Charles Hillier

A pragmatic parallel arm multi-centre randomised controlled trial to assess the effectiveness and cost-effectiveness of a group-based fatigue management programme (FACETS) for people with multiple sclerosis

Background Fatigue is a common and troubling symptom for people with multiple sclerosis (MS). Aim To evaluate the effectiveness and cost-effectiveness of a six-session group-based programme for managing MS-fatigue (Fatigue: Applying Cognitive behavioural and Energy effectiveness Techniques to lifeStyle (FACETS)). Methods Three-centre parallel arm randomised controlled trial with economic evaluation. Patients with MS and significant fatigue were randomised to FACETS plus current local practice (FACETS) or current local practice alone (CLP), using concealed computer-generated randomisation. Participant blinding was not possible. Primary outcomes were fatigue severity (Fatigue Assessment Instrument), self-efficacy (Multiple Sclerosis-Fatigue Self-Efficacy) and disease-specific quality of life (Multiple Sclerosis Impact Scale (MSIS-29)) at 1 and 4 months postintervention (follow-up 1 and 2). Quality adjusted life years (QALYs) were calculated (EuroQoL 5-Dimensions questionnaire and the Short-form 6-Dimensions questionnaire). Results Between May 2008 and November 2009, 164 patients were randomised; primary outcome data were available for 146 (89%). Statistically significant differences favour the intervention group on fatigue self-efficacy at follow-up 1 (mean difference (MD) 9, 95% CI (4 to 14), standardised effect size (SES) 0.54, p=0.001) and follow-up 2 (MD 6, 95% CI (0 to 12), SES 0.36, p=0.05) and fatigue severity at follow-up 2 (MD −0.36, 95% CI (−0.63 to −0.08), SES −0.35, p=0.01) but no differences for MSIS-29 or QALYs. No adverse events reported. Estimated cost per person for FACETS is £453; findings suggest an incremental cost-effectiveness ratio of £2157 per additional person with a clinically significant improvement in fatigue. Conclusions FACETS is effective in reducing fatigue severity and increasing fatigue self-efficacy. However, it is difficult to assess the additional cost in terms of cost-effectiveness (ie, cost per QALY) as improvements in fatigue are not reflected in the QALY outcomes, with no significant differences between FACETS and CLP. The strengths of this trial are its pragmatic nature and high external validity. Trial registration: Current Controlled Trials ISRCTN76517470.

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

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.

Multi-centre parallel arm randomised controlled trial to assess the effectiveness and cost-effectiveness of a group-based cognitive behavioural approach to managing fatigue in people with multiple sclerosis

BackgroundFatigue is one of the most commonly reported and debilitating symptoms of multiple sclerosis (MS); approximately two-thirds of people with MS consider it to be one of their three most troubling symptoms. It may limit or prevent participation in everyday activities, work, leisure, and social pursuits, reduce psychological well-being and is one of the key precipitants of early retirement. Energy effectiveness approaches have been shown to be effective in reducing MS-fatigue, increasing self-efficacy and improving quality of life. Cognitive behavioural approaches have been found to be effective for managing fatigue in other conditions, such as chronic fatigue syndrome, and more recently, in MS. The aim of this pragmatic trial is to evaluate the clinical and cost-effectiveness of a recently developed group-based fatigue management intervention (that blends cognitive behavioural and energy effectiveness approaches) compared with current local practice.Methods/DesignThis is a multi-centre parallel arm block-randomised controlled trial (RCT) of a six session group-based fatigue management intervention, delivered by health professionals, compared with current local practice. 180 consenting adults with a confirmed diagnosis of MS and significant fatigue levels, recruited via secondary/primary care or newsletters/websites, will be randomised to receive the fatigue management intervention or current local practice. An economic evaluation will be undertaken alongside the trial. Primary outcomes are fatigue severity, self-efficacy and disease-specific quality of life. Secondary outcomes include fatigue impact, general quality of life, mood, activity patterns, and cost-effectiveness. Outcomes in those receiving the fatigue management intervention will be measured 1 week prior to, and 1, 4, and 12 months after the intervention (and at equivalent times in those receiving current local practice). A qualitative component will examine what aspects of the fatigue management intervention participants found helpful/unhelpful and barriers to change.DiscussionThis trial is the fourth stage of a research programme that has followed the Medical Research Council guidance for developing and evaluating complex interventions. What makes the intervention unique is that it blends cognitive behavioural and energy effectiveness approaches. A potential strength of the intervention is that it could be integrated into existing service delivery models as it has been designed to be delivered by staff already working with people with MS. Service users will be involved throughout this research.Trial registrationCurrent Controlled Trials ISRCTN76517470

Testing the feasibility and acceptability of using the Nintendo Wii in the home to increase activity levels, vitality and well-being in people with multiple sclerosis (Mii-vitaliSe): protocol for a pilot randomised controlled study

