Using the Medical Research Council framework and public involvement in the development of a communication partner training intervention for people with primary progressive aphasia (PPA): Better Conversations with PPA

To use the Medical Research Council framework for developing complex interventions to define and refine a manual and an online training resource for speech and language therapists to deliver BCPPA to people with PPA and their CPs.

Literature was selected following discussion with the research team to identify papers known to explore the theoretical underpinnings of interventions for dementia and CPT. The author then conducted searches of the reference lists of the articles to identify any other relevant articles. This included literature on existing models of dementia, principles of applied Conversation Analysis, behaviour change theory and frameworks for chronic disease self- management were explored. This informed the preliminary contents and focus of the intervention.

There remains a lack of guidance on undertaking Public Involvement with people with communication difficulties [32] This work was therefore informed by information from the INVOLVE website [28] and bespoke advice from a co-author (K.S.) and expert on Public Involvement with people with stroke aphasia but modified to meet the needs of people in the group. Four people with PPA and their spouses, two expert speech and language therapists, a neuropsychologist and the group facilitator (A.V.) took part in 12 formal BCPPA Public Involvement steering group meetings. Public Involcement work to co-produce the BCPPA intervention materials and training modules was informed by feedback from people with PPA who had previously received CPT [32], research undertaken by A.V [10‐12]. and research into the BCA program for people with stroke aphasia [33]. Discussion focused on identifying what distinct training modules would be required for the BCPPA training program and what the session plans and handouts would need to include for the manual. Once identified, a timeline for development was agreed and work undertaken to coproduce the content in steering group meetings. In order to support communication, steering group members were informed of the topic for discussion in advance of each meeting and invited to contribute in advance, during or after meetings using verbal, written or visual means, e.g. bringing photos, drawing pictures writing brainstorms or assembling and re-assembling draft materials.

A draft of the BCPPA manual was developed using PowerPoint software. In order to upload these to the UCLeXtend website an online software package called Articulate was used to adapt the PowerPoint slides to an appropriate format. The work was undertaken with assistance from speech and language therapist researchers and four postgraduate researchers in speech and language sciences who were paid for their time.

The Nominal Group Technique was carried out at one of the Royal College of Speech and Language Therapy, Dementia and Mental health Clinical Excellence Network meetings. Draft one of the manual was made available to attendees (speech and language therapists). In order to gain an understanding of the clinical experiences and reality of speech and language therapists a qualitative research method was identified as appropriate. Speech and language therapists were encouraged to review the resource and pilot it with their clients. To ensure the BCPPA intervention reflected a consensus view of the most important components to include in a CPT intervention for people with PPA and their families a Nominal Group Technique method was chosen. Given that many of the speech and language therapists participating in the meeting had pre-existing professional relationships that could result in certain voices being represented over others in discussions, the Nominal Group Technique method was also chosen to provide opportunities to consider ideas and experiences equally yet allowing for clarification and discussion prior to rating [34].

Six weeks prior to attending the meeting speech and language therapists were sent an email inviting them to anonymously complete a 12-item feedback survey comprising all open questions (supplementary document 1), hosted online on the Google Forms platform. Survey questions were developed by A.V. in consultation with the steering group and included questions about speech and language therapists’ experiences and views on the content and format of the manual.

The Nominal Group Technique meeting itself comprised a two-stage ranking process commencing with a 90-min group session (stage one), followed by email consultation (stage two). Meeting facilitators (AV and SB) agreed the session plan and central question for discussion in advance (see supplementary document 2), in line with guidelines for conducting Nominal Group Technique meetings [14]. At stage two, results of the group session were circulated via email to all participants, providing information on scores and mean rankings for each item. As per guidelines for conducting Nominal Group Technique meetings [32], items describing the same ideas from the two groups were merged, following discussion and agreement between A.V. and S.B. Participants were asked to reply via email identifying and ranking their top eight items from this list (by placing a number from 1 to 8 to reflect which is most important - 8 and least important - 1). Following Nominal Group Technique guidelines [34], scores were tallied and mean rankings calculated to identify the top eight ranked items overall.

Two focus groups took place, to provide people with PPA and their families the choice of attending with or without partners. Discussion was guided by the question ‘How can speech and language therapists support people with PPA to live well and maintain relationships?’. The focus groups were jointly facilitated by A.V., alongside volunteer student speech and language therapists from UCL (one per focus group). A topic guide was co-produced with the BCPPA steering group and attendees of the PPA branch of the Rare Dementia Support Group at UCL (see supplementary document 3).

