Françoise Ducimetière

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.

Incidence of soft tissue sarcoma and beyond: A population-based prospective study in 3 European regions.

The objectives of this study were to measure the incidence of sarcomas, including viscerally sited tumors that are not reported in cancer statistics, and to draw explanatory clues from a large and reliable sarcoma incidence data set.

Clinicians' adherence versus non adherence to practice guidelines in the management of patients with sarcoma: a cost-effectiveness assessment in two European regions

BackgroundAlthough the management of sarcoma is improving, non adherence to clinical practice guidelines (CPGs) remains high, mainly because of the low incidence of the disease and the variety of histological subtypes. Since little is known about the health economics of sarcoma, we undertook a cost-effectiveness analysis (within the CONnective TIssue CAncer NETwork, CONTICANET) comparing costs and outcomes when clinicians adhered to CPGs and when they did not.MethodsPatients studied had a histological diagnosis of sarcoma, were older than 15 years, and had been treated in the Rhône-Alpes region of France (in 2005/2006) or in the Veneto region of Italy (in 2007). Data collected retrospectively for the three years after diagnosis were used to determine relapse free survival and health costs (adopting the hospitals perspective and a microcosting approach). All costs were expressed in euros (€) at their 2009 value. A 4% annual discount rate was applied to both costs and effects. The incremental cost-effectiveness ratio (ICER) was expressed as cost per relapse-free year gained when management was compliant with CPGs compared with when it was not. To capture uncertainty surrounding ICER, a probabilistic sensitivity analysis was performed based on a non-parametric bootstrap method.ResultsA total of 219 patients were included in the study. Compliance with CPGs was observed for 118 patients (54%). Average total costs reached 23,571 euros when treatment was in accordance with CPGs and 27,313 euros when it was not. In relation to relapse-free survival, compliance with CPGs strictly dominates non compliance, i.e. it is both less costly and more effective. Taking uncertainty into account, the probability that compliance with CPGs still strictly dominates was 75%.ConclusionsOur findings should encourage physicians to increase their compliance with CPGs and healthcare administrators to invest in the implementation of CPGs in the management of sarcoma.

Transferability of health cost evaluation across locations in oncology: cluster and principal component analysis as an explorative tool

BackgroundThe transferability of economic evaluation in health care is of increasing interest in today’s globalized environment. Here, we propose a methodology for assessing the variability of data elements in cost evaluations in oncology. This method was tested in the context of the European Network of Excellence “Connective Tissues Cancers Network”.MethodsUsing a database that was previously aimed at exploring sarcoma management practices in Rhône-Alpes (France) and Veneto (Italy), we developed a model to assess the transferability of health cost evaluation across different locations. A nested data structure with 60 final factors of variability (e.g., unit cost of chest radiograph) within 16 variability areas (e.g., unit cost of imaging) within 12 objects (e.g., diagnoses) was produced in Italy and France, separately. Distances between objects were measured by Euclidean distance, Mahalanobis distance, and city-block metric. A hierarchical structure using cluster analysis (CA) was constructed. The objects were also represented by their projections and area of variability through correlation studies using principal component analysis (PCA). Finally, a hierarchical clustering based on principal components was performed.ResultsCA suggested four clusters of objects: chemotherapy in France; follow-up with relapse in Italy; diagnosis, surgery, radiotherapy, chemotherapy, and follow-up without relapse in Italy; and diagnosis, surgery, and follow-up with or without relapse in France. The variability between clusters was high, suggesting a lower transferability of results. Also, PCA showed a high variability (i.e. lower transferability) for diagnosis between both countries with regard to the quantities and unit costs of biopsies.ConclusionCA and PCA were found to be useful for assessing the variability of cost evaluations across countries. In future studies, regression methods could be applied after these methods to elucidate the determinants of the differences found in these analyses.

Economic Impact of Centralized Histological Reviews in Patients with Sarcoma, Gist, and Desmoid Tumors

Lionel Perrier, PhD1,2; Samuel Kembou Nzale, MSc1; Pauline Rascle, MSc1; Binh Bui Nguyen, MD3; Magali Morelle, MSc1,2; Dominique Ranchère Vince, MD4; Philippe Terrier, MD5; Agnès Neuville, MD6; Anne-Valérie Decouvelaere, MD4; Axel Le Cesne, MD7; Frédéric Gomez, MD8; Christelle de la Fouchardière, MD9; Pierre Meeus, MD10; Olivier Tredan, MD9; Maurice Pérol, MD9; Jérôme Fayette, MD9; Eve-Marie Neidhardt, MD9; Pierre Biron, MD9; Helen Boyle, MD9; Perrine Marec-Bérard MD11, Fadila Farsi MD12; Françoise Ducimetière, PhD13; Jean-Yves Blay, MD, PhD9; Isabelle Ray-Coquard, MD, PhD9,13Jean-Michel Coindre, MD, PhD6

CA1 Discordant Diagnoses in Sarcoma, GIST and Desmoide Tumour in France: Results From the Network RREPS

CA1 DISCORDANT DIAGNOSES IN SARCOMA, GIST AND DESMOIDE TUMOUR IN FRANCE: RESULTS FROM THE NETWORK RREPS Perrier L1, Ranchr̀e Vince D1, Terrier P2, Neuville A3, Decouvelaere AV1, Bui B3, Le Cesne A2, Ray Coquard I1, Ducimetière F1, Jean-Denis M1, Courrèges JB3, Mesli N2, Morelle M1, Plommet N4, Blay JY1, Coindre JM3 1Leon Berard Cancer Centre, Lyon, France, 2Insitut Gustave Roussy, Villejuif, France, 3Institut Bergonié, Bordeaux, France, 4University Lyon 2, Lyon, France OBJECTIVES: Major discordant diagnoses may have strong impact on therapeutic management. So, identification of major discordant diagnoses and predictive factors were conducted in sarcoma patients. METHODS: A multicenter analysis was performed retrospectively from the prospective cohort of sarcoma patients. Inclusion criteria were patients with a diagnosis of sarcoma in 2010 and with a second opinion performed within the network RRePS (Réseau de Référence en Pathologie des Sarcomes supported be the French NCI). Major discordant diagnoses were defined as: sarcoma vs benign lesion, sarcoma vs malignant non sarcoma tumor, gastrointestinal stromal tumors (GIST) vs non GIST, and desmoid tumor vs non desmoid tumor. Patient and disease characteristics were described. Logistic regressions were used in order to define predictive factors of major discordance. RESULTS: 3621 patients were included in the study. 438 patients (12%) had a major discordant diagnoses: sarcoma versus benign lesion (or conversely) in 155 patients (58%); sarcoma instead of malignant non sarcoma tumor (or conversely) in 103 patients (24%); gastrointestinal stromal tumors (GIST) instead of non GIST in 48 patients (11%); desmoid tumor instead of non desmoid tumor in 28 patients (6%) and other (4%). Major diagnostic discordances risks were higher (i) for malignant non sarcoma tumors compared to GIST, liposarcoma, and other sarcoma histological subtypes (p 0.004); (ii) for patients who had a previous cancer (p 0.03); (iii) for limb localization compared to trunk (p 0.004); (iv) when the second opinion was requested by the initial pathologist (p 0.01). CONCLUSIONS: This study reported that sarcoma instead of benign lesion (or conversely) is the major discordant diagnosis in sarcoma patients implying that: (i) patients who should not be treated received anticancer therapy; (ii) treatments are potentially delayed for patients who should be rapidly treated. Economics evaluations are in progress in order to advise health care administrators regarding systematic second reviews in the management of sarcoma.