Crawley – Page 3 – American ME and CFS Society

Defining the minimally clinically important difference of the SF-36 physical function subscale for paediatric CFS/ME: triangulation using three different methods

Abstract:

BACKGROUND: Defining the minimally clinically important difference (MCID) is important for the design and analysis of clinical trials and ensures that findings are clinically meaningful. Studies in adult populations have investigated the MCID of The Short Form 36 physical function sub-scale (SF-36-PFS). However, to our knowledge no studies have defined the MCID of the SF-36-PFS in a paediatric population. We aimed to triangulate findings from distribution, anchor and qualitative methods to identify the MCID of the SF-36-PFS for children and adolescents with CFS/ME.

METHODS: Quantitative methods: We analysed routinely-collected data from a specialist paediatric CFS/ME service in South-West England using: 1) the anchor method, based on Clinical Global Impression (CGI) outcomes at 6 months’ follow-up; 2) the distribution method, based on the standard deviation of baseline SF-36-PFS scores. Qualitative methods: Young people (aged 12-17 years) and parents were asked to complete the SF-36-PFS, marking each question twice: once for where they would currently rate themselves/their child and a second time to show what they felt would be the smallest amount of change for them/their child to feel treatment had made a difference. Semi-structured interviews were designed to explore what factors were deemed important to patients and to what extent an improvement was considered satisfactory. We thematically analysed qualitative interviews from 21 children and their parents.

RESULTS: Quantitative results: Six-month follow-up data were available for 198 children with a mean age of 14 years. Most were female (74%, 146/198) and 95% gave their ethnicity as “White British”. Half the standard deviation of the baseline SF-36-PFS scores was 11.0. “A little better” on the CGI equated to a mean difference on the SF-36-PFS from baseline to 6-month follow-up of 9.0. Qualitative results: Twenty-one children with CFS/ME participated: 16 females (76.2%) with a mean age of 14.4 years. Twenty mothers and two fathers were also interviewed. The median minimal improvement in the SF-36-PFS was 10. Participants indicated that small changes in physical function can lead to important improvements in valued social and family function. Patients and parents were positive about improvement even in the presence of persisting symptoms. Triangulation: The MCID based on the mean score from the three methods was 10.

CONCLUSIONS: Converging evidence indicates future studies in paediatric CFS/ME should use an MCID of 10 on the SF-36-PFS.

Source: Brigden A, Parslow RM, Gaunt D, Collin SM, Jones A, Crawley E. Defining the minimally clinically important difference of the SF-36 physical function subscale for paediatric CFS/ME: triangulation using three different methods. Health Qual Life Outcomes. 2018 Oct 19;16(1):202. doi: 10.1186/s12955-018-1028-2. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6194701/ (Full article)

Using the internet to cope with chronic fatigue syndrome/myalgic encephalomyelitis in adolescence: a qualitative study

Abstract:

BACKGROUND: Adolescents are increasingly using online resources for health purposes. Previous studies suggest that online provision of information about chronic fatigue syndrome or myalgic encephalomyelitis (CFS/ME) is neither balanced nor consistent with evidence-based practice. However, little is known about how adolescents with CFS/ME use the internet for their condition and whether this is helpful or harmful.

METHODS: Nine indepth, semistructured, qualitative interviews were conducted with young people (aged 12-17) recruited from a specialist paediatric CFS/ME service. Interviews explored the types of online resources accessed, motivations for doing so and how resource use related to patterns of coping.

RESULTS: Around the time of diagnosis, participants focused on gathering facts about CFS/ME and therefore used official resources (eg, National Health Service sites) that were considered reliable. This transitioned to exploring patient-led and peer-led spaces: health forums, Facebook and YouTube. Participants accessed these regularly, over the long term, and valued these sites for the personal stories, emotional content and interactive technology. Patient-led and peer-led sites supported coping, encouraging active behavioural management, providing social support and addressing stigmatised aspects of the condition. CFS/ME put a strain on normal adolescent life, such as identity and friendships. Online resources allowed participants to adapt and maintain a sense of normality.

CONCLUSIONS: Adolescents who use the internet find online resources helpful in seeking information and social support for their condition. Healthcare services should improve their online resources to meet the needs of younger users, providing evidence-based content in ways that are relevant to adolescents and that can meet the needs for social support, as well as providing information.

