Health, Wellbeing, and Prognosis of Australian Adolescents with Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS): A Case-Controlled Follow-Up Study

Abstract

Background: The purpose of this study was to follow-up an Australian cohort of adolescents newly-diagnosed with ME/CFS at a tertiary paediatric ME/CFS clinic and healthy controls over a mean period of two years (range 1–5 years) from diagnosis. Objectives were to (a) examine changes over time in health and psychological wellbeing, (b) track ME/CFS symptomatology and fulfillment of paediatric ME/CFS diagnostic criteria over time, and (c) determine baseline predictors of ME/CFS criteria fulfilment at follow-up. Methods: 34 participants aged 13–18 years (25 ME/CFS, 23 controls) completed standardised questionnaires at diagnosis (baseline) and follow-up assessing fatigue, sleep quality and hygiene, pain, anxiety, depression, and health-related quality of life. ME/CFS symptomatology and diagnostic criteria fulfilment was also recorded. Results: ME/CFS patients showed significant improvement in most health and psychological wellbeing domains over time, compared with controls who remained relatively stable. However, fatigue, pain, and health-related quality of life remained significantly poorer amongst ME/CFS patients compared with controls at follow-up. Sixty-five percent of ME/CFS patients at baseline continued to fulfil ME/CFS diagnostic criteria at follow-up, with pain the most frequently experienced symptom. Eighty-two percent of patients at follow-up self-reported that they still had ME/CFS, with 79% of these patients fulfilling criteria. No significant baseline predictors of ME/CFS criteria fulfilment at follow-up were observed, although pain experienced at baseline was significantly associated with criteria fulfilment at follow-up (R = 0.6, p = 0.02). Conclusions: The majority of Australian adolescents with ME/CFS continue to fulfil diagnostic criteria at follow-up, with fatigue, pain, and health-related quality of life representing domains particularly relevant to perpetuation of ME/CFS symptoms in the early years following diagnosis. This has direct clinical impact for treating clinicians in providing a more realistic prognosis and highlighting the need for intervention with young people with ME/CFS at the initial diagnosis and start of treatment.

What lies beneath: White matter microstructure in pediatric myalgic encephalomyelitis/chronic fatigue syndrome using diffusion MRI

“…A key strength of our study was the inclusion of a healthy control sample which enabled us to account for the impact of brain development on chronic illness during the adolescent period. Certainly, our study highlighted the importance of considering ME/CFS in a developmental context, and this will be explored further in the future as part of a wider longitudinal study in this cohort of young patients and controls where participants underwent further clinical assessment and brain MRI approximately 2 years following their diagnosis (Josev et al, 2021). Our study was able to provide novel insights into ME/CFS in children and adolescents, an understudied group whose illness is likely to be at an earlier stage (i.e., during adolescence) and uncomplicated by factors associated with chronicity of illness.…”

“…Our study was able to provide novel insights into ME/CFS in children and adolescents, an understudied group whose illness is likely to be at an earlier stage (i.e., during adolescence) and uncomplicated by factors associated with chronicity of illness. Another strength of our study was that diagnosis was made by pediatricians specializing in ME/CFS using stringent clinical consensus criteria, which allowed for a well‐characterized cohort of adolescents with moderate to severe ME/CFS (see Josev et al, 2021). Our results were also consistent across the multiple analytic methods employed in this study, supporting the robustness of our findings.…”

“…The latter includes neurological symptoms such as pain, sleep, cognition, and post-exertional malaise, as well as autonomic, neuroendocrine, and immune system dysfunction (Jason et al, 2006(Jason et al, , 2010. Symptom severity may improve over time with clinical management, yet a large majority of adolescents continue to meet diagnostic criteria for ME/CFS at least 2 years following initial diagnosis, with a particular persistence of symptoms in the domains of fatigue, pain, and quality of life (Josev et al, 2021). The impact on academic, social, and emotional development of affected adolescents is cause for concern among clinicians, educational staff, and families (Crawley et al, 2011;Crawley & Sterne, 2009), and there are clear financial consequences for patients' families and wider society (Jason et al, 2008;Missen et al, 2012;Velleman et al, 2016).…”

“…More longitudinal investigations have been recommended to determine whether microstructural white matter changes develop with length of illness(Almutairi et al, 2020), especially since symptoms can persist for several years after diagnosis of pediatric ME/CFS(Josev et al, 2021). In particular, increased fatigue duration may relate to reduced white matter volume in adults, particularly in the midbrain(Barnden et al, 2011), but this is yet to be investigated at the axonal level with diffusion tractography and fixel-based analysis.Comparison of our findings with previous research was complicated by the limited number of studies investigating white matter microstructure in ME/CFS, and to date, none have been conducted in a pediatric ME/CFS population.…”

Special Issue “Chronic Fatigue Syndrome/Myalgic Encephalomyelitis: Diagnosis and Treatment”

“…It is known that CFS/ME affects young adults, but there are little studies in pediatric and adolescent age. Australian colleagues Elisha K. Josev and colleagues have carried out a case-controlled follow-up study on the health, wellbeing and prognosis of Australian adolescents with CFS/ME on the comprehension of the important relation between physical and psychological health factors to adolescent’ long-term outcome for approaching future prevention, management and treatment [ 5 ]. Concerning epidemiological data, there is little information for Asian countries such Korea and Japan.…”