Exploring anhedonia in adolescents with Chronic Fatigue Syndrome (CFS): A mixed-methods study

Abstract:

Background: Chronic Fatigue Syndrome (CFS/ME) may get in the way of enjoying activities. A substantial minority of adolescents with CFS/ME are depressed. Anhedonia is a core symptom of depression. Anhedonia in adolescents with CFS/ME has not been previously investigated.

Method: One hundred and sixty-four adolescents, age 12 to 18, with CFS/ME completed a diagnostic interview (K-SADS) and questionnaires (HADS, RCADS). We used a mixed-methods approach to explore the experience of anhedonia and examine how common it is, comparing those with clinically significant anhedonia to those without.

Results: Forty-two percent of adolescents with CFS/ME reported subclinical or clinical levels of anhedonia. Fifteen percent had clinically significant anhedonia. Thematic analysis generated two themes: (1) stopping activities that they previously enjoyed and (2) CFS/ME obstructs enjoyment. Most (72%) of those who reported clinically significant anhedonia met the depression diagnostic criteria. Those who were depressed used more negative language to describe their experience of activities than in those who were not depressed, although the themes were broadly similar.

Conclusions: Experiencing pleasure from activities may be affected in CFS/ME, particularly in those who are depressed. Anhedonia may get in the way of behavioural strategies used within CFS/ME treatments.

Source: Smith L, Crawley E, Riley M, McManus M, Loades ME. Exploring anhedonia in adolescents with Chronic Fatigue Syndrome (CFS): A mixed-methods study. Clin Child Psychol Psychiatry. 2021 Apr 16:13591045211005515. doi: 10.1177/13591045211005515. Epub ahead of print. PMID: 33863235. https://pubmed.ncbi.nlm.nih.gov/33863235/

Recovery from chronic fatigue syndrome: a systematic review-heterogeneity of definition limits study comparison

Abstract:

Background: Paediatric chronic fatigue syndrome or myalgic encephalomyelitis (CFS/ME) is a common illness with a major impact on quality of life. Recovery is poorly understood. Our aim was to describe definitions of recovery in paediatric CFS/ME, the rate of recovery and the time to recovery.

Methods: This systematic review included a detailed search of MEDLINE, EMBASE, PsycInfo and Cochrane Library between 1994 and July 2018. Inclusion criteria were (1) clinical trials and observational studies, (2) participants aged <19 years with CFS/ME, (3) conducted in Western Healthcare systems and (4) studies including a measure of recovery and time taken to recover.

Results: Twelve papers (10 studies) were identified, involving 826 patients (range 23-135). Recovery rates were highly varied, ranging between 4.5% and 83%.Eleven distinct definitions of recovery were used; six were composite outcomes while five used unidimensional outcomes. Outcome measures used to define recovery were highly heterogeneous. School attendance (n=8), fatigue (n=6) and physical functioning (n=4) were the most common outcomes included in definition of recovery. Only five definitions included a personal measure of recovery.

Implications: Definitions of recovery are highly variable, likely secondary to differences in study design, outcomes used, follow-up and study populations. Heterogeneous definitions of recovery limit meaningful comparison between studies, highlighting the need for a consensus definition going forward. Recovery is probably best defined from the child’s own perspective with a single self-reported measure. If composite measures are used for research, there should be agreement on the core outcome set used.

Source: Moore Y, Serafimova T, Anderson N, King H, Richards A, Brigden A, Sinai P, Higgins J, Ascough C, Clery P, Crawley EM. Recovery from chronic fatigue syndrome: a systematic review-heterogeneity of definition limits study comparison. Arch Dis Child. 2021 Apr 12:archdischild-2020-320196. doi: 10.1136/archdischild-2020-320196. Epub ahead of print. PMID: 33846138. https://pubmed.ncbi.nlm.nih.gov/33846138/

A report on comorbidity of chronic fatigue syndrome, postural tachycardia syndrome, and narcolepsy with 5,10-methylenetetrahydrofolate reductase (MTHFR) mutation in an adolescent

