Is rare cancer care organized at national health system level? Multiple case study in six EU countries

Background: As a system of European Reference Networks (ERNs) emerges, we aimed to shed light on the processes through which reference centres (RCs) for rare cancers are embedded in national health systems, and to formulate hypotheses about which national care models favour equitable access for patients. Methods We used a multiple-case-study design based on the experiences of Czechia, Finland, France, Italy, Lithuania and Spain. Using sarcoma as an example of rare cancer, 52 semi-structured interviews were conducted during six on-site visits. Results The comparative analysis showed substantial heterogeneity in the processes for formalising RCs status and in their levels of integration in the different health systems, but two models, namely, the centre-based and the network-based, can be envisaged at national level. RCs for rare cancers were legally established only in France and Spain. Expert clinicians cooperate in a structured way, using network mechanisms, in France and Italy, and these countries, plus Finland and Lithuania, had a referral system to facilitate patients' access from non-expert centres to RCs. Comparative analysis of the cases enabled the identification of key healthcare planning principles in instituting RCs at the national level, among them the need to stipulate the involvement of expert professionals in steering the rare cancer care system