Participatory priority setting of research questions for improving hospital care of adults with childhood-onset intellectual disabilities (PRISMA): a study protocol for a mixed-methods study

Abstract

Introduction

Persons with childhood-onset intellectual or complex disabilities have an elevated probability of encountering delayed or erroneous diagnoses during hospital treatment. It is imperative to consider the possibility that mistreatment may worsen their health. Persons with intellectual disabilities face numerous challenges in accessing and using healthcare. These challenges include structural and communication barriers, as well as a dearth of competencies among health professionals in interacting with persons with intellectual disabilities. Nevertheless, there is a paucity of studies that analyse the challenges faced by persons with intellectual disabilities in hospital and even fewer that involve persons with intellectual disabilities in the research process. Therefore, the objective of the study described in this protocol is to identify the salient research questions for improving hospital care of adults with childhood-onset intellectual disabilities by collecting and prioritising the different perspectives of patients, caregivers and clinicians.

Methods and analysis

The study design is based on the Priority Setting Partnership procedure of the James Lind Alliance, encompassing four steps to identify and prioritise issues with patients, caregivers and clinicians. Initially, problems and issues pertinent to those affected are collated using an open online questionnaire and subsequently clustered into topics. In the second step, the topics are transformed into potential research questions and reviewed by available scientific literature. Subsequently, research questions that cannot yet be answered by current literature are prioritised by participants in a second online questionnaire. Finally, the 25 questions rated most relevant are to be discussed in a one-day workshop with participants reflective of all target groups. The salient 10 research questions are to be determined using nominal group technique.

Ethics and dissemination

The study has received a positive ethics vote from the Ethics Committee at the Ludwig Maximilian University of Munich in accordance with the Declaration of Helsinki on 21 March 2025 (reference 25-0106). This study’s findings will be shared in academic conferences and published in scientific peer-reviewed journals.

Trial registration number

DRKS00037347.

STRENGTHS AND LIMITATIONS OF THIS STUDY.

• This study is limited to participants residing or working in Bavaria; thus, the findings may not be generalised to whole Germany or other European countries.

• The study offers valuable insights into the hospital experiences of persons with intellectual disabilities by exploring both quantitative and qualitative data.

• Patients, clinicians and caregivers are not only questioned but also involved in the research process to improve the accessibility to the study and the study materials.

• The 10 final research questions are subjective and depend on the workshop participants, so that 10 different questions could be chosen if the same workshop were held again.

• By surveying clinicians, patients and caregivers, the study forms the basis for future participatory clinical research concerning hospital care for adults with intellectual disabilities.

Introduction

Persons with intellectual disabilities often encounter challenges when receiving treatment in hospital. These challenges may arise due to various barriers, substandard or inadequate treatment, which in turn have a detrimental effect on the health of persons with intellectual disabilities.1,3 Such inadequate treatment has been shown to reinforce a poorer health status and a higher mortality rate among people with disabilities than without disabilities.4,6 Challenges people with disabilities face in the healthcare system include structural barriers, for example, such as the inaccessibility of certain equipment,² and communication barriers, including challenges in interpersonal interaction or insufficient sharing of information.^{7 8} During hospital stays, persons with intellectual disabilities frequently encounter adverse experiences, with their care often falling short of the standards received by individuals without disabilities.⁹ For instance, persons with intellectual disabilities face an elevated probability of delayed or erroneous diagnoses,¹⁰ consequently precipitating a deterioration in their health due to potential inaccurately administered treatment.³

Clinicians, defined in this study as any professional providing hospital care, also report uncertainties and challenges in the interaction with persons with intellectual disabilities.¹¹ For example, nurses feel underprepared when caring for patients with intellectual disabilities and experience challenges in communicating.¹¹ Communication techniques acquired during their training are often inadequate or insufficient in addressing the needs of persons with intellectual disabilities.¹² Insufficient availability of training on healthcare for persons with intellectual disabilities contributes to uncertainties and difficulties.^{13 14} This leads to barriers that have been shown to result in misunderstandings regarding diagnostic and therapeutic aspects.¹

