Making rare illness visible: engaging people through novel methods of participatory research – Guest Blog

BMJ Open encourages greater involvement of patients in the research process from a study’s conception to the dissemination of its results. In this guest post, Kathy McKay, Veronica Ranieri, and Eilis Kennedy from the Tavistock & Portman NHS Foundation Trust discuss the valuable role that novel participatory methods can play in qualitative health research.

Researchers are often criticised for being stuck in an ivory tower with no sense of what it is like outside of academia. And sometimes this can be the case. We can become stuck in questions of theory, gold-standard methodology, and different statistical tests. Sometimes researchers have forgotten that real people are living what they are studying – that what we do can have lasting consequences on the lives of others.

In order to never forget the real lives involved in research, it is vital to always place them at the centre. This is the spirit of participatory research – of research that is person-centred and person-led. Researchers and people with lived experience working in partnership towards a common goal, rather than the researcher automatically taking the lead. Whilst the researcher can provide the experience to conduct a rigorous study, it is those with lived experience who impart the wisdom to do the study meaningfully. When research is undertaken in partnership, then the findings are always grounded in the everyday life of people – they are intrinsically practical and appropriate.

Various labels have been attached to participatory research but the spirit remains the same – how the study is conducted is guided by people with lived experience. This can happen in many ways, from including those with lived experience in the very development of the study to co-creating data collection tools and analytical frameworks. It can entail discussions around study findings or different challenges that may have arisen. It can also include involvement towards the end of the project as outputs are finalised and dissemination pathways are being decided. Or it can involve all of these aspects.

Let us offer an example.

Primary Sclerosing Cholangitis (PSC) is a rare and chronic liver disease resulting from continuous and worsening inflammation and scarring of an individual’s hepatic bile ducts. Its cause is still unknown and symptoms, like fatigue and itching, can be debilitating. At its most extreme, PSC can lead to end-stage liver disease and cancer. With no known cure, the only way to prevent these extreme outcomes is surgery, often a liver transplant. However, PSC’s progress is unpredictable; people may have few and manageable symptoms for a long time and then suddenly worsen to the point of transplant, or they may be severely ill from the very beginning. In addition, PSC is co-morbid with other illnesses, like Crohn’s Disease, which makes diagnosis more difficult. The majority of people are diagnosed in their late 30s and early 40s, although younger people can also be at risk, and the uncertainty that comes with such a rare and difficult diagnosis can lead to significant anxiety and hopelessness.

The majority of research into PSC is medical and for good reason – it is vital to better understand its causes and progression and to find a cure. However, for people living with PSC, their quality of life is just as important. While people wait for medical advances, they need to be able to live as well as their symptoms allow.

This is where our work with PSC Support comes in. We are currently working on the ‘PSC Wellbeing Study’*, exploring the experiences of people living with PSC, and caregivers, in terms of their mental health and emotional wellbeing. From the very beginning, we have discussed and co-designed every aspect of the study with members of PSC Support. The members of our Steering Committee, all of whom have lived experience of PSC, have been tremendously supportive in guiding us on issues where simply reading existing literature could never help us with – like how scary it can be to suddenly be handed a diagnosis with such severe outcomes and such an uncertain future, and then often be left to your own devices as the doctor may not know more than outlined in the pamphlet they just handed you.

Hearing these stories made understanding how people with PSC can live well feel all the more important, especially when medical professionals are unsure of how to help. However, the rarity of PSC meant we needed to re-think standard data collection measures because geographical distance was likely a factor. And it was important that a study looking at wellbeing didn’t do anything to make people feel more uncomfortable or inconvenienced, and took potential participants’ symptomatology into account.

So we collectively decided on a method that people could do in their own homes, in their own time, in their pyjamas, with a cup of tea in hand. Asynchronous Virtual Focus Groups (AVFGs) fit all those criteria, and have been used successfully before in other studies around the world looking at sensitive issues. Essentially, AVFGs are group discussions in a virtual space. After checking our questions with our Steering Committee, we decided on four questions in total, one question a week. We would post a question on Monday morning and participants had a week to post their response to it and talk to others in their group; we moderated twice a day and would occasionally ask questions to clarify a point or offer support. We also had helplines listed if people needed extra support. This online method was secure and anonymised – people picked their pseudonyms from the very beginning.

As people shared their stories, they found issues of commonality and strength in each other. Moreover, they started to take on the role of the researcher, by probing each other about their experiences. Their engagement, as authors of their own narratives, reinforced the appropriateness and possible therapeutic element of the method. We would have never been able to have these people together in one room at the same time so easily in real life. As we prepare for the next stage of the ‘PSC Wellbeing Study’, we are again being guided by our Steering Committee and know that our work is all the richer for it.

*The PSC Wellbeing Study protocol was concurrently published in BMJ Open. For a copy of this, please visit

(Visited 812 times, 1 visits today)