Skip to main content
Top
Published in: Clinical and Translational Oncology 8/2019

Open Access 01-08-2019 | Breast Cancer | Research Article

Cost–utility analysis of germline BRCA1/2 testing in women with high-grade epithelial ovarian cancer in Spain

Authors: Carlota Moya-Alarcón, Almudena González-Domínguez, Susana Simon, Inés Pérez-Román, Antonio González-Martín, Eloisa Bayo-Lozano, Ana Beatriz Sánchez-Heras

Published in: Clinical and Translational Oncology | Issue 8/2019

Login to get access

Abstract

Purpose

Germline mutations in BRCA1 and/or BRCA2 genes (gBRCA1/2m) are associated with an increased risk of breast cancer (BC) and ovarian cancer (OC). The aim of this study was to estimate the efficiency of providing germline BRCA1/2 testing to high-grade epithelial ovarian cancer (HGEOC) patients without family history of OC or BC and the subsequent testing and management of their relatives with gBRCA1/2m in Spain.

Methods/patients

Incident HGEOC patients without family history of OC or BC who were gBRCA1/2m carriers and their relatives were simulated in a 50-year time horizon. The study compared two scenarios: BRCA1/2 testing vs no testing, using the perspective of the Spanish National Health Service. Cancer risk among gBRCA1/2m carriers was estimated based on their age and whether they had undergone risk-reducing surgeries. Direct healthcare costs and utilities of patients who developed EOC and BC were also included. A probabilistic sensitivity analysis (PSA) with 5 thousand simulations was developed considering ± 25% of the base-case value.

Results

The BRCA1/2-testing scenario amounted to €13,437,897.43 while the no-testing scenario amounted to €12,053,291.17. It was estimated that the screening test improved the quality of life among the patients’ relatives by 43.8 quality-adjusted life years (QALYs). The incremental cost–utility ratio (ICUR) was €31,621.33/QALY in the base case. The PSA showed that 89.12% of the simulations were below the €50,000/QALY threshold.

