Development of a conceptual framework to underpin a health-related quality of life outcome measure in paediatric CFS/ME - Crawley et al Jan 2020

[Sly Saint]

Development of a conceptual framework to underpin a health-related quality of life outcome measure in paediatric chronic fatigue syndrome/myalgic encephalopathy (CFS/ME): prioritisation through card ranking

• Roxanne M. Parslow,
• Nina Anderson,
• Danielle Byrne,
• Kirstie L. Haywood,
• Alison Shaw &
• Esther Crawley

Abstract
Purpose

Chronic 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.

Methods
We 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.

Results
We 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.

Conclusions
An 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.

https://link.springer.com/article/10.1007/s11136-019-02399-z?shared-article-renderer

 

[Skycloud]

it’s actually not a bad thing to formally identify subjective outcome measures that are meaningful to patients lives.

Edit bad joke deleted because it was bad

 

[Hoopoe]

There doesn't seem to be anything obviously wrong, although there could be some bias in the selection of topics that patients were asked to rank, as well as other kinds of more subtle bias.

So I asked myself what important topic might be missing here, and I think it's disbelief. This causes terrible suffering to patients. Not all of them, but a significant number.

The outcome cards patients were asked to rank

Tiredness
Symptoms (pain, headaches, feeling sick, brain fog)
Sleep problems
Daily activities (getting up, getting dressed, going out)
Payback and crashing (tired after activity)
Fluctuation (changing symptoms—good day vs. bad day)
School (attendance, concentrating, keeping up with work)
Activities and hobbies (sports, clubs)
Spending time with friends
Family activities
Mood (feeling down, worrying)
How you feel about yourself (confidence, personality)
Your future (GCSEs, college, jobs)
Independence (doing things without your parents)
Seeing your boyfriend/girlfriend

Maybe it's missing because the children that are facing disbelief are probably undiagnosed and not taken seriously and therefore cannot access healthcare or participate in research.

Maybe it's missing because it's actually rare. But I doubt that. It seems to be a major theme for many patients.

Perhaps it's missing because the disbelief is fueled by the BPS theories and activities and they don't want to acknowledge that they are harming patients by disseminating their narrative.

 

[Trish]

They recruited patients and parents from their outpatient clinic with mild to moderate symptoms. No severe or housebound patients included. Done by interview, mostly in patient's home. Only about 20 kids and 20 parents interviewed.

This is how they diagnose CFS/ME:

Paediatric chronic fatigue syndrome/myalgic encephalopathy (CFS/ME) is relatively common (0.6–2.4% of children), 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

That doesn't seem to conform to any definition I'm aware of except perhaps the NICE guidelines.

Note no mention of PEM. I've bolded their version of PEM in the list below that they were asked to rank. Note it describes it as 'tired after activity'. So not PEM.

And here's the list they asked the kids and parents to put in order of things that matter most to them to improve:

Tiredness
Symptoms (pain, headaches, feeling sick, brain fog)
Sleep problems
Daily activities (getting up, getting dressed, going out)
Payback and crashing (tired after activity)
Fluctuation (changing symptoms—good day vs. bad day)
School (attendance, concentrating, keeping up with work)
Activities and hobbies (sports, clubs)
Spending time with friends
Family activities
Mood (feeling down, worrying)
How you feel about yourself (confidence, personality)
Your future (GCSEs, college, jobs)
Independence (doing things without your parents)
Seeing your boyfriend/girlfriend

It's mildly interesting to see the differences in order by kids and parents, but the numbers are so small it could be just random fluctuations.

Not surprisingly both parents and kids ranked the physical stuff highest - tiredness, symptoms, payback and crashing and what they were able to do.

Perhaps that should tell them something. When someone is physically sick, they want to get better. The rest would sort itself out with a return to physical health.

I can't help thinking they add the rest of the items to the list for 'quality of life' so they can use their 'therapy' to persuade people to put a more positive spin on the stuff like mood, hobbies and family life, so they can claim a patient has improved even if they are still just as physically ill.

