Interventions in pediatric chronic fatigue syndrome/myalgic encephalomyelitis: a systematic review

Abstract:

PURPOSE: A range of interventions have been used for the management of Chronic Fatigue Syndrome/Myalgic Encephalomyelitis (CFS/ME) in children and adolescents. Currently, debate exists as to the effectiveness of these different management strategies. The objective of this review was to synthesize and critically appraise the literature on interventions for pediatric CFS/ME.

METHOD: CINAHL, PsycINFO and Medline databases were searched to retrieve relevant studies of intervention outcomes in children and/or adolescents diagnosed with CFS/ME. Two reviewers independently selected articles and appraised the quality on the basis of predefined criteria.

RESULTS: A total of 24 articles based on 21 studies met the inclusion criteria. Methodological design and quality were variable. The majority assessed behavioral interventions (10 multidisciplinary rehabilitation; 9 psychological interventions; 1 exercise intervention; 1 immunological intervention). There was marked heterogeneity in participant and intervention characteristics, and outcome measures used across studies. The strongest evidence was for Cognitive Behavioral Therapy (CBT)-based interventions, with weaker evidence for multidisciplinary rehabilitation. Limited information exists on the maintenance of intervention effects.

CONCLUSIONS: Evidence for the effectiveness of interventions for children and adolescents with CFS/ME is still emerging. Methodological inadequacies and inconsistent approaches limit interpretation of findings. There is some evidence that children and adolescents with CFS/ME benefit from particular interventions; however, there remain gaps in the current evidence base.

Copyright © 2013 Society for Adolescent Health and Medicine. Published by Elsevier Inc. All rights reserved.

 

Source: Knight SJ, Scheinberg A, Harvey AR. Interventions in pediatric chronic fatigue syndrome/myalgic encephalomyelitis: a systematic review. J Adolesc Health. 2013 Aug;53(2):154-65. doi: 10.1016/j.jadohealth.2013.03.009. Epub 2013 May 1. https://www.ncbi.nlm.nih.gov/pubmed/23643337

 

Fear of movement and avoidance behaviour toward physical activity in chronic-fatigue syndrome and fibromyalgia: state of the art and implications for clinical practice

Abstract:

Severe exacerbation of symptoms following physical activity is characteristic for chronic-fatigue syndrome (CFS) and fibromyalgia (FM). These exacerbations make it understandable for people with CFS and FM to develop fear of performing body movement or physical activity and consequently avoidance behaviour toward physical activity. The aims of this article were to review what measures are available for measuring fear of movement and avoidance behaviour, the prevalence fear of movement and avoidance behaviour toward physical activity and the therapeutic options with fear of movement and avoidance behaviour toward physical activity in patients with CFS and FM.

The review revealed that fear of movement and avoidance behaviour toward physical activity is highly prevalent in both the CFS and FM population, and it is related to various clinical characteristics of CFS and FM, including symptom severity and self-reported quality of life and disability. It appears to be crucial for treatment (success) to identify CFS and FM patients displaying fear of movement and avoidance behaviour toward physical activity.

Individually tailored cognitive behavioural therapy plus exercise training, depending on the patient’s classification as avoiding or persisting, appears to be the most promising strategy for treating fear of movement and avoidance behaviour toward physical activity in patients with CFS and FM.

Comment in: Fear of movement and avoidance behaviour toward physical activity in chronic-fatigue syndrome and fibromyalgia: state of the art and implications for clinical practice. [Clin Rheumatol. 2013]

 

Source: Nijs J, Roussel N, Van Oosterwijck J, De Kooning M, Ickmans K, Struyf F, Meeus M, Lundberg M. Fear of movement and avoidance behaviour toward physical activity in chronic-fatigue syndrome and fibromyalgia: state of the art and implications for clinical practice. Clin Rheumatol. 2013 Aug;32(8):1121-9. doi: 10.1007/s10067-013-2277-4. Epub 2013 May 3. https://www.ncbi.nlm.nih.gov/pubmed/23639990

 

The role of the therapeutic relationship in cognitive behaviour therapy for chronic fatigue syndrome

Abstract:

Cognitive behaviour therapy (CBT) for chronic fatigue syndrome (CFS) can reduce fatigue and impairment. Recently, it was found that changes in fatigue-perpetuating factors, i.e. focusing on symptoms, control over fatigue, perceived activity and physical functioning, are associated with and explain up to half of the variance in fatigue during CBT for CFS.

