Professor Crawley BPSU study - Chronic fatigue syndrome (CFS) Severe children Feb 2019 - 2020

[Andy]

Couldn't find the REC details but did find this, which is part of the funding for this 'study'

Career Development Award
Investigating the treatment of paediatric chronic fatigue syndrome or myalgic encephalomyelitis (CFS/ME).

Plain English Summary:
Paediatric chronic fatigue syndrome or myalgic encephalomyelitis (CFS/ME) is common and disabling yet little is known about recovery, whether Graded Exercise Therapy (GET) is effective, or what treatment strategies might work in children who are severely affected by CFS/ME. This programme of work will: determine what is known about recovery and how to define it; investigate the Minimal Clinically Important Difference in outcomes; study how to deliver GET; conduct a multicentre trial investigating the effectiveness of GET compared with activity management for mildly and moderately affected children, and start the work needed to establish a trial for severely affected children.

Award ID:SRF-2013-06-013

Active Award
The Senior Research Fellowship is a senior training award over 5 years for outstanding researchers aiming towards a Chair with the potential to become academic and research leaders within the award time.

https://www.fundingawards.nihr.ac.uk/award/SRF-2013-06-013

 

[Snowdrop]

In the abstract the Avon Longitudinal study is referenced. I don't know what this is about but thought I'd link to it: http://www.bristol.ac.uk/alspac/

I feel great sympathy for all parents with children who have to navigate these shark infested waters.
Historically it is normal for a paediatrician to report things like measles and was for the good but this 'reporting' of children has such a sinister undertone it's quite horrible.

I also wondered while trying to read about the study how many children were to be included as a full research compliment. I did not see any numbers. And given that the research is always at Bristol U where do they come up with all the participants for all the studies. Can they reuse previous study participants?

If not then I would presume that there is a regular set of newly ill children in Bristol and given that so many remain ill this should be alarming even to those who think they can help.

I find the focus on functioning to be unhelpful and even cruel. There is no concern for the child and their health just a particular narrow focus on getting them back to school. Being in school is important but when a child is so severely ill it becomes a secondary issue to the well-being of the child. Their concern is not well being but functioning according to what is expected of them. Seriously mean people.

 

[Trish]

I also wondered while trying to read about the study how many children were to be included as a full research compliment. I did not see any numbers. And given that the research is always at Bristol U where do they come up with all the participants for all the studies. Can they reuse previous study participants?

As I understand it, it is paediatricians from all over the UK and Ireland who will be asked to fill in information about every severe CFS patient they see in a one month period. So they can't set a number, as the study is designed to find out how many there are. It's not a clinical trial, it's an epidemiological study to find out numbers, what treatment they are getting and how they are a year later.

 

[Snowdrop]

As I understand it, it is paediatricians from all over the UK and Ireland who will be asked to fill in information about every severe CFS patient they see in a one month period. So they can't set a number, as the study is designed to find out how many there are. It's not a clinical trial, it's an epidemiological study to find out numbers, what treatment they are getting and how they are a year later.

Ah, thanks for that @Trish . Will have to go back and have another look.

 

[It's M.E. Linda]

As I understand it, it is paediatricians from all over the UK and Ireland who will be asked to fill in information about every severe CFS patient they see in a one month period. So they can't set a number, as the study is designed to find out how many there are. It's not a clinical trial, it's an epidemiological study to find out numbers, what treatment they are getting and how they are a year later.

Apologies - Working with a depleted Braincell at present:

But if the study is ‘designed to find out how many (severe patients) there are’ in the U.K. - how can they limit this research to a single ‘one month period’? How many children are they going to miss from every clinic because they did not have (/could not attend) an appointment in that particular month?

Am I being dim?

 

[Tilly]

As I understand it, it is paediatricians from all over the UK and Ireland who will be asked to fill in information about every severe CFS patient they see in a one month period. So they can't set a number, as the study is designed to find out how many there are. It's not a clinical trial, it's an epidemiological study to find out numbers, what treatment they are getting and how they are a year later.

But to anyone that would not make sense at all ever? How on earth do they expect to get correct figures and a follow up in what?

If they are bedbound the GP or Paediatric would not see them. Then we have a problem with Drs not understanding the severity and then what if no one turns up in that month anywhere else and only her figures, which she can manipulate as she has done in the past. How can we be sure; due to no safeguarding of information by the patient or parent that true understanding is going to be given?

 

[Andy]

"The fatigue is made worse by activity and is not relived by rest."

Don't know about anyone else, but I relive my fatigue every day, regardless of rest. If fatigue is not "relived" by rest, doesn't that mean that it goes away with rest, and therefore by definition the patient would not have ME/CFS? I'm confused.

