Assessing severity of illness and outcomes of treatment in children with Chronic Fatigue Syndrome/Myalgic Encephalomyelitis (CFS/ME): a systematic review of patient‐reported outcome measures (PROMs)

Abstract

Chronic Fatigue Syndrome or Myalgic Encephalomyelitis (CFS/ME) in children is characterized by persistent or recurrent debilitating fatigue which results in a substantial reduction in activity. There is a growing interest in the use of questionnaires, or patient-reported outcome measures (PROMs), to assess how patients function and feel in relation to their health and associated healthcare. However, guidance for PROM selection for children with CFS/ME does not exist. We reviewed the quality and acceptability of PROMs used with children with CFS/ME to inform recommendations for practice. We conducted a systematic review of PROMs completed by children with CFS/ME. The quality of the evaluative studies and the reviewed measures were assessed against recommended criteria using an appraisal framework and the COnsensus-based Standards for the selection of health Measurement INstruments (COSMIN) checklist. We sought evidence of measurement (reliability, validity, responsiveness, interpretability, data quality) and practical properties (acceptability, relevance, feasibility). Sixteen articles were included in the review, providing evidence of reliability and/or validity for 13 PROMs. Of these, five were child-specific (one health-related quality-of-life; four emotional well-being) and eight were not (four emotional well-being, three fatigue-specific; and one generic). All measures had limited evidence of measurement properties and no evidence of practical properties. Recommendations for patient-reported assessment are difficult to make because of limited evidence of the quality and acceptability of PROMs for children with CFS/ME. The appraisal method highlighted significant methodological and quality issues which must be addressed in future research. There is a lack of qualitative evidence describing the outcomes of healthcare that are important to children with CFS/ME, and the relevance or appropriateness of available measures. Future PROM development and evaluation in this group must seek to involve children collaboratively to ensure that the outcomes that children care about are assessed in an acceptable way.bs_bs_banner Child: care, health and development

Quality and acceptability of measures of exercise adherence in musculoskeletal settings: a systematic review

“…evidence of minimal important change); data precision (data quality, end effects); and evidence of where Item Response Theory models were applied. Extraction for practical properties included acceptability (relevance and respondent burden) and feasibility (clinician burden, including cost, time to complete and score) [30, 31, 34]. The extent of patient involvement in measurement development and/or application was also sought [35].…”

“…Data were qualitatively synthesized to determine the overall quality and acceptability of each measure [30, 33]. Synthesis considered the following: study methodological quality (COSMIN scores); number of studies reporting specific evidence per measure; results for each measurement and practical property per measure; and consistency between studies [33].…”

“…Where large numbers of assessment approaches exist, structured reviews of the quality and acceptability of different approaches are essential to informing selection [30, 31]. This review seeks to identify all clearly reported and reproducible measures of exercise adherence applied in published studies of patients with MSK disorders, and to evaluate these measures against a transparent appraisal framework.…”

Patient and public engagement in health-related quality of life and patient-reported outcomes research: what is important and why should we care? Findings from the first ISOQOL patient engagement symposium

“…As observed in the reporting of PROs in clinical trials [25], reviews of patient involvement in health and social research [16], across healthcare services [22], and in HRQL and PRO research [26,27] have highlighted significant inadequacies in the reporting of PE and PPI activities. In response, the Guidance for Reporting Involvement of Patients and Public (GRIPP) checklist was devised to provide a consensus-based approach to enhance the quality of PPI (PE) reporting [23].…”

Research Waste in ME/CFS

“…children)(Nacul et., al., 2011; Haywood, Staniszewska & 226 Chapman, 2012;Johnston et. al., 2014;Haywood, Collin & Crawley, 2014; 227 Hvidberg et. al., 2015;Murdock et.…”