Inclusion of adults with conditions that have the potential to affect capacity and or communication in research: triangulation from a mixed-methods study of current practice and values across multiple stakeholders.

OBJECTIVES: We aimed to: (A) describe researcher decision-making when including or excluding adults with conditions that have the potential to affect capacity and/or communication in research and (B) explore the underlying values and reasoning of stakeholders in research which falls under the provisions of the Mental Capacity Act, 2005. DESIGN: The mixed-methods design included semistructured interviews with adults with conditions that have the potential to affect capacity and/or communication, supporters, researchers, research ethics committee members and an online survey with researchers. Triangulation was used to integrate the data and examine the complementarity of the findings. SETTING: England and Wales. PARTICIPANTS: There were 61 participants who took part in semistructured interviews, of which 39 were adults with conditions with potential to affect capacity and/or communication, 6 were in support roles for adults with conditions with potential to affect capacity and/or communication (including family members and professionals in advocacy organisations), 8 were members of research ethics committees flagged under the Mental Capacity Act to review research where there could be issues of mental capacity and 8 were researchers with experience of working with adults with conditions that have the potential to affect capacity and/or communication. The online survey had 128 participants, researchers with experience of working with adults with conditions that have the potential to affect capacity and/or communication. RESULTS: All stakeholders were supportive of the genuine inclusion of adults with conditions that have the potential to affect capacity and/or communication in research, and exclusion was seen as a form of discrimination. Many researchers were daunted by meeting the threshold within the legislation for including participants who may lack capacity. CONCLUSION: Further training, expertise and resources are required to promote the successful inclusion in research of adults with conditions that have the potential to affect capacity and/or communication.

How are adults with capacity-affecting conditions and associated communication difficulties included in ethically sound research? A documentary-based survey of ethical review and recruitment processes under the research provisions of the Mental Capacity Act (2005) for England and Wales.

OBJECTIVES: This study aimed to determine the characteristics of ethical review and recruitment processes, concerning the inclusion of adults with capacity-affecting conditions and associated communication difficulties in ethically sound research, under the provisions of the Mental Capacity Act (MCA, 2005) for England and Wales. DESIGN: A documentary-based survey was conducted focusing on adults with capacity-affecting conditions and associated communication difficulties. The survey investigated: (1) retrospective studies during the implementation period of the MCA (2007-2017); (2) prospective applications to MCA-approved Research Ethics Committees (RECs) during a 12-month period (2018-19); (3) presentational and linguistic content of participant information sheets used with this population. SETTING: Studies conducted and approved in England and Wales. SAMPLE: Studies focused on adults with the following capacity-affecting conditions: acquired brain injury; aphasia after stroke; autism; dementia; intellectual disabilities; mental health conditions. The sample comprised: (1) 1605 studies; (2) 83 studies; (3) 25 participant information sheets. PRIMARY AND SECONDARY OUTCOME MEASURES: The primary outcome was the inclusion/exclusion of adults with capacity-affecting conditions from studies. The secondary outcome was the provisions deployed to support their inclusion. RESULTS: The retrospective survey showed an incremental rise in research applications post-MCA implementation from 2 (2012) to 402 (2017). The prospective survey revealed exclusions of people on the bases of: 'lack of capacity' (n=21; 25%); 'communication difficulties' (n=5; 6%); 'lack of consultee' (n=11; 13%); and 'limited English' (n=17; 20%). REC recommendations focused mainly on participant-facing documentation. The participant information sheets were characterised by inconsistent use of images, typography and layout, volume of words and sentences; some simplified language content, but variable readability scores. CONCLUSIONS: People with capacity-affecting conditions and associated communication difficulties continue to be excluded from research, with recruitment efforts largely concentrated around participant-facing documentation. There is a need for a more nuanced approach if such individuals are to be included in ethically sound research.

A systematic review and narrative synthesis of the research provisions under the Mental Capacity Act (2005) in England and Wales: Recruitment of adults with capacity and communication difficulties.