Hahaha, perhaps Crawley or a minion of hers is reading this thread, that typo has been corrected now. It now reads "The fatigue is made worse by activity and is not relieved by rest."

 

[Trish]

But if the study is ‘designed to find out how many (severe patients) there are’ in the U.K. - how can they limit this research to a single ‘one month period’? How many children are they going to miss from every clinic because they did not have (/could not attend) an appointment in that particular month?

Am I being dim?

But to anyone that would not make sense at all ever? How on earth do they expect to get correct figures and a follow up in what?

Just to be clear, I was simply trying to clarify what the study was claiming to do. I was not defending the study.

I think it looks so flawed as to be useless. First, it doesn't define ME properly, second, it doesn't define severe ME properly, third, it will miss the really severe cases never seen by paediatricians, fourth, it's depending on paediatricians who may not recognise ME at all to carry it out properly, fifth, it relies on them doing a thorough and accurate follow up appointment with the patient a year later, etc etc.

Doing it for just a month is probably standard practice for an epidemiology study. If every paediatrician did this for a single month then it should smooth out any individual variations and get a fairly good picture of prevalence of new severe ME patients (as defined by Crawley) seen by paediatricians in a one month period - multiply by 12 to get the annual incidence.

 

[Lacey Diamond]

In the abstract the Avon Longitudinal study is referenced. I don't know what this is about but thought I'd link to it: http://www.bristol.ac.uk/alspac/

I feel great sympathy for all parents with children who have to navigate these shark infested waters.
Historically it is normal for a paediatrician to report things like measles and was for the good but this 'reporting' of children has such a sinister undertone it's quite horrible.

I also wondered while trying to read about the study how many children were to be included as a full research compliment. I did not see any numbers. And given that the research is always at Bristol U where do they come up with all the participants for all the studies. Can they reuse previous study participants?

If not then I would presume that there is a regular set of newly ill children in Bristol and given that so many remain ill this should be alarming even to those who think they can help.

I find the focus on functioning to be unhelpful and even cruel. There is no concern for the child and their health just a particular narrow focus on getting them back to school. Being in school is important but when a child is so severely ill it becomes a secondary issue to the well-being of the child. Their concern is not well being but functioning according to what is expected of them. Seriously mean people.

Yes it is worrying. Classically, if there is a cure for an illness then you enforce notification e.g. Tb can be cured (in most cases) so you make it obligatory. However e.g. AIDS in the 90s there was no cure or treatment yet and so there was no notification of it because otherwise you are conducting a witch hunt, segregating ill people and creating an underclass shunned by society. EC does not seem to worry about these things.

1. I think we need someone who has the ability to look into the ethics approval that has been obtained because that is wrong, the patients are known due to the NHS number being recorded and the whole study design makes it impossible to give input or deny access.

2. In practice (my daughter is in her clinic and has tutoring from the hospital school) I can confirm that the hospital school have already been asked how many children are able to do less than 1 hour tuition a week (so they have already been collecting data before February 2019). Also just for the record her GP last saw her before diagnosis and has absolutely no idea what is going on in her life. The paediatrician also has not seen her and has given no input. She has been discharged and we have been left to fend for ourselves. Now they are asking these people who have left us to our own devices for their opinions?

 

[Lacey Diamond]

Hahaha, perhaps Crawley or a minion of hers is reading this thread, that typo has been corrected now. It now reads "The fatigue is made worse by activity and is not relieved by rest."

In the abstract the Avon Longitudinal study is referenced. I don't know what this is about but thought I'd link to it: http://www.bristol.ac.uk/alspac/

I feel great sympathy for all parents with children who have to navigate these shark infested waters.
Historically it is normal for a paediatrician to report things like measles and was for the good but this 'reporting' of children has such a sinister undertone it's quite horrible.

I also wondered while trying to read about the study how many children were to be included as a full research compliment. I did not see any numbers. And given that the research is always at Bristol U where do they come up with all the participants for all the studies. Can they reuse previous study participants?

If not then I would presume that there is a regular set of newly ill children in Bristol and given that so many remain ill this should be alarming even to those who think they can help.

I find the focus on functioning to be unhelpful and even cruel. There is no concern for the child and their health just a particular narrow focus on getting them back to school. Being in school is important but when a child is so severely ill it becomes a secondary issue to the well-being of the child. Their concern is not well being but functioning according to what is expected of them. Seriously mean people.

It would be useful to know if they are getting some funding via the Avon Longitudinal Study, does anyone know about this?

 

[Tilly]

Just to be clear, I was simply trying to clarify what the study was claiming to do. I was not defending the study.

