Cognitive behavioural therapy with optional graded exercise therapy in patients with severe fatigue with myotonic dystrophy type 1:a multicentre, single-blind, randomised trial

Background: Myotonic dystrophy type 1 is the most common form of muscular dystrophy in adults and leads to severe fatigue, substantial physical functional impairment, and restricted social participation. In this study, we aimed to determine whether cognitive behavioural therapy optionally combined with graded exercise compared with standard care alone improved the health status of patients with myotonic dystrophy type 1. Methods: We did a multicentre, single-blind, randomised trial, at four neuromuscular referral centres with experience in treating patients with myotonic dystrophy type 1 located in Paris (France), Munich (Germany), Nijmegen (Netherlands), and Newcastle (UK). Eligible participants were patients aged 18 years and older with a confirmed genetic diagnosis of myotonic dystrophy type 1, who were severely fatigued (ie, a score of ≥35 on the checklist-individual strength, subscale fatigue). We randomly assigned participants (1:1) to either cognitive behavioural therapy plus standard care and optional graded exercise or standard care alone. Randomisation was done via a central web-based system, stratified by study site. Cognitive behavioural therapy focused on addressing reduced patient initiative, increasing physical activity, optimising social interaction, regulating sleep–wake patterns, coping with pain, and addressing beliefs about fatigue and myotonic dystrophy type 1. Cognitive behavioural therapy was delivered over a 10-month period in 10–14 sessions. A graded exercise module could be added to cognitive behavioural therapy in Nijmegen and Newcastle. The primary outcome was the 10-month change from baseline in scores on the DM1-Activ-c scale, a measure of capacity for activity and social participation (score range 0–100). Statistical analysis of the primary outcome included all participants for whom data were available, using mixed-effects linear regression models with baseline scores as a covariate. Safety data were presented as descriptives. This trial is registered with ClinicalTrials.gov, number NCT02118779. Findings: Between April 2, 2014, and May 29, 2015, we randomly assigned 255 patients to treatment: 128 to cognitive behavioural therapy plus standard care and 127 to standard care alone. 33 (26%) of 128 assigned to cognitive behavioural therapy also received the graded exercise module. Follow-up continued until Oct 17, 2016. The DM1-Activ-c score increased from a mean (SD) of 61·22 (17·35) points at baseline to 63·92 (17·41) at month 10 in the cognitive behavioural therapy group (adjusted mean difference 1·53, 95% CI −0·14 to 3·20), and decreased from 63·00 (17·35) to 60·79 (18·49) in the standard care group (−2·02, −4·02 to −0·01), with a mean difference between groups of 3·27 points (95% CI 0·93 to 5·62, p=0·007). 244 adverse events occurred in 65 (51%) patients in the cognitive behavioural therapy group and 155 in 63 (50%) patients in the standard care alone group, the most common of which were falls (155 events in 40 [31%] patients in the cognitive behavioural therapy group and 71 in 33 [26%] patients in the standard care alone group). 24 serious adverse events were recorded in 19 (15%) patients in the cognitive behavioural therapy group and 23 in 15 (12%) patients in the standard care alone group, the most common of which were gastrointestinal and cardiac. Interpretation: Cognitive behavioural therapy increased the capacity for activity and social participation in patients with myotonic dystrophy type 1 at 10 months. With no curative treatment and few symptomatic treatments, cognitive behavioural therapy could be considered for use in severely fatigued patients with myotonic dystrophy type 1. Funding: The European Union Seventh Framework Programme

Potential mechanisms involved in the effect of cognitive behavioral therapy on fatigue severity in type 1 diabetes