Introduction The benefits of physical activity for people with multiple sclerosis (pwMS) have been recognised. However, exercise regimens can be difficult to maintain over the longer term and pwMS may face unique barriers to physical activity engagement. Pilot research suggests the Nintendo Wii can be used safely at home by pwMS with minimal mobility/balance issues and may confer benefits. We have developed a home-based physiotherapist supported Wii intervention (‘Mii-vitaliSe’) for pwMS that uses commercial software. This is a pilot study to explore the feasibility of conducting a full scale clinical and cost-effectiveness trial of Mii-vitaliSe. Methods and analysis 30 ambulatory, relatively inactive pwMS will be randomised to receive Mii-vitaliSe immediately, or after 6 months. Outcomes, measured at baseline and 6 and 12 months later, will include balance, gait, mobility, hand dexterity and self-reported physical activity levels, fatigue, self-efficacy, mood and quality of life. Interviews conducted on a purposive sample of participants will explore experiences of participation in the study and barriers and facilitators to using the Wii. Mean recruitment, adherence rate and standard deviations (SDs) of potential primary outcomes for the full trial will be estimated and precision summarised using 95% confidence intervals (CIs). Interview transcripts will be thematically analysed using a generic qualitative approach. Ethics and dissemination National Health Service (NHS; ref 12/SC/0420) and university ethical approvals have been obtained as has NHS Research and Development permission from the relevant trust. A home risk assessment will be undertaken for all potential participants. All adverse events will be closely monitored, documented and reported to the study Safety Monitoring Committee. At least one publication in a peer reviewed journal will be produced and research findings presented at a national and international conference. With service users, we will coproduce a summary of the findings for dissemination on our research units website and elsewhere. Trial registration number ISRCTN 49286846.

Mii-vitaliSe: a pilot randomised controlled trial of a home gaming system (Nintendo Wii) to increase activity levels, vitality and well-being in people with multiple sclerosis

Objectives While the health and well-being benefits of physical activity are recognised, people with multiple sclerosis (MS) often face greater barriers than the general population. The Nintendo Wii potentially offers a fun, convenient way of overcoming some of these. The aim was to test the feasibility of conducting a definitive trial of the effectiveness and cost-effectiveness of Mii-vitaliSe; a home-based, physiotherapist-supported Nintendo Wii intervention. Design A single-centre wait-list randomised controlled study. Setting MS service in secondary care. Participants Ambulatory, relatively inactive people with clinically confirmed MS. Intervention Thirty participants were randomised to receive Mii-vitaliSe either immediately (for 12 months) or after a 6-month wait (for 6 months). Mii-vitaliSe consisted of two supervised Nintendo Wii familiarisation sessions in the hospital followed by home use (Wii Sports, Sports Resort and Fit Plus software) with physiotherapist support and personalised resources. Outcomes Included self-reported physical activity levels, quality of life, mood, self-efficacy, fatigue and assessments of balance, gait, mobility and hand dexterity at baseline, 6 and 12 months. Interviews (n=25) explored participants’ experiences and, at study end, the two Mii-vitaliSe facilitators’ experiences of intervention delivery (main qualitative findings reported separately). Results Mean (SD) age was 49.3 (8.7) years, 90% female, with 47% diagnosed with MS <6 years ago and 60% new to active gaming. The recruitment rate was 31% (95% CI 20% to 44%). Outcome data were available for 29 (97%) at 6 months and 28 (93%) at 12 months. No serious adverse events were reported during the study. Qualitative data indicated that Mii-vitaliSe was well-received. Mean Wii use across both groups over the initial 6-month intervention period was twice a week for 27 min/day. Mean cost of delivering Mii-vitaliSe was £684 per person. Discussion Mii-vitaliSe appears acceptable and a future trial feasible and warranted. These findings will inform its design. Trial registration ISRCTN49286846

Calpainopathy presenting as foot drop in a 41 year old

Mutations in the gene encoding muscle-specific calpain 3 protease cause limb girdle muscular dystrophy type 2A. Calpainopathy is characterised by progressive symmetrical atrophy of pelvic, scapular and trunk muscles with an elevated creatine kinase. Most patients develop symptoms in childhood and lose the ability to walk by the age of 40 years. We describe a man who presented with foot drop at the age of 41 years, together with neurophysiological, histopathological and genetic data. This is the first report of calpainopathy presenting as foot drop, and widens the phenotype associated with this disease.