Conclusion

Providing this screening test to HGEOC patients and their relatives is cost-effective and it allows one to identify a target population with high risk of cancer to provide effective prevention strategies.
Appendix
Available only for authorised users
Literature
1.
go back to reference Galceran J, Ameijide A, Carulla M, Mateos A, Quirós JR, Rojas D, et al. Cancer incidence in Spain, 2015. Clin Transl Oncol. 2017;19:799–825.CrossRefPubMed Galceran J, Ameijide A, Carulla M, Mateos A, Quirós JR, Rojas D, et al. Cancer incidence in Spain, 2015. Clin Transl Oncol. 2017;19:799–825.CrossRefPubMed
2.
go back to reference Sociedad Española de Oncología Médica (SEOM). Las cifras del cáncer en España 2018. 2018. Sociedad Española de Oncología Médica (SEOM). Las cifras del cáncer en España 2018. 2018.
3.
go back to reference Matz M, Coleman MP, Carreira H, Salmerón D, Chirlaque MD, Allemani C. Worldwide comparison of ovarian cancer survival: Histological group and stage at diagnosis (CONCORD-2). Gynecol Oncol. 2017;144:396–404.CrossRefPubMed Matz M, Coleman MP, Carreira H, Salmerón D, Chirlaque MD, Allemani C. Worldwide comparison of ovarian cancer survival: Histological group and stage at diagnosis (CONCORD-2). Gynecol Oncol. 2017;144:396–404.CrossRefPubMed
4.
go back to reference Liede A, Karlan BY, Narod SA. Cancer risks for male carriers of germline mutations in BRCA1 or BRCA2: a review of the literature. J Clin Oncol. 2004;22:735–42.CrossRefPubMed Liede A, Karlan BY, Narod SA. Cancer risks for male carriers of germline mutations in BRCA1 or BRCA2: a review of the literature. J Clin Oncol. 2004;22:735–42.CrossRefPubMed
5.
go back to reference Rebbeck TR, Mitra N, Wan F, Sinilnikova OM, Healey S, McGuffog L, et al. Association of type and location of BRCA1 and BRCA2 mutations with risk of breast and ovarian cancer. JAMA. 2015;313:1347.CrossRefPubMedPubMedCentral Rebbeck TR, Mitra N, Wan F, Sinilnikova OM, Healey S, McGuffog L, et al. Association of type and location of BRCA1 and BRCA2 mutations with risk of breast and ovarian cancer. JAMA. 2015;313:1347.CrossRefPubMedPubMedCentral
6.
go back to reference Mavaddat N, Peock S, Frost D, Ellis S, Platte R, Fineberg E, et al. Cancer risks for BRCA1 and BRCA2 mutation carriers: results from prospective analysis of EMBRACE. JNCI J Natl Cancer Inst. 2013;105:812–22.CrossRefPubMed Mavaddat N, Peock S, Frost D, Ellis S, Platte R, Fineberg E, et al. Cancer risks for BRCA1 and BRCA2 mutation carriers: results from prospective analysis of EMBRACE. JNCI J Natl Cancer Inst. 2013;105:812–22.CrossRefPubMed
7.
go back to reference Graña B, Lastra E, Llort G, Brunet J, Isla D. SEOM clinical guidelines for hereditary cancer. Clin Transl Oncol. 2011;13:580.CrossRefPubMed Graña B, Lastra E, Llort G, Brunet J, Isla D. SEOM clinical guidelines for hereditary cancer. Clin Transl Oncol. 2011;13:580.CrossRefPubMed
8.
go back to reference Alsop K, Fereday S, Meldrum C, deFazio A, Emmanuel C, George J, et al. BRCA mutation frequency and patterns of treatment response in BRCA mutation-positive women with ovarian cancer: a report from the Australian Ovarian Cancer Study Group. J Clin Oncol. 2012;30:2654–63.CrossRefPubMedPubMedCentral Alsop K, Fereday S, Meldrum C, deFazio A, Emmanuel C, George J, et al. BRCA mutation frequency and patterns of treatment response in BRCA mutation-positive women with ovarian cancer: a report from the Australian Ovarian Cancer Study Group. J Clin Oncol. 2012;30:2654–63.CrossRefPubMedPubMedCentral
9.
go back to reference Llort G, Chirivella I, Morales R, Serrano R, Sanchez AB, Teulé A, et al. SEOM clinical guidelines in hereditary breast and ovarian cancer. Clin Transl Oncol. 2015;17:956.CrossRefPubMedPubMedCentral Llort G, Chirivella I, Morales R, Serrano R, Sanchez AB, Teulé A, et al. SEOM clinical guidelines in hereditary breast and ovarian cancer. Clin Transl Oncol. 2015;17:956.CrossRefPubMedPubMedCentral
