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

PLoS One. 2021 Feb 25;16(2):e0246958. doi: 10.1371/journal.pone.0246958. eCollection 2021.

Abstract

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.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Female
  • France / epidemiology
  • Humans
  • Incidence
  • Male
  • Middle Aged
  • Neoplasm Grading
  • Prospective Studies
  • Sarcoma / classification
  • Sarcoma / diagnosis
  • Sarcoma / epidemiology*
  • Sarcoma / pathology*
  • World Health Organization
  • Young Adult

Grants and funding

This work was supported by Institut National Du Cancer (FR) NETSARC+, Institut National Du Cancer (FR) RREPS, Institut National Du Cancer (FR) RESOS, Institut National Du Cancer (FR) NETSARC, Institut National Du Cancer (FR) LYriCAN (INCa-DGOS-INSERM_12563), Agence Nationale de la Recherche (FR) Institut Convergence PLASCA, (17-CONV-0002), Agence Nationale de la Recherche (FR) LabEx DEvweCAN (ANR-10-LABX-0061), Agence Nationale de la Recherche (FR) RHU4 DEPGYN (ANR-18-RHUS-0009) Institut National Du Cancer (FR) INTERSARC, Fondation ARC pour la Recherche sur le Cancer Unicancer, Ligue Contre le Cancer (FR) Unicancer, Ligue Contre le Cancer Comité de l’Ain Canopee, and by EUROPEAN COMMISSION (EU) EURACAN (EC 739521). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.