Opportunistic Infections of the Retina in Patients With Aquaporin-4 Antibody Disease

IMPORTANCE Patients with neuromyelitis optica who have aquaporin-4 antibodies are being identified and receiving immunosuppressant treatment earlier and more aggressively as a result of increasing awareness of the importance of preventing relapses responsible for the high morbidity and mortality associated with the disease. To our knowledge, opportunistic retinal infection in patients with aquaporin-4 antibodies who are receiving immunosuppressants has not been reported to date. OBSERVATIONS We describe 2 patients with aquaporin-4 antibodies who were receiving conventional doses of first-line immunosuppressive therapy. Both patients presented with vision loss that was initially thought to be optic neuritis attacks. The subsequent diagnoses were ocular toxoplasmosis and cytomegalovirus retinitis. CONCLUSIONS AND RELEVANCE Retinal opportunistic infections can occur in patients with aquaporin-4 antibodies who are receiving relatively low levels of immunosuppression, may mimic optic neuritis, and are a potentially reversible cause of vision loss when treated promptly.

Group-based cognitive behavioural approach to managing fatigue in multiple sclerosis is effective: a multi-centre parallel arm randomised controlled trial

Aim Fatigue is one of the most commonly reported symptoms of MS. This trial evaluates the clinical effectiveness of a group-based fatigue management intervention for people with MS. Study design Pragmatic multi-centre parallel arm randomised controlled trial of a group-based fatigue management intervention compared to current local practice. The six session intervention blends cognitive behavioural and energy effectiveness approaches to managing fatigue. Participants were adults with MS and significant fatigue levels. Primary outcomes were self-reported fatigue severity (Fatigue Assessment Instrument), self-efficacy (MS—Fatigue Self-Efficacy scale) and disease-specific quality of life (MS Impact Scale) measured before, and 1 and 4 months after, the intervention. Results 164 people with MS randomised across three centres in England. Primary outcome data were available on 146 (89%), and 74% of participants attended at least five sessions. Mean self-efficacy was higher in the fatigue management intervention arm at 1 month (standardised effect size (ES) 0.54, p=0.001) and 4 months (ES 0.36, p=0.05), and mean fatigue severity was lower at 4 months (ES 0.35, p=0.01). There were no statistically significant improvements in the quality of life scale. The mean cost of the intervention was approximately £450 per person. Conclusion The results show that the intervention improves self-efficacy and reduces fatigue severity in people with MS. The intervention is novel in that it blends cognitive behavioural and energy effectiveness approaches to managing fatigue, and has been designed so that it can be delivered by those health professionals who already work with people with MS.

Exploring strategies used following a group-based fatigue management programme for people with multiple sclerosis (FACETS) via the Fatigue Management Strategies Questionnaire (FMSQ)

Objectives To explore cross-sectional patterns of use of fatigue management strategies in people with multiple sclerosis (MS) who had attended a group-based fatigue management programme, Fatigue: Applying Cognitive behavioural and Energy effectiveness Techniques to lifeStyle (‘FACETS’). In a multicentre randomised controlled trial (RCT) the FACETS programme was shown to reduce fatigue severity and improve self-efficacy and quality of life. Design A questionnaire substudy within a RCT involving the self-completed Fatigue Management Strategies Questionnaire (FMSQ). The FMSQ includes: (1) closed questions about the use and helpfulness of fatigue management strategies taught in FACETS and (2) open items about changes to lifestyle, attitudes or expectations, barriers or difficulties encountered and helpful strategies not covered in FACETS. Participants All had a clinical diagnosis of MS, significant fatigue, were ambulatory and had attended at least 4 of 6 scheduled FACETS sessions. Methods Participants (n=72) were posted the FMSQ with a prepaid return envelope 4 months after the end of the FACETS programme. Results 82% (59/72) of participants returned the FMSQ. The fatigue management strategies most frequently used since attending FACETS were prioritisation (80%), pacing (78%), saying no to others (78%), grading tasks (75%) and challenging unhelpful thoughts (71%). Adding in those participants who were already using the respective strategies prior to FACETS, the three most used strategies at 4 months were prioritisation (55/59), grading (54/59) and pacing (53/58). Free-text comments illustrated the complex interplay between attitudes/expectations, behaviours, emotions and the environment. Issues related to expectations featured strongly in participants’ comments. Expectations (from self and others) were both facilitators and barriers to effective fatigue management. Conclusions Individuals’ comments highlighted the complex, multifaceted nature of fatigue management. Revising expectations and a greater acceptance of fatigue were important shifts following the programme. Findings support the relevance of a cognitive behavioural approach for fatigue management. Booster sessions might be a useful addition to the FACETS programme. Trial registration number Current controlled trials ISRCTN76517470; Results.

Palliative Care in Amyotrophic Lateral Sclerosis: From Diagnosis to Bereavement, second ed., David Oliver, Gian Domenico Borasio, Declan Walsh (Eds.). Oxford University Press, 368 pp., Hardcover £69.50 (also available in soft cover £29.95) ISBN-10: 0199212937 (price quoted www.amazon.co.uk).

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