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Developing a clinical pathway to identify and manage cognitive problems in Multiple Sclerosis: Qualitative findings from patients, family members, charity volunteers, clinicians and healthcare commissioners
School of Medicine, University of Nottingham, Nottingham, NG7 2TU, United KingdomInstitute of Mental Health, Nottinghamshire Healthcare NHS Foundation Trust, Nottingham, NG7 2TU, United Kingdom
No established care pathway exists for screening and managing cognitive problems.
•
Based on stakeholder interviews, we developed a logic model for the pathway.
•
The logic model illustrates how a new clinical care pathway could work.
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To work, the pathway relies on shared responsibility and a person-centred approach.
Abstract
Background
Cognitive problems are a common and debilitating symptom of multiple sclerosis (MS). Screening and treatment for cognitive problems are recommended, however these are not routinely delivered in UK clinics.
We collected and synthesised stakeholder perspectives to develop a care pathway for cognitive problems in MS and produce a logic model, illustrating how this pathway might operate.
Methods
Forty-nine stakeholders, including people with MS and care providers, participated in semi-structured interviews and focus groups. Participants viewed information that illustrated how the pathway might work and provided feedback. Data, transcribed verbatim and analysed using Framework Analysis, were mapped onto a preliminary logic model and accompanying thematic framework.
Results
The proposed pathway was perceived as helpful in providing standardised support for a neglected MS symptom. Training packages, online cognitive screening, and triaging decisions were viewed as crucial activities. Shared responsibility, a person-centred approach, and addressing the complexity of cognitive problems were important engagement mechanisms. Allocating time during clinic appointments and within staff workloads were essential resources for implementation.
Conclusion
Our co-constructed MS cognitive screening and management pathway will be evaluated for clinical and cost-effectiveness in a trial. However, in the interim, clinicians can adapt and implement this pathway in their own services and evaluate it locally.
Despite these calls to action, UK MS services do not have an established care pathway which integrates these recommendations. A UK-wide survey of clinicians found variation in cognitive assessments used, often with inappropriate screening tools used rather than the recommended tests [
]. We also found a lack of consistency in reporting cognitive rehabilitation interventions, particularly regarding the content of interventions and their underlying framework [
]. To address this gap, we aimed to develop a multi-agency, co-constructed, clinical pathway to forge a consensus on screening and managing cognitive problems in MS, and how to go about this.
Medical Research Council (MRC) guidelines stress the importance of theory in developing and evaluating complex interventions [
]. Here we propose an initial logic model (Fig. 1) which depicts the theory underpinning a screening and management pathway for cognitive problems in MS. Our model was informed by literature reviews [
]), Patient and Public Involvement (PPI), clinical experience, and service realities. It adopts a Situation-Inputs-Outputs-Mechanism-Outcome configuration [
Situation describes the contextual features that pre-date the introduction of the pathway; including the high prevalence of cognitive problems, lack of standardised screening and support for cognitive problems (particularly mild-moderate problems [
, and healthcare recommendations which drive the focus of clinical care (e.g., NICE recommends screening and managing cognitive problems, but this is not based on robust evidence and does not refer to a particular assessment or treatment [
]). Inputs are the resources required to support the pathway. Outputs describe the products created by the activities of the pathway. Mechanisms are mediating factors and obstacles between the introduction of the pathway and the resulting outcomes (e.g., stakeholders need to be engaged and supported; and the pathway accessible and flexible for implementation). Outcomes describe what results from the pathway. The specifics of these factors can be found in Fig. 1.
Logic models are dynamic, they represent working hypotheses and are modified iteratively as new insights emerge based on primary (e.g., stakeholder consultation, evaluation studies) or secondary data (e.g., existing literature/policy) [
Here, stakeholder perspectives were used to develop a multi-agency cognitive screening and management pathway. This study is part of the Neuropsychological Evaluation and Rehabilitation in Multiple Sclerosis (NEuRoMS) project [
] which will evaluate the efficacy of this pathway across six UK MS clinics.
2. Material and methods
Ethical approval was granted by the University of Nottingham Faculty of Medicine and Health Sciences Ethics Committee (reference: 263–1903). All participants provided informed consent prior to data collection.
2.1 Recruitment
We sought to interview a broad range of stakeholders with complementary perspectives on a screening and management pathway for cognitive problems, we selected purposively from those willing to be interviewed to generate this.
Eligible participants were 18 years or older, able to communicate in English and provide informed consent. Our stakeholders included pwMS (diagnosed with MS), family members (relatives or carers for pwMS), MS charity volunteers, clinicians (neurologists, MS nurse specialists, neuropsychologists, occupational therapists (OTs) and neuro-physiotherapists working clinically with pwMS), and healthcare commissioners (working within a Clinical Commissioning Group that commission National Health Service [NHS] services).
PwMS, family members and charity volunteers were recruited through PPI networks, social media, MS charities, and by word of mouth. Clinicians and commissioners were recruited through professional networks.