BACKGROUND: The Mental Capacity Act (MCA, 2005) and its accompanying Code of Practice (2007), govern research participation for adults with capacity and communication difficulties in England and Wales. We conducted a systematic review and narrative synthesis to investigate the application of these provisions from 2007 to 2019. METHODS AND FINDINGS: We included studies with mental capacity in their criteria, involving participants aged 16 years and above, with capacity-affecting conditions and conducted in England and Wales after the implementation of the MCA. Clinical trials of medicines were excluded. We searched seven databases: Academic Search Complete, ASSIA, MEDLINE, CINAHL, PsycArticles, PsycINFO and Science Direct. We used narrative synthesis to report our results. Our review follows Preferred Reporting Items for Systematic Reviews and is registered on PROSPERO, CRD42020195652. 28 studies of various research designs met our eligibility criteria: 14 (50.0%) were quantitative, 12 (42.9%) qualitative and 2 (7.1%) mixed methods. Included participants were adults with intellectual disabilities (n = 12), dementia (n = 9), mental health disorders (n = 2), autism (n = 3) and aphasia after stroke (n = 2). We found no studies involving adults with acquired brain injury. Diverse strategies were used in the recruitment of adults with capacity and communication difficulties with seven studies excluding individuals deemed to lack capacity. CONCLUSIONS: We found relatively few studies including adults with capacity and communication difficulties with existing regulations interpreted variably. Limited use of consultees and exclusions on the basis of capacity and communication difficulties indicate that this group continue to be under-represented in research. If health and social interventions are to be effective for this population, they need to be included in primary research. The use of strategic adaptations and accommodations during the recruitment process, may serve to support their inclusion.

Inclusion under the Mental Capacity Act (2005): A review of research policy guidance and governance structures in England and Wales.

OBJECTIVE: To investigate how people with communication and understanding difficulties, associated with conditions such as dementia, autism and intellectual disability, are represented in research guidance supplementary to the Mental Capacity Act (MCA: 2005) in England and Wales. METHODS: A documentary survey was conducted. The sample comprised the MCA Code of Practice (CoP: 2007) and 14 multi-authored advisory documents that were publicly available on the Health Research Authority website. Textual review of key words was conducted followed by summative content analysis. RESULTS: Representation of people with communication and understanding difficulties was confined to procedural information and position statements that focused mainly on risk management and protection. Whilst a need to engage potential participants was recognized, guidance provided was imprecise. CONCLUSIONS: Tensions exist between the protection versus empowerment of people with communication and understanding difficulties in research. The development of structured, evidence-based guidance is indicated. PATIENT OR PUBLIC CONTRIBUTION: People with communication and understanding difficulties and carers participated in a working group to explore, discuss and interpret the findings.

Empowering self-help groups for caregivers of children with disabilities in Kilifi, Kenya: Impacts and their underlying mechanisms.

Bringing up a child with disabilities in a low-income setting is challenged by inadequate resources, limited psycho-social support and poverty. Not surprisingly, many caregivers experience fatigue, distress and isolation. To address and investigate these issues, action was taken to set up twenty self-help groups focusing on caregiver empowerment. A realist evaluation design was adopted to evaluate impacts associated with the self-help process and to identify mechanisms determining the outcomes. Monthly monitoring visits were conducted to the groups during a ten-month set-up period, at the end of which eleven active groups remained, nine having dissolved due to disputes, corruption and extreme environmental conditions. A facilitated intervention was delivered to the active groups (N = 154) over a six-month period. The members were guided to review and discuss topics such as economic empowerment, personal situation, peer support, community inclusion, access to health and education. Evaluation employed mixed methods using questionnaires (n = 75) and semi-structured interviews (n = 36) pre- and post-intervention. At baseline, the burden of caregiving was characterised by aloneness, challenges, stigma and discrimination. Post-intervention, caregiver agency was defined by togetherness, capacity-building, acceptance and well-being. Significant impacts associated with caregiver perceptions included increased social support, reduced severity of child's disability and decreased effects of extrinsic factors affecting the caregiver's role. Mechanisms of 'handling goods and money' and 'social ties and support' appeared to underpin the outcomes. Caregiver empowerment was associated with newly developed skills, social connectedness and resource mobilisation. Documentation of group processes contributes to the evidence on community-based inclusive development.

An open randomized controlled trial of the effects of linguistic simplification and mediation on the comprehension of "easy read" text by people with intellectual disabilities.

BACKGROUND: This trial aimed to measure the effects of extrinsic and intrinsic factors on users' comprehension of health information provided in adapted written "easy read" material. METHOD: Sixty adults with intellectual disabilities undertook The Easy Read Task, randomly allocated with stratification by reading ability to one of four conditions (with and without simplified language/with and without mediation). RESULTS: Neither linguistic complexity of the text nor mediation independently or combined made a significant difference to the understanding of information. Post hoc testing revealed that the group who received simplified language with mediation performed significantly better than the group that had complex text with mediation. None of the other differences between the remaining groups were significant. CONCLUSIONS: Constructing meaning needs to extend beyond a consideration of form as found in "easy read" documents to recognize the role of individual capacity for language processing.

Development of self-help groups for caregivers of children with disabilities in Kilifi, Kenya: Process evaluation.

