Nationwide incidence of sarcomas and connective tissue tumors of intermediate malignancy over four years using an expert pathology review network.


Journal

PloS one
ISSN: 1932-6203
Titre abrégé: PLoS One
Pays: United States
ID NLM: 101285081

Informations de publication

Date de publication:
2021
Historique:
received: 20 06 2020
accepted: 28 01 2021
entrez: 25 2 2021
pubmed: 26 2 2021
medline: 6 8 2021
Statut: epublish

Résumé

Since 2010, nationwide networks of reference centers for sarcomas (RREPS/NETSARC/RESOS) collected and prospectively reviewed all cases of sarcomas and connective tumors of intermediate malignancy (TIM) in France. The nationwide incidence of sarcoma or TIM (2013-2016) was measured using the 2013 WHO classification and confirmed by a second independent review by expert pathologists. Simple clinical characteristics, yearly variations and correlation of incidence with published clinical trials are presented and analyzed. Over 150 different histological subtypes are reported from the 25172 patients with sarcomas (n = 18712, 74,3%) or TIM (n = 6460, 25.7%), with n = 5838, n = 6153, n = 6654, and n = 6527 yearly cases from 2013 to 2016. Over these 4 years, the yearly incidence of sarcomas and TIM was therefore 70.7 and 24.4 respectively, with a combined incidence of 95.1/106/year, higher than previously reported. GIST, liposarcoma, leiomyosarcomas, undifferentiated sarcomas represented 13%, 13%, 11% and 11% of tumors. Only GIST, as a single entity had a yearly incidence above 10/106/year. There were respectively 30, 64 and 66 different histological subtypes of sarcomas or TIM with an incidence ranging from 10 to 1/106, 1-0.1/106, or < 0.1/106/year respectively. The 2 latter incidence groups represented 21% of the patients with 130 histotypes. Published phase III and phase II clinical trials (p<10-6) are significantly higher with sarcomas subtypes with an incidence above 1/106 per. This nationwide registry of sarcoma patients, with exhaustive histology review by sarcoma experts, shows that the incidence of sarcoma and TIM is higher than reported, and that tumors with a very low incidence (1<106/year) are less likely to be included in clinical trials.

Sections du résumé

BACKGROUND
Since 2010, nationwide networks of reference centers for sarcomas (RREPS/NETSARC/RESOS) collected and prospectively reviewed all cases of sarcomas and connective tumors of intermediate malignancy (TIM) in France.
METHODS
The nationwide incidence of sarcoma or TIM (2013-2016) was measured using the 2013 WHO classification and confirmed by a second independent review by expert pathologists. Simple clinical characteristics, yearly variations and correlation of incidence with published clinical trials are presented and analyzed.
RESULTS
Over 150 different histological subtypes are reported from the 25172 patients with sarcomas (n = 18712, 74,3%) or TIM (n = 6460, 25.7%), with n = 5838, n = 6153, n = 6654, and n = 6527 yearly cases from 2013 to 2016. Over these 4 years, the yearly incidence of sarcomas and TIM was therefore 70.7 and 24.4 respectively, with a combined incidence of 95.1/106/year, higher than previously reported. GIST, liposarcoma, leiomyosarcomas, undifferentiated sarcomas represented 13%, 13%, 11% and 11% of tumors. Only GIST, as a single entity had a yearly incidence above 10/106/year. There were respectively 30, 64 and 66 different histological subtypes of sarcomas or TIM with an incidence ranging from 10 to 1/106, 1-0.1/106, or < 0.1/106/year respectively. The 2 latter incidence groups represented 21% of the patients with 130 histotypes. Published phase III and phase II clinical trials (p<10-6) are significantly higher with sarcomas subtypes with an incidence above 1/106 per.
CONCLUSIONS
This nationwide registry of sarcoma patients, with exhaustive histology review by sarcoma experts, shows that the incidence of sarcoma and TIM is higher than reported, and that tumors with a very low incidence (1<106/year) are less likely to be included in clinical trials.

Identifiants

pubmed: 33630918
doi: 10.1371/journal.pone.0246958
pii: PONE-D-20-18848
pmc: PMC7906477
doi:

Types de publication

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

Langues

eng

Sous-ensembles de citation

IM

Pagination

e0246958

Déclaration de conflit d'intérêts

No competing interests.

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Auteurs

Gonzague de Pinieux (G)

Department of pathology, CHU de Tours, Tours, France.

Marie Karanian (M)

Department of Biopathology, Centre Léon Bérard, Lyon, France.

Francois Le Loarer (F)

Department of Biopathology, Institut Bergonié, Bordeaux, France.

Sophie Le Guellec (S)

Department of Biopathology, Institut Claudius Regaud et Institut Universitaire du Cancer de Toulouse-Oncopôle, Toulouse, France.

Sylvie Chabaud (S)

Department of Biopathology, Centre Léon Bérard, Lyon, France.

Philippe Terrier (P)

Department of Biopathology, Gustave Roussy, Villejuif, France.

Corinne Bouvier (C)

Department of pathology, La Timone University Hospital, Marseille, France.

Maxime Batistella (M)

Pathology Department, Saint-Louis Hospital, AP-HP, Université de Paris, Paris, France.

Agnès Neuville (A)

Department of Biopathology, Institut Bergonié, Bordeaux, France.

Yves-Marie Robin (YM)

Pôle de Biologie-Pathologie-Génétique Centre Oscar Lambret, & Institut de Pathologie entre Oscar Lambret & CHU Lille, Lille, France.

