austria.
²⁷ St Anna Children's Cancer Research Institute, Vienna, Austria.
²⁸ Hematology-Oncology and Stem Cell Transplantation, HUS Helsinki University Hospital, New Children's Hospital, Helsinki, Finland.
²⁹ Computational Biology and Systems Medicine Group, Centre for Molecular Medicine Norway, University of Oslo, Oslo, Norway.
³⁰ Pathology, Leiden University Medical Center, Leiden, the Netherlands.
³¹ Leiden Center for Computational Oncology, Leiden University Medical Center, Leiden, the Netherlands.
³² Department of Medical Oncology, Institut Curie, Paris, France.
³³ Department of Pediatrics and AYA Oncology, Centre Oscar Lambret, Lille, France.
³⁴ Institute of Hematology and Pediatric Oncology, Léon Bérard Center, Lyon, France.
³⁵ Portuguese Institute of Oncology of Lisbon, Lisbon, Portugal.
³⁶ Division of Imaging and Oncology, University Medical Center Utrecht, Utrecht, the Netherlands.
³⁷ School of Medicine, Nantes Université, Nantes, France.
³⁸ Bone and Soft Tissue Sarcomas and Innovative Therapies Unit, IRCCS Istituto Orthopedico Rizzoli, Bologna, Italy.
³⁹ Anticancer Fund, Meise, Belgium.
⁴⁰ The George Pantziarka TP53 Trust, London, United Kingdom.
⁴¹ Pediatric Hematology-Oncology, Ippokratio General Hospital of Thessaloniki, Thessaloniki, Greece.
⁴² Oncology and Surgical Oncology for Children and Youth, Institute of Mother and Child, Warsaw, Poland.
⁴³ Children's Cancer Research Group, Leeds Institute of Medical Research, University of Leeds, Leeds, United Kingdom.
⁴⁴ Center for Pediatric Oncology and Hematology, Vilnius University Hospital Santaros Klinikos, Vilnius, Lithuania.
⁴⁵ The Danish Centre for Particle Therapy, Aarhus University Hospital, Aarhus, Denmark.
⁴⁶ Laboratory of Experimental Oncology, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy.
⁴⁷ Orthopaedics and Trauma, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy.
⁴⁸ Department of Oncology, University College London Hospitals NHS Foundation Trust, UCL Cancer Institute, London, United Kingdom.
⁴⁹ Balgrist University Hospital, Faculty of Medicine, University of Zurich (UZH), Zurich, Switzerland.
⁵⁰ Department of Orthopaedic Surgery and Traumatology, Ghent University Hospital, Belgium.
⁵¹ Department of Hematology and Oncology, A. Trousseau Hospital, Sorbonne University, APHP, Paris, France.
⁵² Department of Medical Oncology, Institut Bergonié, Bordeaux, France.
⁵³ Medical Oncology, Vall d'Hebron University Hospital, Barcelona, Spain.
⁵⁴ Orthopaedic Oncology, Leiden University Medical Centre, Leiden, the Netherlands.
⁵⁵ Musculoskeletal Radiology, Klinikum Rechts der Isar, Technical University of Munich, Munich, Germany.
⁵⁶ Great North Children's Hospital, Newcastle upon Tyne Hospitals NHS Foundation Trust, Newcastle upon Tyne, United Kingdom.
⁵⁷ Division of Cancer Sciences, School of Medical Sciences, The University of Manchester, Manchester, United Kingdom.
⁵⁸ The Christie NHS Foundation Trust, Manchester, United Kingdom.
⁵⁹ Children's Cancer Research Center, Klinikum Rechts der Isar, Technical University of Munich, Munich, Germany.
⁶⁰ Pediatric Oncology, Klinikum Kassel, Kassel, Germany.

PMID: 38869831
PMCID: PMC11334773
DOI: 10.1158/1078-0432.CCR-24-0101

Practice Guideline

Biological Sample Collection to Advance Research and Treatment: A Fight Osteosarcoma Through European Research and Euro Ewing Consortium Statement

Darrell Green et al. Clin Cancer Res. 2024.

. 2024 Aug 15;30(16):3395-3406.

doi: 10.1158/1078-0432.CCR-24-0101.