A 16-year-old male adolescent was hospitalized complaining of intermittent dizziness, drowsiness, and fatigue for approximately 2 years. The patient had an episode of fever and pharyngalgia lasting nearly 2 weeks and had undergone appendectomy because of acute appendicitis before the presentation of the above symptoms. He suffered from severe dizziness mostly after switching from a supine to an upright posture when he was getting up in the morning. The symptom of dizziness usually persisted for minutes to hours and could be partially alleviated by recumbency. In addition, he felt drowsy and fatigued all day despite a total sleep duration of 14 to 15 h per day. All the symptoms could be partially mitigated by complete bed rest for 1 or 2 weeks, but he relapsed after taking part in normal school life again. Additionally, the feeling of fatigue was obviously aggravated after exertion or infection. He was unable to focus on his studies and had to withdraw from school for a long time. As a result, there was a decline in academic performance after the onset of illness. He used to benefit from taking carnitine and folate; however, the improvement was limited in enabling him to take part in normal social and school life. He was physically and mentally healthy before the presentation and did not feel disgusted with learning in the past. No family history of cardiovascular or nervous system disease was evident. The study was approved by the Ethics Committee of Peking University First Hospital (No. 2020- 415).

Source: Liao, Ying; Qi, Jian-Guang; Yan, Hui; Zhang, Qing-You; Ji, Tao-Yun; Chang, Xing-Zhi; Yang, Hai-Po; Jin, Hong-Fang; Du, Jun-Bao A report on comorbidity of chronic fatigue syndrome, postural tachycardia syndrome, and narcolepsy with 5,10-methylenetetrahydrofolate reductase (MTHFR) mutation in an adolescent, Chinese Medical Journal: March 25, 2021 – Volume Publish Ahead of Print – Issue – doi: 10.1097/CM9.0000000000001387  https://journals.lww.com/cmj/Citation/9000/A_report_on_comorbidity_of_chronic_fatigue.98688.aspx (Full text)

Who should we ask about mental health symptoms in adolescents with CFS/ME? Parent-child agreement on the revised children’s anxiety and depression scale

Abstract:

Background: One in three adolescents with chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) have mental health problems. Multi-informant perspectives are key to psychological assessment. Understanding parent-child agreement is crucial to accurate diagnosis, particularly where severe fatigue limits self-report.

Methods: Agreement on the revised children’s anxiety and depression scale (RCADs) was assessed between parents and children with CFS/ME (n = 93) using Bland-Altman plots, cross tabulations and regression analyses.

Results: Diagnostic thresholds were met more frequently based on child-report. Parent- and child-report had similar sensitivity and specificity on RCADS compared to gold-standard diagnostic interviews. Regression analysis found similar accuracy between both reports. For anxiety diagnoses, odds ratio (OR) for child-report was 1.10 (CI = 1.06-1.14), and 1.10 (CI = 1.05-1.14) for parent-report. For depression, OR for child report was 1.26 (CI = 1.11-1.43), while for parent-report is was 1.25 (CI = 1.10-1.41). For total score, OR for child-report was 1.10 (CI = 1.05-1.13) while OR for parent-report was 1.09 (CI = 1.05-1.13).

Conclusions: Reasonable agreement was observed between parent- and child-report of mental health symptoms in paediatric CFS/ME. While parent-report can facilitate psychological evaluation in CFS/ME, this is not a substitute for a child’s own report.

Source: Serafimova T, Loades M, Gaunt D, Crawley E. Who should we ask about mental health symptoms in adolescents with CFS/ME? Parent-child agreement on the revised children’s anxiety and depression scale. Clin Child Psychol Psychiatry. 2021 Feb 15:1359104521994880. doi: 10.1177/1359104521994880. Epub ahead of print. PMID: 33586480.  https://pubmed.ncbi.nlm.nih.gov/33586480/

Co-occurrence of immune-mediated conditions and endometriosis among adolescents and adult women

Abstract:

Problem: Associations between immune dysfunction conditions (eg, systemic lupus erythematous, rheumatoid arthritis) and endometriosis have been observed in adult women, but not assessed among a younger population. We investigated the association between immune-mediated conditions and endometriosis among young women.

Method of study: This cross-sectional analysis in the Women’s Health Study: From Adolescence to Adulthood included 551 participants with surgically diagnosed endometriosis (median age=19) and 652 controls without endometriosis (median age=24). Participants completed an expanded Endometriosis Phenome and Biobanking Harmonization Project questionnaire. We used logistic regression to estimate odds ratios (ORs) and 95% confidence intervals (CIs) to investigate the associations between autoimmune/inflammatory, atopic, chronic pain/fatigue, and endocrine disorders with endometriosis, adjusting for confounders.