Background: Caring for a child with disabilities in a resource-poor setting brings many challenges to the caregiver. We examined the development of self-help groups for caregivers in a rural part of Kenya. Objectives: To conduct a process evaluation on the development of self-help groups during a 10-month set-up period, focusing on implementation and mechanisms associated with their functional status. Methods: Using a realist evaluation design, we set up 20 self-help groups for 254 caregivers. An evaluation was conducted to investigate implementation and mechanisms of impact. Implementation focused on caregiver registration, community group support and monitoring visit compliance. Data were collected from group registers, records of meetings and field notes. Mechanisms of impact employed a framework of strengths-weaknesses-opportunities-threats to review the groups at the end of the 10-month set-up period. Results: Recruitment resulted in registration of 254 participants to 18 groups - two groups disbanded early. Post-evaluation included 11 active and 7 inactive groups. Compliance with the monitoring visits was consistent across the active groups. All groups engaged in 'merry-go-round' activities. The active groups were characterised by strong leadership and at least one successful income generation project; the inactive had inconsistent leadership and had dishonest behaviour both within the group and/or externally in the community. Mediators associated with functional status included the following: available literacy and numeracy skills, regular meetings with consistent attendance by the members, viable income generating projects, geographical proximity of membership and strong leadership for managing threats. Conclusion: Self-help groups have the potential to progress in resource-poor settings. However, critical to group progression are literacy and numeracy skills amongst the members, their geographical proximity, regular meetings of the group, viable income generating projects and strong leadership.

Development of self-help groups for caregivers of children with disabilities in Kilifi, Kenya: process evaluation

Background: Caring for a child with disabilities in a resource-poor setting brings many challenges to the caregiver. We examined the development of self-help groups for caregivers in a rural part of Kenya. Objectives: To conduct a process evaluation on the development of self-help groups during a 10-month set-up period, focusing on implementation and mechanisms associated with their functional status. Methods: Using a realist evaluation design, we set up 20 self-help groups for 254 caregivers. An evaluation was conducted to investigate implementation and mechanisms of impact. Implementation focused on caregiver registration, community group support and monitoring visit compliance. Data were collected from group registers, records of meetings and field notes. Mechanisms of impact employed a framework of strengths­weaknesses­opportunities­threats to review the groups at the end of the 10-month set-up period. Results: Recruitment resulted in registration of 254 participants to 18 groups ­ two groups disbanded early. Post-evaluation included 11 active and 7 inactive groups. Compliance with the monitoring visits was consistent across the active groups. All groups engaged in 'merry-go-round' activities. The active groups were characterised by strong leadership and at least one successful income generation project; the inactive had inconsistent leadership and had dishonest behaviour both within the group and/or externally in the community. Mediators associated with functional status included the following: available literacy and numeracy skills, regular meetings with consistent attendance by the members, viable income generating projects, geographical proximity of membership and strong leadership for managing threats. Conclusion: Self-help groups have the potential to progress in resource-poor settings. However, critical to group progression are literacy and numeracy skills amongst the members, their geographical proximity, regular meetings of the group, viable income generating projects and strong leadership

Persons with disabilities as experts-by experience: using personal narratives to affect community attitudes in Kilifi, Kenya.

BACKGROUND: The last decade has seen improved public awareness of disability in sub-Saharan Africa. However, negative and stereotypical views of disability still persist in many communities. We conducted a study to promote awareness of disability in rural Kenya, using a process of reflection and education. This paper reports on the second aspect - education. The research question was: How can personal narratives of living with disability affect community attitudes and responses to disability? METHODS: A qualitative phenomenological approach was adopted. Twenty community-based groups involving 249 participants took part. Each group participated in one focus group discussion at baseline, to explore the members' personal experiences and views of disability. The intervention involved three adults with disabilities sharing their personal narratives with each group. After the intervention, repeat focus group discussions were conducted with each group. Thematic analysis was carried out according to the framework method. RESULTS: The emergent framework consisted of four main themes, organised as opposing constructs: 'burden' and 'agency', 'sub-human' and 'human'. 'Burden' focused on the perceived hopelessness of the situation. Post-intervention revealed greater support for the 'agency' of persons with disabilities, evidenced by what the person could do, rather than their inability, and the relevance of support. The 'sub-human' to 'human' construct captured dehumanising and discriminating practice towards persons with disabilities on one side, and recognition of the person and inclusion in the community on the other. Whilst support and empathy were evident at the pre-intervention stage, post-intervention revealed greater recognition of people with disabilities as fellow human beings. CONCLUSION: This study provides a proof of concept regarding the deployment of persons with disabilities as agents for change. Exposure to experts-by-experience provided community groups with opportunities to reflect on, examine and adjust their views on disability in this rural part of Kenya. The sharing of personal narratives appeared to resonate with group members, to encourage recognition of the person and not just the disability, and to move their resolve toward ideas for collective action. Further research is needed to assess the effects of such interventions.