This situation is causing health inequities for persons with intellectual disabilities. Furthermore, it is incompatible with the United Nations Convention on the Rights of Persons with Disabilities (UN-CRPD). According to the UN-CRPD, persons with disabilities are entitled ‘to the highest attainable standard of health without discrimination on the basis of their disability’.¹⁵ Therefore, the quality and standard of healthcare for persons with disabilities should be comparable to that of persons without disabilities. In addition, healthcare services should be tailored to the specific needs of persons with disabilities, such as regionally available, barrier-free and specialised.¹⁵

In Germany, where this study is conducted and the UN-CRPD was ratified in 2009, several strategies have already been developed to reduce barriers in healthcare. For instance, the adoption of an action plan for an inclusive and accessible healthcare system in 2024 by the Ministry of Health.¹⁶ Nevertheless, the UN-CRPD country report demonstrates that individuals with disabilities in Germany continue to experience unequal treatment.¹⁷ Moreover, the United Nations Committee has repeatedly stated that persons with intellectual disabilities are more likely to receive substandard healthcare in Germany compared with persons without disabilities.¹⁸ As published by the anti-discrimination agency, a review of requests for guidance from 2017 to 2023 discovered that 59% of the requests related to health and social services referred to instances of discrimination related to disability.¹⁹ It is evident that there is an urgent need for further research in this field, given the knowledge of the health inequities experienced by persons with disabilities.

When looking at challenges faced by persons with intellectual disabilities receiving treatment in the hospital, there is a paucity of international studies,^{4 5 8 9 11} and to the best of our knowledge, there are hardly any national studies. Furthermore, a paucity of research has been observed regarding the inclusion of persons with intellectual disabilities and healthcare professionals in the research process.²⁰ The exclusion of persons with disabilities from research processes, however, can be mitigated to a considerable extent by adapting the research process and study materials.²¹

Against this background, this study with the acronym PRISMA has the objective to identify the salient research questions for improving hospital care of adults with childhood-onset intellectual disabilities by collecting and prioritising the different perspectives of patients, caregivers and clinicians.

Methods

The reporting is guided by the Reporting guideline for priority setting of health research (REPRISE).²²

Study design

Priority Setting Partnership (PSP)

The study design is grounded in the methodology for Priority Setting Partnerships (PSP) proposed by the James Lind Alliance (JLA).²³ The JLA PSP is a well-established methodology for identifying and prioritising unanswered research questions related to a specific topic. As a foundational principle, it incorporates the perspectives of patients, carers and clinicians at every stage of the priority setting process. Consequently, future clinical research will concentrate on the most pressing research questions for individuals with intellectual disabilities. In PRISMA, the perspectives of the following groups are given full consideration throughout the entire research process: those affected by hospital care, namely, persons with intellectual disabilities and their formal and informal carers, as well as those responsible for delivering care, namely, hospital staff. Since in Germany federal states are responsible for providing healthcare, this study is conducted in Bavaria, a federal state in the south of Germany. Federal states enforce regulations and adapt national policies set by the federal government to local conditions, including hospital planning and training of health workforce.

Patient and public involvement

Regularly, JLA PSPs are overseen by an expert steering group comprising patients, caregivers and clinicians who possess the necessary expertise in the field.²³ In the present study, patients refer to persons with intellectual disabilities. Caregivers can be categorised into two distinct groups: informal caregivers, which include family members and relatives, and formal caregivers, which include care providers in specialised living arrangements for persons with intellectual disabilities. Clinicians are healthcare professionals who deliver hospital care for persons with intellectual disabilities.

PRISMA is coordinated by the research team that offers expertise in conducting services and coordinating participatory research. A steering group, including caregivers and clinicians, has been constituted at the outset of the research process. To ensure that the perspective of persons with intellectual disabilities themselves is represented within the study, a distinct advisory panel consisting of persons with intellectual disabilities has been established.

