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ETIOSARC study : environmental aetiology of sarcomas from a French prospective multicentric population-based case–control study—study protocol
  1. Aude Lacourt1,
  2. Brice Amadéo1,2,
  3. Céline Gramond1,
  4. Emilie Marrer3,
  5. Sandrine Plouvier4,
  6. Isabelle Baldi1,5,
  7. Jean-Yves Blay6,
  8. Jean-Michel Coindre7,
  9. Gonzague de Pinieux8,
  10. François Gouin9,
  11. Antoine Italiano10,
  12. Axel Le Cesne11,
  13. François Le Loarer7,
  14. Alain Monnereau1,12,
  15. Isabelle Pellegrin13,
  16. Nicolas Penel14,
  17. Isabelle Ray-Coquard6,
  18. Maud Toulmonde10,
  19. Françoise Ducimetière15,
  20. Simone Mathoulin-Pélissier1,16
  21. for the Etiosarc study group
    1. 1 Inserm U1219 - EPICENE team, Université de Bordeaux, BordeauxCedex, Aquitaine, France
    2. 2 Inserm CIC 1401, Registre des cancers de la Gironde, Université de Bordeaux, BordeauxCedex, Aquitaine, France
    3. 3 Registre des cancers du Haut-Rhin, Groupe Hospitalier de la Région de Mulhouse et Sud Alsace, Mulhouse, Grand Est, France
    4. 4 Registre des cancers de Lille et de sa Région, Lille, France
    5. 5 Pôle de Santé Publique, Service de Médecine du Travail et Pathologies professionnelles, CHU de Bordeaux, Bordeaux, Aquitaine, France
    6. 6 Department of Medical Oncology, Centre Léon Berard, Université Claude Bernard Lyon 1, Lyon, Auvergne-Rhône-Alpes, France
    7. 7 Department of Biopathology, Bergonie Institute, Bordeaux, Aquitaine, France
    8. 8 Service d’anatomopathologie, CHRU Tours, Tours, France
    9. 9 Inserm UI957, CHU Nantes, Nantes, Pays de la Loire, France
    10. 10 Department of Medical Oncology, Institute Bergonie, Bordeaux, Aquitaine, France
    11. 11 Medical Oncology Department, Institut Gustave Roussy, Villejuif, France
    12. 12 Gironde Registry of Haematological Malignancies, Institute Bergonie, Bordeaux, Aquitaine, France
    13. 13 Service d’Immunologie-Immunogénétique, Pôle de Biologie et Pathologie, CHU de Bordeaux, Bordeaux, Aquitaine, France
    14. 14 Medical Oncology Department, Ctr Oscar Lambret, Lille, France
    15. 15 Réseau de référence sarcome, Centre Léon Bérard, Lyon, Rhône-Alpes, France
    16. 16 Clinical and Epidemiological Research Unit, Inserm CIC 1401, Institute Bergonie, Bordeaux, Aquitaine, France
    1. Correspondence to Mrs. Aude Lacourt; aude.lacourt{at}inserm.fr

    Abstract

    Introduction Sarcomas are rare tumours of connective tissue. The exact overall incidence of sarcomas is unknown due to diagnostic difficulties and the various histological subtypes (over 80 subtypes). However, the apparent increasing incidence of sarcomas suggests environmental causes such as pesticides. Except for some specific factors (ie, ionising radiation, vinyl chloride, dioxin and genetic predispositions) the scientific knowledge on the aetiology of sarcomas is sparse and inconsistent. France is a particularly appropriate country to set up a study investigating the causes of sarcoma occurrence due to the French organisation in treatment and care of sarcoma patients, which is highly structured and revolved around national expert networks. The main objective of the ETIOlogy of SARcomas (ETIOSARC) project is to study the role of lifestyle, environmental and occupational factors in the occurrence of sarcomas among adults from a multicentric population-based case–control study.

    Methods and analysis Cases will be all incident patients (older than 18 years) prospectively identified in 15 districts of France covered by a general population-based cancer registry and/or a reference centre in sarcoma’s patient care over a 3-year period with an inclusion start date ranging from February 2019 to January 2020 and histologically confirmed by a second review of the diagnosis. Two controls will be individually matched by sex, age (5 years group) and districts of residence and randomly selected from electoral rolls. A standardised questionnaire will be administered by a trained interviewer in order to gather information about occupational and residential history, demographic and socioeconomic characteristics and lifestyle factors. At the end of the interview, a saliva sample will be systematically proposed. This study will permit to validate or identify already suspected risk factors for sarcomas such as phenoxyherbicides, chlorophenol and to generate new hypothesis to increase our understanding about the genetic and environmental contributions in the carcinogenicity process.

    Ethics and dissemination The present study is promoted by the French National Institute of Health and Medical Research (identification number C17-03). This study received National French Ethic committee (CPP Sud Méditerrannée I) approval (identification number 18-31) and French Data Protection Authority (CNIL) approval (identification number 918171). Results of this study will be published in international peer-reviewed journals. Technical appendix, statistical code and dataset will be available in the Dryad repository when collection data are completed.

    Trial registration number NCT03670927.

    • cancer
    • sarcoma
    • case–control study
    • environmental and occupational exposure
    • aetiology

    This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited, appropriate credit is given, any changes made indicated, and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/.

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    Footnotes

    • Contributors AL prepared the first draft for all sections of this manuscript with the help of all coauthors. AL, CG, BA, IB, AM and SM-P participated in the literature review. AL, BA, CG and SM-P are responsible for the national coordination center and planed the first draft of the present protocol. EM, SP, BA and AM will manage the cancer registries network involved in this study. J-YB, GdP, AI, ALC, NP, IR-C and FD will manage the clinical centre from the NetSarc clinical sarcoma network. MT is responsible of the shared databases RRePS/ResOS/NetSarc. J-MC, FG and FLL are responsible of the RRePS network involved in this study. IP is responsible of the biological resources center of Bordeaux hospital university centre ‘Bordeaux Biothèque Santé’. All authors were involved in the development and the writing of the protocol. All authors participated in the editing and correction of the final text. All authors read and approved the final manuscript.

    • Funding This work was supported by the French National Cancer Institute in the framework of the SHS-E-SP grant—grant number 2016-118 and by the French National Institute of health and medical research in the framework of the environment and cancer grant—grant number 17CE037-00.

    • Competing interests None declared.

    • Ethics approval This study received National French Ethic committee approval (identification number 18-31) and French Data Protection Authority (CNIL) approval (identification number 918171).

    • Provenance and peer review Not commissioned; externally peer reviewed.

    • Collaborators Etiosarc study group: A Lacourt; B Amadéo; C Gramond; E Marrer; S Plouvier; I Baldi; S Bara; C Bazille; JY Blay; E Bompas; L Chaigneau; MC Chateau; JM Coindre; G Coureau; D Cupissol; T D’Almeida; G Defossez; P Delafosse; C Delcambre Lair; G De Pinieux; A Di Marco; T Fabre; F Fiorenza; JP Ghnassia; F Gouin; AV Guizard; A Italiano; JE Kurtz; V Lebrun-Ly; A Le Cesne; F Le Loarer; LR Le Nail; C Maynou; G Missenard; F Molinié; A Monnereau; A Moreau; N Penel; D Ranchère-Vince; I Ray-Coquard; YM Robin; P Terrier; M Toulmonde; B Tretarre; M Velten; AS Woronoff; F Ducimetière; S Mathoulin-Pélissier.

    • Patient consent for publication Obtained.

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