Patient Perception, Preference and Participation

Development and preliminary evaluation of a cognitive behavioural approachto fatigue management in people with multiple sclerosis

S. Thomas a,*, P.W. Thomas a,b, A. Nock c, V. Slingsby c, K. Galvin b, R. Baker a,d, N. Moffat d, C. Hillier c

a Dorset Research and Development Support Unit, Poole Hospital NHS Foundation Trust, Poole, Dorset, UKb The School of Health and Social Care, Bournemouth University, Bournemouth, Dorset, UKc Dorset MS Service, Poole Hospital NHS Foundation Trust, Poole, Dorset, UKd Dorset HealthCare NHS Foundation Trust, Bournemouth, Dorset, UK

Patient Education and Counseling 78 (2010) 240–249

A R T I C L E I N F O

Article history:

Received 18 September 2008

Received in revised form 29 June 2009

Accepted 3 July 2009

Keywords:

Fatigue

Multiple sclerosis

Intervention

Cognitive behavioural

Energy conservation

Self management

Group-based

A B S T R A C T

Objectives: (i) To develop a group-based intervention for the management of multiple sclerosis (MS)

fatigue incorporating energy effectiveness and cognitive behavioural approaches and (ii) to undertake a

process and preliminary evaluation.

Methods: Drawing upon a literature search, a local model of good practice and the views of service users

and health professionals, a manualised group-based fatigue management programme was developed,

designed to be delivered by health professionals. A process and preliminary outcome evaluation was

undertaken. Sixteen participants attended across two iterations. Participant feedback, obtained via a

focus group and evaluation questionnaires, was used to refine the programme. Outcomes were collected

pre- and post-programme (including fatigue severity, quality of life, self-efficacy).

Results: Focus group feedback suggested the programme was well received, reflected in high attendance

and positive ratings on evaluation questionnaires. At follow-up, despite the small sample size, there

were significant improvements in perceived self-efficacy for managing fatigue.

Conclusion: An evidence-based fatigue management intervention has been developed and preliminary

findings look promising. In the next phase we will examine whether the programme transfers

satisfactorily to other centres and collect data in preparation for a randomised controlled trial (RCT).

Practice implications: Implications for practice will emerge when the results of our RCT are published.

� 2009 Elsevier Ireland Ltd. All rights reserved.

Contents lists available at ScienceDirect

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1. Introduction

Multiple sclerosis (MS) is a chronic, unpredictable, incurable,demyelinating disease of the central nervous system affectingapproximately 2.5 million people [1–3]. It is more common inwomen and onset peaks between 20 and 40 years. The causes andearly development of the disease are not fully understood butprobably involve immune, genetic, and environmental factors[2,3]. Fatigue is one of the most commonly reported and disablingsymptoms of MS, often occurring daily [4] and with a variablecourse [5,6]. Up to 86% of individuals with MS experience fatigue atany one time; 65% consider it to be one of their three mosttroubling symptoms [4].

Fatigue has been defined as ‘‘a subjective lack of physical and/ormental energy that is perceived by the individual or caregiver tointerfere with usual or desired activities, p. 2’’ [7]. The pathophysiol-

* Corresponding author at: Dorset Research and Development Support Unit,

Poole Hospital NHS Foundation Trust, Longfleet Road, Poole, Dorset BH15 2JB, UK.

Tel.: +44 (0) 1202 448617; fax: +44 (0) 1202 448490.

E-mail address: sarah.thomas@poole.nhs.uk (S. Thomas).

0738-3991/$ – see front matter � 2009 Elsevier Ireland Ltd. All rights reserved.

doi:10.1016/j.pec.2009.07.001

ogy of MS-fatigue is unclear [8] but likely to be multi-factorial [9,10].Findings on the relationships between fatigue and other clinicalvariables (such as age, gender, disease duration and clinical activity)have been equivocal [11]. Fatigue may limit or prevent participationin everyday activities, work, leisure and social pursuits, restrict rolefulfilment and reduce psychological well-being [12,13] and is one ofthe key precipitants of early retirement [13,14]. Its ‘invisible’ naturemay lead to difficulties in personal and work relationships [15,16].

Fatigue is highly related to an individual’s sense of control overMS and psychological well-being [12,17,18]. Sense of control hasbeen found to predict lower fatigue severity and chronicity [19],suggesting that increasing self-efficacy related to fatigue couldimprove quality of life (QoL).

Studies exploring the effectiveness of energy conservationprogrammes for MS-fatigue have tended to be small and uncon-trolled [20–24]. Two Occupational Therapy (OT) based fatiguemanagement initiatives have been developed in the UK [25,26]. Onlythe former has been evaluated and numbers were small.

A randomised controlled trial (RCT) [27] of an energyconservation course [28] found evidence for its effectiveness inreducing fatigue impact, increasing self-efficacy and aspects of

S. Thomas et al. / Patient Education and Counseling 78 (2010) 240–249 241

QoL; benefits were maintained at 1 year [29]. Results from aGerman adaptation were also promising [30]. A RCT that evaluateda multi-disciplinary fatigue management programme [31] demon-strated no reduction in fatigue impact compared with a placebointervention.