The steering group

In PRISMA, the steering group provides advice on a quarterly basis on the research process and study materials. Furthermore, it functions as gatekeeper to relevant actors, organisations and institutions. The group consists of representatives of families, caregivers and clinicians, as well as additional stakeholders who possess expertise in the development and delivery of disability-inclusive care for persons with intellectual disabilities.

When identifying the relevant stakeholders for the steering group, the following characteristics were taken into consideration: Representatives from various locations throughout Bavaria, including those from primary care, hospitals and specialised hospital settings. Experts from these healthcare settings have been identified who have developed and implemented specialised healthcare programmes in the past. Furthermore, interest groups representing individuals living with disabilities and their families were also invited to participate. Since institutionalised long-term care is still common for adults with childhood-onset disabilities in Germany, representatives from such institutions were also invited.

The steering group currently consists of seven members, four who work in healthcare delivery representing medicine, nursing and management (SA, JS, AW, MW) and three experts who work in different institutions representing the interests of people with intellectual disabilities and their families (TA, BB, BD). The group is free to invite new members to enrich the process when considered appropriate during the process.

The advisory panel

Persons with intellectual disabilities contribute unique insights to the research process. To explicitly include their perspectives, an advisory panel consisting of persons with intellectual disabilities has been established through a collaboration with a regional association representing the interests of persons with cognitive impairments, predominantly children, youth and adults with Down syndrome and their families (VE). The research team engages with three distinct groups that meet on a regular basis. Whenever relevant topics arise, that need to be discussed, the research team joins one of their regular meeting to receive their feedback on research materials and evolving results. The advisory panel provides, for instance, advice on the accessibility and comprehensibility of study materials. The research team integrates the feedback into the research and informs the steering group about important decisions.

Study population

Study participants are also patients, caregivers and clinicians, more concretely, (1) adults with self-identified childhood-onset intellectual disabilities, (2) their families, (3) their formal caregivers and (4) health professionals delivering hospital care for persons with intellectual disabilities.

As persons with intellectual disabilities often experience other impairments as well, those with both intellectual and complex impairments are considered to be within the target group for this study. The general inclusion criteria for participation in the study are being of legal age, being able to give consent and living or being employed in Bavaria. There is no specific upper/lower limit for the number of participants, but the aim is to achieve a broad range of issues and that the answers are from a good balance of the participating groups. It is aimed at a balance between persons with disabilities and their families and professionals in hospitals and their living environments.

Recruitment

This study will use a sampling strategy involving gatekeepers and the snowball principle. All members of the research team and the steering group are asked to distribute invitations to participate in the study within their networks. The effectiveness of the snowball method is predicated on the assumption that effective networking within relevant institutional contexts has already been established.^{24 25} Participants provide informed consent before participating in the surveys online by clicking an embedded consent statement. To attend the workshop, which will take place in person, participants provide informed consent through signing a consent form. No reimbursement will be paid for participation in the study. Travel expenses will be reimbursed for the in-person workshop.

Design and data collection

The methodology proposed by the JLA PSP is a mixed-methods study design containing four steps: the first step is a survey comprising open-ended questions. The second step is evidence-mapping, and the third step is a survey containing closed questions. The fourth step is a priority setting workshop to identify and prioritise issues and research questions for persons with intellectual disabilities. Throughout the entire study process, participants are asked to indicate whether they are persons with disabilities, family members, formal caregivers or clinicians. These groups are also taken into consideration when analysing the data, enabling any differences between groups in the questions and problems identified to be examined. All study materials are tailored to the respective target group. Scarce personal data are collected to ensure anonymity while still allowing basic description of the study population.