The therapy relationship, e.g. outcome expectations and working alliance, may also contribute to treatment outcome. We aimed to examine the role of the therapy relationship in CBT and determine whether it exerts its effect independently of changes in fatigue-perpetuating factors. We used a cohort of 217 CFS patients in which the pattern of change in fatigue-perpetuating factors was examined previously. Fatigue, therapy relationship and fatigue-perpetuating factors were measured at the start of therapy, three times during CBT and at the end of therapy. Baseline outcome expectations and agreement about the content of therapy predicted post therapy fatigue.

A large part of the variance in post-treatment fatigue (25%) was jointly explained by outcome expectations, working alliance and changes in fatigue-perpetuating factors. From this, we conclude that positive outcome expectations and task agreement seem to facilitate changes in fatigue-perpetuating factors during CBT for CFS. It is therefore important to establish a positive therapy relationship early in therapy.

Copyright © 2013 Elsevier Ltd. All rights reserved.

 

Source: Heins MJ, Knoop H, Bleijenberg G. The role of the therapeutic relationship in cognitive behaviour therapy for chronic fatigue syndrome. Behav Res Ther. 2013 Jul;51(7):368-76. doi: 10.1016/j.brat.2013.02.001. Epub 2013 Apr 9. https://www.ncbi.nlm.nih.gov/pubmed/23639303

 

Neurocognitive impairment in childhood chronic fatigue syndrome

Abstract:

Neurocognitive impairment is a feature of childhood chronic fatigue syndrome (CCFS). Several studies have demonstrated reduced attention control in CCFS patients in switching and divided attention tasks. In students, the extent of deterioration in task performance depends on the level of fatigue.

Poor performance in switching and divided attention is common in both fatigued students and CCFS patients. Additionally, attentional functions show dramatic development from childhood to adolescence, suggesting that abnormal development of switching and divided attention may be induced by chronic fatigue.

The brain structures associated with attentional control are situated in the frontal and parietal cortices, which are the last to mature, suggesting that severe fatigue in CCFS patients and students may inhibit normal structural and functional development in these regions. A combination of treatment with cognitive behavioral therapy and antidepressant medication is effective to improve attentional control processing in CCFS patients. Studies identifying the features of neurocognitive impairment in CCFS have improved our current understanding of the neurophysiological mechanisms of CCFS.

 

Source: Mizuno K, Watanabe Y. Neurocognitive impairment in childhood chronic fatigue syndrome. Front Physiol. 2013 Apr 19;4:87. doi: 10.3389/fphys.2013.00087. ECollection 2013. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3630366/ (Full article)

 

Depression in paediatric chronic fatigue syndrome

Abstract:

OBJECTIVE: To describe the prevalence of depression in children with chronic fatigue syndrome (CFS)/myalgic encephalomyelitis (ME) and investigate the relationship between depression in CFS/ME and clinical symptoms such as fatigue, disability, pain and school attendance.

DESIGN: Cross-sectional survey data using the Hospital Anxiety and Depression Scale (HADS) collected at assessment.

SETTING: Specialist paediatric CFS/ME service in the South West.

PATIENTS: Children aged 12-18 years with CFS/ME.

MAIN OUTCOME MEASURE: Depression was defined as scoring >9 on the HADS depression scale.

RESULTS: 542 subjects had complete data for the HADS and 29% (156/542) (95% CI 25% to 33%) had depression. In a univariable analysis, female sex, poorer school attendance, and higher levels of fatigue, disability, pain, and anxiety were associated with higher odds of depression. Age of child and duration of illness were not associated with depression. In a multivariable analysis, the factors most strongly associated with depression were disability, with higher scores on the physical function subscale of the 36 item Short Form (SF-36).

CONCLUSIONS: Depression is commonly comorbid with CFS/ME, much more common than in the general population, and is associated with markers of disease severity. It is important to screen for, identify and treat depression in this population.

 

Source: Bould H, Collin SM, Lewis G, Rimes K, Crawley E. Depression in paediatric chronic fatigue syndrome. Arch Dis Child. 2013 Jun;98(6):425-8. doi: 10.1136/archdischild-2012-303396. Epub 2013 Apr 25. https://www.ncbi.nlm.nih.gov/pubmed/23619200

 

Could mitochondrial dysfunction be a differentiating marker between chronic fatigue syndrome and fibromyalgia?

Abstract:

Chronic fatigue syndrome (CFS) and fibromyalgia (FM) are complex and serious illnesses that affect approximately 2.5% and 5% of the general population worldwide, respectively. The etiology is unknown; however, recent studies suggest that mitochondrial dysfunction has been involved in the pathophysiology of both conditions. We have investigated the possible association between mitochondrial biogenesis and oxidative stress in patients with CFS and FM.

