TY - JOUR T1 - Cost-effectiveness model of renal cell carcinoma (RCC) surveillance in hereditary leiomyomatosis and renal cell carcinoma (HLRCC) JF - Journal of Medical Genetics JO - J Med Genet SP - 41 LP - 47 DO - 10.1136/jmedgenet-2021-108215 VL - 60 IS - 1 AU - Alexander J Thompson AU - Yousef M Alwan AU - Vijay A C Ramani AU - D Gareth Evans AU - Eamonn R Maher AU - Emma R Woodward Y1 - 2023/01/01 UR - http://jmg.bmj.com/content/60/1/41.abstract N2 - Purpose 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 (11 years, 18 years, 40 years, 60 years), 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.Data are available on reasonable request. The data analysed and example code for the decision model used in this study are available from the corresponding author on request. ER -