Developing a fatigue programme: Protocol for the Nottingham Fatigue After Stroke (NotFAST2) study

Abstract

Introduction

Post-stroke fatigue is common and is one of the most distressing symptoms after stroke. It has a negative impact on physical, social and psychological functioning; it is also associated with poor outcomes and increased mortality. The effective management of post-stroke fatigue is therefore regarded as a clinical priority.

Method

This study will use a mixed-methods design with three overlapping phases. Phase 1 will be a survey of existing fatigue management within the United Kingdom. In phase 2, interviews and focus groups will be conducted with stroke survivors with fatigue, carers and clinicians to determine strategies used to manage fatigue successfully. In phase 3, data from phases 1 and 2 will contribute to the co-design of a fatigue management programme with the NotFAST2 study Patient and Public Involvement group. This will be further refined through subsequent focus groups, which will include those with fatigue associated with other health conditions.

Results

Survey data will be analysed using descriptive statistics. Interview and focus group data will be analysed using a framework approach.

Conclusion

Post-stroke fatigue requires a comprehensive management programme necessitating input from key stakeholders. A post-stroke fatigue programme will be developed which will be tested in a future randomised controlled trial.

Keywords

Protocol post-stroke fatigue fatigue management stroke self-management community

Introduction

Post-stroke fatigue (PSF) is an ‘overwhelming feeling of exhaustion or tiredness’ that is not related to exertion, and that does not typically improve with rest (De Groot et al., 2003). PSF is common, and affects 50% of people after their stroke (Cumming et al., 2016; Hinkle et al., 2017). It is distressing for the stroke survivor (Van der Werf et al., 2001), is linked to poor outcomes such as disability, depression, lower quality of life (Lerdal and Gay, 2017) and increased mortality (Glader et al., 2002), and contributes to significant carer burden (Mandliya et al., 2016). PSF can occur in those who have made an otherwise good recovery, can occur early after stroke or later (Hawkins et al., 2017) and can affect all aspects of daily life (Worthington et al., 2017). However, the effect of fatigue is most likely to be noticed when people are discharged from hospital and are back in their own homes. The management of PSF is recognised as an ongoing national and international research priority (Norrving et al., 2018).

Although researchers have endeavoured to explain PSF mechanisms (De Doncker et al., 2018), these remain unclear. This may be due, in part, to the lack of a single accepted and widely adopted definition of post-stroke fatigue (Barker-Collo et al., 2007; Thomas et al., 2019) and also because of misunderstanding around the use of key terms such as fatigue and tiredness (Thomas et al., 2019). It is known that there are many contributing factors (Aali et al., 2020; Cumming et al., 2016; Mandliya et al., 2016) and it would seem that each previous research team has concentrated on specific issues: exercise, sleep, nutrition, medication and psychological interventions. In an attempt to evaluate the overall evidence, several comprehensive reviews (Aali et al., 2020; Hinkle et al., 2017) and Cochrane systematic reviews (Kennedy and Kidd, 2018; Wu et al., 2015) have compared findings from randomised controlled trials (RCTs) and clinical studies of PSF to draw overarching conclusions. All the reviews to date have concluded that there is insufficient evidence of the efficacy of the tested interventions in trials, and that more robust research with adequate sample sizes is required. For example, in the review conducted by Wu et al. (2015) the authors reported a pooled standardized mean difference (SMD) of –1.07 (95% confidence interval (CI) –1.93 to –0.21) in favour of reducing fatigue with interventions in the studies they included. However, this benefit was not found when data from the more rigorous studies were examined: SMD –0.38 (95% CI –0.80 to 0.04) with adequate allocation concealment and SMD –1.10 (95% CI –2.31 to 0.11) with appropriate blinding of assessors. The results from the studies and reviews demonstrate the need for more research into PSF and underline the need for this research to be more robust and better designed.

Current clinical practice guidelines rely on low levels of evidence, such as expert consensus, to make recommendations for PSF. There are therefore few specific recommendations in United Kingdom (UK) (Royal College of Physicians, 2016) or other international clinical guidelines (for example Australia: National Stroke Foundation, 2010) for its management. Broadly, international expert consensus supports assessment and information provision, the provision of rehabilitation during periods of lower fatigue, pacing of activity, exercise and advice on sleep (Lanctot et al., 2019). It is suggested that this improves the outcome and quality of life of stroke survivors as well as reducing caregiver burden.