The perception of disability by community groups: Stories of local understanding, beliefs and challenges in a rural part of Kenya.

Cultural narratives on disability have received much attention over the past few decades. In contexts of poverty, limited information and everyday challenges associated with having, or caring for someone with a disability, different understandings have emerged. A project was set up to promote disability awareness in neighborhood communities in a rural part of Kenya, using a process of reflection and education. This paper reports on the first aspect-reflection. The aim was to investigate local understanding of disability as a co-constructed concept. The research questions were: 1. What cultural beliefs shape local understanding of disability? 2. What challenges are perceived to be associated with disability? A phenomenological approach was adopted. Focus group discussions were conducted with twenty-one community groups involving 263 participants and audio-recorded. The data were transcribed and thematic analysis was carried out. Visual maps were created to illustrate any interconnections, before establishing the final conclusions. Local beliefs attributed disability to: human transgression of social conventions, particularly concerning inappropriate family relations, which invoked a curse; supernatural forces affecting the child; the will of God; unexplained events; and biomedical factors. Challenges associated with disability related to the burden of caregiving and perceived barriers to inclusion, with stress as a shared bi-product. Local understanding of disability in this rural part of Kenya demonstrated overlapping explanations and plurality of beliefs. Two possible interpretations are offered. Firstly, oscillation between explanatory lines demonstrated instability, affecting broader acceptance of disability. Secondly, and more positively, in the face of challenges, the desire to make sense of the existing situation, reflected a healthy pluralism.

Developing the personal narratives of children with complex communication needs associated with intellectual disabilities: What is the potential of Storysharing® ?

BACKGROUND: Sharing personal experience in narrative is challenging for individuals with intellectual disabilities. The aim was to investigate the potential of Storysharing® (Storysharing is an innovative communication method based on personal narrative, which has been developed to support conversations with people who have severe difficulties in communication) intervention. MATERIALS AND METHODS: The study involved eleven pupil-educational supporter dyads at a special school. Storysharing® was implemented over a 15-week period. Personal narratives were captured on video pre- and post-intervention. The data were analysed for discourse and narrative. RESULTS: Significant differences revealed a decline in 'query-answer' sequences and an increase in supporter use of 'prompts'. After intervention, there were fewer story episodes. Narrative structure showed gains in action sequences leading to climax, and in closing elements, indicating a more complete narrative. CONCLUSIONS: The Storysharing® intervention appears to be associated with changes to the dyadic, personal narratives illustrating its potential.

Caregiver perceptions of children who have complex communication needs following a home-based intervention using augmentative and alternative communication in rural Kenya: an intervention note.

A high level of unmet communication need exists amongst children with developmental disabilities in sub-Saharan Africa. This study investigated preliminary evidence of the impact associated with a home-based, caregiver-implemented intervention employing AAC methods, with nine children in rural Kenya who have complex communication needs. The intervention used mainly locally-sourced low-tech materials, and was designed to make use of the child's strengths and the caregiver's natural expertise. A pretest-posttest design was used in the study. Data were gathered using an adapted version of the Communication Profile, which was based on the International Classification of Functioning, Disability, and Health (ICF) framework. The non-parametric Wilcoxon signed-rank test was applied to data from the first two sections of the Communication Profile-Adapted. Qualitative analysis was conducted on the final section. The data provided evidence of statistically significant positive changes in caregiver perceptions of communication at the levels of Body Structure and Function, and Activities for Communication. Also, analysis of the Participation for Communication section revealed some expansion to the children's social activities. The potential impact of the home-based intervention would benefit from investigation on a larger scale. Limitations of the study are discussed.

Survey of rehabilitation support for children 0-15 years in a rural part of Kenya.