First step: gathering uncertainties

First, relevant topics, also termed ‘uncertainties’ by the JLA,²³ are identified using an online survey with open-ended questions tailored to patients, (informal and formal) caregivers and clinicians. The questionnaire was developed by the research team with reference to other JLA PSPs and discussed with the steering group (onlinesupplemental files 1 2). Some members of the steering group shared the preliminary survey with individuals with different areas of expertise (eg, speech therapists) and forwarded the feedback to the research team. The collected expertise was then incorporated into the survey by the research team.

The survey asks participants to describe any challenges they encounter in hospital care for persons with intellectual disabilities, and which questions they would like future research to address. They are also asked to report on their demographic characteristics. The survey is tailored to the respective target group in terms of language and demographic data. For persons with intellectual disabilities, information materials and the survey are set up in Easy Read.

The online questionnaire has been piloted by the steering group and will be sent to all gatekeepers in the target group. The software REDCap (Vanderbilt University 2025) is used to administer the survey.

Second step: data processing and verifying uncertainties

Themes are identified, based on the data, aligned with the PSP guidelines.²³ The answers to the open-ended survey questions will first be coded and thematically clustered using the software MAXQDA. Based on the generated themes, an initial draft of the questions will be created by the local research team. Early in this stage, the process is presented to the steering group and illustrated with concrete examples of the data revealed. This discussion will enhance clarity and transparency about the further course of the process. To gain feedback from the advisory panel on the uncertainties, a concrete example resulting from the second step will also be presented to the advisory panel to verify common understanding of the emerging uncertainties. Adjustments will be made throughout the process and transparently documented. The steering group will receive the final draft for their review. Based on this set of final questions, available evidence will be identified using a systematic literature search in relevant databases to determine whether the respective research questions have been already answered fully or partially by previous research. Questions that can be answered by research that has been published within the past 5 years will be documented and made publicly available.¹⁶ Research questions that cannot be answered by literature will then be incorporated into the next stage of the process and are considered the initial ‘long list’ of priorities.

Third step: interim priority setting

Third, research questions for which there is little or no evidence available in the literature are presented to all four target groups respectively in an online survey for prioritisation. This prioritisation process will consolidate the ‘long list’ of questions into a shorter list which will be discussed at the final priority setting workshop. The JLA recommends a maximum of around 60 questions for the shorter list.²³ As this number of questions is still likely to be excessive for persons with intellectual disabilities, a shorter list of up to 25 questions is envisioned. This will be agreed with the steering group once the second step of the process is completed. Step 3 will be carried out using an online survey. Once a first draft of the survey is available, the questions will be discussed with the steering group and reframed, added or removed so they align with the research topic. The draft will then be revised and discussed with the advisory panel. The dissemination of the final survey will be done in the same way as in step 1, including the provision of information materials and survey in an Easy Read version and piloting of the questionnaire by the steering group and the advisory panel. Participants will be asked to rank the 10 questions out of all questions they consider to be the most critical priorities for research. Questions will be scored according to their relative rankings and a shortlist of the top 25 questions will be created.¹⁶

Fourth step: final priority setting

Fourth, the 25 salient research questions identified in the third step are then prioritised in a prioritisation workshop. This workshop will be held to reach a consensus on the top 10 research priorities for the hospital care of persons with intellectual disabilities in Bavaria. The workshop will take place in person to minimise communication barriers that could arise in a virtual meeting. Up to 30 participants, with an equal representation from the different target groups, will be invited to participate in the workshop. The adapted nominal group technique, supported by JLA,²³ will be used since it encourages the participation of all members, including those less articulate. Individuals will be recruited from partner organisations, the Steering Committee’s and advisory panel’s networks, as well as from contact information voluntarily provided by participants at the end of the surveys conducted in steps 1 and 3. The results of the discussions are presented in a plenary session and then discussed in small groups with different constellations of participants. At least one person from each of the four target groups will be represented in each group. The results of the small groups are subsequently presented and discussed in a plenary session to find agreement on the 10 salient research questions. If no agreement can be reached in the final discussion, a vote will be held to determine the final prioritisation.