In the absence of definitive guidance, and with an incomplete evidence base, our aim is to conduct a study to develop a fatigue management programme for stroke survivors with PSF. We will not concentrate on one aspect of management but instead will take a comprehensive but pragmatic view and collect data from experts: stroke survivors, carers (that is, unpaid carers who are likely to be family or friends) and clinicians. We will collect data on both PSF strategies and on approaches used in fatigue management more broadly. We have conducted a comprehensive scoping review (Aali et al., 2020) on PSF to inform our research proposal. The specific objectives are to:

Understand and document existing key management practices for PSF.

Gain insights from the experiences and expertise of those who have fatigue, those who have had help to manage it or those who offer support.

Utilise findings to co-design a community post-stroke fatigue management intervention.

We envisage that the proposed programme could be evaluated in a future, appropriately powered randomised controlled trial.

Methods

Ethics

Ethical approval was granted by University of Nottingham, Faculty of Medicine and Health Sciences Research Ethics Committee (Reference 480-2001, 10 March 2020).

Design

This is a mixed-methods study, with three overlapping phases.

Phase 1: survey

We will conduct a survey of fatigue management provision within the UK. We will particularly focus on stroke but will collect relevant data on other conditions where fatigue is a key symptom (for example multiple sclerosis, rheumatoid arthritis). We will examine eligibility criteria for referral to a health professional or a service for fatigue management and examine the format and content of interventions currently available. We will collect data on whether fatigue assessments are routinely used, whether there is any routine follow-up provided and whether there are any differences in management for post-stroke fatigue as compared to fatigue management in other conditions.

Data will be collected by contacting allied health professionals (AHPs), psychologists, doctors and nurses through professional networks, special interest groups and via social media. We will ask colleagues to forward details of the survey to others with an interest in fatigue (snowballing method). The research team will also use professional contacts to alert clinicians to the survey.

Participants will be eligible if they are clinicians from hospital or community fatigue services or who have an interest/expertise in fatigue management in PSF or in other long-term conditions. The survey will be available to complete online or can be returned by post. The aim will be to obtain as many responses as possible over a 3-month period.

Phase 2: interviews and focus groups

Interviews and focus groups will be conducted with stroke survivors with fatigue, carers and clinicians about their experiences and strategies used to successfully manage fatigue. We will use both face to face interview and focus groups because both formats have been found to provide different and enriched data (Lambert and Loiselle, 2008) and also provide participants with choice. Participants will either participate in focus groups or semi-structured interviews.

For stroke survivors and carers, we intend to use a multi-pronged approach to advertising the study locally and regionally: posters and flyers in the community and in GP surgeries, community hospitals, stroke units, outpatient departments, local stroke clubs, community centres and through existing national UK patient networks (such as stroke lay conferences), and through social media. A maximum variation sampling strategy (Palinkas et al., 2015) will be used to ensure we capture a broad range of experiences such as age, gender, time since stroke, ethnicity and geographical location. We want to be as inclusive as possible, and we will therefore include people with aphasia.

Interviews and focus group discussions, based on participant preference, will be conducted with stroke survivors with fatigue, carers and clinicians. We expect to achieve data adequacy (based on similar previous studies) by conducting interviews with up to 20 stroke survivors with fatigue, 10 carers and 20 clinicians. However, our sample size will ultimately be guided by achieving data saturation. Two focus groups with six to eight participants in each will be conducted. Participants will be eligible if they are willing to give written informed consent and meet the following study criteria:

Stroke survivors: have a diagnosis of stroke, are aged 18 years or over and have (or have had) self-defined PSF. They will be excluded if they have a previous significant history of fatigue before stroke, as defined by themselves.

Carers: lives with and provides care (physical or psychological) for a stroke survivor with fatigue and is 18 years or over. These are non-paid carers who are likely to be family members or friends.

Clinicians: are a registered nurse, doctor, psychologist or AHP, working in a service providing fatigue management and/or have a recognised interest/expertise in fatigue management broadly and/or in post-stroke fatigue specifically. We will include international experts where possible for the interviews.

