Top

Published in:

Open Access 01-12-2018 | Meeting Report

Report from the 4th European Bone Sarcoma Networking meeting: focus on osteosarcoma

Authors: Sandra J. Strauss, Jakob Anninga, Rubina Baglio, Daniel Baumhoer, Sam Behjati, Stefan Bielack, Kjetil Boye, Javier M. Broto, Anne-Marie Cleton-Jansen, Andrea Degasperi, Abigail Evans, Franca Fagioli, Marta Fiocco, Nathalie Gaspar, Dominique Heymann, Nadia Hindi, Carlo Lancia, Ola Myklebost, Michaela Nathrath, Francoise Redini, Katia Scotlandi, Elisa Tirtei, Michel Vanden Eynden, Jeremy Whelan

Published in: Clinical Sarcoma Research | Issue 1/2018

Abstract

This report summarizes the proceedings of the 4th European Bone Sarcoma Networking Meeting, held in London, England, on 21 June 2017. The meeting brought together scientific and clinical researchers and representatives from sarcoma charities from 19 countries representing five networks across Europe, to present and discuss new developments on bone sarcoma. In view of the challenges is poses, the meeting focussed primarily on osteosarcoma with presentations on developments in our understanding of osteosarcoma genetics and immunology as well as results from preclinical investigations and discussion of recent and ongoing clinical trials. These include studies examining the efficacy of multi-targeted tyrosine kinase inhibitors and checkpoint inhibitors, as well as those with molecular profiling to stratify patients for specific therapies. Discussion was centred on generation of new hypotheses for collaborative biological and clinical investigations, the ultimate goal being to improve therapy and outcome in patients with bone sarcomas.

Kager L, Whelan J, Dirksen U, Hassan B, Anninga J, Bennister L, et al. The ENCCA-WP7/EuroSarc/EEC/PROVABES/EURAMOS 3rd European Bone Sarcoma Networking Meeting/Joint Workshop of EU Bone Sarcoma Translational Research Networks; Vienna, Austria, September 24–25, 2015. Workshop Report. Clin Sarcoma Res 2016;6:3.

Martin-Broto J, Redondo A, Valverde C, Vaz MA, Mora J, Garcia Del Muro X, et al. Gemcitabine plus sirolimus for relapsed and progressing osteosarcoma patients after standard chemotherapy: a multicenter, single-arm phase II trial of Spanish Group for Research on Sarcoma (GEIS). Ann Oncol. 2017;28(12):2994–9.CrossRefPubMed

Phase II multicenter trial of palbociclib in second line of advanced sarcomas with CDK4 overexpression: https://ClinicalTrials.gov/show/NCT03242382. Accessed 16 Jul 2018.

GEIS 52 Phase I-II trial of sunitinib plus nivolumab after standard treatment in advanced soft tissue and bone sarcomas. https://www.clinicaltrialsregister.eu/ctr-search/search?query=2016-004040-10. Accessed 16 Jul 2018.

Piperno-Neumann S, Le Deley MC, Redini F, Pacquement H, Marec-Berard P, Petit P, et al. Zoledronate in combination with chemotherapy and surgery to treat osteosarcoma (OS2006): a randomised, multicentre, open-label, phase 3 trial. Lancet Oncol. 2016;17(8):1070–80.CrossRefPubMed

Gaspar N, Occean BV, Pacquement H, Bompas E, Bouvier C, Brisse HJ, et al. Results of methotrexate–etoposide–ifosfamide based regimen (M-EI) in osteosarcoma patients included in the French OS2006/sarcome-09 study. Eur J Cancer. 2018;88:57–66.CrossRefPubMed

Meyers PA, Schwartz CL, Krailo M, Kleinerman ES, Betcher D, Bernstein ML, et al. Osteosarcoma: a randomized, prospective trial of the addition of ifosfamide and/or muramyl tripeptide to cisplatin, doxorubicin, and high-dose methotrexate. J Clin Oncol. 2005;23(9):2004–11.CrossRefPubMed

A phase II study evaluating efficacy and safety of regorafenib in patients with metastatic bone sarcomas. https://ClinicalTrials.gov/show/NCT02389244. Accessed 16 Jul 2018.

Cabozantinib-s-malate in treating patients with relapsed osteosarcoma or Ewing sarcoma. https://ClinicalTrials.gov/show/NCT02243605. Accessed 16 Jul 2018.

10.

Study of lenvatinib in children and adolescents with refractory or relapsed solid malignancies and young adults with osteosarcoma. https://ClinicalTrials.gov/show/NCT02432274. Accessed 16 Jul 2018.

11.