10.
go back to reference Paluch-Shimon S, Cardoso F, Sessa C, Balmana J, Cardoso MJ, Gilbert F, et al. Prevention and screening in BRCA mutation carriers and other breast/ovarian hereditary cancer syndromes: ESMO clinical practice guidelines for cancer prevention and screening. Ann Oncol. 2016;27:v103–10.CrossRefPubMed Paluch-Shimon S, Cardoso F, Sessa C, Balmana J, Cardoso MJ, Gilbert F, et al. Prevention and screening in BRCA mutation carriers and other breast/ovarian hereditary cancer syndromes: ESMO clinical practice guidelines for cancer prevention and screening. Ann Oncol. 2016;27:v103–10.CrossRefPubMed
11.
go back to reference Eccleston A, Bentley A, Dyer M, Strydom A, Vereecken W, George A, et al. A cost-effectiveness evaluation of germline BRCA1 and BRCA2 testing in UK women with ovarian cancer. Value Health. 2017;20:567–76.CrossRefPubMedPubMedCentral Eccleston A, Bentley A, Dyer M, Strydom A, Vereecken W, George A, et al. A cost-effectiveness evaluation of germline BRCA1 and BRCA2 testing in UK women with ovarian cancer. Value Health. 2017;20:567–76.CrossRefPubMedPubMedCentral
12.
go back to reference Gabaldó Barrios X, Sarabia Meseguer MD, Marín Vera M, Sánchez Bermúdez AI, Macías Cerrolaza JA, Sánchez Henarejos P, et al. Molecular characterization and clinical interpretation of BRCA1/BRCA2 variants in families from Murcia (south-eastern Spain) with hereditary breast and ovarian cancer: clinical–pathological features in BRCA carriers and non-carriers. Fam Cancer. 2017;16:477–89.CrossRefPubMed Gabaldó Barrios X, Sarabia Meseguer MD, Marín Vera M, Sánchez Bermúdez AI, Macías Cerrolaza JA, Sánchez Henarejos P, et al. Molecular characterization and clinical interpretation of BRCA1/BRCA2 variants in families from Murcia (south-eastern Spain) with hereditary breast and ovarian cancer: clinical–pathological features in BRCA carriers and non-carriers. Fam Cancer. 2017;16:477–89.CrossRefPubMed
14.
go back to reference Chirlaque MD, Salmerón D, Ardanaz E, Galceran J, Martínez R, Marcos-Gragera R, et al. Cancer survival in Spain: estimate for nine major cancers. Ann Oncol. 2010;21:iii21–9.CrossRefPubMed Chirlaque MD, Salmerón D, Ardanaz E, Galceran J, Martínez R, Marcos-Gragera R, et al. Cancer survival in Spain: estimate for nine major cancers. Ann Oncol. 2010;21:iii21–9.CrossRefPubMed
16.
go back to reference Petrucelli N, Daly MB, Pal T. BRCA1- and BRCA2-associated hereditary breast and ovarian cancer. In: Adam MP, Ardinger HH, Pagon RA, Wallace SE, Bean LJ, Stephens K, et al., editors. GeneReviews®. Seattle: University of Washington; 2016. http://www.ncbi.nlm.nih.gov/books/NBK1247/. Accessed 22 Mar 2018. Petrucelli N, Daly MB, Pal T. BRCA1- and BRCA2-associated hereditary breast and ovarian cancer. In: Adam MP, Ardinger HH, Pagon RA, Wallace SE, Bean LJ, Stephens K, et al., editors. GeneReviews®. Seattle: University of Washington; 2016. http://​www.​ncbi.​nlm.​nih.​gov/​books/​NBK1247/​. Accessed 22 Mar 2018.
17.
go back to reference Alonso-Babarro A, Bruera E, Varela-Cerdeira M, Boya-Cristia MJ, Madero R, Torres-Vigil I, et al. Can this patient be discharged home? Factors associated with at-home death among patients with cancer. J Clin Oncol. 2011;29:1159–67.CrossRefPubMedPubMedCentral Alonso-Babarro A, Bruera E, Varela-Cerdeira M, Boya-Cristia MJ, Madero R, Torres-Vigil I, et al. Can this patient be discharged home? Factors associated with at-home death among patients with cancer. J Clin Oncol. 2011;29:1159–67.CrossRefPubMedPubMedCentral
18.