Although the important relationships between physical andpsychological aspects of MS-fatigue are recognised [32], highquality RCTs of psychological interventions are rare [33]. InChronic Fatigue Syndrome (CFS) individual Cognitive BehaviouralTherapy (CBT) is effective in treating fatigue [34,35]. A systematicreview [36] of psychological interventions for MS identified justone RCT that used a CB approach to manage MS-fatigue [37]. Theintervention consisted of individual CBT conducted by a clinicalpsychologist and was shown to be effective in reducing fatigue.However, in the UK National Health Service (NHS), and elsewhere,psychologists working with people with MS are scarce, thus thisapproach may prove impractical. A number of pilot studies haveused group-based CB approaches, either in the context of copingwith MS [38–40], or specifically with MS-fatigue [41]. Group-based

Fig. 1. Research

approaches are potentially more cost effective than one-to-oneand offer opportunities for peer support.

Locally, a group-based fatigue management interventiondelivered by OTs that incorporates a CB approach has been foundhelpful in CFS [42]. The current research draws upon this model,using health professionals routinely involved in the managementof MS, supported by clinical psychologists, to facilitate theintegration of psychology-based approaches into patient care.

The aims of this research were:

1. T

flo

o develop and manualise [43] a group-based intervention forthe management of MS-fatigue blending CB and energyeffectiveness approaches that can be integrated into existingNHS structures (Phase 1).

2. T

o undertake a process and preliminary evaluation [44–46](Phase 2).

Mixed methodologies were used [47], following the MedicalResearch Council (MRC) guidance for developing and evaluating

wchart.

S. Thomas et al. / Patient Education and Counseling 78 (2010) 240–249242

complex interventions [44]. An overview is presented in Fig. 1.Separate ethical approval was obtained from the Local ResearchEthics Committee for Phases 1 and 2. Poole Hospital NHSFoundation Trust provided governance approval.

2. Phase 1: adaptation and development of the fatiguemanagement programme

The programme draws upon a local model of good practice inCFS [42]. The content required adaptation to reflect current theoriesabout the causes and treatment of MS-fatigue. To ensure theintervention was grounded in the literature, a search of relevantpapers and books was conducted. In addition, the views of peoplewith MS were sought via focus groups, and those of health pro-fessionals via an Intervention Development Advisory Group. Thesethree strands are described below. A Senior Research Psychologisttook the lead in developing the programme, and producingstandardised facilitator and participant materials. The process fordeveloping the programme was iterative, returning to the literatureand the Intervention Development Advisory Group as required.

2.1. Literature review

The multi-stranded literature review encompassed:

� P

sychological/OT interventions in MS [36,24] and other chronicconditions [35,48–51]. � G uidelines/self-help materials about MS-fatigue [7,52–54]. � B ooks about group approaches [55–60]. � M anualised CB approaches in chronic conditions [61–67]. � M anualised fatigue management for MS [25,26,68] and other

chronic conditions [42,69].

� In ternet-based CBT interventions [70].

The programme was based upon a conceptual frameworkdrawing upon cognitive behavioural [71–74], social cognitive[75,76], self-efficacy [77,78] and self-management [79,80] the-ories.

2.2. Observing a local model of good practice

The Dorset CFS Service has developed and evaluated a group-based Lifestyle Management Programme [42] grounded in Cox andco-worker’s model [81,82]. It is delivered by OTs and comprises six2 h sessions plus several review sessions. A research team memberobserved one programme iteration. While much was relevant to MS,certain modifications were required (e.g. the underlying objective ofa return to a near normal lifestyle was not deemed appropriate).

2.3. Intervention Development Advisory Group

Members of this multi-disciplinary group (including OTs,clinical psychologists, neurologist, service users) met regularlyand contributed to the theoretical and practical aspects ofprogramme development.

2.4. Focus group

Once a provisional structure for a six session Fatigue Manage-ment Programme was developed, focus groups were held to elicitfeedback and inform programme development.

2.5. Methods

Participants had a clinically definite diagnosis of relapsing–remitting or progressive MS [83–85] and had received guidance on

energy effectiveness in their own home from an OT. They were sentgeneral information about focus groups, an outline of the proposedprogramme and a topic guide.

The 90 min focus group (led by an experienced facilitator,supported by a co-facilitator) included the following topics:

� E

xperiences/impact of fatigue. � C oping with the invisibility of fatigue. � E xperiences of receiving fatigue management advice. � H elpful versus unhelpful fatigue management strategies. � A ttitudes towards scheduled rests. � E xperiences of using a fatigue diary. � O pinions about the proposed programme. � P ractical aspects.

Discussions were audio-recorded and transcribed verbatim.Data were analysed qualitatively by reading and re-reading thetranscript to identify emergent themes [86].

2.6. Results

Of 11 people invited, 8 provided informed consent and attended(5 with relapsing–remitting and 3 with secondary progressive MS;6 female). All had mean Fatigue Severity Scale (FSS) [87] scores >4(x ¼ 5:9; range: 4.4–7.0) indicating significant fatigue. Mean agewas 44 years (range: 39–54 years) and mean time since diagnosiswas 6 years (range: 1.5–13 years).

One of the salient emergent themes was the variable andoverwhelming nature of MS-fatigue. It was often a major limitingfactor in participants’ lives and poorly understood by others (Box1). Fatigue, and the strategies employed to manage it, often had anemotional impact.

Participants were satisfied with the proposed number andduration of sessions in the programme and felt that holding it at anaccessible, ‘‘neutral’’ venue was important. There was a generalpreference for morning sessions (when levels of fatigue are oftenlower [88]).

3. Draft fatigue management programme

3.1. Content and structure

The programme objectives are (i) to normalise the experience offatigue; (ii) to learn how to use available energy more effectively;and (iii) to develop ‘‘helpful thinking styles’’ about fatigue. Adetailed breakdown of content is provided in Table 1.