Objective: To identify mediators of the beneficial effect of cognitive-behavioral therapy (CBT) on fatigue severity in chronically fatigued patients with Type 1 diabetes. Method: We performed secondary analyses of a randomized controlled trial testing the efficacy of CBT. Primary outcome was fatigue severity assessed with the Checklist Individual Strength, subscale fatigue severity. We used multiple mediation analysis to determine potential mediators of the treatment effect. Proposed mediators were symptom focusing, self-efficacy concerning fatigue and pain, perceived activity, sleep disturbances, confidence in diabetes self-care, diabetes distress and discrepancy regarding social support. Actigraphy was used to assess the level of physical activity. The analysis was repeated with depressive symptoms as potential mediator to test whether this caused a change in the other fatigue-related mediators. Results: The effect of CBT on fatigue severity was partly mediated by a change in symptom focusing (-1.39, 95% CI [-3.32, -0.19]), fear avoidance (-1.10, 95% CI [-2.49, -0.22]), self-efficacy concerning fatigue (-1.95, 95% CI [-4.51, -0.40]), and perceived physical activity (-2.44, 95% CI [-4.53, -1.07]). Depressive symptoms were also a mediator (-1.22, 95% CI [-2.56, -0.38]), but the aforementioned fatigue-perpetuating factors still explained part of the treatment effect. Conclusions: Changes in cognitions about fatigue and activity, and a change in depressive symptoms partly mediated the treatment effect of CBT on fatigue severity in Type 1 diabetes. The mediators found are similar to those explaining the effects of CBT in other chronic diseases. This provides knowledge for the development of more effective and efficient interventions for fatigue

Measurement of Treatment Burden in Patients with Multimorbidity in the Netherlands:translation and Validation of the Multimorbidity Treatment Burden Questionnaire (NL-MTBQ)

Background. Multimorbidity is a growing problem. The number and complexity of (non-)pharmaceutical treatments creates a great burden for patients. Treatment burden refers to the perception of the weight of these treatments, and is associated with multimorbidity. Measurement of treatment burden is of great value for optimizing treatment and health-related outcomes. Objective. We aim to translate and validate the Multimorbidity Treatment Burden Questionnaire (MTBQ) for use in the Dutch population with multimorbidity and explore the level of treatment burden. Methods. Translating the MTBQ into Dutch included forward-backward translation, piloting and cognitive interviewing (n=8). Psychometric properties of the questionnaire were assessed in a cross-sectional study of patients with multimorbidity recruited from a panel in the Netherlands (n=959). We examined item properties, dimensionality, internal consistency reliability and construct validity. The level of treatment burden in the population was assessed. Results. The mean age among 959 participants with multimorbidity was 69.9 (17–96) years. Median global NL-MTBQ score was 3.85 (IQR 0-9.62), representing low treatment burden. Significant floor effects were found for all 13 items of the instrument. Factor analysis supported a single-factor structure. The NL-MTBQ had high internal consistency (α=0.845), and provided good evidence on the construct validity of the scale.Conclusion. The Dutch version of the 13-item MTBQ is a single-structured, valid and compact patient-reported outcome measure to assess treatment burden in primary care patients with multimorbidity. It could identify patients experiencing high treatment burden, with great potential to enhance shared-decision making and offer additional support.<br/

Severe fatigue in type 1 diabetes:Exploring its course, predictors and relationship with HbA(1c) in a prospective study

Item does not contain fulltextAIMS: To prospectively identify the course of severe fatigue, its predictors and the relationship with HbA1c in patients with type 1 diabetes. METHODS: 214 adult patients completed questionnaires on fatigue severity and fatigue-related factors at baseline. HbA1c was retrieved from medical records. After 43months, fatigue severity and HbA1c were reassessed in 194 patients. A logistic regression analysis was used to determine predictors of severe fatigue at follow-up with various cognitive-behavioral and clinical factors as potential predictors. The relationship between fatigue and HbA1c was investigated in a sub-analysis by differentiating between patients with suboptimal glucose control [HbA1c>7% (53mmol/mol)] and optimal glucose control [HbA1c7% (53mmol/mol)]. RESULTS: The prevalence of severe fatigue was 40% at baseline and 42% at follow-up. In three out of four severely fatigued patients at baseline (76%), severe fatigue persisted over time. More depressive symptoms, more pain, sleep disturbances, lower self-efficacy concerning fatigue, less confidence in diabetes self-care, more fatigue severity at baseline and more diabetes complications predicted severe fatigue at follow-up. Over time, HbA1c at baseline was positively associated with fatigue severity at follow-up in both groups (suboptimal glucose control: r=.18, p<.05; optimal glucose control: r=.09, p<.05). CONCLUSIONS: About three quarters of patients with type 1 diabetes suffer from persistent fatigue. Aside from the number of diabetes complications, no clinical factors explained the persistence of fatigue. HbA1c and fatigue were weakly associated in a sub-analysis. Since the strongest predictors of severe fatigue were cognitive-behavioral factors, behavioral interventions might be effective in decreasing fatigue.8 p