Source: Brigden A, Barnett J, Parslow RM, Beasant L, Crawley E. Using the internet to cope with chronic fatigue syndrome/myalgic encephalomyelitis in adolescence: a qualitative study. BMJ Paediatr Open. 2018 Aug 23;2(1):e000299. doi: 10.1136/bmjpo-2018-000299. eCollection 2018. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6109806/ (Full article)

Childhood sleep and adolescent chronic fatigue syndrome (CFS/ME): evidence of associations in a UK birth cohort

Abstract:

OBJECTIVE/BACKGROUND: Sleep abnormalities are characteristic of chronic fatigue syndrome (CFS, also known as ‘ME’), however it is unknown whether sleep might be a causal risk factor for CFS/ME.

PATIENTS/METHODS: We analysed data from the Avon Longitudinal Study of Parents and Children (ALSPAC) birth cohort. We describe sleep patterns of children aged 6 months to 11 years, who were subsequently classified as having (or not having) ‘chronic disabling fatigue’ (CDF, a proxy for CFS/ME) between the ages 13 and 18 years, and we investigated the associations of sleep duration at age nine years with CDF at age 13 years, as well as sleep duration at age 11 years with CDF at age 16 years.

RESULTS: Children who had CDF during adolescence had shorter night-time sleep duration from 6 months to 11 years of age, and there was strong evidence that difficulties in going to sleep were more common in children who subsequently developed CDF. The odds of CDF at age 13 years were 39% lower (odds ratio (OR) = 0.61, 95% CI = 0.43, 0.88) for each additional hour of night-time sleep at age nine years, and the odds of CDF at age 16 years were 51% lower (OR = 0.49, 95% CI = 0.34, 0.70) for each additional hour of night-time sleep at age 11 years. Mean night-time sleep duration at age nine years was 13.9 (95% CI = 3.75, 24.0) minutes shorter among children who developed CDF at age 13 years, and sleep duration at age 11 years was 18.7 (95% CI = 9.08, 28.4) minutes shorter among children who developed CDF at age 16 (compared with children who did not develop CDF at 13 and 16 years, respectively).

CONCLUSIONS: Children who develop chronic disabling fatigue in adolescence have shorter night-time sleep duration throughout early childhood, suggesting that sleep abnormalities may have a causal role in CFS/ME or that sleep abnormalities and CFS/ME are associated with a common pathophysiological cause.

Source: Collin SM, Norris T, Gringras P, Blair PS, Tilling K, Crawley E. Childhood sleep and adolescent chronic fatigue syndrome (CFS/ME): evidence of associations in a UK birth cohort. Sleep Med. 2018 Jun;46:26-36. doi: 10.1016/j.sleep.2018.01.005. Epub 2018 Jan 31. https://www.ncbi.nlm.nih.gov/pubmed/29773208

Pediatric chronic fatigue syndrome: current perspectives

Abstract:

Pediatric chronic fatigue syndrome is an important illness as it is relatively common and also very disabling with a wide range of impacts on the child, the family, and health care systems. It is a complicated illness but the majority of children get better with specialist treatment. This literature review provides an update on the epidemiology of chronic fatigue syndrome/myalgic encephalomyelitis, including factors associated with it, and discusses the current evidence for treatment.

Source: Esther Crawley. Pediatric chronic fatigue syndrome: current perspectives. Dove Press. 29 March 2018 Volume 2018:9 Pages 27—33 https://www.dovepress.com/pediatric-chronic-fatigue-syndrome-current-perspectives-peer-reviewed-article-PHMT (You can download a PDF file of the full article.)

UK Research Collaborative Adopts Biomedical Approach

The CMRC’s new biomedical focus and big ambitions

by Simon McGrath

In a dramatic move last week, the UK CFS/ME Research Collaborative (CMRC) committed itself to a new, biomedical direction. It has started taking concrete action to engage with patients and also announced ambitious plans to enable much more biomedical research in the UK. These changes are enshrined in a statement of purpose, objectives and values (PDF) that replaces the Collaborative’s former charter.

At the same time, Professor Esther Crawley, the CMRC’s controversial deputy chair, is stepping down from that role and from the board, due to a change in her role at her university. From this April, she will be replaced by Chris Ponting, Professor of Medical Bioinformatics at the University of Edinburgh. He heads the multi-million-pound Biomedical Genomics research programme at the Medical Research Council (MRC) Institute of Genetics and Molecular Medicine.

