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CFS or ME is different in children compared to in adults: a study of UK and Dutch clinical cohorts

Dolphin

Senior Member
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17,567
Free full text: http://bmjopen.bmj.com/content/5/10/e008830.full

BMJ Open 2015;5:e008830 doi:10.1136/bmjopen-2015-008830
  • Epidemiology
Chronic fatigue syndrome (CFS) or myalgic encephalomyelitis (ME) is different in children compared to in adults: a study of UK and Dutch clinical cohorts
  1. Simon M Collin1,
  2. Roberto Nuevo1,
  3. Elise M van de Putte2,
  4. Sanne L Nijhof2,
  5. Esther Crawley1
+Author Affiliations

  1. esther.crawley@bristol.ac.uk
  • Received 19 May 2015
  • Accepted 24 July 2015
  • Published 28 October 2015
Abstract
Objective To investigate differences between young children, adolescents and adults with chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME).

Study design Comparison of clinical cohorts from 8 paediatric and 27 adult CFS/ME services in the UK and a paediatric randomised controlled trial from the Netherlands. Outcome measures include: fatigue (the UK—Chalder Fatigue Scale); Disability (the UK—SF-36 physical function subscale; the Netherlands—CHQ-CF87); school attendance, pain, anxiety and depression (the UK—Hospital Anxiety & Depression Scale, Spence Children's Anxiety Scale; the Netherlands—Spielberger State-Trait Anxiety Inventory for Children, Children's Depression Inventory); symptoms; time-to-assessment; and body mass index. We used multinomial regression to compare younger (aged <12 years) and older (aged 12–18 years) children with adults, and logistic regression to compare UK and Dutch adolescents.

Results Younger children had a more equal gender balance compared to adolescents and adults. Adults had more disability and fatigue, and had been ill for longer. Younger children were less likely to have cognitive symptoms (OR 0.18 (95% CI 0.13 to 0.25)) and more likely to present with a sore throat (OR 1.42 (1.07 to 1.90). Adolescents were more likely to have headaches (81.1%, OR 1.56 (1.36% to 1.80%)) and less likely to have tender lymph nodes, palpitations, dizziness, general malaise and pain, compared to adults. Adolescents were more likely to have comorbid depression (OR 1.51 (1.33 to 1.72)) and less likely to have anxiety (OR 0.46 (0.41 to 0.53)) compared to adults.

Conclusions Paediatricians need to recognise that children with CFS/ME present differently from adults. Whether these differences reflect an underlying aetiopathology requires further investigation.

Trial registration numbers FITNET trial registration numbers are ISRCTN59878666 andNCT00893438. This paper includes secondary (post-results) analysis of data from this trial, but are unrelated to trial outcomes.
 

Dolphin

Senior Member
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17,567
All the symptom data from the 8 paediatric and 27 adult CFS/ME services in the UK from August 2004 to October 2014.

The following questions may be unreliable because of how they were assessed: "In the UK, adult and paediatric services also prospectively asked patients whether they had received a diagnosis of depression or anxiety, migraine, irritable bowel syndrome, Fibromyalgia or Chronic Regional Pain Disorder." This means people could have had them but not been marked down as having them. The HADS and SCAS scores were part of the assessment so weren't based on past assessments.

Patients were diagnosed with the Fukuda or NICE criteria.

Collin Table 1.png
 

Dolphin

Senior Member
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17,567
They also compared UK and Dutch adolescents. However, this comparison is messy because:
(i) Apart from the presence of symptom questions, the questions used different questionnaires.
(ii) The Dutch patients were part of a trial so if it was felt they had a psychiatric disorder, they could b excluded from the study

Here's what the paper said on the comparison:
Adolescents from the UK and the Netherlands presented with similar symptoms, although headaches were less common, and postexertional malaise, severe pain and mood disorders were more common in UK patients. The difference in postexertional malaise could be explained by the fact that this is considered a cardinal symptom in the UK. The lower prevalence of anxiety and depression in the Dutch cohort could be because high scores for mood disorder were a reason for further psychological assessment to exclude primary mood disorders, leading to possible exclusion from the study. Also, the UK and Dutch cohorts used different instruments to measure mood (and pain), and this is likely to contribute to discrepancies when these measures are categorised to indicate presence/absence (or degree) of symptoms.
 

