Developing a Patient and Public Involvement Training Course for People with Intellectual Disabilities: The Leicestershire Experience

There is a lack of meaningful involvement of people with intellectual disability in research, outside of the context of study participation. There is a need to develop adapted means of facilitating Patient and Public Involvement when working with people with intellectual disability, that takes account for their level of neurodevelopmental functioning. In this article we describe a Patient and Public Involvement course developed for people with intellectual disability, developed by professionals working with this group, which was subsequently piloted in Leicestershire UK. Different versions of the course were developed for student participants with different levels of neurodevelopmental functioning and communication impairment. For Part 1 of the course (‘What is Research?’), students from Pathway 1 (mild intellectual disability) and Pathway 2 (moderate intellectual disability) both had significantly increased (p ≤ 0.05) mean knowledge test scores post-course relative to pre-course, with non-significant increases in mean confidence test scores for both pathway groups. For Part 2 of the course (‘Your Involvement in Research’), most participants agreed or strongly agreed with the statement ‘I found this interesting’ (Pathway 1: 8 of 9 students [89%]; Pathway 2: 8 of 10 students [80%]), but less students provided equivalent responses to the statement ‘I want to be involved in research in the future’ (Pathway 1: 2 of 9 students [22%]; Pathway 2: 8 of 10 students [80%]). This training course provides a template to provide basic training for people with intellectual disability with respect to research involvement, but requires further evaluation in larger student samples of diverse demographic and neurodevelopmental characteristics.


Introduction
Approximately 1% of the population has intellectual disability (ID) (Maulik et al., 2011), which is characterised by significant impairments in both intellectual and adaptive functioning, with onset during the developmental period (American Psychiatric Association, 2013).Historically, a view widely held among researchers was that the cognitive impairment of people with ID (PwID) impeded them from participating in meaningful research (McDonald et al., 2016).
However, in recent decades, there has been considerable interest and intent among researchers to involve PwID in research, advocating for their coparticipatory role, and to ensure outcomes are aligned with their priorities (Di Lorito et al., 2018;Tavecchio et al., 2019).The United Nations have asserted that patients have a democratic right to be involved in everything that affects their life, including scientific research (Nierse & Abma, 2011;United Nations, 2023).
Patient and public involvement (PPI) is defined as ways in which 'patients or members of the public can draw on their lived experiences to ensure that the evaluation, development, organisation, and delivery of health services are appropriately prioritised' (Tritter, 2009).Whilst PPI is based on the premise that it will improve the quality of research such as its prioritisation, commissioning, undertaking, and dissemination of its findings, most PPI activity in the United Kingdom is limited to feedback and information sharing, with a relative lack of meaningful collaboration (Oliver et al., 2008).This is more prevalent for PwID and their carers, whose experiences are often ignored (Aldridge, 2014;Gibbs et al., 2008).
For PwID, collaboration should be more than just being the subjects of research and providers of information.Embracing participatory and emancipatory approaches has been termed as 'inclusive research'.The participatory approach refers to researchers working alongside the PwID in an active collaboration (Andersson, 2018).The emancipatory research model advocates that PwID, rather than researchers, control the research agenda, hence including the principles of capacity-building, mutuality, and benefit (Barton, 2005;Chappell, 2000).Inclusive research, which is the collective term for participatory and emancipatory research should have five key characteristics (Beighton et al., 2019;Johnson & Walmsley, 2003): • The ownership of the problem should lie with PwID • The interests of the PwID should be furthered by it • The involvement of the PwID should remain throughout the process • The power to exert control over the processes and outcomes should remain with the PwID • The entire process -from the research question to the reports, should be accessible to PwID Though inclusive research is identified improve alignment of outcomes and practice, a shared knowledge base regarding it is still deficient (Embregts et al., 2018).
Reasons why inclusive research still remains challenging have been identified.The aforementioned belief among researchers that PwID lack the understanding and capacity continues to be held.Ethical concerns may additionally prevent researchers from including PwID due to their vulnerability (Beresford, 2005;Thompson et al., 2009).Protection by wellmeaning carers or family may restrict their access to research.Therefore, the existing literature surrounding inclusive research provides a limited insight into the experiences of PwID, resulting in a lack of conceptual clarity (El Enany et al., 2013;Grundy et al., 2016;Paul & Holt, 2017;Rose, 2014;Thompson et al., 2012).
Another element influencing the quality of inclusive research is the 'myth of heterogeneity;' that every person with ID is representative of the entire population (Burack et al., 2021;Frankena et al., 2015).PwID who are more accessible to researchers are more likely to be chosen for inclusive research (O'Brien et al., 2014).As a result, it has been observed that researchers fail to account for the diversity in the ID population -for example, the level of ability, and co-existence of other disabilities and/or mental disorders (de Haas et al., 2022;Hatton, 2020;Read et al., 2021).This suggests a considerable gap between the most appropriate versus the most likely participants; there is a near total absence of people with severe or profound ID in inclusive research, or their presence is only by proxy (Cluley, 2017;Ward et al., 2016).Walmsley (2001) reported that current communication methods employed with people with severe or profound ID when they are included in research are insufficient, making their involvement even more challenging.As observed by Brookes et al (2012) 'at the root of issues about language use are issues of power', when diversity in the ID population is represented in inclusive research, it is imperative that researchers can pitch information to research partners at a level that can be well-understood and provide a platform for further collaboration.
There is evidence in the literature of PwID being trained or supported in other ways to improve their skills, confidence and motivation in being involved in research (Fullana et al., 2017).This would support and empower the implementation of inclusive research for PwID.Whilst there is evidence available supporting the usefulness and acceptability of this approach, it is limited mostly to people with mild ID.However, a team based at Kingston and St George's University (London, United Kingdom) previously developed an eight-session training course to teach research skills to people with mild to moderate ID, including 'collecting, analysing and presenting research data,' which was well-received (Tuffrey-Wijne et al., 2020).Nevertheless, in general there is a dearth of literature on the use of an approach to support people with mild, moderate and severe ID.