Focus group discussions were video recorded and transcribed by UCL student speech and language therapists (using transcription guidance [35]). Given the researchers objectives to understand the lived experiences of people with PPA and their families, and gather opinions from them, qualitative methods employing a realist approach to reflexive thematic analysis was undertaken [36, 37]. Initial codes were generated by systematically coding interesting features (phase 2), collating these into potential themes (phase 3) and reviewing them in relation to the coded extracts (phase 4). Potential themes were refined to generate definitions and names (phase 5), further inspected to identify and report any additional key elements (phase 6). In addition, to improve reliability of analysis, four speech and language therapist researchers with experience of thematic analysis independently extracted data from a randomly selected section of transcript, discussed and reached agreement on the coding of themes arising from the data.

Results of work in stages 4 and 5 of intervention development were presented to the project steering group. Refinements were jointly identified and agreed by the group members.

Existing literature comprising the bio-psychosocial model of dementia, applied Conversation Analysis, behaviour change theory and self-management and self-efficacy theory was examined.

The bio-psychosocial model [38] proposes that there are factors other than the organic causes of dementia that influence the nature and speed of deterioration in daily functioning. These include some factors that are fixed, such as PPA variant, that cannot be changed. The BCPPA manual therefore provides practice tasks, to maximise generalisation for people with semantic PPA, for whom this is more difficult than those with non-fluent PPA. Tractable factors, such as the way a CP interacts with a person with PPA, may be amenable to change and are directly targeted in the BCPPA intervention. Adaptive mechanisms used by the CP, such as multiple questions or test questions, may result in the person with PPA feeling incompetent [13]. On the other hand, the use of gesture and enactment (whole body gesture and pantomime) by a person with PPA when they are having difficulty retrieving a spoken word [39] could be described as an effective coping strategy. The BCPPA intervention seeks to take account of fixed factors whilst targeting tractable factors to support the dyad (person with PPA and their CP) achieve their potential function.

Conversation Analysis is an approach to the study of human social interaction through the analysis of spontaneous, naturally occurring talk [40]. A number of Conversation Analysis informed stroke aphasia intervention studies and clinical resources have been developed [41] such as Supported Conversation for adults with Aphasia [18] Supporting Partners of People with Aphasia in Relationships and Conversation [41] and BCA [42]. These have in common the analysis of video recordings of natural conversations between the person with aphasia and their CP, and providing these as video feedback, as a foundation for targeting therapy [41]. The speech and language therapist (who typically delivers such an intervention) analyses 10-15 min video-recorded interaction to identify behaviours resulting in conversational breakdown, known as barriers, and ways in which members of a dyad successfully resolve or circumvent troubles to maintain interaction, known as facilitators. The aim of video feedback is to increase awareness in one or both members of the dyad of the impact of their behaviours, and jointly agree on goals for therapy. Once the goals of therapy are agreed upon, a process of practice, through supported conversations, role play and reflection, is commonly employed [41]. The BCPPA intervention is informed by this well-described [43], CA-underpinned approach to CPT.

Recognising conversational barrier behaviours in video recordings of oneself and setting a goal to cease these, or adopt facilitative strategies instead, does not guarantee that a change in behaviour will occur [44]. Behaviour change theory, specifically the COM-B model [33] accounts for an individual’s behaviour change as the product of three equally weighted components namely Capability, Opportunity and Motivation. Researchers examined video recordings of Conversation Analysis-underpinned CPT being delivered to people with stroke aphasia and their CPs [45] and used the COM-B model [33] to identify the essential change processes and the core procedures that serve them [46]. The BCPPA intervention incorporates the seven core mechanisms that have been identified as essential to behaviour change in a CPT [45], specifically the processes to motivate change and those that embed changes (See supplementary material 4).

Central to self-management is the concept of the client as an active participant whose current status is influenced not only by diagnosis but by psychological responses and experiences. This implies interventions should address the ability to self-manage daily activities and the emotional journey, not just medical symptoms [46, 47]. Taking action to accomplish a plan to self-manage their condition is more likely to succeed if a person has the confidence or self-efficacy to achieve it [48]. Self-efficacy is a mechanism that directs behaviour change, for if one feels in control of a behaviour it becomes easier to make a change to it [49]. Five core self-management skills and four key self-efficacy mechanisms have been highlighted for inclusion in speech and language therapist interventions with people with progressive communication difficulties [48] and these have been considered in the development of the BCPPA intervention (see supplementary file 4).