The programme comprises six 90 min weekly sessions (with amid-session break) and is designed to be delivered by two healthprofessionals with experience in MS (such as OTs, nurses,physiotherapists) in groups of 6–10.

Participants are encouraged to bring a Supportive Other toSession 1. This could help to foster and support lifestyle andattitudinal changes. Informal contact between participants outsidethe group setting is encouraged.

In the programme there is a gradual transition from a practicalto a more psychological orientation. However, CB elements areintroduced early, enabling participants to explore the reciprocallinks, before the CB model is formally introduced in Session 4. Eachsession follows the same general format; namely, facilitator-delivered presentations, flipchart discussions, group activities andhomework.

3.2. The manual

The facilitator manual (�100 pages) provides detailed sessioncontent, guidance on preparation and delivery, a checklist of

Box 1. Phase one focus group.

(a) Topics explored and examples

Variability and impact ‘‘I don’t think you can . . . you can’t describe fatigue. It’s different every day, it’s different for

every part of your body . . . Psychologically it can bring you down’’

Invisibility ‘‘Well, my family forget . . .. I think they genuinely forget coz I just walk into the house as I am.

I haven’t got a label on my head, they just forget all the time & I just sit there & think ‘oh I’m

so tired’’’

Emotions ‘‘I feel worse, if I rest I feel worse. I feel guilty coz I haven’t done anything that day’’

Practical issues

� Activity diary ‘‘For me using the diary it was really good because I just didn’t really think about it, didn’t take

a step back and sort of quantify when my fatigue times were, but it enabled me to identify

when I’m gonna feel fatigued or worse than others then plan my time accordingly

and it’s all individual’’

� Involvement of Supportive Other ‘‘I think it’s a very good idea for your partner to come along, if you have a partner that is.

For information and feedback so they understand more about what’s going on’’

� Group format ‘‘That’s why I think a group like you’re planning is good because then a lot of MS people don’t

get to see any other MS people . . . and you sort of think ‘oh it’s just me’ so if you get a group

of people together and you’re all having the same sort of symptoms and you

can all talk about what’s going on . . .’’

� Is programme appropriate for newly diagnosed? ‘‘Newly diagnosed they should be aware about the symptoms and understand why they’re

tired . . . but it’s got to be timed at the right time for that individual person’’

(b) Examples of fatigue management strategies described by participants

Learning to say ‘no’ to others’ demands ‘‘It’s the added things, the unexpected things that happen that you didn’t plan for and

then learning to say ‘no’ to that’’

Pleasant activity scheduling ‘‘. . . you’ve just got to try and, you know, keep your energy for things that you enjoy . . . and if

you’re just kind of going round in circles getting tireder & tireder & you know doing things

which someone else can do it’s stupid really but it takes you a while to work that out’’

Planning ‘‘Planning my day rather than sort of going for as long as I can and putting myself on

my knees before I succumb to rest’’

Prioritising tasks and activities ‘‘And there’s a whole lot of things in lives that you kind of, you know, they can wait till

tomorrow, they can wait till next week and it’s sort of getting that mentality’’

Incorporating scheduled rests, naps or relaxation periods ‘‘that’s why I use a personal CD player so I can actually block everybody else out. . . . and

people are quite nice they just leave you be because they think ‘Oh, they’re listening’

and it just means that you can just, you don’t have to cope with any conversation’’

Delegation ‘‘I think it’s also using your support network as much as you can really’’

S. Thomas et al. / Patient Education and Counseling 78 (2010) 240–249 243

facilitator objectives and signposts to additional resources.Sessions are delivered via PowerPoint; hence can be easilyreplicated. A companion participant handbook (�100 pages),along with existing information booklets [53,54], reinforcesprogramme content.

4. Phase 2: process evaluation and preliminary outcomeevaluation

Aims: (a) to collect outcome data from participants 1 week pre-and 1 month post-fatigue management programme; (b) to conducta preliminary test of the hypotheses that the programme increases

Table 1Description of the six session fatigue management programme.

Session Title Content

1 What is MS-related fatigue? General introduct

types of fatigue; c

model of fatigue i

2 Opening an ‘‘energy account’’ Rest – functions;

types and techniq

exercise; sleep hy

3 Budgeting energy and smartening

up goals

Types of activity;

moderating activi

factors; goal settin

4 The stress response; Introducing

the cognitive behavioural model

The stress respons

with stress; introd

5 Putting unhelpful thoughts on trial Unhelpful though

thoughts; levels o

6 Recapping and taking the programme

forward

Revisiting expecta

group activity to r

rationale of ‘Keep

self-efficacy and QoL and reduces fatigue; and (c) to obtainfeedback from participants via semi-structured evaluation ques-tionnaires and a focus group.

4.1. Methods

4.1.1. Participants

Inclusion criteria: (a) �18 years; (b) diagnosis of MS [83–85];(c) mean FSS [87] score >4; (d) ambulatory with/without mobilityaid(s) assessed using the lower limb disability subscale of the Guy’sNeurological Disability Scale (GNDS) [89,90]; and (e) Englishspeaking.

