Developing and pretesting a new patient reported outcome measure for paediatric CFS/ME: cognitive interviews with children, 2019, Crawley et al

[Sly Saint]

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

Abstract
Background

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. This study aimed to pre-test the new measure through cognitive interviews with children with CFS/ME.

Methods
Cognitive 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.

Results
Twenty-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.

Conclusions
Cognitive 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.

full paper
https://jpro.springeropen.com/articles/10.1186/s41687-019-0156-8

eta:

Availability of data and materials


The datasets used and/or analysed during the current study are available from the corresponding author on reasonable request.

 

[Snowdrop]

One conclusion of this study from an outside POV is that it took this paediatric "ME expert" at least 15 years of research to decide that patients and their carers should have some input into subjective measuring of outcomes in their illness.

With the caveat as we well know that even this would not be happening if not for the scrutiny of the shit pile of previous research using useless measurements designed to provide the researcher with quality 'see I'm so good and I knew I was right' kind of data.

Yes this post is rather umm, unflattering of our intrepid researcher.

Since it seems she will continue research output for this illness it will be interesting to see what kind of conclusions future research data will draw about the nature of paediatric ME and what treatments are useful.

 

[rvallee]

There is not a single reason why such a measure should be developed with children. Especially given Crawley's lack of ethics, she should not even be allowed to work with minors anymore. As a pediatrician this is a bit of a pickle but she brought this on herself. Here the failure is on the regulators and oversight.

The very first sentence is like scraping a chalkboard with nails. There is indeed a lack of PROMs, because of hacks like Crawley peddling the ideological and self-contradictory nonsense of ME being a problem of perception, which has lead to systematically dismiss the substance of what we report. The end result is as likely to be a genuine PROM as recovery norge is a patient organization. Too much bias involved.

The questionnaire itself is not in the paper so it's impossible to judge anything. From the questions and the presence of a frequency response, it appears at least more competent than the CFQ. But there is still no reason why it should be developed with children rather than with adults. The consequences may be slightly different but the circumstances translate very easily. It's a valid eventual step, not a first.

The time to think about outcome measures was 30 years ago, way before promoting sham treatments as safe and effective without even having a competent definition and measurement of outcomes. No one who has been involved in the promotion of sham treatments should be involved in this subject matter. Regulators, get your damn affairs in order.

 

[Snow Leopard]

This is potentially useful research, the discussion of the concerns of the children illustrates this.

In round one and two, children had difficulty answering items in the participation domain as most are ‘able to do activities’ (e.g. go to the park or meet friends) but would get payback (e.g. tired after activity) which was not captured in the frequency response options (never-always).

School response options: days changed to hours
Some children struggled to indicate their school attendance in days as many moderately affected children only attended school for a few hours a day

And so on...

But I think the relevance/understandability etc still needs to be replicated by outside research groups before the measure is considered relevant. 67 items still seems like too many questions, especially for children though... (questions are in the additional file)

 

[Amw66]

I find this sad.
That research has been done for years with no patient informed context.

That so little has been appreciated re effects of this illness, as demonstrated by the many changes throughout the process.

Any clinician who listens to patients would have had a handle on most of the points ( eg being g at school and being capable of learning at school, doing things v doing things without payback etc) . This is basic to the condition.

That assumptions still inform parts ( my daughter is 18, not 8, and would struggle to recall 4 weeks ago)

That moderate/ severely affected seem to have been ignored again. Paediatrics has a very poor track record for this group.

Heartening to see progress like this.
Can't see the scale itself?
Is there a suggestion that this should be applied in conjunction with any objective measures , or is this too big a jump.

How does this compare with lenny Jason's s paediatric work?

 

[Amw66]

Perhaps that this process had not been done before, and that simple aspects had not been appreciated explains the viewpoint informing the terms of engagement for NICE review for children and young people.
Not a clue .

 

[Esther12]

This is potentially useful research, the discussion of the concerns of the children illustrates this.

To me, this whole PROM project looks hopelessly corrupted by the researchers involved. A waste of time and money imo.

This is of some interest given the SMILE trial's outcome switching, and the way verified school attendance was entirely dropped. @dave30th

School or college attendance (including missing lessons, taking breaks and problems concentrating) is one of the most important outcomes for children with CFS/ME (reference currently under review).

The paper cites SMILE, and some other junk trials, but only to for the claim treatments often take months with CFS... and to boost their citation count?!

 

[Amw66]

To me, this whole PROM project looks hopelessly corrupted by the researchers involved. A waste of time and money imo.

This is of some interest given the SMILE trial's outcome switching, and the way verified school attendance was entirely dropped. @dave30th



The paper cites SMILE, and some other junk trials, but only to for the claim treatments often take months with CFS... and to boost their citation count?!

I haven't looked at references...bristol/ holland/ Denmark as a guess?
When will this narrative change?