Category: Pediatric ME/CFS

Activity measurement in pediatric chronic fatigue syndrome

Abstract:

Objectives: Individuals with myalgic encephalomyelitis (ME) and chronic fatigue syndrome (CFS) experience debilitating symptoms, including post-exertional malaise, an intensification of symptoms after physical or cognitive exertion. Previous studies found differences in the activity levels and patterns of activity among individuals with ME and CFS, compared to healthy controls; however, limited research exists on the activity levels of pediatric patients. The objective of this study was to examine differences in activity between healthy children and youth with ME and CFS.

Methods: The present study examines the objective (i.e., ActiGraphy) and self-reported levels of activity among children (ages 5 to 17) enrolled in a community-based study of pediatric CFS.

Results: Children with ME and CFS evidenced lower activity levels than healthy control children. Moreover, participants with ME and CFS evidenced increased nighttime activity and delayed initiation of daytime activity. Participants’ self-reported activity data significantly correlated with their ActiGraph data, suggesting that children with ME and CFS are able to accurately describe their activity level.

Discussion: This study highlights differences in activity level and diurnal/nocturnal activity patterns between healthy children and those with ME and CFS. These differences should be considered in identifying appropriate supports and accommodations for children with ME and CFS.

Source: Loiacono B, Sunnquist M, Nicholson L, Jason LA. Activity measurement in pediatric chronic fatigue syndrome [published online ahead of print, 2020 Aug 17]. Chronic Illn. 2020;1742395320949613. doi:10.1177/1742395320949613 https://pubmed.ncbi.nlm.nih.gov/32806955/

Severe ME in Children

Abstract:

A current problem regarding Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS) is the large proportion of doctors that are either not trained or refuse to recognize ME/CFS as a genuine clinical entity, and as a result do not diagnose it. An additional problem is that most of the clinical and research studies currently available on ME are focused on patients who are ambulant and able to attend clinics and there is very limited data on patients who are very severe (housebound or bedbound), despite the fact that they constitute an estimated 25% of all ME/CFS cases.

This author has personal experience of managing and advising on numerous cases of severe paediatric ME, and offers a series of case reports of individual cases as a means of illustrating various points regarding clinical presentation, together with general principles of appropriate management.

Source: Speight N. Severe ME in Children. Healthcare (Basel). 2020;8(3):E211. Published 2020 Jul 14. doi:10.3390/healthcare8030211 https://www.mdpi.com/2227-9032/8/3/211/htm (Full text)

The Relationship Between Childhood Trauma and the Response to Group Cognitive-Behavioural Therapy for Chronic Fatigue Syndrome

Abstract:

Objective: To examine the relationship between childhood trauma and the response to group cognitive-behavioural therapy (GCBT) for chronic fatigue syndrome (CFS).

Methods: A single cohort study conducted in an outpatient university referral center for CFS including a well-documented sample of adult patients meeting the CDC criteria for CFS and having received 9 to 12 months of GCBT. A mixed effect model was adopted to examine the impact of childhood trauma on the treatment response in general and over time. The main outcome measures were changes in fatigue, as assessed with the Checklist Individual Strength (total score), and physical functioning, as gauged with the Short Form 36 Health Survey subscale, with the scales being completed at baseline, immediately after treatment completion and after 1 year.

Results: We included 105 patients with CFS. Childhood trauma was not significantly associated with the response to GCBT over time on level of fatigue or physical functioning.

Conclusion: Childhood trauma does not seem to have an effect on the treatment response to dedicated GCBT for CFS sufferers over time. Therefore, in the allocation of patients to this kind of treatment, a history of childhood trauma should not be seen as prohibitive.

Source: De Venter M, Illegems J, Van Royen R, Sabbe BGC, Moorkens G, Van Den Eede F. The Relationship Between Childhood Trauma and the Response to Group Cognitive-Behavioural Therapy for Chronic Fatigue Syndrome. Front Psychiatry. 2020;11:536. Published 2020 Jun 12. doi:10.3389/fpsyt.2020.00536 https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7304305/ (Full text)

“The Child’s Got a Complete Circle Around Him”. The Care of Younger Children (5-11 Years) With CFS/ME. A Qualitative Study Comparing Families’, Teachers’ and Clinicians’ Perspectives’

Abstract:

Society needs to improve the care of children with complex needs. Guidelines recommend integrating care across health and educational settings, however, there is little research on whether this is achieved or how this can be done in practice. Our aim was to address this gap by examining how the care of children (aged 5-11 years) with Chronic Fatigue Syndrome/ Myalgic Encephalomyelitis (CFS/ME) is shared across home, education and health settings, in order to generate recommendations for integrating care.

