The avoiding late diagnosis of ovarian cancer (ALDO) project; a pilot national surveillance programme for women with pathogenic germline variants in and .
Abstract
Background: Our study aimed to establish 'real-world' performance and cost-effectiveness of ovarian cancer (OC) surveillance in women with pathogenic germline BRCA1/2 variants who defer risk-reducing bilateral salpingo-oophorectomy (RRSO).
Methods: Our study recruited 875 female BRCA1/2-heterozygotes at 13 UK centres and via an online media campaign, with 767 undergoing at least one 4-monthly surveillance test with the Risk of Ovarian Cancer Algorithm (ROCA) test. Surveillance performance was calculated with modelling of occult cancers detected at RRSO. The incremental cost-effectiveness ratio (ICER) was calculated using Markov population cohort simulation.
Results: Our study identified 8 OCs during 1277 women screen years: 2 occult OCs at RRSO (both stage 1a), and 6 screen-detected; 3 of 6 (50%) were ≤stage 3a and 5 of 6 (83%) were completely surgically cytoreduced. Modelled sensitivity, specificity, Positive Predictive Value (PPV) and Negative Predictive Value (NPV) for OC were 87.5% (95% CI, 47.3 to 99.7), 99.9% (99.9-100), 75% (34.9-96.8) and 99.9% (99.9-100), respectively. The predicted number of quality-adjusted life years (QALY) gained by surveillance was 0.179 with an ICER cost-saving of -£102,496/QALY.
Conclusion: OC surveillance for women deferring RRSO in a 'real-world' setting is feasible and demonstrates similar performance to research trials; it down-stages OC, leading to a high complete cytoreduction rate and is cost-saving in the UK National Health Service (NHS) setting. While RRSO remains recommended management, ROCA-based surveillance may be considered for female BRCA-heterozygotes who are deferring such surgery.
Author
Philpott, Sue
Raikou, Maria
Manchanda, Ranjit
Lockley, Michelle
Singh, Naveena
Scott, Malcolm
Evans, D Gareth
Adlard, Julian
Ahmed, Munaza
Edmondson, Richard
Woodward, Emma Roisin
Lamnisos, Athena
Balega, Janos
Brady, Angela F
Sharma, Aarti
Izatt, Louise
Kulkarni, Anjana
Tripathi, Vishakha
Solomons, Joyce S
Hayes, Kevin
Hanson, Helen
Snape, Katie
Side, Lucy
Skates, Steve
McGuire, Alistair
Rosenthal, Adam N
Raikou, Maria
Manchanda, Ranjit
Lockley, Michelle
Singh, Naveena
Scott, Malcolm
Evans, D Gareth
Adlard, Julian
Ahmed, Munaza
Edmondson, Richard
Woodward, Emma Roisin
Lamnisos, Athena
Balega, Janos
Brady, Angela F
Sharma, Aarti
Izatt, Louise
Kulkarni, Anjana
Tripathi, Vishakha
Solomons, Joyce S
Hayes, Kevin
Hanson, Helen
Snape, Katie
Side, Lucy
Skates, Steve
McGuire, Alistair
Rosenthal, Adam N
Citations
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Date
2022-11-01
Type
Article
Subject
Gynaecology
Collections
Citation
Philpott S, Raikou M, Manchanda R, Lockley M, Singh N, Scott M, Evans DG, Adlard J, Ahmed M, Edmondson R, Woodward ER, Lamnisos A, Balega J, Brady AF, Sharma A, Izatt L, Kulkarni A, Tripathi V, Solomons JS, Hayes K, Hanson H, Snape K, Side L, Skates S, McGuire A, Rosenthal AN. The avoiding late diagnosis of ovarian cancer (ALDO) project; a pilot national surveillance programme for women with pathogenic germline variants in BRCA1 and BRCA2. J Med Genet. 2023 May;60(5):440-449. doi: 10.1136/jmg-2022-108741. Epub 2022 Nov 1.
Journal / Source Title
Journal of Medical Genetics
DOI
10.1136/jmg-2022-108741
PMID
36319079
Publisher
BMJ Publishing Group