Tawbi HA, Burgess M, Bolejack V, Van Tine BA, Schuetze SM, Hu J, D'Angelo S, Attia S, Riedel RF, Priebat DA, Movva S, Davis LE, Okuno SH, Reed DR, Crowley J, Butterfield LH, Salazar R, Rodriguez-Canales J, Lazar AJ, Wistuba II, Baker LH, Maki RG, Reinke D, Patel S. Pembrolizumab in advanced soft-tissue sarcoma and bone sarcoma (SARC028): a multicentre, two-cohort, single-arm, open-label, phase 2 trial. Lancet Oncol. 2017;18(11):1493–501. https://doi.org/10.1016/S1470-2045(17)30624-1 CrossRefPubMedPubMedCentral

12.

Geoerger B, Karski EE, Zwaan M, Casanova M, Marshall LV, DuBois SG, et al. A phase I/II study of atezolizumab in pediatric and young adult patients with refractory/relapsed solid tumors(iMATRIXAtezolizumab). J Clin Oncol. 2017;35(15_suppl):10524.CrossRef

13.

Combination of MK3475 and metronomic cyclophosphamide in patients with advanced sarcomas: multicentre phase II trial. https://ClinicalTrials.gov/show/NCT02406781. Accessed 16 Jul 2018.

14.

Proof-of-concept study to stratify targeted therapies adapted to molecular profiling. https://ClinicalTrials.gov/show/NCT02613962. Accessed 16 Jul 2018.

15.

Can cancer cells be found counted in blood samples from patients with bone sarcoma? https://www.isrctn.com/ISRCTN29619083?q=29619083&filters=&sort=&offset=1&totalResults=1&page=1&pageSize=10&searchType=basic-search. Accessed 16 Jul 2018.

16.

Behjati S, Tarpey PS, Haase K, Ye H, Young MD, Alexandrov LB, et al. Recurrent mutation of IGF signalling genes and distinct patterns of genomic rearrangement in osteosarcoma. Nat Commun. 2017;8:15936.CrossRefPubMedPubMedCentral

17.

Kovac M, Blattmann C, Ribi S, Smida J, Mueller NS, Engert F, et al. Exome sequencing of osteosarcoma reveals mutation signatures reminiscent of BRCA deficiency. Nat Commun. 2015;6:8940.CrossRefPubMed

18.

Robson M, Im SA, Senkus E, Xu B, Domchek SM, Masuda N, et al. Olaparib for metastatic breast cancer in patients with a germline BRCA mutation. N Engl J Med. 2017;377(6):523–33.CrossRefPubMed

19.

Mateo J, Carreira S, Sandhu S, Miranda S, Mossop H, Perez-Lopez R, et al. DNA-repair defects and olaparib in metastatic prostate cancer. N Engl J Med. 2015;373(18):1697–708.CrossRefPubMedPubMedCentral

20.

Tan DS, Kaye SB. Chemotherapy for patients with BRCA1 and BRCA2-mutated ovarian cancer: same or different?. Am Soc Clin Oncol Educ Book. 2015. https://doi.org/10.14694/EdBook_AM.2015.35.114 CrossRefPubMed

21.

Engert F, Kovac M, Baumhoer D, Nathrath M, Fulda S. Osteosarcoma cells with genetic signatures of BRCAness are susceptible to the PARP inhibitor talazoparib alone or in combination with chemotherapeutics. Oncotarget. 2017;8(30):48794–806.CrossRefPubMed

22.

Davies H, Glodzik D, Morganella S, Yates LR, Staaf J, Zou X, et al. HRDetect is a predictor of BRCA1 and BRCA2 deficiency based on mutational signatures. Nat Med. 2017;23(4):517–25.CrossRefPubMedPubMedCentral

23.

Ottaviano L, Schaefer KL, Gajewski M, Huckenbeck W, Baldus S, Rogel U, et al. Molecular characterization of commonly used cell lines for bone tumor research: a trans-European EuroBoNet effort. Genes Chromosomes Cancer. 2010;49(1):40–51.CrossRefPubMed

24.

Baglio SR, Lagerweij T, Perez-Lanzon M, Ho XD, Leveille N, Melo SA, et al. Blocking tumor-educated MSC paracrine activity halts osteosarcoma progression. Clin Cancer Res. 2017;23(14):3721–33.CrossRefPubMed

25.

Sundara YT, Kostine M, Cleven AH, Bovee JV, Schilham MW, Cleton-Jansen AM. Increased PD-L1 and T-cell infiltration in the presence of HLA class I expression in metastatic high-grade osteosarcoma: a rationale for T-cell-based immunotherapy. Cancer Immunol Immunother. 2017;66(1):119–28.CrossRefPubMed

26.

Rustichelli D, Castiglia S, Gunetti M, Mareschi K, Signorino E, Muraro M, et al. Validation of analytical methods in compliance with good manufacturing practice: a practical approach. J Transl Med. 2013;11:197.CrossRefPubMedPubMedCentral

27.