Authors

Affiliations

¹ Biomedical Research Centre, Norwich Medical School, University of East Anglia, Norwich, United Kingdom.
² Princess Maxima Center for Pediatric Oncology, Utrecht, the Netherlands.
³ Pediatric Oncology, Regina Margherita Children's Hospital, Turin, Italy.
⁴ Department of Public Health and Pediatrics, University of Turin, Turin, Italy.
⁵ Department of Orthopaedics and Trauma, Medical University of Graz, Graz, Austria.
⁶ Pediatric Oncology Unit, Karolinska University Hospital, Stockholm, Sweden.
⁷ Institute of Medical Genetics and Pathology, University Hospital Basel, Basel, Switzerland.
⁸ Center for Pediatric, Adolescent and Women's Medicine, Klinikum Stuttgart-Olgahospital, Stuttgart Cancer Centre, Stuttgart, Germany.
⁹ Department of Musculoskeletal Radiology, Royal Orthopaedic Hospital NHS Foundation Trust, Birmingham, United Kingdom.
¹⁰ Department of Oncology, Oslo University Hospital, Oslo, Norway.
¹¹ Paediatric Oncology, Royal Manchester Children's Hospital, Central Manchester University Hospital NHS Foundation Trust, Manchester, United Kingdom.
¹² Haematology/Oncology, Children's Health Ireland at Crumlin, Dublin, Ireland.
¹³ Department of Pathology, UCL Cancer Institute, University College London, London, United Kingdom.
¹⁴ Pediatrics III, West German Cancer Center, University Hospital Essen, German Cancer Consortium (DKTK) Site Essen, Cancer Research Center (NCT) Cologne-Essen, University of Duisburg-Essen, Essen, Germany.
¹⁵ Patient and Parent Advocacy Group, FOSTER, Washington, District of Columbia.
¹⁶ Histopathology, The Royal National Orthopaedic Hospital NHS Trust, Stanmore, United Kingdom.
¹⁷ Department of Pathology, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy.
¹⁸ Department of Oncology for Child and Adolescent, Gustave Roussy Cancer Campus, Université Paris-Saclay, Villejuif, France.
¹⁹ U1015, Université Paris-Saclay, Villejuif, France.
²⁰ Medical Oncology, Leiden University Medical Center, Leiden, the Netherlands.
²¹ Orthopaedic Oncology, The Royal National Orthopaedic Hospital NHS Trust, Stanmore, United Kingdom.
²² Department of Pathology, Institut Universitaire du Cancer de Toulouse-Oncopole, Toulouse, France.
²³ EQ ONCOSARC, CRCT Inserm/UT3, ERL CNRS, Toulouse, France.
²⁴ Tumour Orthopaedics, University Hospital Essen, German Cancer Consortium (DKTK) Site Essen, Cancer Research Center (NCT) Cologne-Essen, University of Duisburg-Essen, Essen, Germany.
²⁵ Paediatric Haematology and Oncology, University Hospital Motol, Prague, Czech Republic.
²⁶ Pediatrics, St Anna Children's Hospital, Medical University Vienna, Vienna, Austria.
²⁷ St Anna Children's Cancer Research Institute, Vienna, Austria.
²⁸ Hematology-Oncology and Stem Cell Transplantation, HUS Helsinki University Hospital, New Children's Hospital, Helsinki, Finland.
²⁹ Computational Biology and Systems Medicine Group, Centre for Molecular Medicine Norway, University of Oslo, Oslo, Norway.
³⁰ Pathology, Leiden University Medical Center, Leiden, the Netherlands.
³¹ Leiden Center for Computational Oncology, Leiden University Medical Center, Leiden, the Netherlands.
³² Department of Medical Oncology, Institut Curie, Paris, France.
³³ Department of Pediatrics and AYA Oncology, Centre Oscar Lambret, Lille, France.
³⁴ Institute of Hematology and Pediatric Oncology, Léon Bérard Center, Lyon, France.
³⁵ Portuguese Institute of Oncology of Lisbon, Lisbon, Portugal.
³⁶ Division of Imaging and Oncology, University Medical Center Utrecht, Utrecht, the Netherlands.
³⁷ School of Medicine, Nantes Université, Nantes, France.
³⁸ Bone and Soft Tissue Sarcomas and Innovative Therapies Unit, IRCCS Istituto Orthopedico Rizzoli, Bologna, Italy.
³⁹ Anticancer Fund, Meise, Belgium.
⁴⁰ The George Pantziarka TP53 Trust, London, United Kingdom.
⁴¹ Pediatric Hematology-Oncology, Ippokratio General Hospital of Thessaloniki, Thessaloniki, Greece.
⁴² Oncology and Surgical Oncology for Children and Youth, Institute of Mother and Child, Warsaw, Poland.
⁴³ Children's Cancer Research Group, Leeds Institute of Medical Research, University of Leeds, Leeds, United Kingdom.
⁴⁴ Center for Pediatric Oncology and Hematology, Vilnius University Hospital Santaros Klinikos, Vilnius, Lithuania.
⁴⁵ The Danish Centre for Particle Therapy, Aarhus University Hospital, Aarhus, Denmark.
⁴⁶ Laboratory of Experimental Oncology, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy.
⁴⁷ Orthopaedics and Trauma, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy.
⁴⁸ Department of Oncology, University College London Hospitals NHS Foundation Trust, UCL Cancer Institute, London, United Kingdom.
⁴⁹ Balgrist University Hospital, Faculty of Medicine, University of Zurich (UZH), Zurich, Switzerland.
⁵⁰ Department of Orthopaedic Surgery and Traumatology, Ghent University Hospital, Belgium.
⁵¹ Department of Hematology and Oncology, A. Trousseau Hospital, Sorbonne University, APHP, Paris, France.
⁵² Department of Medical Oncology, Institut Bergonié, Bordeaux, France.
⁵³ Medical Oncology, Vall d'Hebron University Hospital, Barcelona, Spain.
⁵⁴ Orthopaedic Oncology, Leiden University Medical Centre, Leiden, the Netherlands.
⁵⁵ Musculoskeletal Radiology, Klinikum Rechts der Isar, Technical University of Munich, Munich, Germany.
⁵⁶ Great North Children's Hospital, Newcastle upon Tyne Hospitals NHS Foundation Trust, Newcastle upon Tyne, United Kingdom.
⁵⁷ Division of Cancer Sciences, School of Medical Sciences, The University of Manchester, Manchester, United Kingdom.
⁵⁸ The Christie NHS Foundation Trust, Manchester, United Kingdom.
⁵⁹ Children's Cancer Research Center, Klinikum Rechts der Isar, Technical University of Munich, Munich, Germany.
⁶⁰ Pediatric Oncology, Klinikum Kassel, Kassel, Germany.