All interviews and focus groups will be semi-structured and will use interview and focus group topic guides with key issues relating to fatigue informed by our recent, wide ranging review of the relevant literature (Aali et al., 2020). The interviews will be conducted remotely via Skype/video conference/telephone or face to face, depending on location, time availability and preferences of participants and the research team. Focus groups will be held in an easily accessible location such as on university premises or using virtual groups using telephone or video conferencing. In order to keep an accurate record, all interviews and focus groups will be audio recorded, transcribed verbatim and anonymised. The research team will also produce a contemporary field note after completion of each interview and focus group.

For stroke survivors and carers our focus will be to collect data on: individual experiences of having fatigue; history and onset of fatigue; characteristics of the fatigue; what helped and what made it worse; and exploring any professional support and/or information and whether it was useful or not. We are also keen to determine what, in the participants’ opinions, should constitute best practice for addressing PSF in the absence of clinical recommendations. In any instance where we have a survivor–carer dyad, we will not have both in the same group as previous experiences have suggested this may prevent individuals from speaking freely.

We will also interview 20 clinicians with an interest or recognised expertise in fatigue. We have opted largely for interviews for this group because it may be difficult to bring clinicians together for focus groups. However, we will endeavour to do this as well. We also want to include those with expertise in other fatigue-related conditions (such as MS) and international experts. We will recruit via targeted personal emails and will aim to get a balance of different professionals, different condition expertise, and national and international views. For professionals, our focus will be to collect data on whether participants work as part of a fatigue service or not, their thoughts and experiences of specific advice and interventions, and their use of the fatigue research evidence.

Phase 3: development of fatigue management programme

In this phase, data and findings from key research literature (specifically from the most recent reviews, that is, Aali et al., 2020; Hinkle et al., 2017) and from phases 1 and 2 of the study will help guide priority-setting in the co-design of a new fatigue management programme. We will determine what should constitute best practice for addressing PSF in the absence of research-based clinical recommendations in terms of the programme content, programme format, overall design and presentation. Our co-design is informed by an experience-based co-design (EBCD) methodology utilising the findings from earlier phases to trigger ideas and solutions for the new fatigue management programme and inform individual and joint priority-setting (King’s Fund, 2018). Ideas on preliminary content and delivery will be explored through focus groups with stroke survivors, carers, people with fatigue from other conditions and clinicians, and further refined through staged discussions with the study Patient and Public Involvement (PPI) group.

Using a co-design approach, we will aim to take an equitable approach to priority-setting and intervention design aiming for a balance and ratio of professionals, stroke survivors and carers. We will also include those with experience of fatigue from other health conditions (for example multiple sclerosis, rheumatoid arthritis) where the literature suggests the features of the presenting fatigue are similar to those in stroke or where services seem to be addressing similar symptoms. We will be flexible, depending on the data collected, as to how the groups are configured. We may also recruit some participants from phase 2 to take part in these focus groups. These participants would be selected on the basis that they had key experiences or novel ideas around fatigue management. The overall aim is that the participants will have the opportunity to shape the programme and consider how it might be best delivered.

Informed consent

In phase 1, completion and return of the questionnaires (either online or by post) will imply informed consent to participate. Each questionnaire will have a front page detailing information about the study and contact details for obtaining further information. A separate information sheet will also be provided.

In phases 2 and 3 all participants will provide informed consent. The researcher will explain the details of the study and provide a participant information sheet, ensuring that the participant has sufficient time to consider participating or not. The researcher will answer any questions that the participant has concerning study participation.

No personal information will be collected until participants have given their consent. For patients who are physically unable to sign the consent form (for example weakness in dominant hand due to stroke), consent will be given using a mark or line in the presence of an independent witness (who has no involvement in the study), who will then corroborate by signing the consent form.

An accessible information sheet will be provided to those with aphasia. This will follow standard aphasia-friendly principles, with one idea presented per page in short simple sentences in a large font. Key words will be emboldened and each idea will be represented by a pictorial image to support understanding of what the study is about. The researcher will be trained to support understanding further by reading parts of the information aloud and using supportive gestures/actions. Participants who have used the accessible information provision will be provided with an aphasia-friendly consent form and asked to initial all boxes before signing.

Analysis

Data from the survey will be analysed using SPSS and Excel using descriptive statistics. Free text data will be analysed using broad categories and used to illustrate the quantitative data.