Participants were contacted by email or phone and invited to a focus group or an interview (in-person, or via telephone/video conferencing), based on their preference.
2.2 Data collection
Semi-structured interviews were conducted by authors JMM, RdN, NE and HE (see Supplementary Materials for interview schedules). The focus group with pwMS was co-facilitated by our PPI partner CB, to enhance data richness [
]. Interviews were audio-recorded and transcribed verbatim.
Participants were shown several resources to illustrate how the pathway might work and were asked to provide feedback. These included brief video demonstrations of digital cognitive screening tests (Paced Auditory Serial Addition Task [PASAT] [
]. The logic model informed a working analytical framework (see Fig. 1 and Supplementary Materials for coding scheme) and data were mapped onto this. Review of the mapped and organised data informed a revision of the logic model based on the findings below.
Yardley's evaluative characteristics for good qualitative research were applied [
]. Summary data were presented to a PPI group to sense-check our interpretations.
3. Results
3.1 Study participants
Forty-four participants were interviewed (25–75 min), and 5 pwMS participated in the focus group (125 min). Characteristics for non-clinician stakeholders are presented in Table 1. Participating clinicians included four neurologists, MS nurse specialists, OTs and neuropsychologists each, three healthcare commissioners, and one neuro-physiotherapist. Recruitment stopped when we fulfilled our purposive sampling criteria and established data from a range of different stakeholders.
Table 1Participant characteristics for non-clinician stakeholders.
People with MS- Interviews (n = 15)
People with MS- Focus group (n = 5)
Family Members (n = 5)
Charity Volunteer (n = 4)
Age M(±)
48(9.5)
49.6(7.1)
54.2(19.9)
42.3(7.6)
Gender Female
12(80%)
4(80%)
1(20%)
3(75%)
Ethnicity White
14(93%)
5(100%)
4(100%)
3(75%)
Education level GCSE A Level Degree Higher degree Other
3 2 4 4 2
2 0 0 2 1
0 1 3 1 0
0 0 3 1 0
Employment Full-time Part-time Not employed Retired Voluntary full/part-time Full-time education
4 3 4 4 0 0
2 3 0 0 0 0
1 1 0 1 1 1
0 2 0 0 4* 0
Time since diagnosis M(±)
12.9(10.3)
12(8.7)
MS sub-type Relapsing-remitting Primary progressive Secondary progressive Unknown
9 1 4 1
4 0 1 0
Note: * 2 of these participants work part-time and volunteer part-time.
There was consensus on the current situation - cognitive problems were reported as prevalent and disruptive, and stakeholders recognised the need to address these problems using a standardised pathway. Discussions therefore focused on how the pathway would work.
Data were organised into overarching themes (pre-defined logic model configurations), themes (core patterns) and sub-themes (further depth). Key results are presented for each overarching theme (inputs, outputs, mechanisms, outcomes) and supporting data are displayed in Table 2, Table 3, Table 4, Table 5, with superscript numbers linking relevant quotes to the text. When multiple participant groups endorsed a sub-theme the term ‘stakeholders’ is used, otherwise the relevant group of stakeholders is specified (e.g., pwMS).
3.3 Inputs (Resources)
Clinical Staff were recognised as an important resource. Stakeholders pointed to ‘competition’ between symptoms during clinic appointments, with limited time to address multiple MS symptoms1. Clinicians commented on the need to monitor drug treatments during the appointment, which took precedence over dealing with cognitive problems2. Physical symptoms including spasticity, bladder and bowel issues were often prioritised over cognitive problems3.
Pressured workloads were also raised, particularly those of MS nurses4. Stakeholders reflected that any new pathway would impact on staff time5 and that it is unclear who might have the capacity to deliver this6.
People with MS thought they would need to expend mental and physical resources to engage in the pathway. Travelling to hospital requires time, effort and planning7. Digital technologies are also needed to access cognitive screening8. Further time commitments may be necessary from those attending multiple rehabilitation sessions9. Clinic facilities, including technology to host the cognitive screening (e.g., computer tablet, WiFi access10), and a clinic room where screening and support sessions could be completed11, was also identified as a key resource.
Clinicians and commissioners thought understanding existing costing and commissioning frameworks and having a strategy in place to show how the proposed pathway addresses these drivers12 would help ensure the pathway is adequately resourced.
Table 2Key Input (Resource) themes and sub-themes with supporting data.