Homework

ion; expectations; icebreaker (quiz);

ontributing factors, conceptual

n MS

Activity/fatigue diary

barriers, techniques; relaxation

ues; diaphragmatic breathing

giene

Rest/activity/sleep planner

balancing activity and rest;

ty using the toolbox; lifestyle

g

S.M.A.R.T. goal setting exercise

e (fight-or-flight); ways of coping

ucing the cognitive behavioural model

Unhelpful thoughts

(related to fatigue) diary

t patterns; challenging unhelpful

f belief

Thought challenge sheet

tions; introducing the ‘forcefield’;

evisit themes of the programme;

ing on Track’ planner

‘Keeping on Track’ planner

S. Thomas et al. / Patient Education and Counseling 78 (2010) 240–249244

Exclusion criteria: (a) judged by the OTs to have cognitivedeficits such that the group format would be unsuitable; (b)relapse in the last 3 months requiring hospital admission/corticosteroids; (c) taking pharmacological treatments for fatigue;(d) started on a disease modifying/anti-depressant drug therapywithin past 3 months; and (e) receiving treatment for a severemental health problem/addiction.

Identification and initial screening of potential participants wasundertaken by multi-disciplinary team members from a hospitalMS Service. Potential participants were sent information sheetsand asked to contact the study coordinator or return a reply slip ifthey wished to find out more.

One month pre-programme a second screening was undertakenand participants were sent the GNDS lower limb disability subscaleand the FSS to complete. Those for whom the programme wasconsidered unsuitable were seen, as required, on a one-to-onebasis by an OT in their own home (standard practice). Programmeplaces were allocated sequentially upon receipt of a completedconsent form.

4.1.2. Intervention and setting

The programme was delivered by two MS specialist OTs in ahotel meeting room.

4.1.3. Measures

One week pre- and 1 month post-programme participants weresent a booklet containing demographic questions (see Table 2), andthe primary and secondary outcome measures to complete over 1

Table 2Demographic and clinical characteristics of the sample.

Characteristics Iteration 1

(n = 7)

Iteration 2

(n = 9)

Total

(n = 16)a

Gender [n (%)]

Female 4 (57%) 7 (78%) 11 (69%)

Male 3 (43%) 2 (22%) 5 (31%)

Age (years)

Mean 45.4 45.6 45.5

S.D. 10.2 5.5 7.6

Range 34–59 38–53 34–59

Disease type [n (%)]

Relapsing–remitting (R–R) 4 (57%) 7 (78%) 11 (69%)

Secondary progressive 1 (14%) 2 (22%) 3 (19%)

Primary progressive 2 (29%) – 2 (12%)

Level of education [n (%)]b

Highest qualification achieved

One or more GCSE (or equiv.) 1 (14%) 1 (11%) 2 (13%)

One or more A level (or equiv.) 2 (29%) – 2 (13%)

First degree 1 (14%) 2 (22%) 3 (19%)

Vocational qualification 3 (43%) 6 (67%) 9 (56%)

Marital status [n (%)]

Married/cohabiting 4 (57%) 8 (89%) 12 (75%)

Single 2 (29%) 1 (11%) 3 (19%)

Divorced 1 (14%) – 1 (6%)

Time since diagnosis (years)

<1 year – 2 (22%) 2 (13%)

1–5 years 6 (86%) 2 (22%) 8 (50%)

6–10 years – 3 (33%) 3 (19%)

11–15 years 1 (14%) 2 (22%) 3 (19%)

16–20 years – – –

>20 years – – –

Percentages are rounded up to the nearest integer and, thus, may not sum exactly to

100%.a Data from iterations 1 and 2 have been combined.b The GCSE (General Certificate of Secondary Education) is the name of a set of

qualifications, generally taken by students at age 14–16 in England, Wales, and

Northern Ireland. The A level (advanced level) is a qualification in England, Wales

and Northern Ireland, usually taken at age 16–18.

week. Cronbach’s alpha (a) (with 95% C.I.) for outcomes is reportedfor the current sample.

4.1.3.1. Primary outcome measures. These are all self-reportedquestionnaire measures, specified a priori.

Fatigue. The 29-item Fatigue Assessment Instrument (FAI) [91]is an expanded version of the 9-item FSS [87]. Responses are madeon Likert-type scales ranging from 1 (completely disagree) to 7(completely agree). A total and four subscale scores can becalculated (see Table 3). Alpha for subscales ranged from 0.57(�0.01, 0.81) to 0.91 (0.81, 0.96) (pre) and from 0.68 (0.29, 0.85) to0.93 (0.84, 0.97) (post).

MS specific QoL. The MS Impact Scale (MSIS-29) [92,93]measures the physical (20 items) and psychological impact (9items) of MS on daily life. It uses 5-point Likert-type scales (from‘not at all’ to ‘extremely’) and focuses on quality of life in the last 2weeks. High scores indicate high impact. Alpha for the physicalsubscale was 0.92 (0.84, 0.96) (pre) and 0.90 (0.80, 0.95) (post) andfor the psychological subscale was 0.75 (0.47, 0.88) (pre) and 0.76(0.50, 0.88) (post).

General QoL. The SF-36 [94] (standard recall version) consists of36 items comprising eight subscales (see Table 3). Scores rangefrom 0 (lowest well-being) to 100 (highest well-being). In addition,a Physical Component Summary (PCS) and a Mental HealthComponent Summary (MCS) were calculated. Alpha for subscalesranged from 0.74 (0.42, 0.88) to 0.96 (0.92, 0.98) (pre) and from0.69 (0.33, 0.86) to 0.93 (0.82, 0.97) (post).