PMID: 38869831
PMCID: PMC11334773
DOI: 10.1158/1078-0432.CCR-24-0101

Abstract

Osteosarcoma and Ewing sarcoma are bone tumors mostly diagnosed in children, adolescents, and young adults. Despite multimodal therapy, morbidity is high and survival rates remain low, especially in the metastatic disease setting. Trials investigating targeted therapies and immunotherapies have not been groundbreaking. Better understanding of biological subgroups, the role of the tumor immune microenvironment, factors that promote metastasis, and clinical biomarkers of prognosis and drug response are required to make progress. A prerequisite to achieve desired success is a thorough, systematic, and clinically linked biological analysis of patient samples, but disease rarity and tissue processing challenges such as logistics and infrastructure have contributed to a lack of relevant samples for clinical care and research. There is a need for a Europe-wide framework to be implemented for the adequate and minimal sampling, processing, storage, and analysis of patient samples. Two international panels of scientists, clinicians, and patient and parent advocates have formed the Fight Osteosarcoma Through European Research consortium and the Euro Ewing Consortium. The consortia shared their expertise and institutional practices to formulate new guidelines. We report new reference standards for adequate and minimally required sampling (time points, diagnostic samples, and liquid biopsy tubes), handling, and biobanking to enable advanced biological studies in bone sarcoma. We describe standards for analysis and annotation to drive collaboration and data harmonization with practical, legal, and ethical considerations. This position paper provides comprehensive guidelines that should become the new standards of care that will accelerate scientific progress, promote collaboration, and improve outcomes.

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Conflict of interest statement

D. Andreou reports other support from PharmaMar outside the submitted work. S.S. Bielack reports personal fees from Y-mAbs, MAP BioPharma, SERB SAS, and Zschimmer and Schwarz Mohsdorf GmbH & Co. KG outside the submitted work. K. Boye reports personal fees from Bayer, GSK, Deciphera, Incyte, and NEC Oncoimmunity, as well as grants and personal fees from Eli Lilly and Company, personal fees from Novartis, and nonfinancial support from Merck outside the submitted work. N. Gaspar reports other support from ISPEN, Eisai, and Innovative Therapies for Children with Cancer outside the submitted work. J. Hardes reports grants from implantcast, Buxtehude, outside the submitted work. S. Hecker-Nolting reports grants from Deutsche Kinderkrebsstiftung, Bonn, Germany, and Foerderverein Krebskranke Kinder, Stuttgart, Germany, outside the submitted work. E. Palmerini reports personal fees from Deciphera Pharmaceutical, SynOX Therapeutics, Daiichi Sankyo, and Eusa Pharma outside the submitted work. S.J. Strauss reports personal fees from Ceridwen Oncology, Boehringer Ingelheim, Tessellate BIO, Inhibrx, and Adaptimmune outside the submitted work. C. Valverde reports grants and personal fees from PharmaMar, personal fees from Roche, GSK, Mundipharma, Eli Lilly and Company, Boehringer Ingelheim, and Ipsen, other support from Adaptimmune, Karyopharm, Foghorn Therapeutics, Ayala therapeutics, and Inhibrx, grants from Bayer, and nonfinancial support and other support from GEIS (Spanish Sarcoma Group) outside the submitted work. M.A.J. van de Sande reports grants from CarboFix and other support from SynOX and Deciphera outside the submitted work. M.G. McCabe reports other support from Eisai outside the submitted work. No disclosures were reported by the other authors.

Figures

**Figure 1.**
Overview of the sample types to be collected. To maximize the advantages of sample collection, in order to obtain a comprehensive biological understanding of bone sarcoma and host-related factors, different sample types at sequential stages of the clinical pathway should be collected. (Created with BioRender.com.)

See this image and copyright information in PMC

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Biological Sample Collection to Advance Research and Treatment: A Fight Osteosarcoma Through European Research and Euro Ewing Consortium Statement

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Biological Sample Collection to Advance Research and Treatment: A Fight Osteosarcoma Through European Research and Euro Ewing Consortium Statement

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