Developing a patient care pathway for emotional support around the point of multiple sclerosis diagnosis: A stakeholder engagement study

Abstract Background Diagnosing multiple sclerosis (MS) can be a lengthy process, which can negatively affect psychological well‐being, condition management, and future engagement with health services. Therefore, providing timely and appropriate emotional support may improve adjustment and health outcomes. Purpose To develop a patient care pathway for providing emotional support around the point of diagnosing MS, and to explore potential barriers and facilitators to delivery and implementation. Methods Focus groups were conducted with 26 stakeholders, including 16 people living with MS, 5 carers/family members and 5 professionals working with people living with MS (3 MS nurses, 1 psychiatrist, and 1 charity staff member). Discussions were audio‐recorded, transcribed verbatim and analyzed using framework analysis. Results Participants suggested that a patient care pathway should include comprehensive information provision as a part of emotional support at diagnosis, and follow‐up sessions with a healthcare professional. Barriers including increasing staff workloads and financial costs to health services were acknowledged, thus participants suggested including peer support workers to deliver additional emotional support. All participants agreed that elements of a care pathway and embedded interventions should be individually tailored, yet provided within a standardized system to ensure accessibility. Conclusions A patient care pathway was developed with stakeholders, which included an embedded MS Nurse support intervention supplemented with peer support sessions. Participants suggested that the pathway should be delivered within a standardized system to ensure equity of service provision across the country. Patient or Public Contribution This research was conceptualized and designed collaboratively with Nottingham Multiple Sclerosis Patient and Public Involvement and Engagement (PPIE) group members. One member is a co‐author and was actively involved in every key stage of the research process, including co‐design of the pathway and research protocol, data collection (including presenting to participants and moderating group discussions), analysis and write‐up. Authors consulted with PPIE members at two meetings (9 and 11 PPIE attendees per meeting) where they gave feedback on the research design, findings and the resulting pathway. People living with MS and carers of people with MS were included in the focus groups as participants.