Self-efficacy – fatigue. The MS Self-Efficacy Scale (MSSE) [95](Control Subscale) consists of nine items modified for fatigue (byreplacing the phrases ‘MS’ and ‘MS symptoms’ with ‘fatigue’ –see below). High scores indicate high perceived control overfatigue. Alpha was 0.83 (0.64, 0.92) (pre) and 0.92 (0.84, 0.96)(post). The correlation between the two versions of the MSSEwas 0.83 (pre) and 0.79 (post). The preliminary validation of theMSSE (Control-fatigue adapted) subscale will be reportedseparately.

4.1.3.2. Secondary outcome measures. Fatigue. The Brief FatigueInventory (BFI) [96] measures severity of fatigue ‘last week’ (using11-point Likert-type scales; endpoints ‘no fatigue’ versus ‘fatigueas bad as it can be’) and level of interference in six domains (ratedon 11-point Likert-type scales; endpoints ‘does not’ versus‘completely’ interferes.). A Global Fatigue score was calculated.Alpha was 0.93 (0.84, 0.96) (pre) and 0.90 (0.80, 0.95) (post). Forthe severity subscale a was 0.81 (0.55, 0.92) (pre) and 0.92(0.82,0.97) (post) and for the impact subscale 0.94 (0.86, 0.97) (pre)and 0.87 (0.73, 0.94) (post).

Mood. The 14-item Hospital Anxiety and Depression Scale(HADS) [97] consists of anxiety (HADS-A) and depression (HADS-D) subscales rated on 4-point Likert-type scales. Higher scoresindicate increased severity. For HADS-D a was 0.84 (0.63, 0.92)(pre) and 0.74 (0.43, 0.88) (post) and for HADS-A was 0.52 (�0.01,0.77) (pre) and 0.64 (0.22, 0.83) (post).

Self-efficacy – MS symptoms. The MS Self-Efficacy (MSSE)Control Subscale [95] consists of nine items (rated on a scale from10 ‘very uncertain’ to 100 ‘very certain’) that measure confidencein ability to manage symptoms/cope with the demands of MS.Alpha was 0.91 (0.80, 0.96) (pre) and 0.92 (0.84, 0.96) (post).

4.1.3.3. Post session evaluation questionnaire. After sessions, parti-cipants were asked to complete a brief anonymised, semi-structured evaluation questionnaire containing eight items on a1–5-point scale measuring content, pace, format, duration andusefulness of sessions, and amount, level of difficulty, andusefulness of homework tasks. Anchor labels are shown inTables 4 and 5.

Table 3Primary and secondary outcome measures (n = 16).

Pre Post Mean diff (95% C.I.) t (d.f.) p value Effect size

(Cohen’s d)

FAI – I (fatigue severity)a 5.8 (1.0) 5.6 (1.1) 0.3 (�0.1, 0.7) 1.63 (15) 0.13 0.27

FAI – II (situation specific fatigue)a 4.5 (1.1) 4.5 (1.1) �0.02 (�0.6, 0.6) �0.07 (15) 0.94 �0.02

FAI – III (fatigue consequences)a 5.8 (1.0) 5.8 (1.1) 0.04 (�0.6, 0.7) 0.13 (15) 0.90 0.04

FAI – IV (responsiveness to rest/sleep)b 5.4 (1.1) 5.6 (1.5) �0.2 (�1.2, 0.8) �0.40 (15) 0.69 �0.14

FAI – totala 5.2 (0.7) 5.1 (0.8) 0.1 (�0.2, 0.4) 0.96 (15) 0.35 0.19

MSSE (Control Subscale – fatigue adapted)b 42 (12) 58 (17) �16 (�25, �8) �4.34 (15) 0.001 �1.13

MSIS-29 (physical)a 2.9 (0.9) 3.1 (0.8) �0.2 (�0.5, 0.1) �1.53 (15) 0.15 �0.26

MSIS-29 (psychological)a 3.0 (0.7) 2.8 (0.7) 0.2 (�0.1, 0.6) 1.31 (15) 0.21 0.33

MSIS-29 (total)a 2.9 (0.7) 3.0 (0.7) �0.1 (�0.4, 0.2) �0.54 (15) 0.60 �0.12

MSSE (Control Subscale – MS symptoms)b 44.9 (13.9) 61 (16) �16 (�24, �8) �4.08 (15) 0.001 �1.05

SF-36 (physical functioning)b 37.8 (22.6) 33.2 (24.7) 4.7 (�1.5, 10.8) 1.62 (15) 0.13 0.20

SF-36 (role physical)b 39.8 (23.4) 40.2 (22.5) �0.4 (�9.1, 8.3) �0.10 (15) 0.93 �0.02

SF-36 (bodily pain)b 58.7 (27.7) 54.7 (31.4) 4.0 (�13.3, 21.3) 0.49 (15) 0.63 0.14

SF-36 (general health)b 47.3 (24.3) 47.2 (21.2) 0.1 (�6.1, 6.3) 0.04 (15) 0.97 0.01

SF-36 (vitality)b 32.0 (15.1) 35.5 (14.6) �3.5 (�10.1, 3.0) �1.15 (15) 0.27 �0.24

SF-36 (social functioning)b 56.3 (26.2) 57.8 (27.0) �1.6 (�13.4, 10.3) �0.28 (15) 0.78 �0.06

SF-36 (role emotional)b 65.1 (32.4) 66.7 (24.9) �1.6 (�20.7, 17.6) �0.17 (15) 0.86 �0.05

SF-36 (mental health)b 64.1 (20.9) 60.6 (18.3) 3.4 (�7.2, 14.1) 0.69 (15) 0.50 0.17