Dolphin

Senior Member
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17,567
Here's what they say on the differences between younger and older children:
The differences in symptom presentation that we found between younger and older children were not found in our previous comparison of these age groups,10 probably because this earlier study was insufficiently powered to detect such differences. It is striking that the gender balance is more equal in younger children (57% female), compared to adolescents (74.1%) and adults (77.9%). This shift in gender balance is consistent with population data, where an equal gender balance (49.6% female) has been described in a population cohort at age 13 years.32 Studies recruiting teenagers from secondary schools have consistently shown a female-to-male ratio of approximately 3:1,33 ,34 suggesting that, during adolescence, the prevalence of CFS/ME increases in females but not in males.35
 

Dolphin

Senior Member
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17,567
Judging by the pre-publication comments, in an earlier version they said such differences could mean adults and children should get different treatments but this was dropped from the final version.

One of the authors, Esther Crawley, said this previously as justification for testing the lightning process in children.
 
Last edited:

Dolphin

Senior Member
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17,567
One potentially interesting thing is they resisted talking about CFS or ME as a functional somatic syndrome/similar. From the pre-publication history:

12. Overall, the discussion is quite short focusing on the strengths and some of the limitations of the study. The authors need to spend more time critically discussing their findings compared to the current literature as well as possible clinical implications of their findings. E.g., the authors describe the more equal gender balance in younger children as striking. However, this result is very similar to what has been reported for other functional syndromes such as for instance functional abdominal pain or medically unexplained symptoms as such. Here an interpretation of the results based on the broader research area of various functional syndromes could be relevant. Also, I miss more discussion of why there might be these different phenotypes of CFS across the life span - could it be due to developmental issues including biological factors etc.

Response: We intentionally avoided over-speculation regarding what are, in essence, descriptive findings. This is because the causes of diverging prevalence of even well-characterised illnesses such as depression are purely speculative. We are also not so keen on CFS being lumped in under the ‘functional somatic syndromes’ umbrella. However, this is an aspect of our Discussion that we would be willing to re-visit if necessary.
 

Dolphin

Senior Member
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17,567
They don't adjust for multiple testing, which may be reasonable:

From the pre-publication history:

7. In the section on the statistical analyses, no information on the chosen significance level is given -please add this information. Also, handling of multiple testing is not commented on; please elaborate on this potential multiple testing problem with the performed analyses.

Our results are presented with 95% confidence intervals (implying a 5% level of significance), and we do not use a P-value threshold to discriminate (statistically) ‘significant’ from ‘non-significant’ results. Instead, we interpret our results on the basis of the magnitude of differences and the precision of our estimates (as quantified by a 95% confidence interval).
 

daisybell

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New Zealand
I would be interested to know how they can be sure that the incidence of cognitive dysfunction is lower in children <12 years old. How many children would accurately be able to describe cognitive change or even be aware of change?? I'm not sure that I would have been able to report cognitive change as a child, particularly if asked as a young child.... I can't see any measure of cognition being used, so presumably this is self-report?
 

Dolphin

Senior Member
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17,567
I would be interested to know how they can be sure that the incidence of cognitive dysfunction is lower in children <12 years old. How many children would accurately be able to describe cognitive change or even be aware of change?? I'm not sure that I would have been able to report cognitive change as a child, particularly if asked as a young child.... I can't see any measure of cognition being used, so presumably this is self-report?
Good point, I think. I think this sort of problem could affect lots of their comparisons.

No tests of cognition mentioned so self-report (perhaps parents also commented in assessment but no mention).
 

Denise

Senior Member
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1,095
Assessing cognitive function and accurately characterizing PEM are both difficult.

Several young people (with ME) who I know cannot describe their physical or cognitive impairments - they may only be able to say they don't feel well/at baseline.

One young person I know, for instance, one day said "My head feels different."
When asked for more details, they couldn't describe what was different, they could only say that it felt much better. But they had never identified their head as hurting before this one instance where it felt better.
The following day it was back to usual and has remained.
Sadly, because no influencing factors could be identified, no reasonable steps could be taken to alleviate the headache.

Similarly, some young people can only say that their ability to think/process info is off - they are unable to be more specific.

And time likely plays a role also. Many young people who have been sick for a long time, forget what normal feels like which also makes it difficult to say what does/doesn't feel right.
 