We undertook semi-structured interviews with families (22 participants), teachers (11 participants) and healthcare providers (9 participants), analysing the data thematically and comparatively. Our analysis of the data was informed by a socio-ecological perspective as we sought to understand the complexity of the relationships and systems around the child. The first theme focuses on the child (“individual level”); child-centred care is seen as essential whilst acknowledging that the child has limited capacity to manage their own care. The second theme presents the distinct roles of parents, teachers and clinicians (“interpersonal and organisational levels”). The third describes how these three levels interact in the management of the child’s care, in the context of the health and education systems and policies (“policy levels”). The fourth explores optimal ways to integrate care across home, school and clinical settings.

In conclusion, there is opportunity to support a child with complex health needs by targeting the systems around the child; parents, teachers and clinicians, as well as education and health policy that can enable shared-care. Involving schools in assessment, communicating diagnosis across settings and using a stepped-care approach to integrated care may be beneficial. Further work is needed to explore these recommendations, with attention to the policy factors that may act as barriers and enablers.

Source: Brigden A, Shaw A, Barnes R, Anderson E, Crawley E. “The child’s got a complete circle around him”. The care of younger children (5-11 years) with CFS/ME. A qualitative study comparing families’, teachers’ and clinicians’ perspectives’ [published online ahead of print, 2020 Jun 9]. Health Soc Care Community. 2020;10.1111/hsc.13029. doi:10.1111/hsc.13029 https://pubmed.ncbi.nlm.nih.gov/32519359/

Health-related Quality of Life in Norwegian Adolescents Living With Chronic Fatigue Syndrome

Abstract:

Purpose: The primary aim was to measure health related quality of life (HRQoL) in a Norwegian cohort of adolescents with Chronic Fatigue Syndrome (CFS/ME). A secondary aim was to identify factors before diagnosis, at time of diagnosis and after diagnosis that were associated with HRQoL.

Methods: In this cross-sectional population-based study, HRQoL was measured by Pediatric Quality of Life Inventory™ Generic Core scale version 4.0 (PedsQL4.0) in 63 adolescents with CFS/ME. In addition, fatigue was measured by PedsQL Multidimensional Fatigue scale (PedsQL-MFS), depressive symptoms were measured by the Short Mood and Feelings Questionnaire (SMFQ), and disruption in school activities was measured by The De Paul Pediatric Health Questionnaire (DPHQ-N). Data were also collected from medical records and patient interviews.

Results: Age at diagnosis was 15 (2) years (mean (SD)), and four out of five participants were female. Time from diagnosis to reply was 39 (22) months. Adolescents with CFS/ME reported PedsQL4.0 score 50 (17), and boys reported a better score than girls (64 vs 47, 95% Confidence Interval (CI) for difference (- 27; – 6)). There were positive associations between overall HRQoL and support from a schoolteacher, school attendance or participation in leisure activities. There were negative associations between overall HRQoL and delayed school progression, having been to rehabilitation stay and depressive symptoms.

Conclusion: HRQoL in adolescents diagnosed with CFS/ME was low. The associations between reported HRQoL, healthcare previously provided, support from a schoolteacher, school attendance and participation in leisure activity may provide information of value when developing refined strategies for healthcare among adolescents with CFS/ME. Possible causal relationships must however be explored in future studies.

Source: Similä WA, Halsteinli V, Helland IB, Suvatne C, Elmi H, Rø TB. Health-related quality of life in Norwegian adolescents living with chronic fatigue syndrome. Health Qual Life Outcomes. 2020;18(1):170. Published 2020 Jun 5. doi:10.1186/s12955-020-01430-z https://hqlo.biomedcentral.com/articles/10.1186/s12955-020-01430-z (Full text)

Paediatric Chronic Fatigue Syndrome Patients’ and Parents’ Perceptions of Recovery

Abstract:

Objectives: Chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) is common in children and adolescents; however, little is known about how we should define recovery. This study aims to explore perceptions of recovery held by paediatric patients with CFS/ME and their parents.