SF-36 (PCS)b 34.6 (9.4) 33.6 (9.1) 1.0 (�2.5, 4.5) 0.60 (15) 0.56 0.11

SF-36 (MCS)b 43.3 (12.9) 43.8 (11.0) �0.6 (�7.6, 6.4) �0.17 (15) 0.87 �0.05

BFI (severity)a 6.4 (1.6) 5.7 (2.3) 0.8 (�0.2, 1.7) 1.67 (15) 0.12 0.21

BFI (impact)a 6.0 (2.2) 5.8 (2.2) 0.2 (�1.0, 1.4) 0.41 (15) 0.69 0.10

BFI (total)a 6.1 (1.8) 5.7 (2.1) 0.4 (�0.6, 1.4) 0.83 (15) 0.42 0.37

HADS (depression)a 7.4 (4.3) 6.8 (3.2) 0.6 (�0.9, 2.0) 0.87 (15) 0.40 0.15

HADS (anxiety)a 9.5 (3.1) 8.3 (3.4) 1.2 (�0.1, 2.5) 1.95 (15) 0.07 0.36

a Positive difference indicates improvement.b Negative difference indicates improvement.

S. Thomas et al. / Patient Education and Counseling 78 (2010) 240–249 245

4.1.4. Focus group

Led by an experienced facilitator (supported by a co-facilitator),this was held a week after the first iteration to elicit participantfeedback (recruitment process, practical arrangements, pro-gramme content/format/materials).

4.1.5. Data analysis

SPSS (Version 12.0) was used. Pre and post differences (with95% C.I.) in outcomes were compared using paired t-tests with a 5%

Table 4Mean rating scores on the post session evaluation questionnaires.

Session No. (%) of

participants

who attended

No. of

questionnaires

completed

x (S.D.)

Content: 1 = not v.

relevant; 5 = very

relevant

1 16 (100%) 16 4.9 (0.3)

1* 6 (*Supportive Others) 6 4.7 (0.5)

2 15 (94%) 15 4.9 (0.3)

3 15 (94%) 15 4.6 (0.8)

4 13 (81%) 12 4.8 (0.6)

5 14 (88%) 14 4.6 (0.7)

6 16 (100%) 15 4.8 (0.6)

1–6 combined – 87 4.8 (0.6)

Table 5Participant ratings of homework tasks.

Session Description of homework task x (S.D.); range,

Amount: too m

too little = 5

1 Activity/fatigue diary 2.9 (0.8); 2–5, n

2 Rest/activity/sleep planner 3.4 (0.5); 3–4, n

3 S.M.A.R.T. goal setting exercise 3.1 (0.5); 2–4, n

4 Unhelpful thoughts diary 2.8 (0.7); 1–4, n

5 Thought challenge sheet 3.2 (0.4); 3–4, n

1–5 combined All homework 3.1 (0.6); 1–5, n

2-tailed significance level. Table 3 contains 25 p values, thus weused Bonferroni’s correction to aid interpretation. Standardisedeffect sizes (Cohen’s d) have been calculated using ‘betweensubject’ variability as the denominator [98]. Pre and postdifferences in outcomes were compared for those who broughta Supportive Other versus those who did not using the interactioneffect in repeated measures ANOVA. As no major changes to theprogramme were made between the two iterations, data have beencombined.

Pace: 1 = too

slow; 5 = too

fast

Format: 1 = did

not work at all;

5 = worked well

Length: 1 = too

short; 5 = too

long

Usefulness: 1 = not

at all useful;

5 = very useful

3.2 (0.4) 4.8 (0.6) 3.0 (0.6) 4.5 (0.9)

3.0 (0.6) 4.7 (0.5) 2.8 (1.3) 4.8 (0.4)

3.1 (0.6) 4.6 (0.6) 3.0 (1.0) 4.7 (0.6)

3.3 (0.6) 4.7 (0.5) 3.0 (0.8) 4.5 (0.8)

3.3 (0.5) 4.3 (0.8) 2.8 (0.7) 4.7 (0.7)

3.1 (0.4) 4.2 (1.0) 2.9 (0.8) 4.5 (0.9)

3.3 (0.6) 4.8 (0.4) 3.0 (0.8) 4.8 (0.4)

3.2 (0.5) 4.6 (0.7) 3.0 (0.8) 4.6 (0.7)

n

uch = 1; Level: too difficult = 1;

too easy = 5

Usefulness: not at all

useful = 1; very useful = 5

= 15 2.9 (0.6); 2–4, n = 15 4.4 (1.0); 2–5, n = 15

= 11 3.3 (0.6); 3–5, n = 11 4.2 (1.1); 2–5, n = 13

= 12 2.9 (0.7); 2–4, n = 11 4.0 (1.0); 2–5, n = 11

= 12 2.8 (0.6); 2–4, n = 12 4.0 (1.0); 2–5, n = 12

= 11 2.5 (0.9); 1–4, n = 13 4.0 (1.1); 2–5, n = 11

= 61 2.9 (0.7); 1–5, n = 62 4.1 (1.0); 2–5, n = 62

S. Thomas et al. / Patient Education and Counseling 78 (2010) 240–249246

As before, data from the focus group were analysed themati-cally by repeatedly re-reading the transcripts to identify generalcategories [86]. Responses to structured questions on theevaluation questionnaires were summarised using descriptivestatistics. Facilitator feedback was discussed among the Interven-tion Development Advisory Group and changes made to manualcontent and wording, where necessary.