Correction: Severe fatigue in type 1 diabetes:Exploring its course, predictors and relationship with HbA(1c) in a prospective study (vol 121, pg 127, 2016)

Item does not contain fulltextThe authors have unfortunately detected an error in their above-mentioned article. In the abstract section of the paper, they wrote: "About three quarters of patients with type 1 diabetes suffer from persistent fatigue". However, the text should correctly read as follows: "About three quarters of fatigued patients with type 1 diabetes suffer from persistent fatigue". The authors would like to apologize for this error and the inconvenience caused.1 p

Assessing the experience of person‐centred coordinated care of people with chronic conditions in the Netherlands: Validation of the Dutch P3CEQ

Background Countries are adapting their health and social care systems to better meet the needs of growing populations with (multiple) chronic conditions. To guide this process, assessment of the ‘patient experience’ is becoming increasingly important. For this purpose, the Person-Centred Coordinated Care Experience Questionnaire (P3CEQ) was developed in the United Kingdom, and translated into several languages. Aim This study aimed to assess the internal and construct validity of the Dutch P3CEQ to capture the experience of person-centred coordinated care of people with chronic conditions in the Netherlands. Participants and Methods Adults with chronic conditions (N = 1098) completed the Dutch P3CEQ, measures of health literacy and patient activation, and reported the use and perceived quality of care services. Data analysis included Principal Component and reliability analysis (internal validity), analysis of variance and Student's T-tests (construct validity). Results The two-component structure found was pretty much the same as in the UK validation study. Sociodemographic correlates also resembled those found in the United Kingdom. Women, persons who were less educated, less health-literate or less activated experienced less person-centred coordinated care. P3CEQ scores correlated positively with general practitioner performance scores and quality ratings of the total care received. Conclusion The Dutch P3CEQ is a valid instrument to assess the experience of person-centred coordinated care among people with chronic conditions in the Netherlands. Awareness of inequity and more attention to communication skills in professional training are needed to ensure that care professionals better recognize the needs of women, lower educated or less health-literate persons, and improve their experiences of care. Patient Contribution The P3CEQ has been developed in collaboration with a range of stakeholders. Eighteen persons with (multiple) chronic conditions participated as patient representatives and codesign experts in (four) codesign workshops. Other patient representatives participated in cognitive testing of the English-language instrument. The usability of the P3CEQ to capture the experience of person-centred coordinated care of older persons has been examined by interviewing 228 older European service users, including 13 living in the Netherlands, as part of the SUSTAIN project. More than a thousand persons with chronic conditions participated in the validation study of the Dutch P3CEQ

Is the Eating Disorder Questionnaire-Online (EDQ-O) a valid diagnostic instrument for the DSM-IV-TR classification of eating disorders?

Background The Eating Disorder Questionnaire-Online (EDQ-O) is an online self-report questionnaire, which was developed specifically to provide a DSM-IV-TR classification of anorexia nervosa (AN), bulimia nervosa (BN), binge-eating disorder (BED), and eating disorder not otherwise specified (EDNOS), without using a face-to-face clinical interview. Objective The purpose of the present study was to examine the psychometric quality of the EDQ-O. Methods The validity of the EDQ-O was determined by examining the agreement with the diagnoses obtained from the Longitudinal, Expert, and All DATA (LEAD) standard. Participants included 134 new patients of a specialist center for eating disorders located in the Netherlands. Results Assessment of the validity of the EDQ-O yielded acceptable to good AUC (area under the receiver operating characteristic curve) values with a range from 0.72 to 0.83. Most other diagnostic efficiency statistics were also good except for a low sensitivity for AN (0.44), a low positive predictive value for BN (0.50), and a relatively low sensitivity for BED (0.66). Conclusion The results of the present study suggest that the EDQ-O performs acceptably as a diagnostic instrument for all DSM-IV-TR eating disorder classifications. However, suggestions are made to further improve the validity of the EDQ-O