Methods: Children with CFS/ME and their parents were recruited through a single specialist paediatric CFS/ME service. Data were collected through semistructured interviews with children and parents. The interview questions explored how participants would know if they/their child had recovered from CFS/ME. Thematic analysis was used to identify patterns within the data.

Results: Twenty-one children with CFS/ME, twenty mothers and two fathers were interviewed. Some children found it hard to define recovery as the illness had become a ‘new normal’. Others thought recovery would indicate returning to pre-morbid levels of activity or achieving the same activity level as peers (socialising, education and leisure activities). Increased flexibility in routines and the absence of payback after activities were important. The interviews highlighted the concept of recovery as highly individual with wide variation in symptoms experienced, type and level of activity that would signify recovery. Parents describe how changes in mood and motivation would signify their child’s recovery, but children did not reflect on this.

Conclusion: Some parents and children struggle to define what would constitute complete recovery. However, signs of recovery were more easily identifiable. Definitions of recovery went far beyond symptom reduction and were focused towards rebuilding lives.

Source: Harland MR, Parslow RM, Anderson N, Byrne D, Crawley E. Paediatric chronic fatigue syndrome patients’ and parents’ perceptions of recovery. BMJ Paediatr Open. 2019;3(1):e000525. Published 2019 Dec 2. doi:10.1136/bmjpo-2019-000525 https://bmjpaedsopen.bmj.com/content/3/1/e000525 (Full text)

Sleep problems in adolescents with CFS: A case-control study nested within a prospective clinical cohort

Abstract:

Sleep problems have a negative impact on a range of outcomes and are very common in adolescents with chronic fatigue syndrome (CFS). We aimed to (a) establish whether adolescents with CFS have more self-reported sleep problems than illness controls as well as healthy controls, (b) investigate changes in sleep problems and (c) explore the extent to which sleep problems at baseline predict fatigue and functioning at follow-up in adolescents with CFS. The Insomnia Scale was completed by 121 adolescents with CFS, 78 healthy adolescents and 27 adolescents with asthma. Eighty (66%) treatment-naïve adolescents with CFS completed questionnaires approximately 3 months later. Adolescents with CFS reported increased sleep problems compared to healthy controls and adolescents with asthma. In CFS, there was no significant change in sleep problems without treatment over a 3-month follow-up. Sleep problems at baseline predicted a significant proportion of the variance in sleep problems at follow-up. Sleep problems should be targeted in treatment. Regulating the ‘body clock’ via the regulation of sleep could influence outcomes not assessed in this study such as school attainment.

Source: Loades ME, Rimes KA, Chalder T. Sleep problems in adolescents with CFS: A case-control study nested within a prospective clinical cohort [published online ahead of print, 2020 May 22]. Clin Child Psychol Psychiatry. 2020;1359104520918364. doi:10.1177/1359104520918364 https://journals.sagepub.com/doi/abs/10.1177/1359104520918364

Assessing functioning in adolescents with chronic fatigue syndrome: psychometric properties and factor structure of the School and Social Adjustment Scale and the Physical Functioning Subscale of the SF36

Abstract:

BACKGROUND: Chronic fatigue syndrome (CFS) has a major impact on functioning. However, no validated measures of functioning for this population exist.

AIMS: We aimed to establish the psychometric properties of the 5-item School and Social Adjustment Scale (SSAS) and the 10-item Physical Functioning Subscale of the SF-36 in adolescents with CFS.

METHOD: Measures were completed by adolescents with CFS (n = 121).

RESULTS: For the Physical Functioning Subscale, a 2-factor solution provided a close fit to the data. Internal consistency was satisfactory. For the SSAS, a 1-factor solution provided an adequate fit to the data. The internal consistency was satisfactory. Inter-item and item-total correlations did not indicate any problematic items and functioning scores were moderately correlated with other measures of disability, providing evidence of construct validity.

CONCLUSION: Both measures were found to be reliable and valid and provide brief measures for assessing these important outcomes. The Physical Functioning Subscale can be used as two subscales in adolescents with CFS.