4.2. Results

4.2.1. Outcome measures

Table 2 presents participants’ demographic and clinicalcharacteristics. There were no statistically significant differencesin demographic or disease-related variables across iterations 1 and2 with the exception of ‘time since diagnosis’ (see Table 2). Acrossthe two iterations, nine participants attended all sessions and theremaining seven attended five. All attended Session 6. SixSupportive Others attended Session 1 (two mothers, three spouses,one relative).

Box 2. Phase two focus group (topics explored and examples).

Content and structure

� Relevance ‘‘I found it generally very us

� Cognitive behavioural approach ‘‘Unhelpful thoughts, yes you

did for sitting down doing no

� Use of a vignette ‘‘Coz you can relate to exam

� Balance of content ‘‘Yeah it wasn’t doom and g

over’ . . . it was quite positive

� PowerPoint format ‘‘You tend to get the image

Venue ‘‘It’s much better than the ho

Group format ‘‘I think it’s just a relief to fin

exactly the same problems a

Group size ‘‘If there’s a lot of people, ca

Facilitators ‘‘friendly and pleasant and w

Involvement of Supportive Others ‘‘I think it would be useful fo

we all feel. I would love them

got an idea that it’s not just

Homework ‘‘I think it was useful. And it

of your life, so if you know y

really it’s not that much of a

Questionnaire booklet ‘‘Yes, it meant you could pic

week then you’re bound to fi

Accompanying materials ‘‘I think it’s good because w

ourselves over what we’ve b

back up, so you can just rea

Recruitment

� Volume of information ‘‘Sometimes the more inform

� Conflict when deciding whether

or not to participate

‘‘I think my only problem is

always worried about going

difficulties because I don’t w

end it came down to the fac

useful for other people in th

Involving Supportive Others ‘‘The only thing that put me

significant other with us and

Strategies for managing fatigue ‘‘Before I used to battle with

I’ve realised that perhaps I c

just a case of accepting and

‘‘I think it helps you to expla

last a bit longer during the d

keep going basically. And it’

‘‘See I don’t feel as guilty as

some paperwork to back it u

Barriers to change ‘‘I think it’s just regular brea

the back of your mind . . .’’

‘‘I still find it very difficult to

Long term impact ‘‘I think it will because this i

what we’ve learned, so yes’’

‘‘Hopefully it should become

In this pilot research statistical power is limited (n = 16).Nonetheless, there were statistically significant improvements inself-efficacy that remained significant after applying Bonferroni’scorrection (Table 3).

Scores on the other scales did not show significant pre to postchange. Comparing change in outcomes between those whobrought a Supportive Other (SO) (n = 6) with those who did not(n = 10) showed one statistically significant difference (p = 0.03)(n.s. after Bonferroni correction). Participants who brought a SOshowed a 17 point improvement in the Social Functioning scale ofthe SF-36, compared to an 8 point decrease in those who did not.

4.2.2. Post session evaluation questionnaires

The mean rating for relevance aggregated across all six sessionssuggested that participants considered programme contentrelevant (94% of ratings either 4 or 5 – Table 4). In terms of‘usefulness’, 89% of ratings were either 4 or 5. Of the six SupportiveOthers who attended Session 1 across the two iterations, five ratedits usefulness as ‘5’ and one as a ‘4’ (see Table 4).

eful really, every session I sort of went away thinking ‘ah, that was good’’’

suddenly realise that you do feel guilty a lot more often than you thought you

thing and then learning to address that. I thought that was quite useful’’

ples like that . . .’’

loom, definitely . . . I didn’t walk away thinking . . . ‘oh it’s all

’’

in your mind and it will stay there’’

spital’’

d that you’re not the only one . . . that other people have got

nd well it just helps to talk about it with somebody that understands’’

n be quite . . . daunting then to talk’’

elcoming’’

r them to come to a group and see how it’s discussed and what

to be able to know and to come to a group, so that they’ve

me, that there are others that have this problem . . .’’

’s only for a few weeks, it’s not as if you’ve got to do it for the rest

ou’ve only got to do homework once a week for a few weeks,

hassle’’

k it up and do it when you wanted, yes so I think if you’ve got a

t it in but if you’ve got only a couple of days it’s more of a hassle’’

e’ve learnt quite a lot having the material there to just refresh

een over. It’s quite good, I think, having like a manual just for

d through it and just refresh yourself’’

ation you get the more daunting it is as well, isn’t it?’’

that because I’m not afflicted too badly; only with fatigue I’m

to an MS group because I don’t want to see people in real

ant to see how I might end up . . . I was torn . . .. I think in the

t that I thought it might be useful for me and also it might be

e future’’

off initially was that I thought that we had to bring a

I didn’t have one’’

it [fatigue], convinced that I could beat it . . . but since taking this course

an’t beat it, I can manage it but I can’t beat it so it’s not worth that . . . so it’s

seeing if you can do something to make it a little bit easier’’

in to others the reasons that you need to rest so that you can

ay. ‘‘The fact that you need to have structured rest breaks to

s easier to explain when it’s been explained to you.’’

I did with resting now. I feel a little better because there’s . . .

p, so to speak.’’

ks – it’s difficult because life takes over but you still know, it’s in

, you know, the frustration of not being able to do what I used to do’’

s a long term problem and if we can keep . . . on top of it through

second nature . . . theoretically, you shouldn’t have to refer to the paperwork.’’