go back to reference Bennett MI, Ziegler L, Allsop M, Daniel S, Hurlow A. What determines duration of palliative care before death for patients with advanced disease? A retrospective cohort study of community and hospital palliative care provision in a large UK city. BMJ Open. 2016;6:e012576.CrossRefPubMedPubMedCentral Bennett MI, Ziegler L, Allsop M, Daniel S, Hurlow A. What determines duration of palliative care before death for patients with advanced disease? A retrospective cohort study of community and hospital palliative care provision in a large UK city. BMJ Open. 2016;6:e012576.CrossRefPubMedPubMedCentral
19.
go back to reference Alonso-Babarro A, Astray-Mochales J, Dominguez-Berjon F, Genova-Maleras R, Bruera E, Diaz-Mayordomo A, et al. The association between in-patient death, utilization of hospital resources and availability of palliative home care for cancer patients. Palliat Med. 2013;27:68–75.CrossRefPubMed Alonso-Babarro A, Astray-Mochales J, Dominguez-Berjon F, Genova-Maleras R, Bruera E, Diaz-Mayordomo A, et al. The association between in-patient death, utilization of hospital resources and availability of palliative home care for cancer patients. Palliat Med. 2013;27:68–75.CrossRefPubMed
20.
go back to reference Ministerio de Sanidad, Servicios Sociales e Igualdad. Instituto de Información Sanitaria. Registro de altas. 2015. Ministerio de Sanidad, Servicios Sociales e Igualdad. Instituto de Información Sanitaria. Registro de altas. 2015.
21.
go back to reference Gómez-Batiste X, Porta-Sales J, Espinosa-Rojas J, Pascual-López A, Tuca A, Rodriguez J. Effectiveness of palliative care services in symptom control of patients with advanced terminal cancer: a Spanish, multicenter, prospective, quasi-experimental, pre-post study. J Pain Symptom Manag. 2010;40:652–60.CrossRef Gómez-Batiste X, Porta-Sales J, Espinosa-Rojas J, Pascual-López A, Tuca A, Rodriguez J. Effectiveness of palliative care services in symptom control of patients with advanced terminal cancer: a Spanish, multicenter, prospective, quasi-experimental, pre-post study. J Pain Symptom Manag. 2010;40:652–60.CrossRef
22.
go back to reference Alonso-Babarro A, Varela-Cerdeira M, Torres-Vigil I, Rodríguez-Barrientos R, Bruera E. At-home palliative sedation for end-of-life cancer patients. Palliat Med. 2010;24:486–92.CrossRefPubMed Alonso-Babarro A, Varela-Cerdeira M, Torres-Vigil I, Rodríguez-Barrientos R, Bruera E. At-home palliative sedation for end-of-life cancer patients. Palliat Med. 2010;24:486–92.CrossRefPubMed
23.
go back to reference Calvo-Espinos C, Ruiz de Gaona E, Gonzalez C, Ruiz de Galarreta L, Lopez C. Palliative sedation for cancer patients included in a home care program: a retrospective study. Palliat Support Care. 2014;13:619–24.CrossRefPubMed Calvo-Espinos C, Ruiz de Gaona E, Gonzalez C, Ruiz de Galarreta L, Lopez C. Palliative sedation for cancer patients included in a home care program: a retrospective study. Palliat Support Care. 2014;13:619–24.CrossRefPubMed
24.
go back to reference Mediana de los costes de los Boletines Oficiales de las Comunidades Autónomas. 2017. Mediana de los costes de los Boletines Oficiales de las Comunidades Autónomas. 2017.
25.
go back to reference Bastida JL, Oliva J, Antoñanzas F, García-Altés A, Gisbert R, Mar J, et al. Propuesta de guía para la evaluación económica aplicada a las tecnologías sanitarias. Gac Sanit. 2010;24:154–70.CrossRef Bastida JL, Oliva J, Antoñanzas F, García-Altés A, Gisbert R, Mar J, et al. Propuesta de guía para la evaluación económica aplicada a las tecnologías sanitarias. Gac Sanit. 2010;24:154–70.CrossRef
27.
go back to reference García-Gutierrez S, Sarasqueta C, Legarreta MJ, Gonzalez N, Redondo M, Rivero A, et al. Health services research in patients with breast cancer (CAMISS-prospective): study protocol for an observational prospective study. BMC Cancer. 2018;18:54.CrossRefPubMedPubMedCentral García-Gutierrez S, Sarasqueta C, Legarreta MJ, Gonzalez N, Redondo M, Rivero A, et al. Health services research in patients with breast cancer (CAMISS-prospective): study protocol for an observational prospective study. BMC Cancer. 2018;18:54.CrossRefPubMedPubMedCentral