Theme
Sub-theme (Context)
Sample of coded text
Clinical staff
Clinic appointment Time allocated to cognitive symptoms during limited clinic appointment
1. And in our symptom management clinics where they have half an hour appointments, in that appointment we will be looking at bladder, bowels, fatigue, mobility, spasticity – you name it, it's addressed in that. And if you touch on cognition, but again we don't have time to sit there and go through a proforma or anything like that or any kind of referral. MS Nurse MS07
2. There's these new medicine, I give you this medicine, has it got any side effects, let me tell you about the side effects and so on. So, the competition for time is, do I – at the moment because we didn't have the evidence base for the effective rehabilitation, there seem to be some pressure and probably it will take second priority in people's minds. Neurologist N01
3. So I usually really just tell her conversation about medication and that's all there's, you know, we have a bit of a tete a tete about that! [Laughs] And that's all there's time for! Charity Volunteer CV05
Pressured Workloads Capacity of multidisciplinary team members
4. Personally my experience of MS…yeah, I don't know how they would have the bandwidth to do this, it would be great if they do, but you know. Charity Volunteer CV05
5. I would just worry a little bit about timing, if you're doing it with everybody, because if they've got the – they're also very limited on time and they'll also be doing other OT roles as well. Neuropsychologist PS05
6. I think probably the Occupational Therapists are more used to delivering cognitive screening, giving advice on cognition and especially in the sense of how we're using the, you know, mechanisms – so the strategy that you're suggesting – incorporating with everyday life. …I have the impression that they have possibly a bit more time than the MS nurses. Neurologist N01
People with MS
Physical & Mental Requires concentration
7. Because it's hard work when you have to get to a hospital appointment, even for those of us that drive and don't have too much physical problems at any one time. It is hard work. Focus Group Male M2 Planning. Focus Group Male M1 Yeah. To get to [hospital 1], plus the expense, it's a big thing. Focus Group Female F1
Physical Requires travel, time away from work, access to technology, mobility
8. They may not have the money to buy the machinery, machines to access the link, they may not be IT literate and they may just be plain right poorly, you know, too poorly to do it. PwMS P02
9. And also trying to get the timing right as well because obviously a lot of people with MS are working so doing – if it's one off group it's easier, but if it's a group over a number of sessions, doing it during working hours is difficult for people to commit to. Neuropsychologist PS03
Clinic facilities
Technology Availability of Wi-Fi, tablet, etc.
10. We have a Wi-Fi. We have also NHS Wi-Fi which is free. MS Nurse MS05
Clinic rooms Availability and management
11. There is general mismanagement of rooms in the hospital. So once the MS nurses, they always find this space, because they find that the empty room and let's go there for the next half an hour. Neurologist N01
Costing and commissioning
Commissioning frameworks Understanding, awareness and a strategy to address
12. So what you'd need to do is cost out this programme in each of those areas, work out what their commissioning structures are for each of those areas and be very clear about what the key metrics are in terms of patient outcomes, experience, safety, funding. Commissioner CM03
Note: Alpha-numerical codes represent participant ID numbers.
Stakeholders agreed that training was important to ensure that staff feel supported and have the skills to deliver the pathway. Clinicians thought training should address screening (administering and understanding the cognitive tasks)1, triaging (interpretation), and managing cognitive problems (goal setting, rehabilitation philosophy)2. Clinicians raised supervision and ongoing monitoring3, recognising that post-training support4 would be beneficial.
The online screening tool was a key focus for discussion (see Supplementary Materials – Coding Scheme). Stakeholders felt that screening should be administered ahead of a clinic appointment, with the results enabling discussion during the appointment5. Most pwMS felt able to access the screening tool online at home6 and advocated sending a weblink to access the tool on a digital device7.
PwMS understood the relevance of the cognitive measures presented to them and thought these were appropriately challenging8. However, some found the mental arithmetic task (PASAT) unpleasant9. Everyone agreed that cognitive screening should be brief. Clinicians acknowledged the need to balance the sensitivity and brevity of screening tasks10.
Clinicians thought that screening results should be digitised within patients’ medical records11. They felt that the feedback report should include cut-offs to help identify individuals who may require support12 and enable discussions around the type and severity of the cognitive problem13. Most stakeholders thought the results should be communicated face-to-face, at a routine clinical appointment, rather than over the telephone or via letter/email14. Communications should be initiated by a neurologist where problems are severe15.
Stakeholders recognised the complexity of triage decisions and referrals. Where present, concurrent symptoms such as low mood and fatigue needed to be interpreted in relation to cognitive problems16. Stakeholders also highlighted the importance of the perspective of pwMS. Triaging should consider how the person was feeling during cognitive screening and any extenuating circumstances (e.g., relapse, technology problems17). Stakeholders thought that pwMS should engage in these discussions to reflect upon the functional impact of cognitive problems18.
Data relating to the cognitive rehabilitation/management programme reiterated that concurrent symptoms need to be addressed by the pathway, through the provision of relevant information19. Stakeholders felt the content should include compensatory strategies that can be implemented at home20 and gave examples of strategies they thought might work well (e.g., digital technologies21) or be less effective (e.g., abstract visualisation22).
Table 3Key Output (Activity) themes and sub-themes with supporting data.