In the final steering group meeting, a plan for disseminating the results for policy makers will be discussed. The created top 10 list of priorities will be framed in a way that they remain accessible to a broad range of stakeholders; hence, tailored to different audiences. Targeted audience includes persons with disabilities and their interest groups who will be reached by briefing summaries, including versions in Easy Read language, providing the evidence to advocate for research relevant to them; researchers via academic publications; and funders and policy makers, through briefing summaries calling for fundable and actionable research. The translation will be undertaken collaboratively with the steering group and advisory panel.

As the project is planned to last 24 months, with each step up to 6 months of duration, this study is expected to be completed in the first quarter of 2027.

Enabling accessibility of study materials

To ensure that the perspectives of persons with intellectual disabilities are included throughout the entire research process, two principles were established. First, all information materials and the surveys for this target group are written in standard German and Easy Read. Second, the option to complete the survey either together with or on behalf of persons with intellectual disabilities will be offered to ensure that the views of those who are unable to read or complete the survey themselves will also be recorded. The steering group monitors these principles.

Discussion

This study directly addresses two major issues in healthcare services research related to childhood-onset disability. First, the present study addresses the need for a more profound understanding of challenges in hospital care for persons with intellectual disabilities, with the ultimate objective to prevent health inequities among this group. Second, the study process is characterised by its participatory nature, with persons with intellectual disabilities, their families, caregivers and clinicians playing an active role at every stage of the study.

Following discussions with the steering group about the different terms used for people with intellectual disabilities in research, the target group is addressed in direct communication with those with cognitive or complex impairment. This wording aligns with survey results, where individuals with intellectual disabilities identified this term as best reflecting their identity.²¹ However, some may not identify with this term, which could affect participation. The methodology for reaching the whole target group must be continually revised throughout the study and in future research.

The steering group has demonstrated unwavering dedication to the research process and is responsible for ensuring that the study’s accessibility is reflected consistently, and that the study process itself is accessible. It is important to acknowledge that the perspectives of individuals lacking the necessary competencies or resources to access study information can only be adequately assessed through the perspectives of their families and caregivers.

Despite the limitations of the study, it offers insights into the hospital experiences of persons with intellectual disabilities through active involvement in research, emphasising the need for more inclusive healthcare practices and research. It identifies important research questions for improving hospital care for adults with childhood-onset intellectual disabilities by collecting and prioritising the perspectives of patients, caregivers and clinicians in Bavaria, southern Germany. PRISMA provides a foundation for future clinical research about the healthcare of persons with intellectual disabilities, in accordance with identified priorities and the UN-CRPD aims. In order to guarantee the execution of the research questions, the chair of inclusive healthcare will build on the results in deciding on further research.

Study status

The steering group meetings have been convened since December 2024. The first step of the study, gathering uncertainties, started in May 2025 with the launch of the primary survey. The final priority setting workshop is scheduled to take place in autumn 2026. The writing of the reports and the dissemination of the findings will mark the conclusion of the study in spring 2027.

Ethics and dissemination

On 21 March 2025, the Ethics Committee at the Ludwig Maximilian University of Munich approved the study in accordance with the Declaration of Helsinki (reference 25-0106). Participation in the study is in every step of the process voluntary. All participants will be given written information in regular German and Easy Read about the project to assure they provide informed consent for their participation.

The General Data Protection Regulation will be fully adhered to. The data collected from the online survey and workshops will be stored on a password-protected server at the University Hospital in Augsburg (UKA). After completion of the study, the data will be archived by the UKA for a period of 10 years, after which it will be destroyed.

The study was registered at the DRKS on 15 July 2025 (reference DRKS00037347) (https://drks.de/search/en/trial/DRKS00037347). After finalising the data collection with the final priority setting workshop that is scheduled to take place in autumn 2026, the writing of the reports and the dissemination of the findings will mark the conclusion of the study in spring 2027.