| INTRODUCTION
Multiple sclerosis (MS) is a chronic neurological condition, often diagnosed in mid-adulthood, and is the most common cause of nontraumatic neurological disability in working-age adults. 1 Symptoms of MS, which may include 'visible' (e.g., dexterity and mobility problems) and 'invisible' symptoms (e.g., fatigue, cognitive problems), 2 result from inflammation and demyelination of the central nervous system. 3,4 An estimated 2.8 million people live with MS worldwide, with around 107,000 new diagnoses each year 1 -a frequency that emphasizes the importance of delivering diagnostic news which meets the needs of an increasing clinical population. However, as there is no single, simple diagnostic test, diagnosing MS can be a lengthy process for individuals, which can cause confusion, relief, distress, and frustration. [5][6][7] The general well-being of people with MS can be impacted by how they adapt to their changing health circumstances. 8 Psychological adjustment refers to the process of adapting to circumstances such as chronic disease and associated treatment, 9 whereby the individual aims to maintain equilibrium between competing environmental demands and the resulting stress. The way in which the period surrounding diagnosis is managed may determine how successfully a person adjusts to MS, influencing future perceptions of their condition, and may affect subsequent engagement with services. 6 The prediagnosis period in which symptoms are investigated can be particularly distressing due to the perceived uncertainty while awaiting diagnosis. 6 Qualitative evidence shows feelings of being misunderstood before the legitimization of individuals' condition by confirmed diagnosis, such that diagnosis produced feelings of devastation which conflicted with relief at being able to explain symptoms that were previously disbelieved by others. 6 Therefore, ensuring that people receive comprehensive information and support from the beginning of their lifelong MS journey may be crucial to facilitating positive psychological adjustment, while improving treatment outcomes and long-term management.
A recent meta-synthesis showed that many newly diagnosed people with MS had unmet emotional and informational needs during their diagnosis period. 10 In a qualitative study of experiences of adjusting to early-stage MS, many participants described feeling fear at being given a diagnosis and feeling overwhelmed by thoughts of impending doom. 5 However, they also felt that seeking positive, optimistic information increased their ability to accept the diagnosis and their perceived control over MS, while social support was regarded as critical for their adjustment. 5 Moreover, other qualitative research has suggested that providing adequate information about their condition and its treatment options at diagnosis may reduce feelings of anxiety and uncertainty. 6 This suggests that providing positively framed information coupled with social support may be key to facilitating successful psychological adjustment and that informational support is considered a part of emotional support. 5,6 Similarly, a recent metareview of systematic reviews on adjustment to MS suggested that professional support, information provision, continuity of care and peer support are factors throughout the diagnostic process which were linked to better psychological adjustment. 11 The value of providing accessible information, suitable advice and support at diagnosis is well recognized. 12,13 However, current literature demonstrates that poor support and information provision has continued for people with MS throughout their diagnosis period 10,11 and should be part of emotional support. 5,6 A survey of people with MS in the United Kingdom identified that information provision at diagnosis was inconsistent. 14 Inadequate information provision has persisted over time and appears to be a common issue across Europe. 6,7,10,11,15 Findings from a meta-review showed that there are no adequate emotional support interventions that TINDALL ET AL. | 859 specifically target individuals newly diagnosed with MS. 11 Most resources dealt with providing information about MS (its causes, symptoms and treatment options), rather than broader emotional support including advice around living with MS. Furthermore, there are no established care pathways in the United Kingdom that include emotional support around MS diagnosis, and no referral systems to seamlessly incorporate wider 'third-sector' or charity-based support resources or services. 10 The healthcare charity sector is an important source of support for many people with specific health issues. In the United Kingdom, the MS Society (www.mssociety.org.uk) and MS Trust (www.mstrust. org.uk) are trusted resources for many people with MS. Shift.MS (www.shift.ms) offers users peer support through its social network to connect with others with MS, helping people to make sense of their diagnosis and adapt to life with MS. These charities and support groups serve a useful adjuvant to the standard care patients receive from the National Health Service (NHS; e.g., providing emotional and social support) and in some cases, offer support that no one else provides (e.g., help with insurance, support for carers, social connection). In the UK context, however, the NHS and the charity providers for people with MS are somewhat disjointed, resulting in patients not receiving the best care they can receive. To address this gap, we aimed to co-construct a care pathway to provide emotional support to people around the point of MS diagnosis that linked NHS services with those provided by the charity sector.
The importance of theory in developing and evaluating complex interventions is well established. 16 Here, we propose an initial pathway that depicts the theoretical framework for providing emotional support around the point of MS diagnosis ( Figure 1). This initial pathway was informed by reviews of literature, 10,11 pertinent theory, 17 Patient and Public Involvement and Engagement (PPIE), clinical experience and service realities. It proposes a timely referral system to charity-based services for bridging the gap between services the clinic and relevant charities can provide around the time of the diagnosis. However, this pathway is preliminary and needs further exploration to determine its utility and acceptability.
Moreover, exploring the views of key stakeholders enables the production of experience-based co-developed interventions that empower and engage service users. 18,19 This study aimed to develop a patient care pathway to provide emotional support around the point of MS diagnosis (i.e., prediagnosis when investigations are underway when the diagnosis is given, and in the weeks postdiagnosis). The secondary aim was to explore potential barriers and facilitators to the delivery and implementation of this pathway. We followed a person-centred, 19 experience-based co-design approach 18 allowing key stakeholders (i.e., service users and service providers) to inform pathway design collaboratively through group discussions, to identify sustainable changes that meet

| METHOD
To ensure the quality of reporting and transparency, we followed the consolidated criteria for reporting qualitative studies (COREQ) and the reporting recommendations for qualitative methods in communication and patient education research. 21 Point of diagnosis F I G U R E 1 Initial care pathway around the point of diagnosis of MS. 'Point of diagnosis' refers to prediagnosis when investigations are underway or when diagnostic news is given to the patient while 'newly diagnosed' refers to the following period (up to five years, as per PPIE comments suggesting that uncertainty following diagnosis can last this long). The top part of the diagram (grey) demonstrates a need for a timely referral system to charity-based services for bridging the gap between the clinic and the charities around the time of the diagnosis and indicates who could provide the care and where it could be provided. The 'Technology and the Internet' section describes the medium by which emotional support could be provided in the interim. This would serve as a link between clinics and charity-based services, and facilitate the transition of support which is provided by clinics to charity-based services (denoted by the arrows), by offering relevant, accessible and reliable online information and ongoing online support. Referral to Psychological Services could provide individual support during the point of diagnosis, and Psychological Services could also link service users to charity-based services. Support from Family/carers is intended to supplement support from MS charities, as per suggestions from PPIE consultation. The bottom two boxes describe when particular types of emotional support could be provided across time. MS, multiple sclerosis; PPIE, Patient and Public Involvement and Engagement.