Decisions made included:

Module 5: the BCPPA therapy, hosted the BCPPA manual comprising session plans, session handouts and home-based tasks for each of the four BCPPA intervention sessions. The session plans identified intervention components as either core or non-essential components that can be tailored to an individual’s needs.

The draft manual was evaluated by the steering group to ensure information was presented in an accessible way. This included decisions on images and formatting.

The first draft of the manual was uploaded to a secure area on the UCLeXtend website and made available to speech and language therapists participating in the stage 4 consensus work via a bespoke URL. It was not publicly accessible.

Refinements for the BCPPA manual are presented in Table 6. The refined BCPPA program was consequently made available to participating local speech and language therapist collaborators on UCLeXtend as part of their training in preparation for delivering the intervention during the randomised controlled pilot-feasibility study. The final intervention is described in detail, using the Template for Intervention Description and Replication (TiDIER), in the authors PhD thesis which this paper is based on [50], and a published protocol for study which remains currently underway [51]. Further to this, the project steering group made plans to continue working to co-produce the remaining four modules, in anticipation of a future launch of the BCPPA program. This paper is based on work from the authors PhD thesis.

Drawing on the best available evidence and appropriate theory to develop the BCPPA manual, in accordance with Medical Research Council guidance [29], should increase the likelihood that components of the intervention result in behaviour change. Extensive use of theory has been associated with larger effect sizes in a review of online behaviour change interventions [53]. This work has involved new research with those targeted by the intervention as well as those delivering it.

There are, however, some methodological limitations. Nominal Group Technique does not allow for anonymisation in the way that other consensus methods such as Delphi do, and can thus bias the responses of participants. Unfortunately, only 20 of the 36 participants who attended the original meeting completed the final Nominal Group Technique ranking task by email. These numbers may be associated with the fact that some participants did not have experience working with people with PPA. The Nominal Group Technique did nevertheless, provide a method of involving large participant numbers and incorporating mathematical voting techniques to aggregate group judgements equally [34]. Despite only 12 of the participants who attended the Nominal Group Technique meeting having viewed the modules beforehand, making the intervention manual available enabled scrutiny of its practicality for clinical practice in anticipation of the phase II NHS based randomised controlled pilot-feasibility study. Notably, only two males were recruited to the Nominal Group Technique, though this is generally representative of the current speech and language therapy community [54]. Despite being a useful method for eliciting participant’s genuine and honest opinions, a focus group can be a challenging communication environment [55]. The role of the speech and language therapist facilitator and the student speech and language therapist co-facilitators was to mitigate this by enabling participants to contribute to discussion. The option to attend with CPs to support communication was also provided, but instead participants prioritised the convenience of meeting dates and times. Given the steering group was established a number of years prior to the recently published practice standards for Public Involvement [56] it is likely that the methods employed may have limited the effectiveness of the co-produced work. Some have criticised the steering group model for consulting with only a small number of individuals. There were only three couples with PPA in this group and that may have limited its value. PPA is, however, a relatively rare condition and people were approached to reflect the known diversity within the condition. Additionally, new members were sought when others withdrew due to disease progression, and the author sought to gather perspectives of other people and their families through individual telephone contact. Despite approaching professionals from other disciplines, including medicine and social work, interested individuals were not able to attend steering group meetings. The author was able to consult with the research team, including neurology colleagues, to gather feedback and ideas.

A manualised approach enables standardised delivery of the intervention for a future trial. Given that speech and language therapists in clinical practice may have limited experience of working with people with PPA [10, 11], this helps to maximise ease and fidelity of delivery for future implementation. However, a manualised intervention may limit the potential to tailor an intervention to individual clients, for example by deciding not to use video recording or by delivering the intervention to a person accompanied by two CPs. Person-centred components have been identified as important for functional communication interventions for people with PPA, and have been highlighted as important for behaviour change [45, 46]. The development of this intervention took behaviour change theory into account and embedded the core processes and mechanisms that had been identified in previous CPT research as essential components. These were clearly signposted in the manual and distinguished from non-essential components that were amendable to tailoring. Furthermore, expecting four 1-h therapy sessions to result in a change may seem ambitious. However, the decision on dosage was made based on the average number of sessions that speech and language therapists reported having available to deliver functional communication interventions for PPA [11]. Developing an intervention that meets this requirement increases the chance of implementation.