Edit: Crossposted with Strategist.

 

[Trish]

So I asked myself what important topic might be missing here, and I think it's disbelief. This causes terrible suffering to patients. Not all of them, but a significant number.

Perhaps context is important to this one. They recruited families at a CFS outpatient clinic, so they were in a situation where they were getting diagnosis and having their symptoms taken seriously. These particular individuals may not yet have encountered the sort of 'disbelief' many of us suffer.

 

[Amw66]

They recruited patients and parents from their outpatient clinic with mild to moderate symptoms. No severe or housebound patients included. Done by interview, mostly in patient's home. Only about 20 kids and 20 parents interviewed.

This is how they diagnose CFS/ME:

That doesn't seem to conform to any definition I'm aware of except perhaps the NICE guidelines.

Note no mention of PEM. I've bolded their version of PEM in the list below that they were asked to rank. Note it describes it as 'tired after activity'. So not PEM.

And here's the list they asked the kids and parents to put in order of things that matter most to them to improve:


It's mildly interesting to see the differences in order by kids and parents, but the numbers are so small it could be just random fluctuations.

Not surprisingly both parents and kids ranked the physical stuff highest - tiredness, symptoms, payback and crashing and what they were able to do.

Perhaps that should tell them something. When someone is physically sick, they want to get better. The rest would sort itself out with a return to physical health.

I can't help thinking they add the rest of the items to the list for 'quality of life' so they can use their 'therapy' to persuade people to put a more positive spin on the stuff like mood, hobbies and family life, so they can claim a patient has improved even if they are still just as physically ill.

Edit: Crossposted with Strategist.

This is where charities need to get involved in summarising flaws , usually silence reigns.
Given the inadequate definition of PEM this is interesting but of little value.
@Russell Fleming
@Action for M.E.

 

[Unable]

Interesting that there are a mix of positive and negative outcomes in that list. I would find those very hard to rank at one time.

I would also feel suspicious that the number of either positive outcomes, or negative outcomes, that I ranked near the top of my list, would somehow be interpreted in ways that I did not intend.

Now I get that they are saying this is to inform future outcome measures, that would be important to young people, but now that they’ve collected this data, I wonder to what other use it might be put!

Another quick thought, this would definitely be different depending on severity. Those who are mild/moderate may not rank “getting dressed and going out” that highly, if they are already doing that thing.

 

[adambeyoncelowe]

This is how they diagnose CFS/ME:

That doesn't seem to conform to any definition I'm aware of except perhaps the NICE guidelines.

Note no mention of PEM. I've bolded their version of PEM in the list below that they were asked to rank. Note it describes it as 'tired after activity'. So not PEM.

It's not even NICE 2007. That guideline says:

1.3.1.3
The diagnosis of CFS/ME should be reconsidered if none of the following key features are present:

• post-exertional fatigue or malaise
  
  
• cognitive difficulties
  
  
• sleep disturbance
  
  
• chronic pain.

PEM is also listed as a mandatory part of their fatigue definition:

fatigue with all of the following features:

• new or had a specific onset (that is, it is not lifelong)
  
  
• persistent and/or recurrent
  
  
• unexplained by other conditions
  
  
• has resulted in a substantial reduction in activity level
  
  
• characterised by post-exertional malaise and/or fatigue (typically delayed, for example by at least 24 hours, with slow recovery over several days)

That reads like more than just 'tired after activity'. Granted, it's a hodgepodge of descriptions, but it states that it's delayed and has slow recovery.

 

[rvallee]

it’s actually not a bad thing to formally identify subjective outcome measures that are meaningful to patients lives.

Edit bad joke deleted because it was bad

Of course. By someone competent and without an agenda.

Still, there is 100x more benefit to devising a set of objective outcomes instead. This effort is just pointless in the grand scheme of things as it will never be used in a formal setting.