Pancreatic cancer (PC) is the sixth leading cause of cancer death in Australia [¹] and the fourth in the United States, [²] as disease outcomes remain dismal, with single digit 5-year survival rates both in Australia [³] and elsewhere [²]. Despite this, research in PC is lagging behind than in other cancers, [⁴] with only 1% of the research budget of the US National Cancer Institute being spent on PC [⁵].

In the absence of agreed processes to reliably identify research areas that can deliver significant advances in PC research, the Cancer Council NSW (CCNSW) established a strategic partnership with the NSW Pancreatic Cancer Network to systematically define critical research issues and opportunities that could accelerate progress in this field in Australia.

Here, we describe the priority-setting process, the outputs generated at each stage and how they informed PC research funding decisions.

The study commenced with a wide consultation with PC researchers, clinicians, and consumer representatives to identify available options for research prioritisation. Consensus development methods were deemed to be the most likely to be able to provide stakeholder agreement on PC research priorities.

The process consisted of five distinct stages: a literature review on recent progress in PC research, semi-structured expert interviews, a Delphi process, consumer focus groups (FGDs), and a nominal group process (NGP). The literature review together with expert interviews was used to provide a ?level playing field? for all participants, irrespective of their specific area of expertise, to facilitate their contribution to the Delphi process. We decided to use FGDs to ascertain consumer priorities, in order to maintain an independent consumer voice throughout the study. Information about priorities identified by the experts through the Delphi process and by consumers through FGDs were provided to nominal group participants, to provide them with a comprehensive picture of potential research priorities to consider and prioritise (see Fig.?1). The study was approved by the relevant Institutional Ethics Committees.

This paper summarises a process of research prioritisation for a cancer which so far has eluded the attention of funding bodies, despite the significant disease burden it imposes. We used a combination of methods for defining research priorities, utilising the outputs of two consensus development methods (Delphi and nominal group processes) and focus group discussion to arrive at a list of ?actionable? research goals specific to Australian PC research. The reason for combining several consensus building methods was to rule out that findings being influenced by the method chosen, although previous studies comparing the Delphi method to nominal group found that both methods performed equally well for predictive purposes [²⁴]. In our study, research priorities generated through the Delphi process and the NGT were very similar and echoed consumer priorities identified through FGDs, validating our approach. Using a Delphi panel, a modified Delphi panel, and a survey-based panel, Brown et al. [²⁵] identified medical conditions for which the risk of hospitalisation is related to ambulatory care access and found that consensus techniques had good predictive ability in this context.

A retrospective review of pancreatic cancer cases reported to a Cancer Registry in Burgundy (France) made similar recommendations regarding future PC research: focus on a better understanding of genetic and environmental causes of PC, on mechanisms involved in its development and on the role of diet in PC risk identified; key challenges related to detecting early/asymptomatic disease and improvements in therapy to improve outcomes [²⁶].

Given the variety of opinions that may exist when a diverse group considers a highly technical and polarising topic, formal consensus approaches (the Delphi method, the nominal group process, and the consensus development conference) are increasingly used to make complex decision in medicine and health [²⁷]. This is based upon the finding that in situations where the evidence limited, unclear or results diverge widely, consensus methods can provide ?a bridge between clinical reasoning and clinical research? [²⁸].

Traditionally cancer research priorities have been determined using a ?group? model of decision-making, where options are advocated for by spokespersons and a judgement of quality and merit made by the group. While a wide range of experience and knowledge of group participants presents distinct advantages for group models of work [²⁴], the output of large committees may be subverted by domineering personalities, or because group members may be unwilling to take a position before all facts are known, to change it once it is stated publicly, or to publicly contradict senior participants [²⁹].

Our group chose consensus development methods in preference to committee or expert reference panels, due to their perceived limitations and based on the proven track record of formal consensus methods in achieving research prioritisation in the health arena [¹⁶, ¹⁷, ¹⁹, ³⁰?³⁴]. It was encouraging to note also that four of the seven research priorities identified by the nominal group arose also from the consumer FGDs recommendations.