Theme
Sub-theme (Context)
Sample of coded text
Training packages
Screening & Triaging How to support pwMS to complete screening and interpret the results
1. I'm not sure our OTs, even our specialist rehab OTs would have used the digit symbol and some of these, so it's kind of, you know, are introducing something new to people that then will need to interpret that, but I don't think with training, I think that's feasible isn't it. Neuropsychologist PS05
Cognitive management programme How to deliver and set goals
2. Not all nurses have been through rehabilitation unit or rehabilitation training to kind of be aware of the notion of goalsetting and monitoring and motivational – you know. Neurologist N03
Supervision Ongoing monitoring and support
3. So if this is just an MS nurse working on their own then they might find it harder to make some of those decisions unless they've had some solid training and some supervision, ongoing supervision just to, you know, flush it out a bit. Neuropsychologist PS05
4. I think it does definitely need to have a review and monitoring built into that. Neuropsychologist PS03
Screening tool
Administration How to enable completion of screening ahead of routine appointment
5. That would be an ideal, digitalising the assessment pre-appointment. That would be wonderful…so when they come into clinic we have got everything there. MS Nurse MS05
6. Interviewer So you wouldn't have any problems filling that in and accessing the link. Carer C20 No, no, not at all, I'd be alright.
7. Which is obviously brilliant because the whole thing about a paper link is that someone then has to type it in. And as much as possible, isn't it, you want someone to have the link on their device so they just have to click on it. Neurologist N03
Cognitive measures Include short, sharp assessments that avoid mathematics
8. Interviewer Did those tests seem relevant? PwMS P20 Yeah, very much so, yeah. Especially the one with the colours and the - Interviewer The Stroop test. PwMS P20 Yeah, you know, it's quite profound how your brain works because, you know, somebody without MS you still kind of have to really think one thing but you're having to override it. I think that's a really good test
9. Because I've got to think of too many numbers so I can't focus on the number that I need to focus on because I've just done the total. So I will have forgotten that number. I just couldn't do that. Focus Group Female F1
10. Your balances a bit, isn't it, it's finding something which is quick but MS nurses will be able to do with everybody quickly as part of their clinical interview while still being meaningful enough. Neuropsychologist PS03
Screening results
Transfer Electronic transfer to clinical team
11. So probably for us it would be emailing to the MS coordinator, who would upload it onto the patient's EPR so that it was there as an electronic document and then it would remain so, as opposed to lost in someone's email and never available again. Neurologist N03
Feedback report What should this include
12. I think if we could categorise them really in a binary way or in these three categories, I think that would be excellent. That's very practical and I think we all know pretty well who we're thinking of when we're thinking of these categories. Neurologist N02
13. I mean, it might be useful to know in roughly what sort of domain we're talking about the deficit as being, you know, so whether it's a memory problem or whether it's – I don't know – been a processing problem or maybe something roughly categorising it a little bit further rather than just severity. Neurologist N04
Communication How to communicate screening results to pwMS
14. I think face to face is probably better than an email. Carer C20
15. Well it depends what the result is. If it's very severe, you're going to need a consultant I think. Focus Group Male M2
16. You will need somebody who's quite skilled at interpreting the different components you've discussed, so the mood and the cognitive, to think about how best to manage their difficulties. Neuropsychologist PS03
Perspective of person with MS Allow pwMS to indicate how they are feeling
17. Everything's all right, or you might be having a bad day but you picked up this. How do you feel about how this goes for you every day? Is this actually a problem or was it just, you know, that answer on the test? PwMS P27
Functional impact How do cognitive problems affect pwMS
18. I think as long as it's then not prescriptive and it's not taken at that value of it being the person in front of them is sitting there saying ‘I'm struggling at work’ and we turn round and go ‘but the screens don't show us anything’ and then that's taken as part of I suppose a triangulated discussion which you'd hope any clinical team would facilitate. Neuropsychologist PS05
Cognitive management programme
Content What should the programme cover
19. And it could also provide a bit of information, you mentioned fatigue earlier and that having an impact sometimes. Charity Volunteer CV01
20. Skills, different techniques, to help us and then you perhaps try and test it. Focus Group Male M2
21. The way that people are making decisions, and in the way that they're managing their condition or not managing their condition effectively. Occupational Therapist OT02
22. If it's abstract it's very difficult for them to take, say reading something, or being given a handout to applying it. Neuropsychologist PS03
Note: Alpha-numerical codes represent participant ID numbers.
The complexity of cognitive problems was raised as an important mediator. Stakeholders highlighted the interdependence of symptoms and recognised that stress and fatigue can exacerbate cognitive problems1. Similarly, cognitively demanding activities left pwMS feeling fatigued and drained2. Stakeholders thought the pathway should recognise that some cognitive problems will stem from brain damage driven by MS, whilst others are a secondary reaction to living with MS3. Clinicians thought this was an important distinction to observe4. Stakeholders suggested the pathway should also acknowledge that individual differences could influence cognitive performance and the effectiveness of support programs, and the perspective of related informants need to be addressed5,6.
Engaging pwMS addresses their reactions towards the pathway. Stakeholders felt pwMS should be informed about the rationale for the pathway. Explanations should reassure them that the pathway is meaningful7 and clarify how it will inform their clinical care8. Stakeholders thought information should be clearly communicated avoiding medical jargon9.