PURPOSE: Information regarding the nature, availability and distribution of rehabilitation services for children with disabilities across developing countries is scarce, and data that do exist are of variable quality. If planning and development are to progress, information about service provision is vital. The aim was to establish the scope and nature of rehabilitation support available to children with disabilities (0-15 years) and their families in rural Kenya. METHOD: A comprehensive sample comprising service provision in the health and special education sectors was established. Non-governmental and community-based organisations were also included. A survey of rehabilitation services was conducted through examination of service-related documentation and key informant interviews with the heads of services. RESULTS: Rehabilitation comprised hospital-based occupational therapy, physiotherapy and orthopaedic technology; and seven special education establishments plus an education assessment resource centre. There was one non-government organisation and one community-based organisation relevant to children with disabilities. Activities focused on assessment, diagnosis and raising community awareness. Provision was challenged by inadequate staffing, resources and transport. Government funding was supplemented variously by donations and self-sufficiency initiatives. Rehabilitation approaches appeared to be informed by professional background of practitioner, rather than the needs of child. Service documentation revealed use of inconsistent recording methods. CONCLUSIONS: The data highlight the challenges of rehabilitation, demanding greater investment in personnel and their training, more material resources, improved access to the community and better recording mechanisms. IMPLICATIONS FOR REHABILITATION: There needs to be greater investment in rehabilitation provision in developing countries. Consideration of community-based initiatives is required to support better access for all. In order to argue the case for improved resources, better skills and mechanisms for recording, monitoring and evaluating practice are needed.

Investigation of practices to support the complex communication needs of children with hearing impairment and cerebral palsy in a rural district of Kenya: a case series.

BACKGROUND: Rehabilitation services are scarce in low-income countries, where under-representation of some specialist professions has led to the role extension of others. An example of this can be found in Kilifi in Kenya where the role of speech and language therapy has been taken on by occupational therapists and teachers. AIMS: To investigate the communication practices used by these professional groups to support children with complex communication needs in a rural part of Kenya and to explore the ways in which this might be seen to facilitate or obstruct improved communication by asking the following questions: What are the critical features of interactional discourse in practitioner-child dyads with caregiver-child dyads providing a natural comparison? What communicative modalities and practice techniques are invoked? And how does this information relate to extending professional roles? METHODS & PROCEDURES: An in-depth, descriptive study of a case series was conducted in a school for deaf children and the occupational therapy department of a district general hospital. A mixed methodology was used involving naturalistic observation and applied linguistics analysis. A convenience sample was established comprising six practitioner-child dyads assigned to partnership types: (A) three children with hearing impairment and their teachers; and (B) three children with cerebral palsy and their occupational therapists. As a natural comparator, the same three children in B were also observed with their mothers (partnership type C). Dyadic interaction was video recorded on three occasions. The video data were sampled, transcribed into standard orthography and translated. Codes were applied to determine turn structure, linguistic move types and communicative modalities. Sequential analysis was conducted on the move types. OUTCOMES & RESULTS: Partnership type A dyads showed a fairly even turn distribution between teacher and child. A common pattern was teacher-initiated Instruct and Model/Prompt, followed by child response in the form of an Action. The most frequently used modality was Sound Production and Hands-on-Articulators, which corresponded to articulation drill practice. Partnership type B dyads revealed a tendency towards adult domination of turns. The majority of adult-initiated moves required no response from the child. The practice technique Hands-on-Articulators involved manipulating the oral musculature of the child. Partnership type C dyads showed resonances of type B dyads, although focused more on Motor-Action in relation to task performance. CONCLUSIONS & IMPLICATIONS: The assignment of speech and language therapy duties to teachers and occupational therapists has resulted in suboptimal practice for children with complex communication needs.

Assessing and comparing the outcome measures for the rehabilitation of adults with communication disorders in randomised controlled trials: an International Classification of Functioning, Disability and Health approach.

PURPOSE: To identify the assessment instruments and relevant outcome measures used in randomised clinical trials (RCTs) relating to interventions for adults with communication disorders, and then examine and compare the domains of the outcome measures using the International Classification of Functioning, Disability and Health (ICF) as a reference tool. METHOD: Published RCTs with primary focus on the effectiveness of rehabilitation for adults with communication disorders were systematically reviewed. Identified RCTs were examined for all the assessments used as outcome measures. Distinctions were made between the use of standardised assessment tools and non-standardised empirical measures. The key concepts examined by the outcome measures were then linked to the ICF using the established ICF linking rules. RESULTS: The systematic review included 24 RCTs in which 11 trials used non-standardised empirical assessment as the outcome measure, and 18 trials included standardised instruments as the outcome measure. It is clear that all the identified items and meaningful concepts from the assessment used in the included studies can be linked to the ICF categories. Of the 108 linked level-two ICF categories, 53% were linked to 'body functions', 36% to 'activity and participation' and 9% to 'environmental factors'. CONCLUSIONS: A wide range of outcome measures have been used in RCTs of interventions for adults with communications disorders. The ICF provides a clarifying framework for systematically gathering and examining the information about the content of outcome measures and then can be used as a common reference to identify and compare the domains of the outcome measures. The high proportion of elements relating to body functions raises some questions about the purpose and aims of the interventions.