I think it looks so flawed as to be useless. First, it doesn't define ME properly, second, it doesn't define severe ME properly, third, it will miss the really severe cases never seen by paediatricians, fourth, it's depending on paediatricians who may not recognise ME at all to carry it out properly, fifth, it relies on them doing a thorough and accurate follow up appointment with the patient a year later, etc etc.

Doing it for just a month is probably standard practice for an epidemiology study. If every paediatrician did this for a single month then it should smooth out any individual variations and get a fairly good picture of prevalence of new severe ME patients (as defined by Crawley) seen by paediatricians in a one month period - multiply by 12 to get the annual incidence.

I know you were not defending the trial. My remarks were sort of dumbfounded disbelief and trying to understand how on earth this got past any sort of ethics. To say I am flabbergasted is an understatement. I should be used to this by now.

 

[Andy]

It would be useful to know if they are getting some funding via the Avon Longitudinal Study, does anyone know about this?

Listed funding is

Funding
The study is funded by The National Institute for Health Research (Senior Research Fellowship, SRF-2013-06-013) and a grant from the Royal United Hospitals Bath NHS Foundation Trust.

 

[Sly Saint]

Hahaha, perhaps Crawley or a minion of hers is reading this thread

 

[Snow Leopard]

This study could be quite valuable if done correctly, but won't be because the people running the study don't bother to listen to patients when it comes to study design...

 

[Cinders66]

Because people with severe ME tend to drop off the radar and there haven’t been real attempts to identify numbers, presentation and outcomes this might have worth.

I’m concerned that asking about treatment and outcomes might make pediatricians whod other wise just support or do nothing be more proactive in an unhelpful way so we in effect get a trial of rehabilitation intervention with the children as guinea pigs, maybe that’s too dramatic.

I agree her definition of moderate is wrong but her “typical “ might be tired but doing sport?

 

[Unable]

Apologies - Working with a depleted Braincell at present:

But if the study is ‘designed to find out how many (severe patients) there are’ in the U.K. - how can they limit this research to a single ‘one month period’? How many children are they going to miss from every clinic because they did not have (/could not attend) an appointment in that particular month?

Am I being dim?

It’s a form of sampling. A snapshot used to extrapolate to a larger situation.

It might go something like this:
If you know how many patients were seen in total in that month, and you know what proportion of each surgery’s full complement of patients those patients make up, then if you also know the proportion of “patients seen” that are ME patients, in theory you have a means of multiplying the answers up, and so estimating the numbers of ME patients that are being seen nationally.

Of course as others have said the answers are likely to be inaccurate because:

- the method assumes that the frequency with which patients attend their doctors is constant (or at least random)
- ME gets defined variably from place to place
- Severe ME gets defined variably from place to place
- many Severe & Very Severe ME patients will have dropped out of NHS care entirely so NEVER get counted
- the misdiagnosis of ME patients with another condition
- undiagnosed patients are not counted (if doctors “don’t believe in ME” then their patients will not be counted).
- re-diagnosis of ME patients to another condition
- CFS (and hence ME) can sometimes be used as a waste-basket diagnosis

Estimating numbers is never easy, and every estimate made will be forced to make some assumptions in the process. If the assumptions are erroneous so the estimates are also likely to be out.

If all the assumptions are made in such a manner that they tend to skew data in one direction over another, then the estimates produced can become wildly out.

I suspect it is very difficult to ensure that the biases of the researchers concerned are adequately moderated. Hence in many walks of life we see wildly differing estimates of difficult to measure quantities.

ME patients are naturally going to be suspicious of what the estimates from this survey will produce - because of the history of the researcher involved.

 

[Snow Leopard]

Of course as others have said the answers are likely to be inaccurate because:

The problem extends to the followup, namely loss to followup and related biases in the case of the GP who enrolled the patient into the study was not helpful, or worse...

 

[Unable]

The problem extends to the followup, namely loss to followup and related biases because the GP who enrolled the patient into the study was not helpful, or worse...

Indeed! I didn’t even go that far... but YES!

 

[It's M.E. Linda]

Just to be clear, I was simply trying to clarify what the study was claiming to do. I was not defending the study.

Apologies @Trish, I understand.

I was only using your clear words to show how dumbfounded I felt.

The same with @Unable - thank you.

My main concern was all of the Missing Severe ‘patients’ who either currently are not seen by their Consultant or have been discharged to GP.

 

[MEMarge]

Part of this NIH research award work includes:

"investigate the Minimal Clinically Important Difference in outcomes; ..........and start the work needed to establish a trial for severely affected children."

https://www.fundingawards.nihr.ac.uk/award/SRF-2013-06-013

This award was supposed to end in Feb 2019, but I'm sure she will have other funds available.