Aim
The aim of this project was to develop an introductory research course for people with varying levels of ID and communication difficulties, and obtain participant feedback from the course.

Method
A course relating to research and related involvement was developed for PwID, with different variations of the course materials to address the needs of persons with varying levels of ID and communication difficulties.The course was split into three parts; the first part describing what research is, the second part detailing how PwID can get involved in research, and lastly encouraging PwID to explore their own small research project.With respect to the broad content of the course, these were similar that of Tuffrey-Wijne and colleagues (2020), though our course was developed with different pathways according to the level of neurodevelopmental functioning of the student, and people with severe ID were also included (their course was limited to people with mild to moderate ID).(Krathwohl, 2002), the corresponding learning outcomes were principally related to the knowledge and comprehension cognitive levels.Considering that Sense College support students with a wide range of ID and communication difficulties, the project team decided that a 'one size fits all' approach would be unsuitable.Thus, the course was split into three pathways, each designed for people with different levels of ID and communication difficulties: • Pathway 1-Individuals with a mild ID and mild communication difficulties Seven students took part Pathway 1,1 consisting of three males and four females, with an age range of 16-21 years.These participants had a mixture of disabilities, including mild ID (all Pathway 1 students), mild communication difficulties, autism, attention deficit hyperactivity disorder (ADHD), and visual impairment.
Pathway 1 students completed a quiz to test their knowledge and confidence related to research, both prior to the first session, and following the final session.

Pathway 2
Pathway 2 consisted of four weekly sessions delivered over four weeks, with each session being of approximately one hour duration.The session topics were the same as for Pathway 1, with the information and language adjusted to account for the developmental level of the students, in light of their lower level of intellectual functioning and heightened communication difficulties.
Eleven students took part in Pathway 2,2 consisting of eight males and three females, with an age range of 17-21 years.These participants had various disabilities, including moderate ID (all Pathway 2 students), moderate communication difficulties, autism, Fragile X Syndrome, Cerebral Palsy, and Downs Syndrome.
Pathway 2 students similarly completed a quiz both prior to the first session and following the final session, though the questions were adjusted according to the student's severity of ID and communication difficulties.

