Strategies for meaningful patient and public involvement in neuro-oncological research

It is well known how living with a central nervous system tumor can profoundly affect the quality of life, primarily due to the high symptom burden, emotional distress, and poor prognosis. Rogers et al. have demonstrated that a significantly elevated symptom burden, when compared to the absence of symptoms, may not necessarily correlate with a tumor grade or mutational profile.¹ Still, the relationship between symptom burden, tumor classification, and the patient’s perception of quality of life is not fully understood. This raises questions that could potentially be addressed by involving individuals with lived experience from being diagnosed with a central nervous system tumor, not only in providing their perspective into a specific research project but also in participating in the design and planning of meaningful research. Initially, this Patient and Public Involvement (PPI) in research may seem complex. Still, it is both feasible and has been successfully conducted even in challenging populations, such as adolescents and young adults with cancer,² as well as individuals with lived experience of dementia.³. PPI has gained worldwide recognition as a standard practice. In the United States, the Food and Drug Administration has prioritized patient engagement primarily concerning drug development. In recent years, Australia has witnessed a rise in consumer participation in clinical trials. However, there’s a notable anticipation for broader implementation across various disciplines. Therefore, a promising direction for neuro-oncological research may involve PPI in research processes.

In the domain of research, PPI embodies a paradigm where research is conducted “with” or “by” individuals from the public, rather than being done “to,” “about,” or “for” the public. The term “public” encompasses a broad range, including patients, prospective patients, informal caregivers, and individuals who engage with health and social care services. It also extends to individuals representing service users within organizations and the general public.

In recent years, there has been a significant shift in the field of research, recognizing the importance of PPI during the entire research process.^4,5 This paradigm acknowledges that patients bring unique perspectives, experiences, and insights that can greatly enhance the quality and relevance of studies. Thereby, PPI contributes to the credibility of acquired knowledge and data, proactively addressing potential challenges that patients and/or researchers may encounter during a study. Additionally, PPI enhances the dissemination of research findings to relevant stakeholders and promotes implementation in clinical practice. However, despite the increasing practice and recognition of PPI, there is a lack of practical guidance available for researchers to run PPI in clinical trials, particularly in the context of neuro-oncology where there is limited evidence of neuro-specific approaches. There is not a systematic representation of the PPI in cancer research, and there is variation in how at which stages and to what degree the public is involved in the research processes. PPI has increasingly become integral into neuro-oncology, for example, patients with high-grade gliomas, their caregivers, and clinicians have established a shared research agenda,⁶ laying the ground for designing meaningful and patient-relevant studies. Hence, this collaborative approach, which may involve patients’ representatives, organizations, and their caregivers, holds promise for person-centric research, even in a patient population suffering from cognitive deficits and impaired mental capacity.

With this editorial, we seek to inspire researchers who are eager to contribute to establishing evidence on neuro-oncological PPI. Fortunately, we are witnessing a growing trend in neuro-oncological studies, with the establishment of patient panels collaborating closely with research teams. The forthcoming body of evidence, along with guidance and recommendations, will rely on the dissemination of findings from these initiatives.

For the neuro-oncological society, we highlight the significance of building strong relationships with patients and relevant patient advocacy groups. This involves promoting open and transparent communication and ensuring diverse and inclusive patient representation to enhance the relevance of our research. These principles align with shared decision-making initiatives for patients with brain tumors found in the literature.⁷ Moreover, the European Code of Cancer Practice states the core requirements for good patient-centered clinical cancer practice.⁸

While awaiting PPI guidelines in neuro-oncology, the steps and strategies for meaningful PPI in neuro-oncological research processes could be (1) develop a clear PPI plan with specific activities and action points, including collaborative research question formulation with patients^6,9; (2) ensure inclusivity by addressing disparities when inviting the target population in the PPI process; (3) define the patient’s role, level of involvement, the objectives of PPI, its deliverables, and associated costs to incorporate PPI into funding applications; (4) Identify key themes and research priorities arising from the outlined research improvements; (5) provide actionable recommendations while aligning with ethical, regulatory, and research considerations, (6) enhance research dissemination through firsthand experiences and endorsements to maximize impact, reach a wider audience, and drive positive implementation in clinical practice or impact on policy development; and (7) finally, evaluate the impact and effectiveness of PPI in research programs and share with the public and international scientific societies.¹⁰

Moreover, offering early training in PPI to researchers can be beneficial, aiming to support and encourage their engagement with PPI in research. Making PPI a natural part of not only research design but also fundraising is a moving process,⁹ where we must learn and understand new ways of working.

With this editorial, we hope to have inspired other neuro-oncological specialists and researchers to explore, refine, and further develop PPI in neuro-oncological research.

Funding

Pre-application grant from Rising Tide Foundation for Clinical Cancer Research.

Conflict of interest statement

None disclosed.

Authorship statement

All authors contributed to the study’s conception and design. The first draft of the manuscript was written by K.P. and all authors commented on previous versions of the manuscript. All authors read and approved the final manuscript.