Qualitative interview and focus group data will be analysed separately, managed using NVivo software, and will use a framework approach (Gale et al., 2013). A working analytical framework will be developed simultaneously and iteratively with the coding stage, and data chunks will be transferred onto a framework matrix, which will also be informed by theory.

Study management

The chief investigator will have overall responsibility for study management and will oversee all study management. A study steering group, comprising the co-investigators and PPI members, will oversee the study.

Discussion

PSF can have significant consequences for stroke survivors and their recovery: it is not simply a trivial, unimportant symptom. Despite recognition of the need to manage PSF appropriately, it is accepted that the current evidence base around PSF and its management is weak, which may be because previous research has examined potential individual contributing factors rather than focusing on the broader issues. However, given the complex, multi-factorial profile of PSF, a single therapy approach for the management of PSF is unlikely to be appropriate. We therefore have suggested that, given PSF is multi-faceted, it requires a comprehensive management programme. Involving experts (that is, stroke survivors, carers, people with fatigue arising from other conditions, and clinicians working with those with PSF and with fatigue from other conditions) in the co-design of such a programme is vital as they can share the strategies that they believe are successful, from a concrete and informed perspective.

Using experts, that is, both patient and professional experts, in the programme development also has additional benefits. Aside from offering legitimacy to the programme, including a range of participants and a mixed-methods design ensures that evidence is collected from several sources and that key issues are triangulated. Moreover, having a programme that is co-designed provides the best opportunity to develop a programme that is practicable, accessible, which people will engage with and which can be sustained over the longer term. Such a programme also has the added benefit that, in drawing on the experiences of experts, there is the potential to enable stroke survivors to self-manage their own fatigue. If individuals have belief in their capability (self-efficacy) to manage their own condition, this can have positive impacts.

The research is novel given the fact that it uses key stakeholders in the development of the programme. It also uses a mixed-methods design that incorporates survey, interview, focus group and co-design methodologies in order to be able to draw data and findings together from a range of credible sources. It also draws on both knowledge of PSF techniques as well as from non-stroke fatigue sources. However, as with any proposed research study, there could be difficulties in conducting the study. It is possible that there is not a satisfactory response to the survey, which would have implications for the generalisability of our results. It is also possible that the required numbers of participants in the second phase are not achieved, or that we are not successful in including a diverse range of participants. Both of these would limit the value of our results.

It is our intention that this research will lay the foundations for conducting a future definitive RCT to test the efficacy of the programme and to determine whether stroke survivors could indeed self-manage their fatigue. If management of PSF were possible, it would potentially reduce its impact and the concomitant effects on recovery and quality of life. Although our proposed RCT will be conducted in the UK, the results would be relevant worldwide as the issue of managing post-stroke fatigue appropriately is a global issue, as reflected in current national and international clinical guidelines and in the fatigue literature.

Key findings

We will identify successful strategies to be used in developing a comprehensive fatigue management programme.

This programme will need to be fully evaluated in future studies and trials.

What the study has added

This protocol details proposed research to develop a comprehensive programme to manage post-stroke fatigue (PSF) using expert input from key stakeholders to shape the proposed intervention.

Footnotes

Acknowledgements

The authors wish to convey their thanks to the Nottingham Stroke Research Partnership Group, who discussed the study and gave valuable comments during the development of the protocol. Thanks also to our own PPI members.

Research ethics

Ethical approval has been granted by the University of Nottingham, Faculty of Medicine and Health Sciences Research Ethics Committee (Reference 480-2001, 10 March 2020).

Consent

In phase one, return of a completed questionnaire (by post or on line) will imply that the participant has given their informed consent to participate. In phases two and three, all participants will provide written informed consent to participate in the study. For any participant unable to sign (e.g. due to hemiplegia), informed consent will be given using a mark or line in the presence of an independent witness who will corroborate this by also signing the consent form. For any participant who has aphasia, an aphasia friendly information sheet and consent form will be provided, and they will be asked to sign this.

Declaration of conflicting interests

The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Contributorship

AD is the Principle Investigator. AD, ST and RN conceived the initial study and the design was further developed with input from AD, JA, LC, RN, AJ, FJ, NS, and ST. AD wrote the first draft of the protocol, and all authors commented on, and edited, this. All authors read and approved the final manuscript.

Funding

The authors disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This study is funded by the UK Stroke Association. Ref SA PG19\100060.

ORCID iDs

Avril Drummond

Joanne Ablewhite

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