28.
go back to reference Arrospide A, Soto-Gordoa M, Acaiturri T, López-Vivanco G, Abecia LC, Mar J. Cost of breast cancer treatment by clinical stage in the Basque Country, Spain. Rev Esp Salud Pública. 2015;89:93–7.CrossRefPubMed Arrospide A, Soto-Gordoa M, Acaiturri T, López-Vivanco G, Abecia LC, Mar J. Cost of breast cancer treatment by clinical stage in the Basque Country, Spain. Rev Esp Salud Pública. 2015;89:93–7.CrossRefPubMed
29.
go back to reference Luengo-Fernandez R, Leal J, Gray A, Sullivan R. Economic burden of cancer across the European Union: a population-based cost analysis. Lancet Oncol. 2013;14:1165–74.CrossRefPubMed Luengo-Fernandez R, Leal J, Gray A, Sullivan R. Economic burden of cancer across the European Union: a population-based cost analysis. Lancet Oncol. 2013;14:1165–74.CrossRefPubMed
31.
go back to reference Havrilesky LJ, Broadwater G, Davis DM, Nolte KC, Barnett JC, Myers ER, et al. Determination of quality of life-related utilities for health states relevant to ovarian cancer diagnosis and treatment. Gynecol Oncol. 2009;113:216–20.CrossRefPubMedPubMedCentral Havrilesky LJ, Broadwater G, Davis DM, Nolte KC, Barnett JC, Myers ER, et al. Determination of quality of life-related utilities for health states relevant to ovarian cancer diagnosis and treatment. Gynecol Oncol. 2009;113:216–20.CrossRefPubMedPubMedCentral
32.
go back to reference Peasgood T, Ward SE, Brazier J. Health-state utility values in breast cancer. Expert Rev Pharmacoecon Outcomes Res. 2010;10:553–66.CrossRefPubMed Peasgood T, Ward SE, Brazier J. Health-state utility values in breast cancer. Expert Rev Pharmacoecon Outcomes Res. 2010;10:553–66.CrossRefPubMed
33.
go back to reference National Institute for Clinical Excellence (NICE). Cost-effectiveness evidence review. Familial breast cancer: classification and care of women at risk of familial breast cancer and management of breast cancer and related risk in people with a family history of breast cancer. 2013. National Institute for Clinical Excellence (NICE). Cost-effectiveness evidence review. Familial breast cancer: classification and care of women at risk of familial breast cancer and management of breast cancer and related risk in people with a family history of breast cancer. 2013.
35.
go back to reference Grann VR, Patel PR, Jacobson JS, Warner E, Heitjan DF, Ashby-Thompson M, et al. Comparative effectiveness of screening and prevention strategies among BRCA1/2-affected mutation carriers. Breast Cancer Res Treat. 2011;125:837–47.CrossRefPubMed Grann VR, Patel PR, Jacobson JS, Warner E, Heitjan DF, Ashby-Thompson M, et al. Comparative effectiveness of screening and prevention strategies among BRCA1/2-affected mutation carriers. Breast Cancer Res Treat. 2011;125:837–47.CrossRefPubMed
36.
go back to reference Halbert CH, Stopfer JE, McDonald J, Weathers B, Collier A, Troxel AB, et al. Long-term reactions to genetic testing for BRCA1 and BRCA2 mutations: Does time heal women’s concerns? J Clin Oncol. 2011;29:4302–6.CrossRefPubMedPubMedCentral Halbert CH, Stopfer JE, McDonald J, Weathers B, Collier A, Troxel AB, et al. Long-term reactions to genetic testing for BRCA1 and BRCA2 mutations: Does time heal women’s concerns? J Clin Oncol. 2011;29:4302–6.CrossRefPubMedPubMedCentral
37.
