The avoiding late diagnosis of ovarian cancer (ALDO) project; a pilot national surveillance programme for women with pathogenic germline variants in


Journal

Journal of medical genetics
ISSN: 1468-6244
Titre abrégé: J Med Genet
Pays: England
ID NLM: 2985087R

Informations de publication

Date de publication:
05 2023
Historique:
received: 07 06 2022
accepted: 17 08 2022
medline: 24 4 2023
pubmed: 2 11 2022
entrez: 1 11 2022
Statut: ppublish

Résumé

Our study aimed to establish 'real-world' performance and cost-effectiveness of ovarian cancer (OC) surveillance in women with pathogenic germline Our study recruited 875 female 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 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

Sections du résumé

BACKGROUND
Our study aimed to establish 'real-world' performance and cost-effectiveness of ovarian cancer (OC) surveillance in women with pathogenic germline
METHODS
Our study recruited 875 female
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
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

Identifiants

pubmed: 36319079
pii: jmg-2022-108741
doi: 10.1136/jmg-2022-108741
pmc: PMC10176325
doi:

Substances chimiques

BRCA1 Protein 0
BRCA1 protein, human 0
BRCA2 Protein 0
BRCA2 protein, human 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

440-449

Subventions

Organisme : Department of Health
Pays : United Kingdom

Informations de copyright

© Author(s) (or their employer(s)) 2023. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ.

Déclaration de conflit d'intérêts

Competing interests: The project was co-funded by Abcodia Ltd and North Central London Cancer Alliance. Abcodia Ltd had no role in the design of the project, nor in the interpretation of the findings or the drafting/editing of the manuscript. Sue Philpott has previously held a consulting role with Abcodia Ltd Adam Rosenthal has previously held a consulting role with Abcodia Ltd and Everything Genetic Ltd. Ranjit Manchanda has received funding from Yorkshire Cancer Research, GSK, Eve Appeal, Cancer Research UK, NHS Innovation Accelerator (NIA), Barts & the London Charity, Rose Trees Trust outside this work for research related to genetic testing and honorarium for advisory board membership or lectures from Astrazeneca/MSD/GSK/EGL. Naveena Singh has served on advisory boards for Astra-Zeneca-MSD and Glaxo SmithKline. Gareth Evans has a consultancy role with AstraZeneca. Helen Hanson has served on advisory boards for AstraZeneca. Steve Skates works at Massachusetts General Hospital which has co-licensed the software for early detection of ovarian cancer to Abcodia and has served on clinical advisory boards for Guardant Health and LUNGevity, has collaborated on early detection research with Freenome, participates in the Independent Data Monitoring Committee for GRAIL and has stock option for serving on the scientific Advisory Board for SISCAPA Assay Technologies. The other authors declare no competing interests.

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Auteurs

Sue Philpott (S)

North Central London Cancer Alliance, University College London Hospitals NHS Foundation Trust, London, UK.

Maria Raikou (M)

Department of Economics, University of Piraeus, Athens, Greece.
Health Economics, The London School of Economics and Political Science, London, UK.

Ranjit Manchanda (R)

Department of Gynaecological Oncology, Barts Health NHS Trust, London, UK.
London School of Hygiene and Tropical Medicine, London, UK.
Wolfsen Institue of Population Health Medicine, Barts CRUK Cancer Centre, Queen Mary University of London, London, UK.

Michelle Lockley (M)

Centre for Cancer genomics and Computational Biology, Barts Cancer Institute, Queen Mary University of London, London, London, UK.

Naveena Singh (N)

Department of Cellular Pathology, Barts Health NHS Trust, London, London, UK.

Malcolm Scott (M)

Familial Cancer Clinic, Department of Gynaecology, University College London Hospitals NHS Foundation Trust, London, London, UK.

D Gareth Evans (DG)

Division of Evolution and Genomic Medicine, School of Biological Sciences, Faculty of Biology, Medicine and Health, The University of Manchester, Manchester, Manchester, UK.
Manchester Centre for Genomic Medicine, St Mary's Hospital, Manchester University NHS Foundation Trust, Manchester, Greater Manchester, UK.

Julian Adlard (J)

Yorkshire Regional Genetics Service, Chapel Allerton Hospital, Leeds, West Yorkshire, UK.

Munaza Ahmed (M)

North East Thames Regional Genetics Service, Great Ormond Street Hospital for Children, London, London, UK.

Richard Edmondson (R)

Division of Cancer Sciences, Faculty of Biology, Medicine and Health, St Mary's Hospital, University of Manchester, Manchester, UK, Manchester University NHS Foundation Trust, Manchester, Greater Manchester, UK.

Emma Roisin Woodward (ER)

Division of Evolution and Genomic Medicine, School of Biological Sciences, Faculty of Biology, Medicine and Health, The University of Manchester, Manchester, Manchester, UK.
Manchester Centre for Genomic Medicine, St Mary's Hospital, Manchester University NHS Foundation Trust, Manchester, Greater Manchester, UK.

Athena Lamnisos (A)

The Eve Appeal, London, UK.

Janos Balega (J)

Birmingham City Hospital, Birmingham, Birmingham, UK.

Angela F Brady (AF)

North West Thames Regional Genetics Service, London North West University Healthcare NHS Trust, Harrow, London, UK.

Aarti Sharma (A)

University Hospital of Wales Healthcare NHS Trust, Heath Park, Cardiff, UK.

Louise Izatt (L)

Clinical Genetics, Guy's and St Thomas' NHS Foundation Trust, London, London, UK.
Medical and Molecular Genetics, King's College London Faculty of Life Sciences and Medicine, London, UK.

Anjana Kulkarni (A)

Clinical Genetics, Guy's and St Thomas' NHS Foundation Trust, London, London, UK.

Vishakha Tripathi (V)

Department of Clinical Genetics, Guy's and St Thomas' Hospitals NHS Trust, London, London, UK.

Joyce S Solomons (JS)

Oxford Centre for Genomic Medicine (OXGeM), Oxford University Hospitals NHS Trust, Oxford, UK.

Kevin Hayes (K)

St George's University Hospitals NHS Foundation Trust, London, London, UK.

Helen Hanson (H)

23 Clinical Genetics, St George's University Hospitals NHS Foundation Trust, London, UK.

Katie Snape (K)

Clinical Genetics, St George's University Hospitals NHS Foundation Trust, London, UK.
IMBE, University of London St George's, London, UK.

Lucy Side (L)

25, Wesses Clinical Genetics Service, University Hospitals Southampton, Southampton, Southampton, UK.

Steve Skates (S)

Massachusetts General Hospital, Boston, Massachusetts, USA.

Alistair McGuire (A)

Health Economics, The London School of Economics and Political Science, London, London, UK.

Adam N Rosenthal (AN)

Familial Cancer Clinic, Department of Gynaecology, University College London Hospitals NHS Foundation Trust, London, London, UK adam.rosenthal@ucl.ac.uk.
Gynaecological Oncology, University College London EGA Institute for Women's Health Department of Women's Cancer, London, London, UK.

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