The aim of this project is to (1) evaluate the efficiency of early management of sarcoma patients by the sarcoma referral network (NETSARC) vs. outside the network; (2) measure the budgetary impact of the generalization of the most cost-effective strategy across the country; (3) identify the organizational and financial constraints likely to hinder the generalization of the most cost-effective strategy and propose solutions, and finally (4) analyse the budgetary impact of a generalization of sarcoma patient care by the reference network by integrating the organizational and financial solutions proposed.

The study relies on an exhaustive national cohort of all sarcoma patients who underwent primary tumor surgery for the year 2013. Patients will be allocated to four distinct strategies, each representing a different management of sarcoma patients who had a sarcoma-specialized multidisciplinary tumor board (MDTB) before the initial surgery and complete initial management in the network (strategy 1); patients who had a sarcoma-specialized MDTB before the initial surgery and initial management outside the network (strategy 2); patients who had a sarcoma-specialized MDTB after initial surgery and initial management outside the network (strategy 3); patients who had an initial management outside the network, without sarcoma-specialized MDTB neither before nor after the initial surgery (strategy 4).

Matching of the National Health Data System (SNDS) and the NETSARC databases made it possible to include 2431 patients in the study. These databases will allow to obtain information on patients, estimate overall survival and identify healthcare consumption, which will be useful in achieving study's objectives.

Introduction

Sarcomas are a large family of rare and heterogeneous tumors. As a consequence, their diagnosis and management are difficult and complex, which can lead to misdiagnosis and suboptimal patient management. Thus, in 2009, the National Cancer Institute (INCa) and the "Direction Générale de l'Offre de Soins" (DGOS) supported the creation of a sarcoma reference network (NETSARC) with the aim to guarantee optimal care for sarcoma patients. Numerous studies show that initial patients' management in the sarcoma referral network reduces the risk of death and relapse. However, the proportion of sarcoma patients initially managed within the referral network is still too low today. The aim of this project is to (1) evaluate the efficiency of early management of sarcoma patients by the sarcoma referral network vs. outside the network; (2) measure the budgetary impact of the generalization of the most cost-effective strategy across the country; (3) identify the organizational and financial constraints likely to hinder the generalization of the most cost-effective strategy and propose solutions, and finally (4) analyses the budgetary impact of a generalization of sarcoma patient care by the reference network by integrating the organizational and financial solutions proposed.

Methods and analysis

The study relies on an exhaustive national cohort of all sarcoma patients (ICD code 10 C49, C48 or C40-41) who underwent primary tumor surgery for the year 2013. Patients will be allocated to four distinct strategies, each representing a different management of sarcoma patients: patients who had a sarcoma-specialized multidisciplinary tumor board (MDTB) before the initial surgery and complete initial management in the network (strategy 1); patients who had a sarcoma-specialized MDTB before the initial surgery and initial management outside the network (strategy 2); patients who had a sarcoma-specialized MDTB after initial surgery and initial management outside the network (strategy 3); patients who had an initial management outside the network, without sarcoma-specialized MDTB neither before nor after the initial surgery (strategy 4).

Matching of the National Health Data System (SNDS) and the NETSARC databases made it possible to include 2431 patients in the study. These databases will allow to obtain information on patients, estimate overall survival and identify healthcare consumption, which will be useful to achieve study's objectives.

The evaluation of early management of operable sarcoma patients by the sarcoma reference network vs. outside the network will be based on three approaches which are both distinct and complementary in terms of decision support. (1) Firstly, an efficiency assessment based on a cost-effectiveness analysis (CEA) and a cost-utility analysis (CUA) will be carried out. The short-term CEA will use the Incremental Cost-Effectiveness Ratio (ICER) expressed as cost per life year gained, while the long-term CUA will use the ICER expressed as cost per quality-adjusted life year (QALY) gained. The efficiency analysis will be complemented by a Budget Impact Analysis (BIA) based on an estimate of the additional and avoided costs induced by the generalization of the most cost-effective strategy determined in the previous steps. (2) Next, an organizational evaluation focusing on a flow analysis will be carried out. (3) Finally, an innovative financing model will be developed to encourage inter-center cooperation.

Ethics and dissemination

The OPTISARC project will be conducted in accordance with the ethical principles of the latest version of the Declaration of Helsinki developed by the World Medical Association (WMA). This study falls within the framework of research not involving the human person, study or health assessment (RNIPH) and will be conducted in accordance with the French and European laws and regulations in force, as well as any other applicable guidelines.

This Project has obtained a favorable opinion from the "Comité d'Expertise pour les Recherches, les Etudes et les Evaluations dans le domaine de la Santé" (CEREES) and an authorization from the "Comité National Informatique et Liberté" (CNIL) allowing the access to SNDS data (DCIR and PMSI) from 2013 to 2018.

Inclusion Criteria:

• Sarcoma diagnosis between 01/01/2013 and 31/12/2013
• International Classification of Diseases (ICD) code 10: C49 Malignant neoplasm of other connective and soft tissue, or C48 Malignant neoplasm of retroperitoneum and peritoneum, or C40-41 Malignant neoplasm of bone and articular cartilage of limbs)
• Surgery of the primary tumour.

Exclusion Criteria:

• Gastro intestinal stromal tumour
• Diagnosis not confirmed by the a second histological review

Cohort of sarcoma patient diagnosed in 2013 with a surgery of initial tumour.

No sharing of individual data