The CMRC has set out its new purpose as promoting the discovery of the biological mechanisms and causal pathways that underpin ME/CFS, in order to develop targeted new treatments…

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Investigating the effectiveness and cost-effectiveness of FITNET-NHS (Fatigue In Teenagers on the interNET in the NHS) compared to Activity Management to treat paediatric chronic fatigue syndrome (CFS)/myalgic encephalomyelitis (ME): protocol for a randomised controlled trial

Abstract:

BACKGROUND: Paediatric chronic fatigue syndrome or myalgic encephalomyelitis (CFS/ME) is a relatively common and disabling condition. The National Institute for Health and Clinical Excellence (NICE) recommends Cognitive Behavioural Therapy (CBT) as a treatment option for paediatric CFS/ME because there is good evidence that it is effective. Despite this, most young people in the UK are unable to access local specialist CBT for CFS/ME. A randomised controlled trial (RCT) showed FITNET was effective in the Netherlands but we do not know if it is effective in the National Health Service (NHS) or if it is cost-effective. This trial will investigate whether FITNET-NHS is clinically effective and cost-effective in the NHS.

METHODS: Seven hundred and thirty-four paediatric patients (aged 11-17 years) with CFS/ ME will be randomised (1:1) to receive either FITNET-NHS (online CBT) or Activity Management (delivered via video call). The internal pilot study will use integrated qualitative methods to examine the feasibility of recruitment and the acceptability of treatment. The full trial will assess whether FITNET-NHS is clinically effective and cost-effective. The primary outcome is disability at 6 months, measured using the SF-36-PFS (Physical Function Scale) questionnaire. Cost-effectiveness is measured via cost-utility analysis from an NHS perspective. Secondary subgroup analysis will investigate the effectiveness of FITNET-NHS in those with co-morbid mood disorders.

DISCUSSION: If FITNET-NHS is found to be feasible and acceptable (internal pilot) and effective and cost-effective (full trial), its provision by the NHS has the potential to deliver substantial health gains for the large number of young people suffering from CFS/ME but unable to access treatment because there is no local specialist service. This trial will provide further evidence evaluating the delivery of online CBT to young people with chronic conditions.

TRIAL REGISTRATION: ISRCTN registry, registration number: ISRCTN18020851 . Registered on 4 August 2016.

Source: Baos S, Brigden A, Anderson E, Hollingworth W, Price S, Mills N, Beasant L, Gaunt D, Garfield K, Metcalfe C, Parslow R, Downing H, Kessler D, Macleod J, Stallard P, Knoop H, Van de Putte E, Nijhof S, Bleijenberg G, Crawley E. Investigating the effectiveness and cost-effectiveness of FITNET-NHS (Fatigue In Teenagers on the interNET in the NHS) compared to Activity Management to treat paediatric chronic fatigue syndrome (CFS)/myalgic encephalomyelitis (ME): protocol for a randomised controlled trial. Trials. 2018 Feb 22;19(1):136. doi: 10.1186/s13063-018-2500-3. https://www.ncbi.nlm.nih.gov/pubmed/29471861

Note: Update published December 19, 2019. https://trialsjournal.biomedcentral.com/articles/10.1186/s13063-019-3895-1

The international collaborative on fatigue following infection (COFFI)

Abstract:

Background: The purpose of the Collaborative on Fatigue Following Infection (COFFI) is for investigators of post-infection fatigue (PIF) and other syndromes to collaborate on these enigmatic and poorly understood conditions by studying relatively homogeneous populations with known infectious triggers. Utilising COFFI, pooled data and stored biosamples will support both epidemiological and laboratory research to better understand the etiology and risk factors for development and progression of PIF.

Methods: COFFI consists of prospective cohorts from the UK, Netherlands, Norway, USA, New Zealand and Australia, with some cohorts closed and some open to recruitment. The 9 cohorts closed to recruitment total over 3000 participants, including nearly 1000 with infectious mononucleosis (IM), > 500 with Q fever, > 800 with giardiasis, > 600 with campylobacter gastroenteritis (CG), 190 with Legionnaires disease and 60 with Ross River virus. Follow-ups have been at least 6 months and up to 10 years. All studies use the Fukuda criteria for defining chronic fatigue syndrome (CFS).

Results: Preliminary analyses indicated that risk factors for non-recovery from PIF included lower physical fitness, female gender, severity of the acute sickness response, and autonomic dysfunction.

