TRIUMPH-TS

Tell us about your project:

***The project is still ongoing***

Between 1958 and 1961, Thalidomide was widely prescribed in the UK as a safe, non-addictive sedative and tranquiliser. Despite being marketed as an entirely safe treatment for the discomforts of pregnancy (including morning sickness), Thalidomide caused serious damage to the unborn child when taken during the first trimester. Foetal damage typically included upper and/or lower limb malformation (total limb absence, missing bones, etc), eye, ear and facial damage (damaged ears, narrow ear canals, small or damaged eyes, restricted eye movement and facial palsy), and malformation of internal organs (heart, urinary and alimentary tracts, and reproductive organs). Collectively, such birth defects are referred to as Thalidomide Embryopathy or Thalidomide Syndrome.

A charitable trust – the Thalidomide Children’s Trust (now the Thalidomide Trust), was established to oversee compensation payments from Distillers to those children affected by Thalidomide in the UK.

Thalidomide survivors experience significantly worse physical and mental health than the general population of the same age, demonstrating a wide range of secondary health problems, in particular chronic musculoskeletal pain, which is the most reported symptom, as well as movement restrictions, and mental disorders. Such health problems impair the ability of these individuals to remain fully independent, and negatively impact on their health-related quality of life as they limit their overall ability to adopt an active lifestyle and engage or access potentially beneficial treatments.

The project’s aim is to investigate the pain experience of Thalidomide survivors and to identify the best practice approaches to pain management, leading to a tailored service pathway that meets the unique needs of beneficiaries of the Thalidomide Trust.

The project entails two work packages with unique objectives within each:

Work Package 1:

i. [QUALITATIVE/QUANTITATIVE] How beneficiaries of the Thalidomide Trust experience pain, how it affects their lives, and how different their experience is to other unique populations with persistent pain.

ii. [QUALITATIVE] How different stakeholders involved in the healthcare of beneficiaries, as well as the beneficiaries themselves, think their pain and associated symptoms can best be managed, and what provisions are available in their location.

Work Package 2:

i. [CO-DEVELOPMENT] How to develop or modify a pain management service that best meets the needs of those who have the most to gain.

How did you involve people?

To pursue a co-development model for this project, the Thalidomide trust was approached before the submission of the grant application in order to invite its beneficiaries on two sessions (09/08/2023 and 30/08/2023) to co-develop a research project that fits their needs and their priorities. As a result, a smaller Beneficiary Advisory Group (BAG) was formed, designed to be representative of the population under investigation. The BAG co-developed the project by highlighting the importance of interviewing stakeholders additional to the Trust’s beneficiaries, such as family members or carers and clinicians. The group provided invaluable advice on what aspects the research team should investigate (e.g., use of medicines, fear of movement, mental health, pain), what outcome measures it should use, and how to best formulate the questionnaire booklet to be user friendly and sensitive to the needs of Thalidomide survivors.

To complement the group’s activities in more specialised dialogue between researchers and beneficiaries, we have been meeting remotely quarterly (four sessions in total, two so far) to monitor the progress of each work package. Rotating representatives from the group sit also on the steering committee (SC), holding sessions related to individual work packages. All work packages are designed to incorporate close working relationships with the BAG.

After Work Package 1 is completed, I will form a Participatory Expert Panel (PEP), featuring key stakeholders (researchers, beneficiaries, beneficiaries’ family members or carers, clinicians, commissioners, policy makers). In informative workshops (activities set every two weeks in a period of 3 months), the PEP will initially be introduced to the development process. Then, the PEP will consider all evidence produced in Work Package 1 and use the accumulated inference to contextually review the findings. Using the National Institute of Health and Care Excellence’s (NICE) guidelines on the management of chronic pain as frame of reference, the PEP will explore whether existing pain management paradigms are sufficient but certain barriers are hindering engagement or access (e.g., interventions such as physiotherapy or exercise cannot be delivered or accessed due to limb deficits and other co-morbidities).