Pathway 3
Pathway 3 consisted of two weekly sessions delivered over two weeks, with each session being approximately 30 min in duration.It was felt by the team that much of the information delivered in Pathways 1 and 2 would be too complex for the Pathway 3 students to understand.Considering this, their sessions comprised of predominantly practical activities, including a questionnaire and a focus group, to provide an experience of what taking part in research may involve.
Eleven students took part in Pathway 3, consisting of four males and seven females, with an age range of 18-22 years.These participants had various disabilities, including severe ID, autism, ADHD, visual impairment, Down syndrome, and severe communication difficulties.Of the Pathway 3 students, 3 were entirely non-verbal, 5 had very limited verbal communication (e.g., some single words), and 3 were verbal but still with significant communication difficulties.
Considering the difference in course design, as well as intellectual functioning and communication impairment among the Pathway 3 cohort, the students did not complete pre-and post-course quizzes.

Part 2
Part 2 of the educational series was titled 'Your Involvement in Research', and ran from approximately 1st to 26th November 2021.This learning focussed on how PwID can get involved in research, with constituent sessions relating to what NHS research is and why it is important, and sessions on different aspects of patient and public involvement, namely consultations, collaboration, and co-production.With respect to Bloom's taxonomy (Krathwohl, 2002), the corresponding learning outcomes were again principally related to the knowledge and comprehension cognitive levels.
Part 2 also consisted of four sessions.Sessions were split into Pathways 1 and 2; for Part 2, Pathway 3 was omitted as it was felt that the information covered in these sessions would be too complex and not meaningful for students with severe ID and communication difficulties.The information covered in Pathways 1 and 2 was broadly similar, though the information, language, and activities were adjusted to accommodate the developmental level and communication needs of students on the respective pathways.Each session included a PowerPoint presentation covering various pertinent topics, including a recap of what research is, and the ways in which PwID can engage with PPI including consultation, collaboration and co-production.These sessions included activities to engage the students in learning, such as true/false quizzes, and various PPI activities.Feedback questionnaires for Part 2 included Likert-style questions without pictorial representations.

Pathway 1
Nine students took part in Pathway 1,3 consisting of five males and four females, with an age range of 16-21 years.These participants had various disabilities, including mild ID (all Pathway 1 students), and mild communication difficulties, autism, ADHD, and visual impairment.Five students from this cohort had attended all sessions of the Part 1 Pathway 1 course, with the other four students having attended some of these sessions.

Pathway 2
Ten students took part in Pathway 2,4 consisting of eight males of two females, aged 17-21 years.These participants had various disabilities, including moderate ID, moderate communication difficulties, autism, Fragile X Syndrome, cerebral palsy, and Down Syndrome.Seven students from this cohort had attended all sessions of the Part 1 Pathway 2 course, with the other three students only having attended some of these sessions.

Part 3
Part 3 ran as a block of 4 sessions, lasting roughly an hour each, one per week over four weeks.
The Part 3 cohort were a group of ten students who had taken part in Pathway 1.These consisted of six females and 4 males, ranging in age from 16 to 23, with a range of disabilities including mild learning disability, autism, ADHD, attachment disorder and visual impairment.Of these 10 students, 2 had previously completed both part 1 and 2, and 4 had completed at least one part.
During the first week, the students were given a recap of the information they had learnt on part 1 and 2. They were then split into three groups, and were asked to develop their own mini research project.They were asked to think about what they wanted to research and decide how they would do this.During the next week the groups then had the opportunity to look at each other's ideas and provide feedback to them.In the subsequent weeks the groups then had opportunity to have a go at running their project, and then write up their conclusions.With respect to Bloom's taxonomy (Krathwohl, 2002), the corresponding learning outcomes were again principally related to the application, analysis, synthesis, and evaluation cognitive levels (as well as the knowledge and comprehension levels with respect to recap of parts 1 and 2).Feedback questionnaires for Part 3 included Likert-style questions without pictorial representations.

Part 1
Students were asked questions which ascertained their knowledge and confidence surrounding how much they know about research before and after completing the course,

Pathway 1
For Part 1 Pathway 1 students, the percentage of correct answers concerning how much the students felt they knew about research, increased from 20% before completing the course to 41% after completing the course, representing a significant increase (p = 0.04).
With regards to students' confidence surrounding what research is and what taking part involves, there was a non-statistically significant increase from 74 to 82% (p = 0.07) when comparing before and after the delivery of the course (Table 1 and Fig. 1).

Pathway 2
For Part 1 Pathway 2 students, the percentage of correct answers increased from 61 to 82% concerning knowledge-based questions, representing a statistically significant increase (p = 0.05), with a non-statistically significant increase from 61 to 85% (p = 0.06) concerning confidence-based questions (Table 2 and Fig. 2).