Conclusions: COFFI (https://internationalcoffi.wordpress.com/) is an international collaboration which should be able to answer questions based on pooled data that are not answerable in the individual cohorts. Possible questions may include the following: Do different infections trigger different PIF syndromes (e.g. CFS vs. irritable bowel syndrome)?; What are longitudinal predictors of PIF and its severity?

Source: Ben Z Katz, Simon M Collin, Gabrielle Murphy, Rona Moss-Morris, Vegard Bruun Wyller, Knut-Arne Wensaas, Jeannine L.A. Hautvast, Chantal P Bleeker-Rovers, Ute Vollmer-Conna, Dedra Buchwald, Renée Taylor, Paul Little, Esther Crawley, Peter D White & Andrew Lloyd. The international collaborative on fatigue following infection (COFFI). Fatigue: Biomedicine, Health & Behavior Vol. 0, Iss. 0, 2018. http://www.tandfonline.com/doi/abs/10.1080/21641846.2018.1426086?journalCode=rftg20

Chronic fatigue syndrome (CFS/ME) symptom-based phenotypes and 1-year treatment outcomes in two clinical cohorts of adult patients in the UK and The Netherlands

Abstract:

OBJECTIVE: We previously described symptom-based chronic fatigue syndrome (CFS/ME) phenotypes in clinical assessment data from 7041 UK and 1392 Dutch adult CFS/ME patients. Here we aim to replicate these phenotypes in a more recent UK patient cohort, and investigate whether phenotypes are associated with 1-year treatment outcome.

METHODS: 12 specialist CFS/ME services (11 UK, 1 NL) recorded the presence/absence of 5 symptoms (muscle pain, joint pain, headache, sore throat, and painful lymph nodes) which can occur in addition to the 3 symptoms (post-exertional malaise, cognitive dysfunction, and disturbed/unrefreshing sleep) that are present for almost all patients. Latent Class Analysis (LCA) was used to assign symptom profiles (phenotypes). Multinomial logistic regression models were fitted to quantify associations between phenotypes and overall change in health 1year after the start of treatment.

RESULTS: Baseline data were available for N=918 UK and N=1392 Dutch patients, of whom 416 (45.3%) and 912 (65.5%) had 1-year follow-up data, respectively. 3- and 4-class phenotypes identified in the previous UK patient cohort were replicated in the new UK cohort. UK patients who presented with ‘polysymptomatic’ and ‘pain-only’ phenotypes were 57% and 67% less likely (multinomial odds ratio (MOR) 0.43 (95% CI 0.19-0.94) and 0.33 (95% CI 0.13-0.84)) to report that their health was “very much better” or “much better” than patients who presented with an ‘oligosymptomatic’ phenotype. For Dutch patients, polysymptomatic and pain-only phenotypes were associated with 72% and 55% lower odds of improvement (MOR 0.28 (95% CI 0.11, 0.69) and 0.45 (95% CI 0.21, 0.99)) compared with oligosymptomatic patients.

CONCLUSIONS: Adult CFS/ME patients with multiple symptoms or pain symptoms who present for specialist treatment are much less likely to report favourable treatment outcomes than patients who present with few symptoms.

Source: Collin SM, Heron J, Nikolaus S, Knoop H, Crawley E. Chronic fatigue syndrome (CFS/ME) symptom-based phenotypes and 1-year treatment outcomes in two clinical cohorts of adult patients in the UK and The Netherlands. J Psychosom Res. 2018 Jan;104:29-34. doi: 10.1016/j.jpsychores.2017.11.007. Epub 2017 Nov 8. https://www.ncbi.nlm.nih.gov/pubmed/29275782

Clinical and cost-effectiveness of the Lightning Process in addition to specialist medical care for paediatric chronic fatigue syndrome: randomised controlled trial

Editor’s Comment: Numerous critiques of the Lightning Process have been made by physicians, medical practitioners, and patient organizations. There are cases in which LP has resulted in long-lasting physical harm. (You can read about these cases here and here.) Dr. Charles Shepherd, an ME/CFS specialist, does not recommend LP for any ME/CFS patients.

Abstract:

OBJECTIVE: Investigate the effectiveness and cost-effectiveness of the Lightning Process (LP) in addition to specialist medical care (SMC) compared with SMC alone, for children with chronic fatigue syndrome (CFS)/myalgic encephalitis (ME).