S. Thomas et al. / Patient Education and Counseling 78 (2010) 240–249 247

Homework ratings indicated that, for the most part, partici-pants considered the amount of homework acceptable (97%of ratings between 2 and 4 inclusive), the level appropriate (95%of ratings between 2 and 4 inclusive) and that it was useful (71% ofratings either 4 or 5) (Table 5).

Evaluation questionnaire feedback suggested that some wouldhave preferred longer sessions. We increased session lengthslightly (Session 1 from 90 to 120 min and Sessions 2–6 from 90 to105 min) to permit a more relaxed pace and increased opportunityfor social interaction.

4.2.3. Focus group

The focus group was held after the first programme iteration.Six of seven participants attended. The topics explored, along withrelevant quotations, are presented in Box 2.

Participants reported that they found all sessions to be relevantand identified a variety of topics/skills as being the ‘most helpful’:

� E

ducation about MS-related fatigue and structured rest breaks. � B reathing exercises. � P acing – overcoming boom and bust patterns. � G oal setting – sense of achievement. � C hallenging negative thoughts. � M eeting others facing similar challenges.

Participants were positive about the venue (a local hotel),preferring it to the hospital (where parking is problematic). Therewas a general consensus that the CB components of theprogramme were helpful and relevant. Two participants felt thatthey would like more content on these aspects.

Participants valued the group context and considered themaximum viable group size to be 10–12. They reported feeling atease during sessions and were positive about the option of bringinga Supportive Other to a session. However, work commitments canmake it difficult for Supportive Others to attend sessions.

Finding time for the homework was sometimes challenging forparticipants; however, overall it was deemed important andworthwhile. One participant commented that it would be helpfulto receive individualised feedback.

There was evidence of lifestyle changes and altered attitudestowards fatigue and its management. Participants noted certainpractical and emotional barriers. They thought the programmecontent would have continued relevance.

5. Discussion

The fatigue management programme was well received andattendance was high. For some, it was the first time they had talkedto others with MS. Peer interactions were highly valued, evidentfrom the focus group transcripts, evaluation questionnaire feed-back and the sessions themselves. Participants were encouraged toexchange contact details and we observed this happening.

Introducing the CB components early, before formally introdu-cing the CB model in Session 4, seemed to work well. Asparticipants had already explored some of the reciprocal linksbetween the components (via flipchart exercises, ensuing discus-sions and homework) this eased the transition to a morepsychological focus. Having learnt about the CB model in thecontext of fatigue, participants can apply the principles to othersymptoms/aspects [48].

Despite the small sample size (n = 16), there was significantlyhigher perceived ability to control MS/MS-related fatigue 1 monthpost-intervention, and the effect size was large [98]. There were nosignificant differences on any of the other outcomes, and theestimates of effect size for these measures were generally small.However, the confidence intervals are wide and this pilot did not

include a control arm. Thus, a larger, more rigorous study is neededto estimate effect sizes more precisely. The main aims of these twophases were to develop and manualise the programme and assessthe feasibility and acceptability of the outcome measures inpreparation for a RCT. A potential criticism is that the outcomemeasures are all self-reported. In the RCT we will obtain anobjective measure of free-living activity using accelerometers [99].

In addition, in response to participant and facilitator feedback,we have refined the programme as follows: (i) adding anintroductory section to the participant handbook; (ii) providingmore detailed guidance for the unhelpful thoughts diary andthought challenge sheet (Sessions 4 and 5); (iii) providing threeadditional vignettes in the participant handbook (Session 5) toillustrate fatigue ‘vicious cycles’; and (iv) incorporating a relaxa-tion practice into Sessions 3–6.

Self-efficacy has been identified as an important predictor ofMS-fatigue [100]. Perceived self-efficacy can be considered interms of four sources of expectations [78]: During the programmeparticipants are encouraged to provide peer support, in addition tothat provided by the facilitators (verbal persuasion). Participantslearn ways to cope with stress and anxiety in the form of breathingexercises (emotional arousal), how to set realistic goals that can betried out as homework (enactive mastery experience), and hearabout others’ successes (vicarious experience).

A strength of this study is the rigour undertaken in developingthe fatigue management programme. Phases 1 and 2 relate to thedevelopment and modelling stages of the MRC framework [44]. Asnoted by Robinson et al. often new or modified interventionsproceed rapidly to the RCT stage with earlier, equally important,phases of development and evaluation overlooked [101]. Incor-porating these, has ensured the intervention, has a robusttheoretical underpinning, has been fully manualised, and hasundergone a thorough process evaluation. In the next stage we willpilot the intervention on one group in each of three centres to testits transferability and collect data in preparation for a RCT.

5.1. Practice implications

A potential strength of the intervention is that it can beintegrated into existing service delivery models. The possibility ofproviding interventions that have a sound psychological basisusing staff already working with people with MS, could facilitatethe integration of psychology-based approaches into patient care.Specific practice implications will emerge when the results of ourRCT are published.

Acknowledgments

This research was supported by grants from the MS Trust andthe BUPA Foundation. We would like to thank all participantsinvolved in this study. We would like to thank Caroline Chandler,Claire Stuttaford and the Dorset MS Service for their support. Wewould like to thank Dr Selwyn Richards, Michelle Selby andBarbara Goodchild from the Dorset CFS Service for permitting us toobserve the Lifestyle Management Programme and for theirexpertise. We would like to thank Dr Rosie Jones, Dr PaulaKersten, Geoff Linder and Tim Worner for their advice and input.We would like to thank Chris Richards and Dawn Stevens foradministrative support.

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