SOC

Senior Member
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7,849
And time likely plays a role also. Many young people who have been sick for a long time, forget what normal feels like which also makes it difficult to say what does/doesn't feel right.
This is a very good point. My daughter has had ME half her life. She is currently functioning relatively normally -- married, working full-time, etc. I asked her if she felt she was finally back to normal and she replied, "I don't remember what normal feels like anymore. All I can say is that I feel better than I did when I was sicker." It breaks my heart that this is the world my ME kid lives in, even though she's doing very well at the moment. She doesn't remember what it's like to feel truly healthy. :(
 

CantThink

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England, UK
I would be interested to know how they can be sure that the incidence of cognitive dysfunction is lower in children <12 years old. How many children would accurately be able to describe cognitive change or even be aware of change?? I'm not sure that I would have been able to report cognitive change as a child, particularly if asked as a young child.... I can't see any measure of cognition being used, so presumably this is self-report?

When I got ill at age 10, I was able to understand that something was different cognitively, yet I don't remember ever describing it or being asked.

With hindsight years later, I remember feeling very fogged, and my ability to do maths and in particular mental arithmetic completely vanished. I couldn't hold numbers whilst working on the next part of an equation for example. By the time I'd worked out the next part, I'd totally forgotten the answer to the first part. Seemingly overnight, I completely lost the ability to do long division and fractions. It was weird and I remember being upset because I couldn't keep up with peers who I had previously academically equalled.

My brain seemed to have slowed down a lot. I felt as if I was wading through mud while trying to process thoughts when addressing schoolwork questions/problems etc. If more than one person spoke I had trouble following. My mother was a teacher and would give me the schoolwork to do to try to keep up when I was housebound. Half the time I didn't do it. I was so physically shattered that my mind was exhausted too - she'd come in and find me lying asleep on the floor next to the table! Thsnkfully she never tried to make me - she could see I was too ill.

No one ever asked me about it. They were just interested in physical wellness - as in was I well enough to go to school. I'm not so sure I really understood or recognised exactly what was happening. It was only recently that I realised how odd and abnormal it was.
 

Snow Leopard

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South Australia
One of the authors, Esther Crawley, said this previous as justification for testing the lightning process in children.

Skating on thin ethical ice...

I asked her if she felt she was finally back to normal and she replied, "I don't remember what normal feels like anymore. All I can say is that I feel better than I did when I was sicker." It breaks my heart that this is the world my ME kid lives in, even though she's doing very well at the moment. She doesn't remember what it's like to feel truly healthy. :(

Yes, it sucks!

I simply can't remember what it is like to be "normal". I am however sure that it is not normal to have a constant headache, aching muscles and brainfog for 15 years continuously.
 

A.B.

Senior Member
Messages
3,780
I started having abnormal fatigue at age 15 and definitely did not fully realize how abnormal it was. My guess is that younger people have very little awareness about the existence of chronic disease and may not interpret symptoms as disease but rather find them confusing. If the family is not open to the possibility of disease, they may also lack the self confidence needed to fully express how unwell they're feeling.

If my guess is correct, children should make easy targets for symptom denying psychoquackery. It's very sad.
 

Simon

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Location
Monmouth, UK
It is striking that the gender balance is more equal in younger children (57% female), compared to adolescents (74.1%) and adults (77.9%). This shift in gender balance is consistent with population data, where an equal gender balance (49.6% female) has been described in a population cohort at age 13 years.32 Studies recruiting teenagers from secondary schools have consistently shown a female-to-male ratio of approximately 3:1,33 ,34 suggesting that, during adolescence, the prevalence of CFS/ME increases in females but not in males.35

I think the gender imbalance, with roughly 3x as many women as men, is one of the most striking and understudied aspect of the illness. That this new study also finds the difference emerging at around puberty adds weight to the importance of sex differences. Typically, but not always, autoimmune diseases affect more women than men.

See this Norwegian study that found the same thing (ref 1 in above study), again with the sex differencce at around the time of puberty.

Copied from elsewhere:
This graph of new cases from a large Norwegian study (discussed on this thread):
s12916-014-0167-5-1.jpg


[Figure 1 from: Two age peaks in the incidence of chronic fatigue syndrome/myalgic encephalomyelitis: a population-based registry study from Norway 2008-2012]