Research priorities in mesothelioma: A James Lind Alliance Priority Setting Partnership

Our plain-English summary of Research priorities in mesothelioma: A James Lind Alliance Priority Setting Partnership, by R.J. Stephens, C. Whiting, K. Cowan, et al (Lung Cancer, 2015).

The study

• Authors: R.J. Stephens, C. Whiting, K. Cowan, James Lind Alliance Mesothelioma Priority Setting Partnership Steering Committee
• Journal: Lung Cancer
• Published: 2015;89(2):175-80
• Cited by: 31 articles, as counted by NIH iCite
• Read it: PubMed · Publisher

Abstract

Background: In the UK, despite the import and use of all forms of asbestos being banned more than 15 years ago, the incidence of mesothelioma continues to rise. Mesothelioma is almost invariably fatal, and more research is required, not only to find more effective treatments, but also to achieve an earlier diagnosis and improve palliative care. Following a debate in the House of Lords in July 2013, a package of measures was agreed, which included a James Lind Alliance Priority Setting Partnership, funded by the National Institute for Health Research. The partnership brought together patients, carers, health professionals and support organisations to agree the top 10 research priorities relating to the diagnosis, treatment and care of patients with mesothelioma.

Methods: Following the established James Lind Alliance priority setting process, mesothelioma patients, current and bereaved carers, and health professionals were surveyed to elicit their concerns regarding diagnosis, treatment and care. Research questions were generated from the survey responses, and following checks that the questions were currently unanswered, an interim prioritisation survey was conducted to identify a shortlist of questions to take to a final consensus meeting.

Findings: Four hundred and fifty-three initial surveys were returned, which were refined into 52 unique unanswered research questions. The interim prioritisation survey was completed by 202 responders, and the top 30 questions were taken to a final meeting where mesothelioma patients, carers, and health professionals prioritised all the questions, and reached a consensus on the top 10.

Interpretation: The top 10 questions cover a wide portfolio of research (including assessing the value of immunotherapy, individualised chemotherapy, second-line treatment and immediate chemotherapy, monitoring patients with pleural thickening, defining the management of ascites in peritoneal mesothelioma, and optimising follow-up strategy). This list is an invaluable resource, which should be used to inform the prioritisation and funding of future mesothelioma research.

Copyright © 2015 The Authors. Published by Elsevier Ireland Ltd.. All rights reserved.

In plain English

This was a UK research priority-setting exercise, not a study of asbestos exposure or disease rates. A James Lind Alliance partnership asked mesothelioma patients, carers and health professionals which unanswered questions about diagnosis, treatment and care of pleural or peritoneal mesothelioma research should address. Between February and April 2014, 453 responses came in, 103 from patients and 242 from carers, partners or relatives. These became 52 unanswered questions about interventions. An interim survey drew 202 respondents; the top 30 questions went to a 30-person consensus meeting on 10 November 2014, which unanimously agreed a top 10. First was whether boosting the immune system, for example with PD-1 or PD-L1 agents, improves response and survival. Next came individualised chemotherapy, then how best to monitor people with diffuse pleural thickening and a negative biopsy considered at high risk of developing mesothelioma. The authors noted that the Steering Group patients may not have been typical. Many patients and carers at the final workshop felt disadvantaged by not understanding the complex medical issues. The authors concluded that the priorities present clear directions for future interventional research.

We wrote this plain-English summary from the full article with the help of AI, and a second AI model checked each sentence against the article. It is not the authors' wording; the abstract and the article are their own account.

What the study did and found

• Design: Research priority-setting partnership (James Lind Alliance method): an initial survey, an interim ranking survey and a final consensus meeting. It is not a study of exposure or disease outcomes.
• Who was studied: In the UK, 453 initial survey responses: 103 from patients, 242 from carers, partners or relatives of mesothelioma patients, 82 from health professionals, and 26 from support organisations or people who gave no category or more than one. The survey ran February to April 2014.
• Main results:
  • Among initial survey respondents, 79% of patients were male, 86% of carers were female, and only 5% of patients had peritoneal mesothelioma.
  • After merging similar themes, 98 themes remained. Of the draft questions, 52 involved an intervention and were kept; the remaining 46 were considered out of scope.
  • A search found 30 systematic reviews. None of the 52 questions was removed as already answered.
  • The interim survey was sent to 397 people. It drew 202 respondents: 38 patients, 98 carers, 50 health professionals and 16 support organisations.
  • Thirty people, including six patients, four carers, 16 health professionals and four representatives of support organisations, attended the final meeting on 10 November 2014. The meeting closed with a unanimous consensus on the top 10, and three further questions were highlighted.
  • Ranked first: whether boosting the immune system (using new agents such as PD-1 or PD-L1) improves response and survival rates for mesothelioma patients.
  • Ranked second: whether individualised chemotherapy can be given to mesothelioma patients based on predictive factors.
  • Ranked third: the best way to monitor patients with diffuse pleural thickening and a negative biopsy who are considered at high risk of developing mesothelioma.
  • Ranked fifth: the most effective current treatment for ascites in patients with peritoneal mesothelioma.
  • Ranked ninth: whether an annual chest X-ray and/or CT scan and medical examination in high-risk occupations leads to earlier diagnosis of mesothelioma.
• The authors' conclusion: The authors concluded that the priorities identified present clear directions for future interventional research.
• Limitations the authors state:
  • The two patients on the Steering Group were long-term survivors and may not have been typical patients.
  • The questions were hard to put concisely in lay language, which was a major challenge for most patients and carers. Many patients and carers at the final workshop felt disadvantaged by their lack of understanding of the complex medical issues.
  • Adding rank positions together may appear overly simplistic. The authors reported that alternative methods gave very similar results.
  • Ranking every question in order proved difficult, so the top 13 might be better viewed as equally important.
  • Emphasis shifted over the stages away from patient and carer concerns towards more complex interventions.

Topics

The National Library of Medicine indexes this study under subjects our site covers: Lung cancer, Mesothelioma.

Medical terms in this study, explained in our Lexicon: Mesothelioma, Palliative Care, Immunotherapy, Chemotherapy, Pleural Thickening, Ascites, Peritoneal Mesothelioma.

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