Cost-effectiveness model of renal cell carcinoma (RCC) surveillance in hereditary leiomyomatosis and renal cell carcinoma (HLRCC)
AffiliationManchester Centre for Health Economics, Division of Population Health, Health Services Research and Primary Care, The University of Manchester, Manchester, UK. Department of Clinical Radiology, Manchester University NHS Foundation Trust, Manchester, UK. Department of Urology, The Christie NHS Foundation Trust, Manchester, UK. Division of Evolution and Genomic Medicine, The University of Manchester, Manchester, UK. Manchester Centre for Genomic Medicine, Manchester University NHS Foundation Trust, Manchester, UK. Department of Medical Genetics, University of Cambridge and Cambridge University Hospitals NHS Foundation Trust, Cambridge, UK. Division of Evolution and Genomic Medicine, The University of Manchester, Manchester, UK Emma.Woodward@mft.nhs.uk.
MetadataShow full item record
AbstractPurpose To determine the cost-effectiveness of annual renal imaging surveillance (RIS) in hereditary leiomyomatosis and renal cell cancer (HLRCC). HLRCC is associated with a 21% risk to age 70 years of RCC. Presentations with advanced renal cell cancer (RCC) are associated with poor outcomes whereas RIS detects early-stage RCC; however, evidence for the cost-effectiveness of RIS is lacking. Methods We developed a decision-analytic model to compare, at different age starting points (11years, 18years, 40years, 60years), the costs and benefits of lifetime contrast-enhanced renal MRI surveillance (CERMRIS) vs no surveillance in HLRCC. Benefits were measured in life-years gained (LYG), quality-adjusted life years (QALYs) and costs in British Pounds Sterling (GBP). Net monetary benefit (NMB) was calculated using a cost-effectiveness threshold of £20 000/QALY. One-way sensitivity and probabilistic analyses were also performed. Results In the base-case 11-year age cohort, surveillance was cost-effective (Incremental_NMB=£3522 (95% CI −£2747 to £7652); Incremental_LYG=1.25 (95% CI 0.30 to 1.86); Incremental_QALYs=0.29 (95% CI 0.07 to 0.43)] at an additional mean discounted cost of £2185/patient (95% CI £430 to £4144). Surveillance was also cost-effective in other age cohorts and dominated a no surveillance strategy in the 40 year cohort [Incremental_NMB=£12 655 (95% CIs −£709 to £21 134); Incremental_LYG=1.52 (95% CI 0.30 to 2.26); Incremental_QALYs=0.58 (95% CI 0.12 to 0.87) with a cost saving of £965/patient (95% CI −£4202 to £2652). Conclusion Annual CERMRI in HLRCC is cost-effective across age groups modelled.
CitationThompson AJ, Alwan YM, Ramani VAC, Evans DG, Maher ER, Woodward ER. Cost-effectiveness model of renal cell carcinoma (RCC) surveillance in hereditary leiomyomatosis and renal cell carcinoma (HLRCC). Journal of Medical Genetics. BMJ; 2022. p. jmedgenet-2021-108215.
JournalJournal of Medical Genetics
- Hereditary Leiomyomatosis and Renal Cell Cancer: Clinical, Molecular, and Screening Features in a Cohort of 185 Affected Individuals.
- Authors: Forde C, Lim DHK, Alwan Y, Burghel G, Butland L, Cleaver R, Dixit A, Evans DG, Hanson H, Lalloo F, Oliveira P, Vialard L, Wallis Y, Maher ER, Woodward ER
- Issue date: 2020 Dec
- Hereditary leiomyomatosis and renal cell carcinoma: a case series and literature review.
- Authors: Chayed Z, Kristensen LK, Ousager LB, Rønlund K, Bygum A
- Issue date: 2021 Jan 18
- Hereditary leiomyomatosis and renal cell cancer (HLRCC): renal cancer risk, surveillance and treatment.
- Authors: Menko FH, Maher ER, Schmidt LS, Middelton LA, Aittomäki K, Tomlinson I, Richard S, Linehan WM
- Issue date: 2014 Dec
- Renal cell carcinoma in young FH mutation carriers: case series and review of the literature.
- Authors: Hol JA, Jongmans MCJ, Littooij AS, de Krijger RR, Kuiper RP, van Harssel JJT, Mensenkamp A, Simons M, Tytgat GAM, van den Heuvel-Eibrink MM, van Grotel M
- Issue date: 2020 Jan
- Hereditary leiomyomatosis and renal cell carcinoma syndrome associated uterine smooth muscle tumors: Bridging morphology and clinical screening.
- Authors: Garg K, Rabban J
- Issue date: 2021 Mar