| Design
A qualitative, multistakeholder engagement study design was used to gain different perspectives from key stakeholders. The engagement involved consulting with stakeholders through different research activities. This consisted of one activity and two discussions, each addressing a different open question, across five individual focus groups and whole-group feedback sessions following each discussion ( Figure 2). All stakeholders participated in the focus groups, and one representative from each group summarized their groups' reflections within whole-group feedback.

| Participants
We refer to stakeholders as individuals targeted by the proposed pathway or involved in its development and/or delivery. Inclusion criteria for each stakeholder category are provided in Table 1

| Data analysis
Discussions were audio-recorded and transcribed verbatim, omitting participant-identifiable information and analysed using framework analysis. 23,24 The analysis primarily applied a deductive-inductive approach to address our research aims. Thus, we anticipated that   Table 2 describes further demographics.
Following framework analysis, 31 subthemes were interpreted (presented in italics) relating to the development of an emotional support pathway, organized into 5 superordinate categories (Table 3).

| Individualization
Focus groups elaborated on the timing of the pathway's elements, which was also repeated during whole-group feedback. They felt it should depend on the patient's unique needs and preferences. People with MS People referred for possible MS diagnosis (i.e., the period just before receiving a formal diagnosis of MS by a neurologist up to 5 years), or newly diagnosed with MS (up to 5 years postdiagnosis) or have been diagnosed longer than 5 years ago who could comment on their past experiences. We used a 5-year period because, for some people with MS, the uncertainty around diagnosis lasted for this long, and our PPIE group felt that it was important to capture those who were within this period.

| Standardization
While participants emphasized the importance of individualization, they agreed that some standardization is required to ensure accessibility.
Postcode lottery should be avoided, whereby a person's geographical location in the country can determine the service they receive.
Therefore, participants suggested using technology (e.g., the Internet, videoconferencing) to standardize service delivery throughout the pathway to be accessible for everyone. However, some participants felt that this would not be appropriate for all, arguing that some people avoid the Internet or would benefit more from in-person sessions rather than telephone calls. Again, this is linked with individualization and applying a person-centred approach throughout. Groups unanimously agreed that the crucial facilitator of peer support within a pathway was the contribution of shared experiences.
Many participants felt that individuals living with MS would be more relatable and credible, as most clinicians cannot understand the subjective experience of MS. Therefore, sharing relevant experiences may help patients to problem-solve, which many felt was vital for emotional support. Moreover, participants suggested that the peer supporter could act as a befriender to listen and empathize because they felt that sometimes patients need more emotional support than a healthcare professional can provide. Therefore, peers 'on the same wavelength' (Woman with MS, Group 5) could be instrumental during the process of adjustment around the point of diagnosis.
A patient care pathway was subsequently created by the research-active clinicians present during the stakeholder engagement discussions, in light of participants' reflections. This was then presented to a group of seven people with MS and four carers, who helped us to refine the pathway. Figure 3 depicts the co-constructed pathway.

| DISCUSSION
Stakeholder focus groups and whole-group discussions informed the development of a blueprint for a co-constructed 'point of MS diagnosis' patient care pathway to provide emotional support around the point of diagnosing MS (Figure 3). Many participants agreed that the proposed pathway should involve MS Nurses at the forefront, who should be introduced to the patient early during discussions about the diagnosis. Groups also agreed that adequate, appropriately delivered information is a key element of emotional support, equipping patients with reliable advice for dealing with practical issues, which should be positively framed to instil hope. The addition of peer support volunteers to assist during the pathway was suggested and accepted by many participants. This may also improve the health and well-being of the volunteers themselves. 26