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      Surgery in reference centers improves survival of sarcoma patients: a nationwide study

      1 , 2 , 3 , 4 , 5 , 1 , 2 , 3 , 6 , 7 , 1 , 2 , 3 , 8 , 9 , 10 , 11 , 12 , 13 , 14 , 15 , 16 , 16 , 17 , 18 , 19 , 1 , 2 , 3 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 4 , 26 , 8 , 9 , 11 , 12 , 13 , 14 , 13 , 14 , 15 , 19 , 4 , 4 , 1 , 2 , 3 , 1 , 2 , 3 , 1 , 2 , 3 , 1 , 2 , 3 , 1 , 2 , 3 , 1 , 2 , 3 , 10 , 1 , 2 , 3 , 5 , 1 , 2 , 3 , 5 , 4 , 6 , 7 , 31 , NETSARC/REPPS/RESOS and French Sarcoma Group–Groupe d’Etude des Tumeurs Osseuses (GSF-GETO) Networks

      Annals of Oncology

      Oxford University Press

      sarcoma, resection, surgery, relapse, survival, reference center

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          NETSARC (netsarc.org) is a network of 26 sarcoma reference centers with specialized multidisciplinary tumor boards (MDTB) aiming to improve the outcome of sarcoma patients. Since 2010, presentation to an MDTB and expert pathological review are mandatory for sarcoma patients nationwide. In the present work, the impact of surgery in a reference center on the survival of sarcoma patients investigated using this national NETSARC registry.

          Patients and methods

          Patients’ characteristics and follow-up are prospectively collected and data monitored. Descriptive, uni- and multivariate analysis of prognostic factors were conducted in the entire series ( N = 35 784) and in the subgroup of incident patient population ( N = 29 497).


          Among the 35 784 patients, 155 different histological subtypes were reported. 4310 (11.6%) patients were metastatic at diagnosis. Previous cancer, previous radiotherapy, neurofibromatosis type 1 (NF1), and Li–Fraumeni syndrome were reported in 12.5%, 3.6%, 0.7%, and 0.1% of patients respectively. Among the 29 497 incident patients, 25 851 (87.6%) patients had surgical removal of the sarcoma, including 9949 (33.7%) operated in a NETSARC center. Location, grade, age, size, depth, histotypes, gender, NF1, and surgery outside a NETSARC center all correlated to overall survival (OS), local relapse free survival (LRFS), and event-free survival (EFS) in the incident patient population. NF1 history was one of the strongest adverse prognostic factors for LRFS, EFS, and OS. Presentation to an MDTB was associated with an improved LRFS and EFS, but was an adverse prognostic factor for OS if surgery was not carried out in a reference center. In multivariate analysis, surgery in a NETSARC center was positively correlated with LRFS, EFS, and OS [ P < 0.001 for all, with a hazard ratio of 0.681 (95% CI 0.618–0.749) for OS].


          This nationwide registry of sarcoma patients shows that surgical treatment in a reference center reduces the risk of relapse and death.

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          Most cited references 23

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          Incidence of Sarcoma Histotypes and Molecular Subtypes in a Prospective Epidemiological Study with Central Pathology Review and Molecular Testing

          Background The exact overall incidence of sarcoma and sarcoma subtypes is not known. The objective of the present population-based study was to determine this incidence in a European region (Rhone-Alpes) of six million inhabitants, based on a central pathological review of the cases. Methodology/Principal Findings From March 2005 to February 2007, pathology reports and tumor blocks were prospectively collected from the 158 pathologists of the Rhone-Alpes region. All diagnosed or suspected cases of sarcoma were collected, reviewed centrally, examined for molecular alterations and classified according to the 2002 World Health Organization classification. Of the 1287 patients screened during the study period, 748 met the criteria for inclusion in the study. The overall crude and world age-standardized incidence rates were respectively 6.2 and 4.8 per 100,000/year. Incidence rates for soft tissue, visceral and bone sarcomas were respectively 3.6, 2.0 and 0.6 per 100,000. The most frequent histological subtypes were gastrointestinal stromal tumor (18%; 1.1/100,000), unclassified sarcoma (16%; 1/100,000), liposarcoma (15%; 0.9/100,000) and leiomyosarcoma (11%; 0.7/100,000). Conclusions/Significance The observed incidence of sarcomas was higher than expected. This study is the first detailed investigation of the crude incidence of histological and molecular subtypes of sarcomas.
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            Descriptive epidemiology of sarcomas in Europe: report from the RARECARE project.

            Sarcomas are a heterogeneous group of malignant neoplasms arising from mesenchymal cells which encompass dozens of histological types and can occur in virtually any anatomic site. They form one of the principal groups of rare cancers in Europe as defined in the RARECARE project. We analysed 45,568 incident cases diagnosed during 1995-2002 and registered by 76 population-based cancer registries. Total crude incidence was 5.6 per 100,000 per year with an estimated 27,908 new cases per year in the EU27 countries, of which 84% were soft tissue sarcomas and 14% were bone sarcomas. Gastrointestinal stromal tumours (GIST) were only widely recognised as an entity in the late 1990s and consequently were under-registered. Their true incidence is believed to be about 1.5 per 100,000. Age-standardised incidence of soft tissue sarcomas ranged from 3.3 per 100,000 in Eastern Europe to 4.7 per 100,000 in Northern Europe. About 280,000 persons were estimated to be alive at the beginning of 2003 with a past diagnosis of sarcoma, of which 83% were soft tissue sarcomas and 16% were bone sarcomas. Five-year relative survival for 2000-2002 by the period was 58% for soft tissue sarcomas and 62% for bone sarcomas. The diversity and rarity of sarcomas combined with the quite large number of people affected by them mean that they provide a classic example of the importance of networking in diagnosis, therapy and research for rare cancers. Copyright © 2012 Elsevier Ltd. All rights reserved.
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              Soft Tissue Sarcoma, Version 2.2016, NCCN Clinical Practice Guidelines in Oncology.

