The outcomes measured and reported in observational studies of incidental and untreated intracranial meningioma: A systematic review.

Background: The clinical management of patients with incidental intracranial meningioma varies markedly and is often based on clinician choice and observational data. Heterogeneous outcome measurement has likely hampered knowledge progress by preventing comparative analysis of similar cohorts of patients. This systematic review aimed to summarize the outcomes measured and reported in observational studies. Methods: A systematic literature search was performed to identify published full texts describing active monitoring of adult cohorts with incidental and untreated intracranial meningioma (PubMed, EMBASE, MEDLINE, and CINAHL via EBSCO, completed January 24, 2022). Reported outcomes were extracted verbatim, along with an associated definition and method of measurement if provided. Verbatim outcomes were de-duplicated and the resulting unique outcomes were grouped under standardized outcome terms. These were classified using the taxonomy proposed by the "Core Outcome Measures in Effectiveness Trials" (COMET) initiative. Results: Thirty-three published articles and 1 ongoing study were included describing 32 unique studies: study designs were retrospective n = 27 and prospective n = 5. In total, 268 verbatim outcomes were reported, of which 77 were defined. Following de-duplication, 178 unique verbatim outcomes remained and were grouped into 53 standardized outcome terms. These were classified using the COMET taxonomy into 9 outcome domains and 3 core areas. Conclusions: Outcome measurement across observational studies of incidental and untreated intracranial meningioma is heterogeneous. The standardized outcome terms identified will be prioritized through an eDelphi survey and consensus meeting of key stakeholders (including patients), in order to develop a Core Outcome Set for use in future observational studies.

The outcomes measured and reported in intracranial meningioma clinical trials: A systematic review.

Background: Meningioma clinical trials have assessed interventions including surgery, radiotherapy, and pharmacotherapy. However, agreement does not exist on what, how, and when outcomes of interest should be measured. To do so would allow comparative analysis of similar trials. This systematic review aimed to summarize the outcomes measured and reported in meningioma clinical trials. Methods: Systematic literature and trial registry searches were performed to identify published and ongoing intracranial meningioma clinical trials (PubMed, Embase, Medline, CINAHL via EBSCO, and Web of Science, completed January 22, 2022). Reported outcomes were extracted verbatim, along with an associated definition and method of measurement if provided. Verbatim outcomes were deduplicated and the resulting unique outcomes were grouped under standardized outcome terms. These were classified using the taxonomy proposed by the "Core Outcome Measures in Effectiveness Trials" (COMET) initiative. Results: Thirty published articles and 18 ongoing studies were included, describing 47 unique clinical trials: Phase 2 n = 33, phase 3 n = 14. Common interventions included: Surgery n = 13, radiotherapy n = 8, and pharmacotherapy n = 20. In total, 659 verbatim outcomes were reported, of which 84 were defined. Following de-duplication, 415 unique verbatim outcomes remained and were grouped into 115 standardized outcome terms. These were classified using the COMET taxonomy into 29 outcome domains and 5 core areas. Conclusions: Outcome measurement across meningioma clinical trials is heterogeneous. The standardized outcome terms identified will be prioritized through an eDelphi survey and consensus meeting of key stakeholders (including patients), in order to develop a core outcome set for use in future meningioma clinical trials.

Opening up ideas: an advent calendar for patient and public engagement in clinical trials research.

The involvement of patients and the public in research is now an expectation in research with funders requesting a clear plan of involvement and engagement. In the United Kingdom involvement typically focuses on research prioritisation, design and delivery, in contrast activities that share the results of research or research methods more generally are considered to be engagement. Clinical trials tend to concentrate on involvement activities with less emphasis on engagement. To promote engagement activities in the context of clinical trials we asked people attending the 2022 International Clinical Trials Methodology Conference to share ideas on how we can best engage with patients and the public. Responses were reviewed and 22 themes identified. One suggestion was to create an advent calendar and so these 22 themes plus two from the authors were used as a daily tweet from the 1st to the 24th December 2022. Here we share these ideas and draw comparisons between engagement activities in research and traditions of the Christmas period. The ideas shared are not intended as a definitive list but instead a novel way to start discussions between experts by experience, researchers, health professionals and communities to facilitate co-production of meaningful engagement strategies.

Patient and public involvement and engagement are terms used to describe specific activities that have a variety of goals from information giving through topublic co-production of research. Involvement and engagement are important as they can help reduce waste in research by ensuring that the research is relevant, conducted well and that the results are shared to those that will use them to make decisions about treatments, including patients. In the United Kingdom the term "engagement" usually refers to activities that focus mainly on information giving, for example sharing the results of research or information about how research is done in general. In this commentary we share ideas for engagement activities that were collected from people attending the International Clinical Trials Methodology Conference in 2022. One of the ideas shared was to have an advent calendar and we have used this to draw comparisons between traditions surrounding the Christmas period and engagement of patients and the public. We share 24 different ideas in the form of a printable advent calendar and invite the clinical trials community, including experts by experience, to reflect on these to generate more ideas for meaningful engagement activities so that everyone who will use the results of research has the opportunity to shape, share, and benefit from research.

Review finds core outcome set uptake in new studies and systematic reviews needs improvement.

