Management of severe ME/CFS in children and young people in the UK: a British Paediatric Surveillance Unit study, 2024, Royston et al.

SNT Gatchaman

SNT Gatchaman

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Management of severe ME/CFS in children and young people in the UK: a British Paediatric Surveillance Unit study
Alexander Peter Royston; Sarah Burge; Ilaria Idini; Amberly Brigden; Katharine Claire Pike

OBJECTIVES
Severe myalgic encephalomyelitis or chronic fatigue syndrome (ME/CFS) in children and young people (CYP) is a little-understood condition which significantly impacts education, development and quality of life. We used data from a population-wide surveillance study to explore the screening investigation, referral and management of suspected cases of paediatric severe ME/CFS.

METHODS
A British Paediatric Surveillance Unit (BPSU) study reported cases of CYP with suspected severe ME/CFS between February 2018 and February 2019. Paediatricians reporting cases to BPSU and allied healthcare professionals in two large specialist paediatric ME/CFS centres were invited to complete questionnaires for CYP meeting the surveillance case definition. The study focused primarily on CYP with confirmed severe ME/CFS and the extent to which their care met NICE (The National Institute for Health and Care Excellence) recommendations but also considered separately those with probable or possible severe ME/CFS.

RESULTS
This study includes a total of 92 CYP with suspected severe ME/CFS; 33 meeting criteria for severe ME/CFS and an additional 59 classified as probable or possible severe ME/CFS. For 16 possible cases, incomplete investigation to exclude alternative diagnoses prevented confirmation of a severe ME/CFS diagnosis. Only 21 of 33 (64%) confirmed severe ME/CFS cases had been referred to specialist services. The management provided varied considerably between patients and four received nothing at all. Of the management provided, the most frequent approaches were medication (67%), activity management (61%) and physiotherapy (61%). Domiciliary assessments and support, and social services referrals were received by 12% and 6% of confirmed severe cases. Similar proportions of management approaches were seen in probable/possible severe ME/CFS.

CONCLUSIONS
Full investigation is frequently incomplete in CYP with suspected severe ME/CFS and recommendations for referral and management are poorly implemented, in particular the needs of CYP who are unable to leave their home might be poorly met.


Link | PDF (BMJ Paediatrics Open) [Open Access]
 
The finding that a substantial number of CYP with suspected severe ME/CFS have incomplete screening investigations not only affects the accuracy of prevalence estimates but also has diagnostic implications. Treatable conditions that resemble ME/CFS might not be identified, and this can lead to inappropriate management. This potential for harm is recognised in the NICE guidance and underlies the recommendation that prescribing should be initiated under the supervision of a paediatrician with expertise in ME/CFS. This is a particular concern given that a sizeable proportion of CYP are either without a confirmed diagnosis or under management in non-specialist centres.
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NICE’s 2021 guidance for ME/CFS of all grades of severity, including those with severe forms of the condition, recommends clinicians “refer children and young people who have been diagnosed with ME/CFS after assessment by a paediatrician directly to a paediatric ME/CFS specialist team”.
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Our results show that only two-thirds of patients with severe ME/CFS were under specialist management when receiving physiotherapy and activity management. NICE recommends specialist physiotherapy advice and additional care for those with severe or very severe ME/ CFS when an approach involving energy management has been chosen. While it is encouraging that CYP with severe ME/CFS have access to physiotherapy, it is possible that they may not be receiving optimal care. It is not known why specialist centres appear to recommend activity management and physiotherapy less often.
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The scarcity of domiciliary visits is of concern for this patient group. The proportion of patients receiving home assessments (12%) falls short of NICE’s recommendations that home visits should be offered to all with severe or very severe ME/CFS. Social services assessment and support was similarly rare in our cohort (6%), raising the possibility that CYP and their parents or carers are not receiving the support that they are entitled to.
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"Case definition
A staged process was used to confirm severe ME/CFS; this required screening blood tests to exclude alternative diagnoses and assessment of symptom characteristics, duration and impact.12 Patients with abnormal blood tests suggestive of another cause for the fatigue were classified as not severe ME/CFS and excluded from further analysis. Urinalysis is recommended within the NICE guideline, but this was removed as a mandatory criterion by the study team because it was so often missed. Patients otherwise meeting the case definition but for whom it was unclear whether they had experienced a significant reduction in activities of daily living were classified as having probable severe ME/CFS. Those meeting most criteria but with incomplete screening tests or information on function were classified as having possible severe ME/CFS.