We studied 23 CFS patients, 20 FM patients, and 15 healthy controls. Peripheral blood mononuclear cell showed decreased levels of Coenzyme Q10 from CFS patients (p<0.001 compared with controls) and from FM subjects (p<0.001 compared with controls) and ATP levels for CFS patients (p<0.001 compared with controls) and for FM subjects (p<0.001 compared with controls).

On the contrary, CFS/FM patients had significantly increased levels of lipid peroxidation, respectively (p<0.001 for both CFS and FM patients with regard to controls) that were indicative of oxidative stress-induced damage. Mitochondrial citrate synthase activity was significantly lower in FM patients (p<0.001) and, however, in CFS, it resulted in similar levels than controls. Mitochondrial DNA content (mtDNA/gDNA ratio) was normal in CFS and reduced in FM patients versus healthy controls, respectively (p<0.001). Expression levels of peroxisome proliferator-activated receptor gamma-coactivator 1-alpha and transcription factor A, mitochondrial by immunoblotting were significantly lower in FM patients (p<0.001) and were normal in CFS subjects compared with healthy controls.

These data lead to the hypothesis that mitochondrial dysfunction-dependent events could be a marker of differentiation between CFS and FM, indicating the mitochondria as a new potential therapeutic target for these conditions.

 

Source: Castro-Marrero J, Cordero MD, Sáez-Francas N, Jimenez-Gutierrez C, Aguilar-Montilla FJ, Aliste L, Alegre-Martin J. Could mitochondrial dysfunction be a differentiating marker between chronic fatigue syndrome and fibromyalgia? Antioxid Redox Signal. 2013 Nov 20;19(15):1855-60. doi: 10.1089/ars.2013.5346. Epub 2013 May 29. https://www.ncbi.nlm.nih.gov/pubmed/23600892

 

Moderators of the treatment response to guided self-instruction for chronic fatigue syndrome

Abstract:

OBJECTIVE: The efficiency and efficacy of guided self-instruction for chronic fatigue syndrome (CFS) can be enhanced if it is known which patients will benefit from the intervention. This study aimed to identify moderators of treatment response.

METHODS: This study is a secondary analysis of two randomized controlled trials evaluating the efficacy of guided self-instruction for CFS. A sample of 261 patients fulfilling US Center for Disease Control and Prevention criteria for CFS was randomly allocated to guided self-instruction or a wait list. The following potential treatment moderators were selected from the literature: age, fatigue severity, level of physical functioning, pain, level of depressive symptoms, self-efficacy with respect to fatigue, somatic attributions, avoidance of activity, and focus on bodily symptoms. Logistic and linear regression analyses were used with interaction term between treatment response and the potential moderator.

RESULTS: Age, level of depression, and avoidance of activity moderated the response to guided self-instruction. Patients who were young, had low levels of depressive symptoms, and who had a low tendency to avoid activity benefited more from the intervention than older patients and patients with high levels of depressive symptoms and a strong tendency to avoid activity.

CONCLUSION: Guided self-instruction is exclusively aimed at cognitions and behaviours that perpetuate fatigue. Patients with severe depressive symptom may need more specific interventions aimed at the reduction of depressive symptoms to profit from the intervention. Therefore we suggest that patients with substantial depressive symptoms be directly referred to regular cognitive behaviour therapy.

Copyright © 2013 Elsevier Inc. All rights reserved.

 

Source: Tummers M, Knoop H, van Dam A, Bleijenberg G. Moderators of the treatment response to guided self-instruction for chronic fatigue syndrome. J Psychosom Res. 2013 May;74(5):373-7. doi: 10.1016/j.jpsychores.2013.01.007. Epub 2013 Mar 5. https://www.ncbi.nlm.nih.gov/pubmed/23597323

 

Cognitive behavior therapy in patients with chronic fatigue syndrome: the role of illness acceptance and neuroticism

Abstract:

OBJECTIVE: Increasing the quality of life (QoL) of patients with chronic fatigue is challenging because recovery is seldom achieved. Therefore, it is important to identify processes that improve QoL. This study examined the extent of improvement related to cognitive behavior group therapy (CBT), and whether improvement is affected by initial levels of acceptance and neuroticism.

METHODS: Eighty CFS patients followed CBT, and self-reported (pre-post design) on mental and physical QoL (MQoL and PQoL), fatigue, acceptance, and neuroticism. The extent of improvement was analyzed using t-tests, effect sizes, and clinically significant change criteria. Whether acceptance and neuroticism at baseline predicted changes was analyzed by means of correlation and regression analyses.

