Title: Estimating health-related quality of life for people with learning disabilities; an adaptation of the EQ-5D
Funder: Department of Health and Social Care

Abstract: Background: People with LD have poorer health, experience delays in diagnosis and treatment, are more likely to have poorer health outcomes and significantly shorter life expectancy than people without LD. NHS England's response to the Government's mandate of addressing poor outcomes and inequalities has placed transforming healthcare services and reducing inequalities at the centre of their agenda. Integral to an improvement in services is the need for research to accurately reflect the Health-Related Quality of Life (HRQoL) benefits, and thus the cost effectiveness, i.e. value for money, of potential improvements to inform decision making. In LD populations there are challenges collecting data specifically HRQoL for use in economic evaluations. NICE guidance states that the preferred measure of HRQoL for cost-effectiveness analysis in adults is the EQ-5D. However, research shows that people with LD have difficulty completing the measure due to the wording/language and understanding terms used. While proxy completion of EQ-5D has been proposed as a valid method, the ethics and reliability of proxy completions has been questioned. The UK Research Governance Framework and Equality Act 2010, require that, where necessary, reasonable adjustments must be made to provide accessible research materials to enable equity of access. This requirement also applies to the design of reliable and valid measures to record patient reported outcomes. Aim: The aim of this research is to adapt the EQ-5D, a preference based measure of health related quality of life, for people with mild or moderate LD for use in economic evaluation. Plan of investigation: The research utilises a mixed methods approach, across four phases. The first phase will consist of a systematic review of literature on the development or use of quality of life measures in different populations with communication difficulties, cognitive impairment or LD, and the methods used to adapt these measures for different patient groups; this will inform the development of the proposed adaptation. The second phase involves people with LD, carers/supporters of people with LD (e.g. family members or third-sector workers) reviewing both the EQ-5D-3L and EQ-5D-5L in individual Think Aloud (TA) sessions and focus groups, with emphasis being placed on the wording/language, domains and structure included in EQ-5D and the mode of its delivery. Results of the systematic review and qualitative research will inform the development of the adapted EQ-5D. The third phase will compare general population values of similar health states using original EQ-5D and adapted EQ-5D in order to assess the extent to which valuations using the adapted EQ-5D correspond to the previously established measure. The final phase will focus on testing the adapted version of EQ-5D in terms of ease of completion, internal consistency and reliability. Potential benefits to patients and the NHS: Service improvements proposed by the current government should be based on robust research evidence. Integral to that is adequately capturing patient-reported health outcomes. The development of a bespoke and robust instrument that allows adults with mild to moderate LD to reflect their opinions on their own health supports these proposals. It is proposed that a more accessible instrument would therefore be a more reliable measure of their own health and could be used in decisions relating to the services and support they receive.

Lay abstract: Background: People with learning disabilities (LD) tend to have worse health than those without. This may be due to delays in diagnosis and treatment, and a higher likelihood of having multiple health problems and poorer outcomes after treatment. In order to address this health inequality we need to be sure that we accurately reflect any benefits that accrue from health care. In the UK, decisions on health care provision are typically subject to being shown to represent value for money (i.e. cost-effective). This is assessed using cost effectiveness analysis, which involves an assessment of the costs and benefits; the latter assessed using a measure of health-related quality of life (HRQoL). The National Institute for Health and Care Excellence (NICE) recommend using the EQ-5D (EuroQol - Five Dimensions), as the measure of HRQoL for cost-effectiveness analyses. The EQ-5D is a simple, one-for-all' measure of HRQoL, where the user chooses the statements which best describes their health today. It has been designed for self-completion by participants and can be used in face-to-face interviews, postal surveys and via telephone using a standard set of wording. It was designed to be of little difficulty, taking only a few minutes for someone to complete. For people with LD however, there can be challenges completing the EQ-5D. A recent study, OK-Diabetes, assessed the EQ-5D with adults with mild to moderate LD and found that over half of the group had difficulty completing the measure. The main difficulty experienced by the study participants was with the wording/language of the statements, and being able to understand what the statements meant. Rephrasing and explanation of terms made completion easier, however this invalidates the questionnaire.Aim: My aim is to adapt the EQ-5D for completion by adults with mild to moderate LD, so that the resulting data accurately reflect the HRQoL of this group.Methods: To inform the development of the adaptation, the research includes a review of existing studies involving people with communication difficulties, cognitive impairment or LD that have developed or used measures of HRQoL as part of the study. The next phase involves individual interviews and two focus groups with people with LD and the carers/supporters of people with LD (e.g. family or staff members who work with people with LD). This phase will be used to explore and understand the key difficulties experienced by people with LD when completing and understanding the EQ-5D. I will ask participants for their suggestions about the structure and the wording/language of EQ-5D. I will also ask them about aspects of the HRQoL of a person with LD that are, or are not, covered by the EQ-5D. Using the findings from the review, interviews and focus groups I will develop an adapted version of EQ-5D for adults with mild to moderate LD.By using an online survey, I will test if using the adapted EQ-5D would provide similar results as the original EQ-5D in terms of assessing HRQoL. People selected from the general population will be used to assess if the wording/language used in the different versions gives the same values in terms of HRQoL. This will confirm if the wording/language used in the adapted EQ-5D and the original EQ-5D have the same meaning. The adapted EQ-5D will be tested against the original version to assess whether it is easier to understand and complete by asking people with LD to complete the questionnaire on two different occasions. I will also ask a carer/supporter, if present, to complete the original EQ-5D from the point of view of the person they care for/support. The answers that I receive from the participant with LD and the carer/supporter will be tested using proven statistical methods to assess if the adapted EQ-5D can be used reliably.Patient and public involvement (PPI): The study will include a group of people with LD, which will meet regularly. The group will contribute their advice, ideas and opinions on materials e.g. participant information sheets, and methods e.g. break period activities, to be used. I will regularly feedback all elements of this proposed research to a group of experts (Scientific Advisory Board) who give advice on the study.Dissemination: The results of this study will be published to ensure that they are seen by researchers with an interest in LD or in how to measure HRQoL. I will present findings from this research at conferences. I will seek opportunities to present findings to people with LD and their representatives; informed through discussions with the PPI group.

Research activity codes: 8.2 HEALTH AND WELFARE ECONOMICS
Health categories: MENTAL HEALTH