The timing of the pathway is an important mediator for pwMS. Stakeholders thought invitations to complete the screening should be aligned with a routine appointment where the results can be communicated without delays10. Most pwMS supported the idea of being told about the screening tool in advance of their appointment11 but acknowledged some might worry about this12. Some thought receiving information about the pathway might overwhelm newly diagnosed patients13, while others felt cognitive problems should form part of these early conversations14.
Stakeholders indicated that home-based approaches (e.g., online screening, telephone follow-ups for the cognitive management programme) would be convenient and less stressful for most pwMS15, particularly those in employment who cannot attend multiple appointments16. However, home-based approaches were not perceived as feasible for all17; stakeholders thought access to additional support (telephone or face-to-face in-clinic) would promote engagement18.
Engaging clinical staff encompassed clinicians’ reflections on which team members they thought responsible for addressing cognitive problems. Some thought MS nurses and neurologists would not consider cognitive problems as part of their role19 or see this as beyond their expertise20. Other clinicians perceived cognitive problems as a shared responsibility and thought the pathway should advocate a team-based approach21,22. Clinicians acknowledged that staff have different priorities and thought willingness to prioritise cognitive problems would promote acceptance of the pathway23.
Stakeholders thought the pathway should foster shared values, including a person-centred approach where care is tailored to the needs of the individual24. The pathway should recognise the differing impact that cognitive problems have25 and ensure support strategies are meaningful to the individual26. Some stakeholders recommended adopting a holistic approach to care27, incorporating physical and psychological wellbeing28.
The pathway should empower pwMS to be proactive and to take actions which target milder cognitive problems29,30. However, stakeholders also acknowledged some pwMS will not be motivated to engage with this because of fatigue and cognitive problems31.
PwMS thought the pathway should promote a positive outlook and inspire them32,33. However, clinicians recognised the need to manage expectations, e.g., manage problems rather than restore abilities34,35. These discussions can be frustrating for pwMS who want their cognitive abilities restored36. Stakeholders thought appropriate information might temper unrealistic expecations37.
Table 4Key Mechanism themes and sub-themes with supporting data.
Theme
Sub-theme (Context)
Sample of coded text
Complexity of cognitive problems
Interdependence of symptoms e.g., cognition mood, fatigue
1. Her cognitive issues come when she's under stress, so she breaks down into this almost jelly like state. Carer C20
2. There's lots of tabs open in my head, that's the only analogy I can put it down to and that creates the stress and then it's almost like I can't think straight and my head is like just all over the place and then that completely fatigues me. PwMS P20
Nature of cognitive problems Brain-based or secondary reaction
3. There's also lots of questions about whether it's associated with their mood or whether it's to do with their MS or some other issues too, so those questions often come up quite a lot. Neuropsychologist PS03
4. Sometimes I think we tend to think about real and not real cognitive problems and give the impression that we think the real ones are the ones related to scanning and your depressed, your tired, your sleepy patients do have cognitive problems, it's just the means to address them are likely to be different. Neurologist N03
Acknowledge individual differences Personal circumstances
5. I guess that's my question, like, how do I know I've got cognitive problems over and above the average 40 something year old. Charity Volunteer CV05
6. We do have people who there can be a temptation sometimes to overegg your cognitive problems. Neurologist N03
Engaging people with MS
Rationale clearly explained Describe pathway and what it involves
7. Someone might look at that and think “I don't see why I'm doing this. I don't understand why I'm being asked to do this. This has no bearing on my life. I'd never do a test like this in my real life”. PwMS P27
8. I think that, no, it's just explaining about the benefits they can get from it and that sort of thing. Carer C20
Instructions Clear, concise instructions
9. Yeah, I think definitely simplicity is definitely, yeah, the way to do it, yeah. PwMS P02
Timing Align screening with an upcoming appointment and avoid point of diagnosis
10. If it's that sort of level and it can get fed back fairly quickly at the next appointment, that – ‘cause you don't want to be sitting worrying about it, you want to have something pretty quick. PwMS P27
11. I think maybe if they'd told me in advance ‘we're going to do a very basic, you know, cognitive test, it's nothing to worry about, we'll explain it, please don't do any preparation, but just be relaxed about it’, I think that probably would have helped. Charity Volunteer CV05
12. I think if I'd known about in advance, you know, I might have then overthought it, to be honest. Charity Volunteer CV05
13. I think as people have said before, it's like what stage you're at and whether you want to look or whether you need to look. Focus Group Female F1
14. I had no idea about the cognitive things until later on and that was a nasty shock. Yeah, I think being more upfront about things might be, would be useful. PwMS P27
Home-based completion Remote screening allows flexibility and convenience