Pathway 3
The professionals running the Pathway 3 course felt that the majority of the student cohort were able to Table 1 Scores on knowledge and confidence questions for Part 1 Pathway 1 students, before and after undertaking the Part 1 course.There were 6 knowledge question items (corre-sponding to a maximum of 6 marks), and confidence question items (corresponding to a maximum of 15 marks).Significance testing was performed using the paired-samples t-test a Comments typed verbatim-This student had completed a sports session prior to the research session    The answers to the Likert statements for the Pathway 1 cohort were predominantly positive, with 8 of 9 students selecting 'agree' or 'strongly agree' for the statement 'I found this interesting', and 7 of 9 students for the statement 'I found it easy to understand.'However, only 2 of 9 students selected 'agree' or 'strongly agree' for the statement 'I want to be involved in research in the future.'Full details of responses to Likert statements for the Pathway 1 cohort are detailed in Table 3. Free text responses for Pathway 1 students indicated a preference for practical activities and discussions (Table 4).Pathway 2 Many answers to the Likert statements for the Pathway 2 cohort, were similarly positive, with 8 of 10 students selecting 'agree' or 'strongly agree' for the statement 'I found this interesting', and 7 of 10 students for the statement 'I found it easy to understand.'However, in contrast to the Pathway 1 cohort, 8 of 10 students selected 'agree' or 'strongly agree' for the statement 'I want to be involved in research in the future.'Pathway 2 students did not provide any free text responses to the course, as it was felt that their developmental level was such that they could not communicate answers to open questions (Table 5).
Part 3 The three Part 3 subgroups decided on three different research questions.These were 'Who exercise more -men or women?', 'Do people eat more fruits or more vegetables?',and 'Do males and females have different hobbies?'All groups decided on creating questionnaires and asking staff and students around the college.Once these had been completed, the students filled in an evaluation form, with most students stating that they found the activity interesting, that it helped them to learn how to run research, that they now know more about running research than before, they think research is important and they want to be involved in helping to run research in the future.A full breakdown of Part 3 student responses is shown in Table 6.