go back to reference Sie AS, Spruijt L, van Zelst-Stams WAG, Mensenkamp AR, Ligtenberg MJL, Brunner HG, et al. High satisfaction and low distress in breast cancer patients one year after BRCA-mutation testing without prior face-to-face genetic counseling. J Genet Couns. 2016;25:504–14.CrossRefPubMed Sie AS, Spruijt L, van Zelst-Stams WAG, Mensenkamp AR, Ligtenberg MJL, Brunner HG, et al. High satisfaction and low distress in breast cancer patients one year after BRCA-mutation testing without prior face-to-face genetic counseling. J Genet Couns. 2016;25:504–14.CrossRefPubMed
38.
go back to reference Pil L, Hoorens I, Vossaert K, Kruse V, Tromme I, Speybroeck N, et al. Cost-effectiveness and budget effect analysis of a population-based skin cancer screening. JAMA Dermatol. 2017;153:147–53.CrossRefPubMed Pil L, Hoorens I, Vossaert K, Kruse V, Tromme I, Speybroeck N, et al. Cost-effectiveness and budget effect analysis of a population-based skin cancer screening. JAMA Dermatol. 2017;153:147–53.CrossRefPubMed
39.
go back to reference D’Andrea E, Marzuillo C, Pelone F, Vito CD, Villari P. Genetic testing and economic evaluations: a systematic review of the literature. Epidemiol Prev. 2015;39:45–50.PubMed D’Andrea E, Marzuillo C, Pelone F, Vito CD, Villari P. Genetic testing and economic evaluations: a systematic review of the literature. Epidemiol Prev. 2015;39:45–50.PubMed
40.
go back to reference Areia M, Spaander MC, Kuipers EJ, Dinis-Ribeiro M. Endoscopic screening for gastric cancer: a cost-utility analysis for countries with an intermediate gastric cancer risk. United Eur Gastroenterol J. 2018;6:192–202.CrossRef Areia M, Spaander MC, Kuipers EJ, Dinis-Ribeiro M. Endoscopic screening for gastric cancer: a cost-utility analysis for countries with an intermediate gastric cancer risk. United Eur Gastroenterol J. 2018;6:192–202.CrossRef
41.
go back to reference Neumann A, Lindholm L, Norberg M, Schoffer O, Klug SJ, Norström F. The cost-effectiveness of interventions targeting lifestyle change for the prevention of diabetes in a Swedish primary care and community based prevention program. Eur J Health Econ. 2017;18:905–19.CrossRefPubMed Neumann A, Lindholm L, Norberg M, Schoffer O, Klug SJ, Norström F. The cost-effectiveness of interventions targeting lifestyle change for the prevention of diabetes in a Swedish primary care and community based prevention program. Eur J Health Econ. 2017;18:905–19.CrossRefPubMed
42.
go back to reference Naber SK, Matthijsse SM, Rozemeijer K, Penning C, de Kok IMCM, van Ballegooijen M. Cervical cancer screening in partly HPV vaccinated cohorts—a cost-effectiveness analysis. PLoS ONE. 2016;11:e0145548.CrossRefPubMedPubMedCentral Naber SK, Matthijsse SM, Rozemeijer K, Penning C, de Kok IMCM, van Ballegooijen M. Cervical cancer screening in partly HPV vaccinated cohorts—a cost-effectiveness analysis. PLoS ONE. 2016;11:e0145548.CrossRefPubMedPubMedCentral
Metadata
Title
Cost–utility analysis of germline BRCA1/2 testing in women with high-grade epithelial ovarian cancer in Spain
Authors
Carlota Moya-Alarcón
Almudena González-Domínguez
Susana Simon
Inés Pérez-Román
Antonio González-Martín
Eloisa Bayo-Lozano
Ana Beatriz Sánchez-Heras
Publication date
01-08-2019
Publisher
Springer International Publishing
Published in
Clinical and Translational Oncology / Issue 8/2019
Print ISSN: 1699-048X
Electronic ISSN: 1699-3055
DOI
https://doi.org/10.1007/s12094-018-02026-2

Other articles of this Issue 8/2019

Clinical and Translational Oncology 8/2019 Go to the issue
Webinar | 19-02-2024 | 17:30 (CET)

Keynote webinar | Spotlight on antibody–drug conjugates in cancer

Antibody–drug conjugates (ADCs) are novel agents that have shown promise across multiple tumor types. Explore the current landscape of ADCs in breast and lung cancer with our experts, and gain insights into the mechanism of action, key clinical trials data, existing challenges, and future directions.

Dr. Véronique Diéras
Prof. Fabrice Barlesi
Developed by: Springer Medicine