              Soft tissue sarcomas (STS) are rare solid tumors of mesenchymal cell origin that display a heterogenous mix of clinical and pathologic characteristics. STS can develop from fat, muscle, nerves, blood vessels, and other connective tissues. The evaluation and treatment of patients with STS requires a multidisciplinary team with demonstrated expertise in the management of these tumors. The complete NCCN Guidelines for Soft Tissue Sarcoma (available at NCCN.org) provide recommendations for the diagnosis, evaluation, and treatment of extremity/superficial trunk/head and neck STS, as well as intra-abdominal/retroperitoneal STS, gastrointestinal stromal tumor, desmoid tumors, and rhabdomyosarcoma. This manuscript discusses guiding principles for the diagnosis and staging of STS and evidence for treatment modalities that include surgery, radiation, chemoradiation, chemotherapy, and targeted therapy.

                Author and article information

                Ann Oncol
                Ann. Oncol
                Annals of Oncology
                Oxford University Press
                July 2019
                09 April 2019
                09 April 2019
                : 30
                : 7 , Targeting the PI3-kinase pathway in triple-negative breast cancer
                : 1143-1153
                [1 ]Department of Medical Oncology and Department of Surgical Oncology, Centre Léon Bérard, Lyon
                [2 ]Université Claude Bernard, Lyon
                [3 ]Institut de Cancerologie L. Neuwirth
                [4 ]Department of Medical Oncology and Department of Surgical Oncology, Gustave Roussy Cancer Campus—Surgery, Villejuif
                [5 ]Department of Medical Oncology and Department of Surgical Oncology, Institut Bergonié, Bordeaux
                [6 ]Department of Medical Oncology and Department of Surgical Oncology, Centre Oscar Lambret, Lille
                [7 ]Department of Surgical Oncology, CHU, Lille
                [8 ]Department of Medical Oncology and Department of Surgical Oncology, Institut de Cancerologie Nantes, Nantes
                [9 ]Department of Orthopedics, CHU Nantes, Nantes
                [10 ]Department of Orthopedics, Hôpital Cochin-Saint-Vincent de Paul, Paris
                [11 ]Department of Medical Oncology and Department of Surgical Oncology, Institut Universitaire de Cancerologie de Toulouse, Claudius Regaud, Toulouse
                [12 ]Department of Medical Oncology and Department of Orthopedics, La Timone University Hospital, Marseille
                [13 ]Medical Oncology Department, Eugene Marquis Comprehensive Cancer Center, Rennes
                [14 ]Department of Orthopedics, CHU, Rennes
                [15 ]Medical Oncology Department, Institut de Cancérologie de Montpellier, Montpellier
                [16 ]Department of Medical Oncology and Department of Surgical Oncology, Centre Alexis Vautrin, Vandoeuvre-lès-Nancy
                [17 ]Department of Surgical Oncology and Department of Medical Oncology, Centre Paul Strauss & CHU Strasbourg, Hôpitaux Universitaires de Strasbourg, Strasbourg
                [18 ]Department of Orthopedics, CHU de Tours, Tours
                [19 ]Department of Medical Oncology and Department of Surgical Oncology, Institut Paoli Calmettes, Marseille
                [20 ]Department of Medical Oncology and Department of Surgical Oncology, Centre Antoine-Lacassagne, Nice
                [21 ]Oncology Unit, Saint Louis Hospital, Paris
                [22 ]Department of Surgery, Centre Georges François Leclerc, Dijon
                [23 ]Department of Surgery, Centre Jean Perrin/ERTICa EA, Clermont-Ferrand
                [24 ]Department of Medical Oncology and Department of Surgical Oncology, CHU Limoges, Limoges
                [25 ]Medical Oncology Department, CHU Besancon, Besançon
                [26 ]Department of Medical Oncology and Department of Surgical Oncology, Institut J Godinot Reims
                [27 ]Department of Surgical Oncology, Centre Francois Baclesse, Caen
                [28 ]Department of Surgery, Centre Henri Becquerel, Rouen
                [29 ]Medical Oncology Department, APHP La Pitié Salpetriere/Tenon/Bicetre, Paris
                [30 ]Medical Oncology Department, CHU La Réunion, Saint-Pierre, La Réunion
                [31 ]Surgery Department, Institut Curie, Paris, France
                Author notes
                Correspondence to: Prof. Jean-Yves Blay, Department of Medical Oncology, Centre Léon Bérard & Université Claude Bernard Lyon I, 28 rue Laënnec, 69373 Lyon Cedex 08, France. Tel: +33-4-78-78-51-26; E-mail: jean-yves.blay@ 123456lyon.unicancer.fr

                The members are listed in the supplementary document S1 (available at Annals of Oncology online).

                © The Author(s) 2019. Published by Oxford University Press on behalf of the European Society for Medical Oncology.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com

                Page count
                Pages: 11
                Funded by: NetSARC
                Funded by: INCa & DGOS
                Funded by: RREPS
                Funded by: RESOS (INCa & DGOS) and LYRICAN (INCa-DGOS-INSERM 12563)
                Funded by: Association DAM’s, Ensemble contre Le GIST
                Funded by: Eurosarc
                Award ID: FP7-278742
                Funded by: la Fondation ARC, Infosarcome
                Award ID: ANR-10-LABX-0061
                Funded by: Ligue de L’Ain contre le Cancer, La Ligue contre le Cancer
                Funded by: EURACAN
                Award ID: EC 739521
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