Jean-Francois Emile (JF)

Department of Biopathology, Hopital Ambroise Paré, Boulogne, France.

Anne Moreau (A)

Department of Pathology, Department of Orthopedy CHU Nantes, Nantes, France.

Frederique Larousserie (F)

Department of Biopathology, Hôpital Cochin-Saint-Vincent de Paul, Paris, France.

Agnes Leroux (A)

Department of Biopathology, Institut de Cancérologie de Lorraine-Alexis Vautrin, Vandoeuvre-lès-Nancy, France.

Nathalie Stock (N)

Department of Biopathology, Eugene Marquis Comprehensive Cancer Center & CHU Rennes, Rennes, France.

Marick Lae (M)

Department of Biopathology, Eugene Marquis Comprehensive Cancer Center & CHU Rennes, Rennes, France.
Department of Biopathology, Institut Curie, Paris, France.

Francoise Collin (F)

Department of Biopathology, Centre Georges François Leclerc, Dijon, France.

Nicolas Weinbreck (N)

Medipath, Frejus, France.

Sebastien Aubert (S)

Pôle de Biologie-Pathologie-Génétique Centre Oscar Lambret, & Institut de Pathologie entre Oscar Lambret & CHU Lille, Lille, France.

Florence Mishellany (F)

Department of Biopathology, Centre Jean Perrin, Clermont-Ferrand, France.

Celine Charon-Barra (C)

Department of Biopathology, Centre Georges François Leclerc, Dijon, France.

Sabrina Croce (S)

Department of Biopathology, Institut Bergonié, Bordeaux, France.

Laurent Doucet (L)

Department of pathology, CHRU Brest, Brest, France.

Isabelle Quintin-Rouet (I)

Department of pathology, CHRU Brest, Brest, France.

Marie-Christine Chateau (MC)

Department of Biopathology, Institut de Cancérologie de Montpellier & CHU Montpellier, Montpellier, France.

Celine Bazille (C)

Department of Biopathology, Centre Francois Baclesse, Caen, France.

Isabelle Valo (I)

Department of Pathology, Institut de Cancerologie de l'Ouest, Angers, France.

Bruno Chetaille (B)

Medipath, Frejus, France.

Nicolas Ortonne (N)

Department of Biopathology, Hopital Henri Mondor, Creteil, France.

Anne Brouchet (A)

Department of Biopathology, Institut Claudius Regaud et Institut Universitaire du Cancer de Toulouse-Oncopôle, Toulouse, France.

Philippe Rochaix (P)

Department of Biopathology, Institut Claudius Regaud et Institut Universitaire du Cancer de Toulouse-Oncopôle, Toulouse, France.

Anne Demuret (A)

Department of pathology, CHU de Tours, Tours, France.

Jean-Pierre Ghnassia (JP)

Department of Biopathology, Centre Paul Strauss, Strasbourg, France.

Lenaig Mescam (L)

Department of Biopathology, Institut Paoli Calmettes, Marseille, France.

Nicolas Macagno (N)

Department of pathology, La Timone University Hospital, Marseille, France.

Isabelle Birtwisle-Peyrottes (I)

Department of Biopathology, Centre Antoine-Lacassagne, Nice, France.

Christophe Delfour (C)

Department of Biopathology, Institut de Cancérologie de Montpellier & CHU Montpellier, Montpellier, France.

Emilie Angot (E)

Department of Biopathology, Eugene Marquis Comprehensive Cancer Center & CHU Rennes, Rennes, France.

Isabelle Pommepuy (I)

Department of Biopathology, CHU Limoges, Limoges, France.

Dominique Ranchere (D)

Department of Biopathology, Centre Léon Bérard, Lyon, France.

Claire Chemin-Airiau (C)

Department of Biopathology, Centre Léon Bérard, Lyon, France.

Myriam Jean-Denis (M)

Department of Biopathology, Centre Léon Bérard, Lyon, France.

Yohan Fayet (Y)

Department of Biopathology, Centre Léon Bérard, Lyon, France.

Jean-Baptiste Courrèges (JB)

Department of Biopathology, Institut Bergonié, Bordeaux, France.

Nouria Mesli (N)

Department of Biopathology, Institut Bergonié, Bordeaux, France.

Juliane Berchoud (J)

Department of Pathology, Department of Orthopedy CHU Nantes, Nantes, France.

Maud Toulmonde (M)

Department of Biopathology, Institut Bergonié, Bordeaux, France.

Antoine Italiano (A)

Department of Biopathology, Institut Bergonié, Bordeaux, France.

Axel Le Cesne (A)

Department of Biopathology, Centre Léon Bérard, Lyon, France.

Nicolas Penel (N)

Pôle de Biologie-Pathologie-Génétique Centre Oscar Lambret, & Institut de Pathologie entre Oscar Lambret & CHU Lille, Lille, France.

Francoise Ducimetiere (F)

Department of Biopathology, Centre Léon Bérard, Lyon, France.

Francois Gouin (F)

Department of Biopathology, Centre Léon Bérard, Lyon, France.

Jean-Michel Coindre (JM)

Department of Biopathology, Institut Bergonié, Bordeaux, France.

Jean-Yves Blay (JY)

Department of Biopathology, Centre Léon Bérard, Lyon, France.
Department of Medicine of Centre Leon Berard, University Claude Bernard Lyon I, Lyon, France.
Headquarters, Unicancer, Paris, France.

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