DESIGN: Pragmatic randomised controlled open trial. Participants were randomly assigned to SMC or SMC+LP. Randomisation was minimised by age and gender.

SETTING: Specialist paediatric CFS/ME service.

PATIENTS: 12-18 year olds with mild/moderate CFS/ME.

MAIN OUTCOME MEASURES: The primary outcome was the the 36-Item Short-Form Health Survey Physical Function Subscale (SF-36-PFS) at 6 months. Secondary outcomes included pain, anxiety, depression, school attendance and cost-effectiveness from a health service perspective at 3, 6 and 12 months.

RESULTS: We recruited 100 participants, of whom 51 were randomised to SMC+LP. Data from 81 participants were analysed at 6 months. Physical function (SF-36-PFS) was better in those allocated SMC+LP (adjusted difference in means 12.5(95% CI 4.5 to 20.5), p=0.003) and this improved further at 12 months (15.1 (5.8 to 24.4), p=0.002). At 6 months, fatigue and anxiety were reduced, and at 12 months, fatigue, anxiety, depression and school attendance had improved in the SMC+LP arm. Results were similar following multiple imputation. SMC+LP was probably more cost-effective in the multiple imputation dataset (difference in means in net monetary benefit at 12 months £1474(95% CI £111 to £2836), p=0.034) but not for complete cases.

CONCLUSION: The LP is effective and is probably cost-effective when provided in addition to SMC for mild/moderately affected adolescents with CFS/ME.

TRIAL REGISTRATION NUMBER: ISRCTN81456207.

Source: Crawley EM, Gaunt DM, Garfield K, Hollingworth W, Sterne JAC, Beasant L, Collin SM, Mills N, Montgomery AA. Clinical and cost-effectiveness of the Lightning Process in addition to specialist medical care for paediatric chronic fatigue syndrome: randomised controlled trial. Arch Dis Child. 2017 Sep 20. pii: archdischild-2017-313375. doi: 10.1136/archdischild-2017-313375. [Epub ahead of print] https://www.ncbi.nlm.nih.gov/pubmed/28931531

What treatments work for anxiety in children with chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME)? Systematic review

Abstract:

OBJECTIVES: Anxiety is more prevalent in children with chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) than in the general population. A systematic review was carried out to identify which treatment methods are most effective for children with CFS and anxiety.

DESIGN: Systematic review using search terms entered into the Cochrane library and Ovid to search the databases Medline, Embase and psychINFO.

PARTICIPANTS: Studies were selected if participants were <18 years old, diagnosed with CFS/ME (using US Centers for Disease Control and Prevention, the National Institute for Health and Care Excellence or Oxford criteria) and had a valid assessment of anxiety.

INTERVENTIONS: We included observational studies and randomised controlled trials.

COMPARISON: Any or none.

OUTCOMES: Change in anxiety diagnostic status and/or change in anxiety severity on a validated measure of anxiety from pretreatment to post-treatment.

RESULTS: The review identified nine papers from eight studies that met the inclusion criteria. None of the studies specifically targeted anxiety but six studies tested an intervention and measured anxiety as a secondary outcome. Of these studies, four used a cognitive behavioural therapy (CBT)-type approach to treat CFS/ME, one used a behavioural approach and one compared a drug treatment, gammaglobulin with a placebo. Three of the CBT-type studies described an improvement in anxiety as did the trial of gammaglobulin. As none of the studies stratified outcomes according to anxiety diagnostic status or severity, we were unable to determine whether anxiety changed prognosis or whether treatments were equally effective in those with comorbid anxiety compared with those without.

CONCLUSION: We do not know what treatment should be offered for children with both anxiety and CFS/ME. Further research is therefore required to answer this question.

TRIAL REGISTRATION NUMBER: This review was registered on Prospective Register of Systematic Review Protocols (PROSPERO) and the protocol is available from http://www.crd.york.ac.uk/PROSPERO/display_record.asp?ID=CRD42016043488.

Source: Stoll SVE, Crawley E, Richards V, Lal N, Brigden A, Loades ME. What treatments work for anxiety in children with chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME)? Systematic review. BMJ Open. 2017 Sep 5;7(9):e015481. doi: 10.1136/bmjopen-2016-015481. https://www.ncbi.nlm.nih.gov/pubmed/28877941