Source: Loades ME, Vitoratou S, Rimes KA, Chalder T. Assessing functioning in adolescents with chronic fatigue syndrome: psychometric properties and factor structure of the School and Social Adjustment Scale and the Physical Functioning Subscale of the SF36. Behav Cogn Psychother. 2020 Apr 1:1-11. doi: 10.1017/S1352465820000193. [Epub ahead of print] https://www.ncbi.nlm.nih.gov/pubmed/32234097

The Prevalence of Pediatric Myalgic Encephalomyelitis/Chronic Fatigue Syndrome in a Community-Based Sample

Abstract:

Background: Most pediatric prevalence studies of myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) have been based upon data from tertiary care centers, a process known for systematic biases such as excluding youth of lower socioeconomic status and those less likely to have access to health care. In addition, most pediatric ME/CFS epidemiologic studies have not included a thorough medical and psychiatric examination. The purpose of this study was to determine the prevalence of pediatric ME/CFS from an ethnically and sociodemographically diverse community-based random sample.

Method: A sample of 10,119 youth aged 5–17 from 5622 households in the Chicagoland area were screened. Following evaluations, a team of physicians made final diagnoses. Youth were given a diagnosis of ME/CFS if they met criteria for three selected case definitions. A probabilistic, multi-stage formula was used for final prevalence calculations.

Results: The prevalence of pediatric ME/CFS was 0.75%, with a higher percentage being African American and Latinx than Caucasian. Of the youth diagnosed with ME/CFS, less than 5% had been previously diagnosed with the illness.

Conclusions: Many youth with the illness have not been previously diagnosed with ME/CFS. These findings point to the need for better ways to identify and diagnose youth with this illness.

Source: Jason, L.A., Katz, B.Z., Sunnquist, M. et al. The Prevalence of Pediatric Myalgic Encephalomyelitis/Chronic Fatigue Syndrome in a Community-Based Sample. Child Youth Care Forum (2020). https://doi.org/10.1007/s10566-019-09543-3. https://link.springer.com/article/10.1007%2Fs10566-019-09543-3

Development of a conceptual framework to underpin a health-related quality of life outcome measure in paediatric chronic fatigue syndrome/myalgic encephalopathy (CFS/ME): prioritisation through card ranking

Abstract:

PURPOSE: Chronic fatigue syndrome (CFS)/myalgic encephalopathy (ME) is relatively common in children and is disabling at an important time in their development. This study aimed to develop a conceptual framework of paediatric CFS/ME using the patient-perspective to ensure that the content of a new outcome measure includes the outcomes most important to young people.

METHODS: We developed a child-centred interactive card ranking exercise that included health-related quality of life (HRQoL) outcomes identified from a previous review of the literature as well as qualitative work. Adolescents and their parents selected and ranked the outcomes most important to them and discussed each outcome in further detail. Adolescents were purposively sampled from a single specialist paediatric CFS/ME service in England. Interviews were audio recorded and transcribed verbatim, and thematic framework analysis was used to develop the final conceptual framework.

RESULTS: We interviewed 43 participants in which there are 21 adolescents, 12-17 years of age with mild-moderate CFS/ME and their parents (20 mothers and 2 fathers). ‘Symptoms’, ‘tiredness’, ‘payback and crashing’ and ‘activities and hobbies’ were ranked most important to improve by both children and parents. Children ranked ‘school’ higher than parents and parents ranked ‘mood’ higher than children. A youth- specific CFS/ME conceptual framework of HRQoL was produced that included 4 outcome domains and 11 subdomains: sleep, tiredness, problems concentrating, individual symptoms, fluctuation and payback, daily and general activities, participation in school, leisure and social life, mood, anxiety and self-esteem.

CONCLUSIONS: An interactive card ranking exercise worked well for adolescents aged 12-17 to elicit the most important outcomes to them and explore each domain in further detail. We developed a final conceptual framework of HRQoL that forms the basis of a new paediatric patient-reported outcome measure (PROM) in CFS/ME.

Source: Parslow RM, Anderson N, Byrne D, Haywood KL, Shaw A, Crawley E. Development of a conceptual framework to underpin a health-related quality of life outcome measure in paediatric chronic fatigue syndrome/myalgic encephalopathy (CFS/ME): prioritisation through card ranking. Qual Life Res. 2020 Jan 6. doi: 10.1007/s11136-019-02399-z. [Epub ahead of print] https://www.ncbi.nlm.nih.gov/pubmed/31907870