OBJECTIVE: Core Outcome Sets (COS) define the minimum outcomes that should be measured and reported in all clinical trials for a specific health condition or health area. The aim was to develop two COS for intracranial meningioma; one for clinical trials (COSMIC: Intervention) and one for observational studies (COSMIC: Observation).
METHODS: A study advisory group was formed with representation from international stakeholders of charities (IBTA, TBTC, brainstust), neurosurgery societies (SBNS, BIMS) and neuro-oncology societies (BNOS, EANO, EORTC, ICOM, RANO-PRO, SNO). Two systematic literature reviews and trial registry searches were performed to identify outcomes reported in published and ongoing 1) meningioma clinical trials, and 2) studies of incidental/untreated meningioma. Outcomes were deduplicated and categorised. Individuals were recruited from stakeholder groups (including meningioma patients and carers, healthcare professionals, researchers) to participate in 2-round, international, modified eDelphi surveys. The final core outcome sets were ratified through online consensus meetings.
RESULTS: The COSMIC: Intervention systematic review identified 660 verbatim outcomes, rationalized into 24 Delphi survey items. Following the consensus meeting, 15 outcomes were included in the final set. For COSMIC: Observation, 267 verbatim outcomes were identified and rationalized into 17 Delphi survey items. 16 outcomes were included in the final set. There were eight core outcomes common to both COSMIC: Intervention and COSMIC: Observation COS, namely tumour growth, physical, emotional, and neurocognitive functioning, overall quality of life, progression-free survival, meningioma specific mortality and overall survival. Role and social functioning were core outcomes in COSMIC Observation but not COSMIC intervention.
CONCLUSIONS: The COSMIC study has defined the core outcome sets for use in future meningioma clinical trials and studies. Role and social functioning were important to incidental meningioma patients and likely reflects the anxiety and uncertainty around clinical treatment. International stakeholder agreement should ensure widespread use and adoption.
Christopher P. Millward (Principal Investigator), Terri S. Armstrong, Heather Barrington, Sabrina Bell, Andrew R. Brodbelt, Helen Bulbeck, Anna Crofton, Linda Dirven, Theo Georgious, Paul Grundy, Abdurrahman I. Islim, Mohsen Javadpour, Sumirat M. Keshwara, Shelli Diane Koszdin, Anthony G. Marson, Michael W. McDermott, Torstein R. Meling, Kathy Oliver, Puneet Plaha, Matthias Preusser, Thomas Santarius, Nisaharan Srikandarajah, Martin Taphoorn, Carole Turner, Colin Watts, Michael Weller, Paula R. Williamson, Gelareh Zadeh, Amir H. Zamanipoor Najafabadi, Michael D. Jenkinson (Chief Investigator)
On behalf of EORTC BTG, ICOM, EANO, SNO, RANO-PRO, BNOS, SBNS, BIMS, IBTA, TBTC, Brainstrust, and Brain Tumour Foundation of Canada.
Institute of Systems, Molecular, & Integrative Biology, University of Liverpool, UK (CPM, ARB, AII, SMK, AGM, NS, MDJ).
Department of Neurosurgery, The Walton Centre NHS Foundation Trust, Liverpool, UK (CPM, ARB, AC, AII, SMK, NS, MDJ).
Neuro-Oncology Branch, Center for Cancer Research, National Cancer Institute, Bethesda, Maryland, USA (TSA).
Institute of Population Health, University of Liverpool, UK (HB, PRW).
The Brain Tumour Charity (SB, TG).
Brainstrust – the brain cancer people (HB).
Department of Neurology, Leiden University Medical Centre, The Netherlands (LD, MT).
Department of Neurology, Haaglanden Medical Center, The Hague, The Netherlands (LD, MT).
Department of Neurosurgery, University Hospital Southampton, UK (PG).
National Centre for Neurosurgery, Beaumont Hospital, Dublin, Ireland (MJ).
Veterans Affairs Healthcare System, Palo Alto, California, USA (SDK).
Department of Neurology, The Walton Centre NHS Foundation Trust, Liverpool, UK (AGM).
Division of Neuroscience, Florida International University, Florida, USA (MWM).
Department of Neurosurgery, Geneva University Hospital, Geneva, Switzerland (TRM).
International Brain Tumour Alliance (KO).
Nuffield Department of Surgical Sciences, University of Oxford, UK (PP).
Division of Oncology, Department of Medicine, Medical University of Vienna, Austria (MP).
Department of Neurosurgery, Addenbrooke’s Hospital & University of Cambridge, UK (TS, CT).
Institute of Cancer and Genomic Sciences, University of Birmingham, UK (CW).
Department of Neurology, University Hospital and University of Zurich, Switzerland (MW).
Department of Surgery, University of Toronto, Canada (GZ).
University Neurosurgical Center Holland, Leiden University Medical Centre, Haaglanden Medical Center, Haga Teaching Hospitals, Leiden and The Hague, The Netherlands (AHZN).
Disease Category: Neurology
Disease Name: Meningiomas
Age Range: 18 - 120
Sex: Either
Nature of Intervention: Any
- Charities
- Clinical experts
- Consumers (caregivers)
- Consumers (patients)
- Patient/ support group representatives
- Researchers
- Epidemiologists
- COS for clinical trials or clinical research
- Consensus meeting
- Delphi process
- Systematic review
Two systematic literature reviews and two trial registry searches will identify outcomes reported in published and ongoing 1) intracranial meningioma clinical effectiveness trials, and 2) clinical studies of incidental/untreated intracranial meningioma. Outcomes include those that are clinician-reported, patient-reported, caregiver-reported, and neurocognitive, as well as measures of progression and survival. Outcomes will be deduplicated and each unique outcome categorised according to the taxonomy presented by the Core Outcome Measures in Effectiveness Trials (COMET) initiative. Two separate long-lists will be generated. Each will be supplemented with additional patient-reported and neurocognitive outcomes identified from two existing systematic literature reviews, along with patient-centred outcomes identified from published, semi-structured interviews with meningioma patients (where applicable). The two long-lists will be prioritised through two separate 2-round, international, modified eDelphi surveys including meningioma patients, healthcare professionals, researchers, and other key stakeholders in a caring or supporting role, to identify those that are considered most relevant. The final COSMIC: Intervention and COSMIC: Observation COS will be ratified through two, one-day consensus meetings, with representation from all key stakeholder groups.