Discussion
This research pilot course was developed to introduce the topic of research to PwID and to develop skills and knowledge that could be used to support inclusive research in the future.Sessions were designed in collaboration with a multidisciplinary team of professionals working at Leicestershire Partnership Trust and Sense College which enabled invaluable input from a breadth of health professionals as well as educators of PwID.The sessions were designed to be flexible in their approach, allowing for a rebalancing of the power dynamic between researcher and pilot participant (Brett et al., 2014) as knowledge was imparted and confidence levels increased, meaning that participants were able to work with increasing levels of empowerment throughout, such that towards the later stages of the course pilot participants took the opportunity to formulate their own research question and attended consultations with peers who provided constructive feedback and suggestions on how to further develop their methodology.This represents the true spirit of emancipatory work (Chappell, 2000).
The delivery method was consistent with that of Brooks and Davies (2008); sessions delivered over time, with understanding reached in 'the doing'.Such a delivery method incurs additional expense, and this needs to be considered when planning for time constraints and funding, as it is an identifiable barrier to inclusive research (Di Lorito et al., 2018;Paul & Holt, 2017;Tuffrey-Wijne & Butler, 2010).Time portioning was considered early in the course design, and a timetable was allocated prior to the  course commencing to ensure that this could be facilitated by the educating team, as well as attended by participants.The course delivery focused on imparting theoretical knowledge and then bolstering this with practical activities.The impact of this was two-fold, allowing attention and interest to be maintained throughout the sessions but also to support and develop skills, moving from the theoretical to empirical arena.
The tendency to work with co-researchers who are readily accessible has been well documented and has been observed to lead to participation bias whereby people who have a mild ID may be overrepresented in PPI in ID research in comparison to those with moderate, severe and profound ID (Di Lorito et al., 2018;Frankena et al., 2015;Tuffrey-Wijne et al., 2020).Additionally, the use of already established research groups or the selection of individuals based on them having previous research experience may introduce bias, as these participants tend to be 'over researched,' 'rehearsed,' and non-representative of the entirety of the ID cohort (Beighton et al., 2019).The pilot addresses such bias by engaging with people who have a range of ID, including severe ID (Pathway 3), to share learning about what is research is and how to get involved.The course design was robust in that inclusive methodologies were used for all pathways.Time portioning (1-h sessions for Pathway 1 and 2, 30-min sessions for Pathway 3) was utilised and adapted for each Pathway.The course content was amended for delivery to each group, with the expertise of educators at Sense College.Activities were amended such that Pathway 3 were engaged in more practical activities, in order to sustain attention and optimise learning owing to the 1-2-word level comprehension of this group.Accessible information was provided where needed.Formal feedback was collated as appropriate for each Pathway, including free-text and a Likert scale for Pathway 1, a Likert scale for Pathway 2 and a choice of preferred activity for Pathway 3.
There was an expected increase in the knowledge base of pilot participants, which was measured with a knowledge-based quiz following Part 1 of the course.Interestingly, this was increased by just over 20 percent in both Pathway 1 and 2 groups, representing statistically significant increases in both groups (p = 0.04 and p = 0.05 respectively).Pathway 3 did not complete the quiz.The baseline knowledge score measured at the beginning of the course was different for both Pathways 1 and 2, however it is not possible to make direct comparisons between groups due to the adjustment of the information and content of knowledge questions on the feedback form.The measurable increase in knowledge base does however evidence the utility of such a pilot scheme, and contradicts stigmatising beliefs that PwID lack the understanding and cognitive capability that would be required to learn the relevant concepts required in research.The retention of knowledge over time was not captured, however this would be useful to know whether 'top-up' sessions would be required in the delivery of a PPI course, or whether a discrete number of sessions would be sufficient.However, for Pathway 3 students, it was less clear for some of the students as to whether they understood the purpose of the activities, principally those who were non-verbal, had complex disabilities, and those with understanding of language at a 1-2-word level.
The available literature notes that participation in PPI based research has resulted in an increase in reported confidence levels for PwID.This is both specific to the topic of research, as well as more generally to increased self-confidence and self-esteem.This has been widely remarked upon by both patients and public involved in research, as well as their supporters and care network (Beighton et al., 2019;Puyalto et al., 2016;Tuffrey-Wijne et al., 2020).Thus far, findings have been principally anecdotal in the literature.This study measures research-based confidence levels in a quantifiable manner by asking confidencebased questions in the questionnaire at the beginning and end of Part 1, showing an increase for both Pathway 1 and 2 groups (Pathway 3 did not complete the quiz).
There were noted limitations of the project.Despite the conscious recruitment of participants with mild, moderate and severe ID, there were challenges associated with the collation of feedback from participants with a lower level of intellectual functioning.It was also felt that owing to the level of function and communication difficulties, the knowledge and confidence-based quiz was not completed.Pathway 3 students were able to state a preference for completing either the questionnaire or the focus group activity, but it was unclear as to whether they understood the purpose of the activities.A preference for a PPI activity can however provide useful information as to what the PwID finds to be valuable, or interesting.Although amendments were made to the delivery of the pilot course, it was felt that the content of Part 2 sessions was too complex, and Pathway 3 were omitted on this basis.It is important for PwID to feel that their participation in PPI in research is not tokenistic (Paul & Holt, 2017) and that their inclusion is for the added value both to the further development of research and to the ID population that is being represented.As Tuffrey-Wijne and Butler (2010) describe, tokenism can be addressed by ensuring that the needs of individuals and/or groups are accommodated for, and that the rationale for their involvement is clear and appropriately resourced.Furthermore, it would have been valuable to obtain more comprehensive feedback from participants, such as via semi-structured interviews specifically adapted for persons with ID.Finally, it is important to ensure that such training is accessible for all groups with ID, and thus such training courses need to be evaluated in other groups, such as people with ID of different ethnic groups.Ideally, more baseline data, such as ethnic group membership, would have been obtained from the study population reported on in this study.Additionally, pictorial representations of Likert-style questions on feedback questionnaires were only used for Part 1 of the course (not Parts 2 or 3); ideally such representations should have been used on all questionnaires as they can improve response rates from PwID as well as aid in distinguishing between different responses (Hartley & MacLean Jr, 2006).
A further consideration is that despite giving attention to the representation of PwID in terms of severity, all participants in the pilot scheme are students at Sense College, which could introduce bias as they are not representative of the ID community in its entirety.Students who attend an educational establishment may be more familiar with engaging in education and training, and may indeed be more likely to have an interest in academic pursuits.For future consideration would be the engagement of PwID from a wider educational background, including those who do not attend a formal college education.
Furthermore, there remains scope to shape this pilot scheme in the future in a truly inclusive fashion.This pilot scheme has been developed and delivered as a service development project rather than as a piece of emancipatory research, however it would certainly benefit with input from PwID at formulation and with improvement of pilot material, delivery of pilot sessions and the collation, interpretation, and dissemination of future data (O'Brien et al., 2014).A truly inclusive approach involves active partnership between PwID, carers, and healthcare professionals, with recognition of each other's respective skillsets (Alexander et al., 2021).There is intent for the data collated from this pilot scheme to be disseminated to PwID, and to work collaboratively in the further development of course material.Though principally designed to impart information and to encourage participation in PPI based research, an additional utility of such a pilot has been the ability to explore and garner interest within the ID cohort attending the sessions.It must be recognised that despite the optimisation of skillset, knowledge base and confidence levels, it is also essential that the person has a genuine interest in participating in research.Being a PwID or having previous experience of research alone does not qualify someone to be able to work as a part of the research team (Williams & Simons, 2005); and nor does it mean that they would like to assume this role by default.Of interest and in contrast to work by Beighton et al. (2019) and the Pathway 2 group, most Pathway 1 participants disagreed with the statement that they want to be involved in research in the future, despite the vast majority finding the sessions interesting and useful, and feeling that research was important.This was not qualified by any associated free text explanation, which is a limitation of this study.It does however highlight an important factor to explore.Is this sentiment fixed or subject to change?What parameters might change this answer?What reasons might people give for not wanting to participate in research?Opening the floor to such questions would not only further understanding as to ways in which the pilot course could be improved, but also to gain insight from co-researchers as to their own agenda, priorities and interests.
As well as the need for further exploration on these matters, it also identifies an additional use for a replicable pilot course that could be disseminated widely.A formal training programme, delivered across a large geographical area may have potential of reducing the cost associated with delivering inclusive research-based training, and will notably identify those who do not have an enduring interest, as well as those who do.An apprenticeship model such as 'on the job' training, mentoring and peer-peer research are other valuable opportunities (Tuffrey-Wijne et al., 2020), but are associated with considerable cost and commitment, thus it may be advisable to explore suitability in advance through more cost-effective measures such as this pilot scheme.
In summary, the implications of the production and delivery of such a scheme are considerable.There is capacity to educate, inspire, and ultimately recruit PwID to the research forum, enabling related research to be directed by their own needs and priorities.PwID are underrepresented in research and PPI, yet this project has enabled doubly underrepresented groups such as those with moderate and severe ID to engage in PPI related work, with substantial engagement and interest.This project furthers the aims of NHS England and the Accelerated Access Collaborative PPI strategy, in that a diverse range of patients and public are involved in PPI, including those with lived experience and those who experience the greatest health inequalities.Such people should be involved in influencing the direction and delivery of PPI, with continual consideration of equality and inclusion (National Health Service, 2022) This service development project has also helped to support the sentiment in current research that engaging in PPI develops both knowledge and confidence, which has been evidenced in a measurable manner.It further supports existing research in that a pilot scheme is feasible in terms of its development, delivery and dissemination, and provides a cost-effective method of imparting information.Formal training courses have been identified as one way of addressing training needs and should be considered as a starting point for exploring interest, developing knowledge, and improving skills.

Fig. 1
Fig. 1 Percentage of correct answers on the knowledge and confidence questions for Part 1 Pathway 1 students, before and after taking the course

Fig. 2
Fig. 2 Percentage of correct answers on the knowledge and confidence questions for Part 1 Pathway 2 students, before and after taking the course

Table 3
Response to Likert-style course evaluation question items for the Part 2 Pathway 1 student cohort

Table 4
Free-text responses to course evaluation question items for the Part 2 Pathway 1 cohort.Please note that the acronym 'NR' stands for 'no response', but that all other responses are copied verbatim from students

Table 5
Response to Likert-style course evaluation question items for the Part 2 Pathway 2 student cohort