Economic Modelling of Screen-and-Treat Strategies for Brazilian Women at Risk of Hereditary Breast and Ovarian Cancer

Applied Health Economics and Health Policy https://doi.org/10.1007/s40258-020-00599-0 ORIGINAL RESEARCH ARTICLE Economic Modelling of Screen‑and‑Treat Strategies for Brazilian Women at Risk of Hereditary Breast and Ovarian Cancer Julia Simoes Correa‑Galendi1 · Maria del Pilar Estevez Diz2 · Stephanie Stock1 · Dirk Müller1 © The Author(s) 2020 Abstract Background Clinical evidence supports the use of genetic counselling and BRCA1/2 testing for women at risk for hereditary breast and ovarian cancer. Currently, screen-and-treat strategies are not reimbursed in the Brazilian Unified Healthcare System (SUS). The aim of this modelling study was to evaluate the cost effectiveness of a gene-based screen-and-treat strategy for BRCA1/2 in women with a high familial risk followed by preventive interventions compared with no screening. Methods Adopting the SUS perspective, a Markov model with a lifelong time horizon was developed for a cohort of healthy women aged 30 years that fulfilled the criteria for BRCA1/2 testing according to the National Comprehensive Cancer Network (NCCN) guideline. For women who tested positive, preventive options included intensified surveillance, risk-reducing bilateral mastectomy and bilateral salpingo-oophorectomy. The Markov model comprised the health states ‘well’, ‘breast cancer’, ‘death’ and two post-cancer states. Outcomes were the incremental costs per quality-adjusted life-year (QALY) and the incremental costs per life-year gained (LYG). Data were mainly obtained by a literature review. Deterministic and probabilistic sensitivity analyses were performed to assess the robustness of the results. Results In the base case, the screen-and-treat strategy resulted in additional costs of 3515 Brazilian reais (R$) (US$1698) and a gain of 0.145 QALYs, compared with no screening. The incremental cost-effectiveness ratio (ICER) was R$24,263 (US$21,724) per QALY and R$27,258 (US$24,405) per LYG. Applying deterministic sensitivity analyses, the ICER was most sensitive to the probability of a positive test result and the discount rate. In the probabilistic sensitivity analysis, a willingness to pay of R$25,000 per QALY gained for the screen-and-treat strategy resulted in a probability of cost effectiveness of 80%. Conclusion Although there is no rigorous cost-effectiveness threshold in Brazil, the result of this cost-effectiveness analysis may support the inclusion of BRCA1/2 testing for women at high-risk of cancer in the SUS. The ICER calculated for the provision of genetic testing for BRCA1/2 approximates the cost-effectiveness threshold proposed by the World Health Organization (WHO) for low- and middle-income countries. Key Points for Decision Makers Electronic supplementary material The online version of this article (https://doi.org/10.1007/s40258-020-00599-0) contains supplementary material, which is available to authorized users. * Julia Simoes Correa‑Galendi julia.simoes‑correa‑galendi@uk‑koeln.de 1 Institute for Health Economics and Clinical Epidemiology, The University Hospital of Cologne (AöR), Sao Paulo, SP, Brazil 2 Insituto Do Cancer Do Estado de Sao Paulo, Hospital das Clinicas HCFMUSP, Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo, SP, Brazil Genetic testing for BRCA1/2 for healthy women with high familiar risk results in more quality-adjusted life years at a moderately higher cost. This economic modelling shows that a screen-and-treat strategy for women at risk for hereditary breast and ovarian cancer might be cost-effective from the perspective of the Brazilian Unified Healthcare System (SUS). Vol.:(0123456789) J. Simoes Correa‑Galendi et al. 1 Introduction Breast cancer is the main cause of cancer-related death in Brazilian women [1]. According to the Brazilian National Cancer Institute, in 2018, the incidence of breast cancer amounted to 56 cases per 100,000 women, resulting in 59,700 new cases annually [1]. Additionally, 6150 incident cases of ovarian cancer were diagnosed in Brazil in 2018 [1]. Although there has been a trend toward stabilization in female breast cancer mortality rates in Brazil, the statespecific mortality rates show considerable inequalities that may indicate disparities in healthcare availability [2]. The risk of breast cancer or ovarian cancer increases with the number of affected relatives in the family and the closeness of the relationship (i.e. first- or second-degree relative). Additionally, the younger a woman is diagnosed with cancer, the more likely a genetic component will be found [3]. Hereditary breast and ovarian cancer (HBOC) are mainly associated with germline mutations in the genes BRCA1 and BRCA2 (collectively named BRCAhereafter). Pathogenic variants in the BRCA genes are important predictors of breast and ovarian cancer, with a 40–80% lifetime risk of breast cancer and an 11–50% lifetime risk of ovarian cancer, respectively [4]. In case of a BRCA mutation, a non-directive counselling session should be provided to inform women of their absolute individual risk. For women who have tested positive, different preventive options are recommended. According to published evidence, risk-reducing surgery (bilateral mastectomy or bilateral salpingo-oophorectomy), chemoprevention and enhanced surveillance are effective for reducing the incidence and mortality of cancer [5, 6]. However, the decision between the available risk-reducing strategies is highly preference-sensitive [7, 8]. In order to identify high-risk women for genetic testing and counselling, the National Comprehensive Cancer Network (NCCN) guideline proposes to select women based on their personal and familial history of cancer [7]. These criteria are currently applied to reimburse genetic testing for privately insured women in Brazil [9]. Whereas private insurance offers supplementary coverage, about 70% of the population is exclusively insured within the Brazilian Unified Healthcare System (SUS). The SUS is organized according to three levels of complexity of care. In the primary level, general practitioners are responsible for basic care. If further laboratory testing and imaging is required, patients are referred to high complexity centres. Cancer care in Brazil is performed on the third level (i.e. specialized care units and hospital complexes), which is largely financed by the federal government and reimbursed according to disease-related package sums. Among the high-complexity oncology centres, only a few with research motivation offer genetic counselling and testing [10]. As a result, in Brazil there is limited access to genetic counselling and BRCA testing for women at increased familial risk. Recently, a Markov model for screen-and-treat strategies offered to Brazilian women at risk for BRCAmutation revealed an incremental cost-effectiveness ratio of R$910 (Brazilian reais) per cancer case avoided. However, the cohort simulated in this study represented only first-degree relatives of women with ovarian ca (...truncated)