RESULTS: Significant improvement was found for all variables. The effect size for MQoL and PQoL was small; for acceptance and fatigue, effect size was moderate. About 20% (MQoL) to 40% (fatigue) of the participants clinically improved. Pre-treatment level of acceptance was negatively correlated with changes in MQoL, not with PQoL changes. Neuroticism pre-treatment was positively related with MQoL changes. Regression analysis showed an effect of acceptance on changes in MQoL beyond the effect of neuroticism.

CONCLUSIONS: Although CBT is an evidence-based treatment, the sizes of the effects are often small regarding QoL. Our study also revealed small effect sizes. Our study showed that patient characteristics at baseline were significantly associated with MQoL outcome; indicating that CFS patients with high neuroticism or with a low acceptance show more improvement in MQoL. We propose to specifically target acceptance and neuroticism before treatment in order to maximize clinical relevance.

Copyright © 2013 Elsevier Inc. All rights reserved.

 

Source: Poppe C, Petrovic M, Vogelaers D, Crombez G. Cognitive behavior therapy in patients with chronic fatigue syndrome: the role of illness acceptance and neuroticism. J Psychosom Res. 2013 May;74(5):367-72. doi: 10.1016/j.jpsychores.2013.02.011. Epub 2013 Mar 25. https://www.ncbi.nlm.nih.gov/pubmed/23597322

 

Couples’ experiences of interacting with outside others in chronic fatigue syndrome: a qualitative study

Abstract:

OBJECTIVES: Social isolation and stigma are frequently reported by patients with chronic fatigue syndrome/myalgic encephalomyelitis and relationships in the home environment with those close to the patients (their ‘significant others’) may thus be particularly important. Rather little attention has yet been paid to the beliefs and experiences of ‘significant others’ themselves in this context. This study sought to explore in-depth the beliefs and experiences of both patients and ‘significant others’ in relation to chronic fatigue syndrome/myalgic encephalomyelitis.

METHODS: In-depth interviews using a semi-structured interview schedule designed around the core constructs of the Common-Sense Model of self-regulation were conducted with two patients with chronic fatigue syndrome/myalgic encephalomyelitis and their spouses. Interpretative Phenomenological Analysis was used to analyse interview data.

RESULTS: Experiences of social interactions in relation to chronic fatigue syndrome/myalgic encephalomyelitis with others outside of the relationship dyad emerged as a key issue for all participants when reflecting on their experiences of living with the condition. These concerns are presented under two themes: interactions with healthcare professionals and interactions with the social world.

CONCLUSIONS: It is evident that significant others play an important role in the lived experience of chronic fatigue syndrome/myalgic encephalomyelitis. For both patients and significant others, the wider social world and interactions with outside others may be important influences on dyadic coping in chronic fatigue syndrome/myalgic encephalomyelitis. Both future research and treatment interventions could usefully include a ‘significant other’ perspective.

 

Source: Brooks J, King N, Wearden A. Couples’ experiences of interacting with outside others in chronic fatigue syndrome: a qualitative study. Chronic Illn. 2014 Mar;10(1):5-17. doi: 10.1177/1742395312474478. Epub 2013 Apr 12. https://www.ncbi.nlm.nih.gov/pubmed/23585635

Examining the energy envelope and associated symptom patterns in chronic fatigue syndrome: does coping matter?

Abstract:

OBJECTIVE/Hypothesis: The objective of this study was to examine sub-types of individuals with chronic fatigue syndrome based on variables that are associated with the energy envelope theory and to examine the role of coping strategies in explaining the differences found between the subtypes.

METHODS: Cluster analysis was used. Grouping variables included physical functioning, post-exertional malaise severity, and the extent to which an individual was outside of the energy envelope. These clusters were evaluated using discriminant function analysis to determine whether they could be differentiated based on coping styles.

RESULTS: Cluster analysis identified three groups. Clusters 1 and 2 were consistent with the energy envelope theory. However, Cluster 3 was characterized by patients with the most impairment, but they were to a lesser extent exceeding their energy envelope. Coping strategies explained a small percentage (10%) of the variance in differentiating the clusters.

DISCUSSION: Energy maintenance may be associated with improved functioning and less severe symptoms for some. However, patients in Cluster 3 were closer to remaining within their energy envelope and also used higher levels of adaptive coping but were more impaired than Cluster 2. This suggests that adaptive coping strategies were not associated with improved health, as members of Cluster 3 were severely limited in functioning.

 

Source: Brown AA, Evans MA, Jason LA. Examining the energy envelope and associated symptom patterns in chronic fatigue syndrome: does coping matter? Chronic Illn. 2013 Dec;9(4):302-11. doi: 10.1177/1742395313478220. Epub 2013 Apr 12. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3893101/ (Full article)