Beneficiaries’ views about implementation and dissemination are critical to ensure that findings reach all stakeholders in the most appropriate manner and in keeping with beneficiaries’ priorities (e.g. press releases, interviews, etc.).

How did you find people to involve in your project?

Initially, we informed The Thalidomide Trust about the research group’s desire to submit a bid to their call for a pain management project. We then requested to meet with a group od

their beneficiaries to discuss our idea and get their feedback. The Thalidomide Trust responded positively and arranged a meeting.

How did you support people in your project?

Throughout this project, I have provided a structured system of support and training. One-to-one support is provided through tailored training events. I have worked and will continue to work with beneficiaries to identify training needs and requirements, so beneficiaries have access to courses enabling them to improve uptake of evidence into the NHS. For example, I have made plans to enable the entire Participatory Expert Panel to undertake the ‘Co-production and health services’ course of the Social Care Institute for Excellence (single day online training), which will allow the panel to train in co-production practices to help achieve the aims of Work Package 2.

Another unofficial training session was held to familiarise the beneficiaries of the BAG with the Microsoft Teams platform as this software is used for online interviews. This way I have created a system of peer support between beneficiaries to ensure my participants are able to use specific platforms and software.

I have also included costs to reimburse time and expenses of all involved beneficiaries in every aspect of the proposed project. Costs reflect current NIHR guidance.

Can you give more examples of how you worked with them?

In Work Package 1 beneficiaries had the opportunity to co-develop the proposed interview questions targeting the various stakeholders and are involved in the interpretation of their responses. They were also able to review the questionnaire booklet developed specifically for their needs. One particular point, highlighted early in our meetings, was how past researchers had approached beneficiaries for participation in their study with questionnaires developed for fully developed adults, which they found inappropriate, and led to recruitment problems. We wished to avoid that given that one of the main questionnaires we wanted to use (Central Aspects of Pain) features a textbook body manikin where people are asked to indicate the areas where they experienced pain over the past month. We requested the advice of beneficiaries on how to modify it to ensure it is not offensive and off-putting. The beneficiaries converted the body manikin into a simple list of body sites that can be simply ticked if the participant felt pain there, thus removing the body manikin, while maintaining the accuracy and integrity of the questionnaire.

For Work Package 2 plans have been made for beneficiaries to work with me and other stakeholders (as members of a Participatory Expert Panel) to refine the information gained from Work Package 1 and incorporate them into an informative suggestion on what the Thalidomide Trust needs to do to offer its beneficiaries a pain management pathway.

Finally, the BAG has significantly aided recruitment by providing details of potential participants and encouraging peers to participate in the study.

What impact did it have?

The modification process of the Central Aspects of Pain questionnaire was highlighted as an extremely positive process that weighed heavily in winning the grant.

The involvement of the BAG has led to the development of a questionnaire booklet with highly relevant questionnaires that participants are findings very interesting to complete. Similarly, the co-development of the interview questions has led to highly informative and time-efficient interviews.

The project has benefited as a whole by the BAG as it represents a unique population with unique needs that are rarely known by the wider public.

What would you change if you could do it again?

I would try to increase the number of people subscribing to the BAG, so I can rotate members more frequently and get a wider perspective. Due to time constraints this was not done to the degree I wished.

What tips would you give another researcher?

In general, I would advise researchers to involve the members of the public as early as possible with an open mind to their ideas. Codeveloping a project can make all the difference between success and failure of a project. Researchers need to create a system of bilateral support between them and the public, but also between individual members of the public.

As researchers, we should not assume that the tools we have available are appropriate for every population out there, and we should at least make the effort to ensure that what we propose to ask participants to do, follows their broader needs and interests.

What challenges did you face and how did you overcome them?

A frequent challenge when dealing with multiple individuals is to be able to find common ground on days and times to schedule activities. It is a challenge not to exclude people who may be less available than others and an effective system of rotation needs to be in place to ensure involvement is always at the desired target.