Results: Participants with any autoimmune and/or inflammatory condition had an increased odds of co-occurring endometriosis (OR: 1.87; CI: 0.92-3.80), as did participants with allergies (OR: 1.76; CI: 1.32-2.36), asthma (OR: 1.35; CI: 0.97-1.88), chronic fatigue syndrome and/or fibromyalgia (OR: 5.81; CI: 1.89-17.9), or previous mononucleosis (OR: 1.75; CI: 1.14-2.68). Odds of endometriosis were lower among participants with eczema (OR: 0.68; CI: 0.44-1.04). We observed a positive trend between the number of immune-mediated conditions and the odds of endometriosis (p-trend=0.0002). Endocrine disorders were not associated with endometriosis.

Conclusions: Among this population of adolescents and adult women, endometriosis was more likely among participants with autoimmune and/or inflammatory diseases, allergies, asthma, previous mononucleosis infection, and chronic fatigue and/or fibromyalgia. We observed that an increasing number of immune-mediated conditions were positively associated with endometriosis risk. It is important for clinicians who care for adolescents and women with these conditions to consider endometriosis as a comorbidity.

Source: Shafrir AL, Palmor MC, Fourquet J, DiVasta AD, Farland LV, Vitonis AF, Harris HR, Laufer MR, Cramer DW, Terry KL, Missmer SA. Co-occurrence of immune-mediated conditions and endometriosis among adolescents and adult women. Am J Reprod Immunol. 2021 Feb 14:e13404. doi: 10.1111/aji.13404. Epub ahead of print. PMID: 33583078. https://pubmed.ncbi.nlm.nih.gov/33583078/

Are there subgroups of chronic fatigue syndrome? An exploratory cluster analysis of biological markers

Abstract:

Background: Chronic fatigue syndrome (CFS) is defined according to subjective symptoms only, and several conflicting case definition exist. Previous research has discovered certain biological alterations. The aim of the present study was to explore possible subgroups based on biological markers within a widely defined cohort of adolescent CFS patients and investigate to what extent eventual subgroups are associated with other variables.

Methods: The Norwegian Study of Chronic Fatigue Syndrome in Adolescents: Pathophysiology and Intervention Trial (NorCAPITAL) has previously performed detailed investigation of immunological, autonomic, neuroendocrine, cognitive and sensory processing functions in an adolescent group of CFS patients recruited according to wide diagnostic criteria. In the present study, hierarchical cluster analyses (Ward’s method) were performed using representative variables from all these domains. Associations between clusters and constitutional factors (including candidate genetic markers), diagnostic criteria, subjective symptoms and prognosis were explored by standard statistical methods.

Results: A total of 116 patients (26.7% males, mean age 15.4 years) were included. The final cluster analyses revealed six clusters labelled pain tolerant & good cognitions, restored HPA dynamics, orthostatic intolerance, low-grade inflammation, pain intolerant & poor cognitions, and high vagal (parasympathetic) activity, respectively. There was substantial overlap between clusters. The pain intolerant & poor cognitions-cluster was associated with low functional abilities and quality of life, and adherence to the Canada 2003 diagnostic criteria for CFS. No other statistically significant cluster associations were discovered.

Conclusion: Within a widely defined cohort of adolescent CFS patients, clusters could be delineated, but no distinct subgroups could be identified. Associations between clusters and constitutional factors, subjective symptoms and prognosis were scarce. These results question the clinical usefulness of searching for CFS subgroups, as well as the validity of the most “narrow” CFS diagnostic criteria.

Source: Asprusten TT, Sletner L, Wyller VBB. Are there subgroups of chronic fatigue syndrome? An exploratory cluster analysis of biological markers. J Transl Med. 2021 Jan 30;19(1):48. doi: 10.1186/s12967-021-02713-9. PMID: 33516248; PMCID: PMC7847574. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7847574/ (Full text)

Pain in adolescent chronic fatigue following Epstein-Barr virus infection

Abstract:

Objectives: Acute Epstein-Barr virus (EBV) infection is a trigger of Chronic Fatigue (CF) and Chronic Fatigue Syndrome (CFS). The aim of this cross-sectional study was to investigate pain symptoms and pressure pain thresholds in fatigued and non-fatigued adolescents six months after acute EBV-infection, and in healthy controls. This study is part of the CEBA-project (CF following acute EBV infection in adolescents).