15. I think so. I think a lot of people would be happier doing that at home. It's more of a – yeah – it's that less stressful situation, you know. Carer C20
16. Doing it during working hours is difficult for people to commit to. Neuropsychologist PS03
17. I can imagine there's people who are really not au fait with computers, with it all becoming paper free and that stuff. PwMS P28
18. I think discussion in person one to one, whether that be a consultant or MS nurse, is probably the way to start it off. PwMS P27
Engaging clinical staff
Perceived responsibility As an individual and within a clinical team
19. I am trained to give medicines. So, why to see patients and make sure that they have all medicine's correctly. So there will be some doctors that will not be keen doing this 2 min in a consultation. Neurologist N01
20. I think that it's not part of their role, they wouldn't see it as part of their role and it's slightly out of their competency I suppose…it's not traditionally seen as a nurse thing, I don't think, it's more a psychology, an occupational therapist, you know, an OT thing, yeah. Neuropsychologist PS01
21. I actually do think it needs to be you know, every symptom and management kind of needs to be sort of kind of responsibility for everybody. Occupational Therapist OT04
22. So it's really useful to have that team approach to cognitive difficulties. Neuropsychologist PS03
Willingness and motivation To adopt an alternative approach
23. Yes, we absolutely value the importance of cognition. And that would be no problem at all from our perspective, in terms of making that a key priority. Occupational Therapist OT02
Foster shared values of the pathway
Person-centred Individualised
24. Not the same thing works for everybody, so I think a kind of basic starting point and then personalise it after that would be a good idea. Charity Volunteer CV01
25. How much it's impacting somebody's life, so you can have, you know, quite mild difficulties but that really impact on somebody's life, or you can have moderate difficulties which aren't really impacting. Neuropsychologist PS03
26. I suppose it's meaningful to that person, rather than it just being a lot of suggestions thrown at them, that it is meaningful to that person. Neuropsychologist PS05
Holistic Care for the person as a whole
27. We want to ensure there's parity, that these people get parity of esteem, that they're whole system. Commissioner CM02
28. So there's always been very holistic, very focused on physical rehab as well as psychological and cognitive. Neuropsychologist PS04
Proactive Initiate and act
29. I feel like that could have quite a protective element to it …and it helps them, it empowers them to speak about their difficulties as well with language that they understand. Neuropsychologist PS05
30. It's to help and it'll help yourself and it'll help others and just go that way. Carer C20
31. So if somebody is really, really depressed, again through struggles with their memory and they will probably struggle with initiation as well because they'll be so low, so actually probably implementing some memory strategies might be difficult for them. Neuropsychologist PS03
Positivity Encourage a positive attitude
32. Somebody who's like inspiring, positive, you know, that's what I would love. Focus Group Female F1
33. It's about retaining a positive mind and a positive mindset. PwMS P20
Manage Expectations Coping with cognitive problems - not retraining cognitive skills
34. It's about this is a way of managing and understanding, not a way of getting rid of difficulties. Neuropsychologist PS05
35. But I think it's very important in terms of the language we're giving MS nurses and other professionals who are then going to be feeding this back and doing cognitive rehabilitation is the idea that coming on these four sessions, it's not going to make this any better, but it may help you to live with it better. Neuropsychologist PS05
36. I want to recover, yes, I do want to compensate and find other strategies in the meantime, but I still want to feel that it's something I can regain and rebuild. Charity Volunteer CV05
37. And there are really difficult conversations to have with lots of people we work with and you're going ‘actually there's no evidence for that’. Neuropsychologist PS05
Note: Alpha-numerical codes represent participant ID numbers.
We elicited ‘short-term’ outcomes of the pathway from the data, including improved access to standardised clinical care for cognitive problems. Clinicians spoke about the lack of formalised guidelines for MS services1 and thought the pathway would address this2. Clinicians and commissioners felt the pathway would facilitate conversations about cognitive problems by providing information about patients’ symptoms3,4. These conversations were also perceived positively by pwMS5, who valued the opportunity to discuss cognitive problems6 and have their concerns validated7,8.
Some stakeholders acknowledged that anticipating the screening results could worry pwMS9. Moreover, being reminded of symptoms and noticing cognitive decline might be upsetting10,11. Consequently, some pwMS may disengage with the pathway12.
‘Longer-term’, stakeholders thought being able to understand and manage cognitive problems would improve quality of life amongst pwMS13,14. Earlier detection and management of problems might even prevent deterioration15 and prolong independence16. Stakeholders indicated that early detection and management could promote efficient use of NHS resources17,18. Home-based cognitive screening was also considered efficient, reducing the time required with a clinician19. Stakeholders reflected on a potential increase in referrals to psychological services20, which could overwhelm already pressured services21.
Table 5Key Outcome themes and sub-themes with supporting data.