Discussions in the planning phase of the research led to a decision not to include consumers in the Delphi process, to safeguard against the potential ?lay disempowerment? that may arise when bringing together experts and consumers. Qualitative researchers have questioned whether the notion of ?empowerment? reflects participants acquiescing researcher?s views, rather than empowerment to express their own views [³⁵]. While studying Australian research priorities in mental health, Griffiths et al. [³⁶] found significant differences in research priorities between the committees responsible for evaluating research grants and consumer and carer groups. We wanted to be able to identify specific consumer and researcher perspectives on research priorities before merging them into a final set of recommendations through the nominal group process.

A telephone-based system can present specific challenges for the conduct of FGDs, as important non-verbal cues may be overlooked in this setting. A key element in obtaining quality data and providing a good experience to participants is to ensure that moderators are experienced in eliciting the views of severely ill patients. In this project, the FGDs were conducted by a pair of specially trained telephone counsellors working with people with cancer. They worked closely with a qualitative researcher with expertise in conducting FGDs, to ensure that participants had a positive experience and that all participants had the opportunity to have their views heard. The FGDs produced a large amount of key information relatively expeditiously and in a way that allowed the participation of consumers who were unwell and would not otherwise have the opportunity to have their voice heard.

While the Delphi process involved a panel of 27 experts, their heavy workloads precluded their participation in every round, so response rates ranged between 63 and 85% per round. However, the inclusion of multiple rounds?including a final opportunity to dispute or add to the group?s consensus?enables us to feel confident that the 14 priorities identified reflect the views of the entire panel.

We appreciate that the findings of the FGDs cannot be used to make statements about what all those affected by PC think about key research priorities, but the level of concordance reached independently by patients and carers would suggest that to some extent at least these results are generalisable to the larger group. While focus groups are intended to encourage participants to talk with each other, rather than answer the moderators? questions, this presented specific challenges for a telephone mode of delivery. Using experienced support group moderators was essential in ensuring group interaction and to provide an in-depth understanding of consumer?s views.

The FGD recommendations, together with the results of the Delphi process were discussed in a nominal group. This enabled the list of research priorities to be discussed in a face-to-face setting, alleviating some of the criticisms levelled at the Delphi methodology, such as the potential of manipulating the consensus, unreliability of the process, and doubts as to what represents acceptable level of consensus [³⁷, ³⁸].

The nominal group defined and ranked seven research directions for Australian PC research. We critically summarised the findings of the consensus process and fed them back to the participants for comment. The priority list was circulated to researchers in the field, inviting applications for a new funding scheme?the Innovator Grants. The grants were open to all applicants who addressed the research priorities and the application process was simplified, in keeping with suggestions received from nominal group participants. Twenty-three applications were received and six were funded through the new scheme, closing the loop from research prioritisation to implementation.

This prioritisation exercise provided a much needed ?road map? for research prioritisation in pancreatic cancer, providing valuable information to researchers applying for PC research grants. Additionally, the consumer FGDs provided insights into what patients and their carers see as critical for PC research: better and earlier diagnostic modalities, improvements in understanding disease aetiology, high quality information provision about all aspects of the disease and its treatment and devising better ways in communication between doctors and patients. It was encouraging to note that many consumer research priorities rose to the top of the priority list and will hopefully provide new research insights in PC.

This project was funded by The Cancer Council NSW and was the product of a collaboration between CCNSW and the NSW Pancreatic Cancer Network (www.pancreaticcancer.net.au). We would like to thank Mr. John Friedsam for his contribution to the conduct of the focus group discussions and to all people affected by pancreatic cancer who participated in this study. Special thanks go to the large group of experts who shared their experience regarding pancreatic cancer research and its challenges and opportunities. This study was funded by the Cancer Council NSW.

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