In the absence of an official paediatric ME/CFS specialist service specification, a list of specialist services was compiled from services containing a paediatrician and treating CYP that were named either in a list published by Great Ormond Street Hospital for Children21 or the 2018 directory22 of the British Association of Clinicians in ME/CFS. The questionnaire asked whether the CYP had received any of the following treatments/assessments: activity management, cognitive–behavioural therapy (CBT), domiciliary assessment or treatment, graded exercise therapy, medication for symptoms, physiotherapy, social services assessment and support, or treatment from Child and Adolescent Mental Health Services (CAMHS). These approaches were in line with NICE guidelines at the time of study design,23 though more recent guidelines also recommend dietetic assessments for certain CYP and prohibit graded exercise therapy."

Well, I've read all of that and I'm still not clear on how they define "severe ME/CFS".
 
Andy said:
I'm still not clear on how they define "severe ME/CFS".
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It's discussed before the case definition section.

Severely affected individuals can have limited mobility, be unable to leave their home and have severe cognitive difficulties.
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Using the British Paediatric Surveillance Unit’s (BPSU) reporting system for rare conditions, between February 2018 and February 2019 paediatricians in the UK and RoI were invited to report CYP aged 5–16 years, meeting a clinical diagnosis of ME/CFS who due to fatigue were able to attend school for an hour or less per week over the last six term-time weeks.
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This seems to be an offshoot of the same study @dx-revision-watch.77 mentioned over here: https://www.s4me.info/threads/grade...-gaunt-crawley-et-al.37488/page-4#post-519144

The 92 patients are the severe cypwME mentioned in this study: https://pubmed.ncbi.nlm.nih.gov/36456114/

They seem to have forgotten the FINE trial in their round-up of previous attempts to treat severe ME.

eta: Here is the RCPCH page for this study: https://www.rcpch.ac.uk/work-we-do/...c-fatigue-syndrome-myalgic-encephalitis-cfsme
 
Lucibee said:
This seems to be an offshoot of the same study @dx-revision-watch.77 mentioned over here: https://www.s4me.info/threads/grade...-gaunt-crawley-et-al.37488/page-4#post-519144

The 92 patients are the severe cypwME mentioned in this study: https://pubmed.ncbi.nlm.nih.gov/36456114/

They seem to have forgotten the FINE trial in their round-up of previous attempts to treat severe ME.

eta: Here is the RCPCH page for this study: https://www.rcpch.ac.uk/work-we-do/...c-fatigue-syndrome-myalgic-encephalitis-cfsme
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https://www.rcpch.ac.uk/work-we-do/...c-fatigue-syndrome-myalgic-encephalitis-cfsme

Lead investigators
Professor Esther Crawley
Centre of Child and Adolescent Health, University of Bristol
1-5 Whiteladies Road
Bristol BS8 1NU
Email: esther.crawley@bristol.ac.uk

And yet she isn't one of the authors.
 
Dolphin said:
And yet she isn't one of the authors.
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"Contributors: This study was conceived and designed by Professor Esther Crawley and AB [Amberly Bridgen]."

Authorship is, and should be, about who actually writes the paper. She probably takes the same line as Wessely - already on sooooo many papers that she doesn't need her name to be on any more thank you very much.
 
So, this is a study of care providers using a now defunct guideline who show no useful knowledge of ME, severe or otherwise.
Our results show that only two-thirds of patients with severe ME/CFS were under specialist management when receiving physiotherapy and activity management
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So this is by definition only patients who are under someone's care, which is a minority, and they are overseen using a harmful care model that was deprecated between the time they started the study and publication. It's not accurate to say that 2/3 severe patients are under specialist management, it's only 2/3 who were seen using this clinical pathway by this group. From people who overuse statistics this much, they don't seem to understand that accurate denominators are important when calculating percentages.
The questionnaire asked whether the CYP had received any of the following treatments/assessments: activity management, cognitive–behavioural therapy (CBT), domiciliary assessment or treatment, graded exercise therapy, medication for symptoms
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CBT was deprecated. GET is contraindicated. No drug is approved or recommended under either NICE guidelines.

Somehow the lead investigator of the study is one of the main actors responsible for this disaster, and even they can't spin this in a positive way. But she'll get more funding regardless, once again presenting a new approach that is the same as the old approach.