Theme
Sub-theme (Context)
Sample of coded text
Short-term: Service
Standardised care Formal guidelines and referral pathways
1. Cognitive problems in people with MS they aren't routinely identified within normal practice, so no standardised way. Neuropsychologist PS03
2. I think if there's a tool there to use, I think it would facilitate, I can see it leading to more! Neuropsychologist PS05
Opportunity to discuss cognitive problems Confidence and competency to discuss
3. The patient would complete this and it would give more information to whoever is seeing them at their next appointment to further their conversation. Commissioner CM03
4. I think if it creates a conversation, I think anything is helpful and if it prompts the conversation in the direction of triangulating that with the patient experience and any observations that the team or families can offer, then it's doing the right thing. Neuropsychologist PS05
Short-term: People with MS
Opportunity to discuss cognitive problems Better able to understand and manage their condition
6. It helps them, it empowers them to speak about their difficulties as well with language that they understand. Neuropsychologist PS05
7. It feels so subtle that I think when those people mention that, it can be so easily dismissed as it's not impacting that much, but actually it can have a huge impact. Neuropsychologist PS05
8. I think it is about facing it on, it's not about keeping it down there, you have to face that you have had some changes and run with it, rather than hiding it, so I think questionnaires are good. PwMS P20
Remind person of their symptoms Being reminded of symptoms could be upsetting
9. In terms of doing a test on your own at home feeling like you've really struggled with it and then not having anyone to talk to about that until you're at your appointment, which is then filled with lots of other things, could feel quite isolating. Neuropsychologist PS05
10. It depends what sort of relationship they've got and obviously their disability as well, you know, how it's impacted so it can be quite emotional for some people I suppose. PwMS P20
11. And it's basically whether it is actually upsetting to some people to realise just where you were to where you are now, and that really upset me, from that point of view. PwMS P28
12. There are some people who, for a number of reasons, do not want things measured or recorded. Neurologist N03
Longer-term
Quality of life Improved confidence and productivity
13. I suspect that if people felt their cognition was better, they would have more confidence and more willingness to go out and do things and try things. PwMS P27
14. And the idea that if you invest in a strategy to make life a bit easier then you probably may not get fatigued as quickly. Neuropsychologist PS05
Prevention Earlier detection and intervention to reduce impact of cognitive problems
15. It's like why wait for the problem, just, you know, if people knew about that then they could get, they could work on it and get better and slow down the progression of cognitive depletion. PwMS P20
16. I I If they understand those changes earlier and they can learn the strategies then potentially that might protect employment. Neuropsychologist PS05
Efficient use of resources Optimise NHS resources
17. I mean, is this going to stop people ending up having unnecessary attendances or admissions into hospital, or is it going to just keep people more able to live at home for longer on their own or whatever it is. Commissioner CM03
18. Yeah – and also the not having to – if they're not going to some services then they don't have to travel and the cost and impact that it has on that. Commissioner CM03
19. Proposal of having patient to carry out assessment online at home, and is not coming to me, to clinician, I think is an excellent idea. MS Nurse MS05
Potential increase in referrals to psychological services Pathway could overwhelm already pressured services
20. This new activity that you are going to be potentially offering here is going to be – this in itself is going to cause more outpatient appointments, isn't it, it's going to cause more – it's going to encourage more appointments, or is this in Primary Care?
- so there'd be more activity in Secondary Care on the back of the fact that people would be called in for cognitive appointments. Commissioner CM03
21. But for us actually we have a massive delay to CBT and talking therapies. Neurologist N03
Note: Alpha-numerical codes represent participant ID numbers.
Stakeholder feedback confirmed that staff time, training packages, brief online screening tasks, and person-centred support for cognitive problems are important elements in the pathway. Improved access to care and clearer referral pathways for cognitive problems were still thought of as possible outcomes (as in Fig. 1). However, our data also offers new insights that challenge the initial logic model and enabled us to consider implementation more directly. Fig. 2 reflects a revised logic model that could inform subsequent service development.
Fig. 2Revised Logic Model Based on Stakeholder Input.
‘Resources’ replace ‘Inputs’ to reflect the importance of stakeholder investment. Our revised model recognises the need for staff to allocate time for the pathway during already stretched clinic appointments and pressured workloads. Stakeholders felt the capacity of MS nurses was particularly limited and this led us to reconsider which team members are best placed to support a new pathway. Other staff (e.g., psychologists, OTs) can be involved with appropriate training. Kalb et al. [
] acknowledge insufficient resources and highlight the lack of adequately trained clinicians as barriers for addressing cognitive problems. Training was therefore retained within the revised model with cognitive screening, triaging, management, and supervision packages now specified to reflect our new insights and explain how services can equip multi-disciplinary team members to deliver the pathway. Improved awareness of the importance of addressing cognitive problems within healthcare drivers/ recommendations and accepting shared responsibility for cognitive problems as part of comprehensive care provision amongst the wider neurology community will also help engage clinical staff with the pathway.
The ‘Activities’ component (previously labelled ‘Outputs’) supports recommendations that computerised testing is a means to implement cognitive screening in routine care (13). Electronic data gathered in this way may be integrated within patient records to promote routine monitoring [
]. Our study builds on this by demonstrating how and when cognitive screening could be undertaken (i.e., remotely prior to an upcoming clinic appointment with the option to access additional support). This may facilitate clinical implementation and sustainability.
Our revised logic model reflects the nature of cognitive problems and individual circumstances as important mediating factors. Medications, age, selection/scoring of cognitive assessments, comorbid health conditions and concurrent symptoms can affect cognitive problems [
]. Interpreting and contextualising results (because of this) might be challenging and staff training is needed to facilitate consultations and aid triage decisions. Psychoeducation focussing on concurrent symptoms should also be provided to pwMS.