Methods: A total of 195 adolescents (12-20 years old) that had undergone an acute EBV infection six months prior to assessment were divided into fatigued (EBV CF+) and non-fatigued (EBV CF-) cases based on questionnaire score. The EBV CF+ cases were further sub-divided according to case definitions of CFS. In addition, a group of seventy healthy controls was included. Symptoms were mapped with questionnaires. Pressure pain thresholds were measured through pressure algometry. One way ANOVA were used for between-group analyses. Linear regression analyses were used to explore associations between Pediatric Quality of Life (dependent variable), pain symptoms and other variables within the EBV (CF+) group.

Results: The EBV CF+ group had significantly higher scores for pain symptoms as compared with the EBV CF- group and healthy controls, but pressure pain threshold did not differ significantly. The number of pain symptoms as well as pain severity were strongly and independently associated with quality of life.

Conclusions: CF and CFS following acute EBV-infection in adolescents is characterized by high pain symptom burden, which in turn is associated with a decline in quality of life. Pain in CF and CFS is of considerable clinical importance, and should be a focal point for further investigation and intervention in these patient groups.

Source: Brodwall EM, Pedersen M, Asprusten TT, Wyller VBB. Pain in adolescent chronic fatigue following Epstein-Barr virus infection [published online ahead of print, 2020 Sep 7]. Scand J Pain. 2020;/j/sjpain.ahead-of-print/sjpain-2020-0031/sjpain-2020-0031.xml. doi:10.1515/sjpain-2020-0031  https://pubmed.ncbi.nlm.nih.gov/32892183/

Chronic Fatigue Syndrome: An Evaluation of a Community Based Management Programme for Adolescents and their Families

Abstract:

Background: Young people with chronic fatigue syndrome (CFS), families and clinicians may differ in their attributions about CFS and consequently in their approach to treatment. Research that clarifies the best treatment approaches is clearly needed. We have sought to develop a model that engages young people and their families in a collaborative way. The approach adopts an optimistic and holistic stance using an active rehabilitation model paying attention to the integrated nature of the physiological and psychological aspects of the illness.

Method: This small study set out to evaluate this approach from a service user perspective. Semi-structured interviews were carried out with young people and their parents separately in order to elicit their views on key treatment elements and their perceived degree of recovery.

Results: Improvements are indicated in all key areas addressed and qualitative information suggests that families value this approach.

Conclusion: Further research is needed to address treatment issues for families who choose not to opt into the service model.

Source: Ashby B, Wright B, Jordan J. Chronic Fatigue Syndrome: An Evaluation of a Community Based Management Programme for Adolescents and their Families. Child Adolesc Ment Health. 2006;11(1):13-18. doi:10.1111/j.1475-3588.2005.00383.x https://pubmed.ncbi.nlm.nih.gov/32811062/

Illness Beliefs in Chronic Fatigue Syndrome: A Study Involving Affected Adolescents and their Parents

Abstract:

Background: The aim of the study was too investigate the beliefs of young people with Chronic Fatigue Syndrome/Myalgic Encephalomyelitis (CFS/ME) and their parents, about illness causes and management.

Method: Twenty-one young people with CFS/ME and their parents participated in an open-ended interview.

Results: Infective causes were identified by the majority of respondents, and psychological ones by a minority. Many highlighted reducing activity and resting in symptom management. Positive and negative experiences of psychiatric and psychological treatments were recorded.

Conclusion: Professionals should carefully explore the illness related beliefs of young people with CFS/ME and parental beliefs in order to agree treatment plans.

Source: Richards J, Chaplin R, Starkey C, Turk J. Illness Beliefs in Chronic Fatigue Syndrome: A Study Involving Affected Adolescents and their Parents. Child Adolesc Ment Health. 2006;11(4):198-203. doi:10.1111/j.1475-3588.2006.00409.x https://pubmed.ncbi.nlm.nih.gov/32810979/

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)