Sangiolo D, Mesiano G, Gammaitoni L, Aglietta M, Grignani G. Activity of cytokine-induced killer cells against bone and soft tissue sarcoma. Oncoimmunology. 2014;3:e28269.CrossRefPubMedPubMedCentral

28.

Ratti C, Botti L, Cancila V, Galvan S, Torselli I, Garofalo C, et al. Trabectedin overrides osteosarcoma differentiative block and reprograms the tumor immune environment enabling effective combination with immune checkpoint inhibitors. Clin Cancer Res. 2017;23(17):5149–61.CrossRefPubMed

29.

Palmerini E, Agostinelli C, Picci P, Pileri S, Marafioti T, Lollini PL, et al. Tumoral immune-infiltrate (IF), PD-L1 expression and role of CD8/TIA-1 lymphocytes in localized osteosarcoma patients treated within protocol ISG-OS1. Oncotarget. 2017;8(67):111836–46.CrossRefPubMedPubMedCentral

30.

Gabriel MT, Calleja LR, Chalopin A, Ory B, Heymann D. Circulating tumor cells: a review of non-EpCAM-based approaches for cell enrichment and isolation. Clin Chem. 2016;62(4):571–81.CrossRefPubMed

31.

Hanahan D, Weinberg RA. Hallmarks of cancer: the next generation. Cell. 2011;144(5):646–74.CrossRefPubMed

32.

Pickett HA, Reddel RR. Molecular mechanisms of activity and derepression of alternative lengthening of telomeres. Nat Struct Mol Biol. 2015;22(11):875–80.CrossRefPubMed

33.

Episkopou H, Draskovic I, Van Beneden A, Tilman G, Mattiussi M, Gobin M, et al. Alternative lengthening of telomeres is characterized by reduced compaction of telomeric chromatin. Nucleic Acids Res. 2014;42(7):4391–405.CrossRefPubMedPubMedCentral

34.

Heaphy CM, Subhawong AP, Hong SM, Goggins MG, Montgomery EA, Gabrielson E, et al. Prevalence of the alternative lengthening of telomeres telomere maintenance mechanism in human cancer subtypes. Am J Pathol. 2011;179(4):1608–15.CrossRefPubMedPubMedCentral

35.

Commission E. European reference networks. https://ec.europa.eu/health/ern/networks_en. Accessed 16 Jul 2018.

Title: Report from the 4th European Bone Sarcoma Networking meeting: focus on osteosarcoma
Authors: Sandra J. Strauss
Jakob Anninga
Rubina Baglio
Daniel Baumhoer
Sam Behjati
Stefan Bielack
Kjetil Boye
Javier M. Broto
Anne-Marie Cleton-Jansen
Andrea Degasperi
Abigail Evans
Franca Fagioli
Marta Fiocco
Nathalie Gaspar
Dominique Heymann
Nadia Hindi
Carlo Lancia
Ola Myklebost
Michaela Nathrath
Francoise Redini
Katia Scotlandi
Elisa Tirtei
Michel Vanden Eynden
Jeremy Whelan
Publication date: 01-12-2018
Publisher: BioMed Central
Published in: Clinical Sarcoma Research / Issue 1/2018
Electronic ISSN: 2045-3329
DOI: https://doi.org/10.1186/s13569-018-0103-0

Webinar | 19-02-2024 | 17:30 (CET)

Keynote webinar | Spotlight on antibody–drug conjugates in cancer

Watch now

Antibody–drug conjugates (ADCs) are novel agents that have shown promise across multiple tumor types. Explore the current landscape of ADCs in breast and lung cancer with our experts, and gain insights into the mechanism of action, key clinical trials data, existing challenges, and future directions.

Dr. Véronique Diéras

Prof. Fabrice Barlesi

Developed by: Springer Medicine

Keynote webinar | Spotlight on sleep in brain health

Springer Medicine

Abstract

Please log in to get access to this content

Other articles of this Issue 1/2018

Regional chemotherapy by isolated limb perfusion prior to surgery compared with surgery and post-operative radiotherapy for primary, locally advanced extremity sarcoma: a comparison of matched cohorts

The use of whole exome sequencing and murine patient derived xenografts as a method of chemosensitivity testing in sarcoma

Extraskeletal myxoid chondrosarcoma with massive pulmonary metastases

Treatment patterns and survival among older adults in the United States with advanced soft-tissue sarcomas

Different quality of treatment in retroperitoneal sarcomas (RPS) according to hospital-case volume and surgeon-case volume: a retrospective regional analysis in Italy

Non-canonical WNT6/WNT10A signal factor expression in EBV+ post-transplant smooth muscle tumors

Keynote webinar | Spotlight on antibody–drug conjugates in cancer