This is a level above being a study of failure, it's a study of people who fail so miserably they don't even realize they are failing. It's failure studying itself, and failing to see that it is failing. Remarkable, in all the worst possible ways.
 
Lucibee said:
"Contributors: This study was conceived and designed by Professor Esther Crawley and AB [Amberly Bridgen]."

Authorship is, and should be, about who actually writes the paper. She probably takes the same line as Wessely - already on sooooo many papers that she doesn't need her name to be on any more thank you very much.
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There could be other reasons why she might not put her name to the paper. For example she might not agree with the contents.
 
Dolphin said:
There could be other reasons why she might not put her name to the paper. For example she might not agree with the contents.
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Having more papers with different author lists creates the idea of a wider movement rather than a single group of researchers pushing forward under the lead of just one high profile academic.

Also even Prof Crawley must be aware that she has become toxic at least to the patient community so it may be felt that having some papers not immediately attracting the adverse scrutiny arising from being associated with her name is helpful.
 
rvallee said:
So, this is a study of care providers using a now defunct guideline who show no useful knowledge of ME, severe or otherwise.

So this is by definition only patients who are under someone's care, which is a minority, and they are overseen using a harmful care model that was deprecated between the time they started the study and publication. It's not accurate to say that 2/3 severe patients are under specialist management, it's only 2/3 who were seen using this clinical pathway by this group. From people who overuse statistics this much, they don't seem to understand that accurate denominators are important when calculating percentages.

CBT was deprecated. GET is contraindicated. No drug is approved or recommended under either NICE guidelines.

Somehow the lead investigator of the study is one of the main actors responsible for this disaster, and even they can't spin this in a positive way. But she'll get more funding regardless, once again presenting a new approach that is the same as the old approach.

This is a level above being a study of failure, it's a study of people who fail so miserably they don't even realize they are failing. It's failure studying itself, and failing to see that it is failing. Remarkable, in all the worst possible ways.
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There is a big and important underlying question too:

Would any of those who had been harmed and were at the brunt end of all of this feel safe thinking 'yes I'll just send back a questionnaire to Crawley and those who treated me saying how I didn't enjoy it and it harmed me' ?

In essence this seems to be a culmination of those who picked the only two other viable options - ignore or reply and say what they want to hear.

There needs, given what we all know has happened with the harm caused by those running the system in the past, to at least be a safe and independent route for collecting data on it.

It's outrageous that after what has been someone can claim this is 'representative' without 'making it safe' for honest replies.

I'd actually be intrigued for us to list here what that 'being safe' would require as a brainstorm - thinking out whether any of that data going to those who delivered the treatment could ever possibly be the way given circumstances?
 
Peter Trewhitt said:
Having more papers with different author lists creates the idea of a wider movement rather than a single group of researchers pushing forward under the lead of just one high profile academic.

Also even Prof Crawley must be aware that she has become toxic at least to the patient community so it may be felt that having some papers not immediately attracting the adverse scrutiny arising from being associated with her name is helpful.
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Similar to Wessely and PACE being his brain-child, having heavily participated in it, but did not put his name anywhere near the published material, then wrote an editorial pretending to be some neutral observer marveling at how great a study it was.

Given her role, Crawley should have been a lead investigator on Clock, and yet she wasn't. Now this. And she wasn't on the Magenta paper either. It's clear that she has lost a bit of influence. If only the money could stop flowing to her now.
 
Sid said:
She is an author on the Magenta paper.
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I mean she wasn't lead author. She has basically been the go-to expert on pediatric "chronic fatigue" for decades, and was probably lead author on more papers than any other research has authorship. She wasn't on Clock either, instead it was some other pediatrician who never did any research on anything to do with ME. I don't know what it means exactly, but it definitely means something.
 
rvallee said:
I mean she wasn't lead author. She has basically been the go-to expert on pediatric "chronic fatigue" for decades, and was probably lead author on more papers than any other research has authorship. She wasn't on Clock either, instead it was some other pediatrician who never did any research on anything to do with ME. I don't know what it means exactly, but it definitely means something.
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Plausible deniability ?
 
Given

that social services involvement is usually a negative experience and can result in children being removed

That specialist ME/CFS services are usually ( and at 2018-19) not a good thing

I wonder at the framing of this .

The problem is not simply availability, it's what's offered

The provision of GET to the severe is simple malpractice

ETA , GET is still there , it's simply been rebranded
 
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