Our findings provided insights into the content of the cognitive rehabilitation programme. This should be person-centred, encompassing psychoeducation and behavioural strategies to help pwMS cope with cognitive problems - not retrain cognitive skills. The rehabilitation programme was therefore relabelled as ‘cognitive management’ to better reflect the content and ethos of the programme. This is consistent with previous research which has demonstrated improved mood and self-reported memory problems, but no restorative cognitive effect following rehabilitation in MS [
]. Online cognitive screening and telephone-based support will be important in engaging stakeholders and in utilising clinic resources efficiently.
Stakeholders thought the pathway would help facilitate conversations about cognitive problems but recognised that implementation could increase referrals to psychological services. Our revised logic model now acknowledges this possibility as a ‘longer-term’ outcome.
Stakeholders also felt the impact of cognitive screening on the wellbeing of pwMS should be considered. Most pwMS valued the opportunity to discuss cognitive problems but acknowledged that some could feel anxious (about screening) and depressed (if the results indicated a problem). These potential outcomes will be monitored as part of NEuRoMS. Ultimately, stakeholders felt referral pathways could help optimise resources (staff can treat those most likely to benefit from their help), prevent mild cognitive problems becoming more severe, and prolong functional independence - all key priorities in managing long-term neurological conditions [
], we took a theory-based approach to intervention development to model the causal processes of the pathway. Conducting new primary research with a varied sample of stakeholders offers an in-depth and diverse understanding into how the pathway might work, recognising important mediating factors for implementation. The resulting pathway is now more relevant and theoretically sound, and therefore more likely to be implemented by stakeholders [
Nonetheless, our results are based on data gained from a research sample. Other stakeholders might offer further nuance to our understanding - for instance, we did not include IT specialists who would be responsible for data linkage from the screening results to the patient notes and ultimately integrating the pathway within existing IT systems. This, and insight from subsequent pathway piloting, will contribute to the iterative development of the logic model.
Our model is thus a blueprint that healthcare professionals could adapt to suit local circumstances, with local pathways being designed in the nexus of our logic model and local needs/resources. We view the training packages, screening tool, triaging and cognitive management programme (resulting in an integrated care pathway) as core elements to be retained across healthcare systems/pathways. However, availability and commissioning of resources will differ across healthcare models. For those low-income and middle-income countries where patients do not routinely receive disease modifying therapies, this might be prioritised in terms of funding [
Time matters in multiple sclerosis: can early treatment and long-term follow-up ensure everyone benefits from the latest advances in multiple sclerosis?.
J. Neurol. Neurosurg. Psychiatry.2018; 89: 844-850
]. Access to technology (e.g., tablet and Wi-Fi access for screening in-clinic) may also be limited here. Transferring the screening results to the clinical team will also require contextual strategies to integrate the results within existing record keeping systems (e.g., digital software, paper-based files) and ensure accessibility. Engagement mechanisms of timing screening to coincide with an upcoming appointment and providing the option of in-clinic support will also vary, with healthcare systems showing considerable variation in the frequency of consultations (e.g., quarterly versus annually [
]), which will influence the timing of appointments. Our model offers MS clinics the flexibility to work within the constraints of their systems and integrate existing successful practices.
5. Conclusions
Existing MS clinical pathways have not been co-constructed with stakeholders and are not based on robust evidence [
. We have developed a multi-stakeholder, co-constructed, clinical pathway for routine screening and management of cognitive problems in MS. Our stakeholders felt that introducing brief online screening tasks (with options to complete in-clinic or at home) and support for self-managing cognitive problems (including mild problems), would improve quality of life for pwMS and streamline NHS resources through earlier detection and intervention. Clinical staff will need to invest their time and require training, while pwMS should be supported to actively participate in the pathway. As part of the NEuRoMS programme, the clinical and cost-effectiveness of the pathway will be evaluated in clinical trials encompassing intervention fidelity, health economics, and process evaluations.
Funding information
This report is independent research funded by the National Institute for Health Research (Programme Grants for Applied Research, Neuropsychological Evaluation and Rehabilitation in Multiple Sclerosis – Developing, evaluating and implementing a clinical management pathway (NEuRoMS), RP-PG-0218–20002). The views expressed in this publication are those of the author(s) and not necessarily those of the NHS, the National institute for Health Research or the Department of Health and Social Care.
Disclosure
The Authors declare that there is no conflict of interest.
Data Statement
Anonymised data that support the findings of this study are available from the corresponding author upon reasonable request.
To facilitate reproducibility and date reuse, we also share our logic models (Figs. 1 and 2), coding schema (Supplementary Materials) and interview schedules (Supplementary Materials).
Time matters in multiple sclerosis: can early treatment and long-term follow-up ensure everyone benefits from the latest advances in multiple sclerosis?.
J. Neurol. Neurosurg. Psychiatry.2018; 89: 844-850
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