Developing and pretesting a new patient reported outcome measure for paediatric Chronic Fatigue Syndrome/ Myalgic Encephalopathy (CFS/ME): cognitive interviews with children

Roxanne M Parslow,Kirstie L Haywood,Alison Shaw,Esther Crawley

doi:10.1186/s41687-019-0156-8

Abstract

BackgroundThere is a lack of patient derived, child specific outcome measures to capture what health outcomes are important to children with Chronic Fatigue Syndrome/ Myalgic Encephalopathy (CFS/ME). We developed a new Patient Reported Outcome Measure (PROM) for paediatric CFS/ME through qualitative research with children. This study aimed to pre-test the new measure through cognitive interviews with children with CFS/ME.MethodsCognitive interviews were undertaken in children’s homes or over Skype. The Three-Step Test-Interview (TSTI) method was used to assess the quality of the draft PROM with children with CFS/ME to identify problems with initial content and design and test modifications over subsequent interview rounds. Children were purposively sampled from a single specialist paediatric CFS/ME service in England.ResultsTwenty-four children and their parents took part. They felt the new measure captured issues relevant to their condition and preferred it to the generic measures they completed in clinical assessment. Changes were made to item content and phrasing, timeframe and response options and tested through three rounds of interviews.ConclusionsCognitive interviews identified problems with the draft PROM, enabling us to make changes and then confirm acceptability in children aged 11–18. Further cognitive interviews are required with children 8–10 years old to examine the acceptability and content validity and provide evidence for age related cut offs of the new PROM to meet FDA standards. This study demonstrates the content validity of the new measure as relevant and acceptable for children with CFS/ME. The next stage is to undertake a psychometric evaluation to support the reduction of items, confirm the structure of the PROM and provide evidence of the data quality, reliability and validity.

Highlights

There is a lack of patient derived, child specific outcome measures to capture what health outcomes are important to children with Chronic Fatigue Syndrome/ Myalgic Encephalopathy (CFS/ME)
Healthy children as well as children with CFS/ME were consulted on their views on the structure and formatting of existing generic child Patient Reported Outcome Measure (PROM) with the new measure designed around their preferences
Our study revealed that children with CFS/ME found interference response options (With no difficulty- Not able to do) helpful to describe how they are able to do an activity but this may be ‘difficult’ and result in payback which is a key feature of CFS/ME

Summary

Introduction

There is a lack of patient derived, child specific outcome measures to capture what health outcomes are important to children with Chronic Fatigue Syndrome/ Myalgic Encephalopathy (CFS/ME). We developed a new Patient Reported Outcome Measure (PROM) for paediatric CFS/ME through qualitative research with children. Measuring HRQoL through Patient Reported Outcome Measures (PROMs) is important to describe the impact of an illness on a patient’s daily functioning. The first key stage in PROM development [17,18,19] is the content validity phase, during which qualitative methods are used to provide evidence that domains measured in the final instrument are important to patients [17, 19, 20]. There is currently no validated paediatric CFS/ME-specific PROM [27] informed by children’s views

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