Abstract
PurposeChronic 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.MethodsWe 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.ResultsWe 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.ConclusionsAn 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.
Highlights
Paediatric chronic fatigue syndrome/myalgic encephalopathy (CFS/ME) is relatively common (0.6–2.4% of children) [1,2,3,4,5,6], diagnosed by extreme disabling fatigue and one or more physical and/or cognitive symptoms such as sleep problems, pain, problems concentrating, headaches, sore throat and dizziness persisting for 3 months in children [7, 8]
The domains outlined in the conceptual framework as part of this study are similar to those found in generic health-related quality of life (HRQoL) models [56,57,58] and consistent with outcomes measured in clinical trials: pain, fatigue, physical functioning, social role participation and emotional distress [59]
This study revealed the specific types of anxieties adolescents with Chronic fatigue syndrome (CFS)/ME have: worries about making symptoms worse after activity or coping with symptoms in social situations
Summary
Paediatric chronic fatigue syndrome/myalgic encephalopathy (CFS/ME) is relatively common (0.6–2.4% of children) [1,2,3,4,5,6], diagnosed by extreme disabling fatigue and one or more physical and/or cognitive symptoms such as sleep problems, pain, problems concentrating, headaches, sore throat and dizziness persisting for 3 months in children [7, 8]. Children can become bedbound [9], miss school [10, 11] and develop mood disorders [12, 13]. In a large cross sectional study, adolescents with CFS/ME were found to have significantly lower health-related quality of life (HRQoL) ( in physical and school functioning) compared to healthy controls [10]. Evidence of the effectiveness of treatment is hindered by the lack of well-developed patientreported outcome measures (PROMs) for children. A review of PROMs completed by children with CFS/ME identified 13 PROMs, six were child specific and seven were not. The authors failed to recommend a PROM due inadequate evidence of quality and acceptability to children with CFS/ME [14]
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