OBJECTIVES: To review evidence about the uptake of core outcome sets (COS). A COS is an agreed standardized set of outcomes that should be measured and reported, as a minimum, in all clinical trials in a specific area of health or healthcare. STUDY DESIGN AND SETTING: This article provides an analysis of what is known about the uptake of COS in research. Similarities between COS and outcomes recommended by stakeholders in the evidence ecosystem is reviewed and actions taken by them to facilitate COS uptake described. RESULTS: COS uptake is low in most research areas. Common facilitators relate to trialist awareness and understanding. Common barriers were not including in the development process all specialties that might use the COS and the lack of recommendations for how to measure the outcomes. Increasingly, COS developers are considering strategies for promoting uptake earlier in the process, including actions beyond traditional dissemination approaches. An overlap between COS and outcomes in regulatory documents and health technology assessments is good. An increasing number and variety of organizations are recommending COS be considered. CONCLUSION: We suggest actions for various stakeholders for improving COS uptake. Research is needed to assess the impact of these actions to identify effective evidence-based strategies.

Development of 'Core Outcome Sets' for Meningioma in Clinical Studies (The COSMIC Project): protocol for two systematic literature reviews, eDelphi surveys and online consensus meetings.

INTRODUCTION: Meningioma is the most common primary intracranial tumour in adults. The majority are non-malignant, but a proportion behave more aggressively. Incidental/minimally symptomatic meningioma are often managed by serial imaging. Symptomatic meningioma, those that threaten neurovascular structures, or demonstrate radiological growth, are usually resected as first-line management strategy. For patients in poor clinical condition, or with inoperable, residual or recurrent disease, radiotherapy is often used as primary or adjuvant treatment. Effective pharmacotherapy treatments do not currently exist. There is heterogeneity in the outcomes measured and reported in meningioma clinical studies. Two 'Core Outcome Sets' (COS) will be developed: (COSMIC: Intervention) for use in meningioma clinical effectiveness trials and (COSMIC: Observation) for use in clinical studies of incidental/untreated meningioma. METHODS AND ANALYSIS: Two systematic literature reviews and trial registry searches will identify outcomes measured and reported in published and ongoing (1) meningioma clinical effectiveness trials, and (2) clinical studies of incidental/untreated meningioma. Outcomes include those that are clinician reported, patient reported, caregiver reported and based on objective tests (eg, neurocognitive tests), as well as measures of progression and survival. Outcomes will be deduplicated and categorised to generate two long lists. The two long lists will be prioritised through two, two-round, international, modified eDelphi surveys including patients with meningioma, healthcare professionals, researchers and those in caring/supporting roles. The two final COS will be ratified through two 1-day online consensus meetings, with representation from all stakeholder groups. ETHICS AND DISSEMINATION: Institutional review board (University of Liverpool) approval was obtained for the conduct of this study. Participant eConsent will be obtained prior to participation in the eDelphi surveys and consensus meetings. The two systematic literature reviews and two final COS will be published and freely available. TRIAL REGISTRATION NUMBER: COMET study ID 1508.

Opportunities and challenges for the development of "core outcome sets" in neuro-oncology.

Core Outcome Sets (COS) define minimum outcomes to be measured and reported in clinical effectiveness trials for a particular health condition/health area. Despite recognition as critical to clinical research design for other health areas, none have been developed for neuro-oncology. COS development projects should carefully consider: scope (how the COS should be used), stakeholders involved in development (including patients as both research partners and participants), and consensus methodologies used (typically a Delphi survey and consensus meeting), as well as dissemination plans. Developing COS for neuro-oncology is potentially challenging due to extensive tumor subclassification (including molecular stratification), different symptoms related to anatomical tumor location, and variation in treatment options. Development of a COS specific to tumor subtype, in a specific location, for a particular intervention may be too narrow and would be unlikely to be used. Equally, a COS that is applicable across a wider area of neuro-oncology may be too broad and therefore lack specificity. This review describes why and how a COS may be developed, and discusses challenges for their development, specific to neuro-oncology. The COS under development are briefly described, including: adult glioma, incidental/untreated meningioma, meningioma requiring intervention, and adverse events from surgical intervention for pediatric brain tumors.

Patient participation in Delphi surveys to develop core outcome sets: systematic review.

OBJECTIVES: To describe the design and conduct of core outcome set (COS) studies that have included patients as participants, exploring how study characteristics might impact their response rates. DESIGN: Systematic review of COS studies published between 2015 and 2019 that included more than one patient, carer or representative as participants (hereafter referred to as patients for brevity) in scoring outcomes in a Delphi. RESULTS: There were variations in the design and conduct of COS studies that included patients in the Delphi process, including differing: scoring and feedback systems, approaches to recruiting patients, length of time between rounds, use of reminders, incentives, patient and public involvement, and piloting. Minimal reporting of participant characteristics and a lack of translation of Delphi surveys into local languages were found. Additionally, there were indications that studies that recruited patients through treatment centres had higher round two response rates than studies recruiting through patient organisations. CONCLUSIONS: Variability was striking in how COS Delphi surveys were designed and conducted to include patient participants and other stakeholders. Future research is needed to explore what motivates patients to take part in COS studies and what factors influence COS developer recruitment strategies. Improved reporting would increase knowledge of how methods affect patient participation in COS Delphi studies.