Long Term Follow up of Young People With Chronic Fatigue Syndrome Attending a Pediatric Outpatient Service, 2019, Rowe

Andy

Andy

Retired committee member
Aim: To determine the reported duration of illness, the functional and educational long-term outcomes, predictive factors for recovery and seek feedback regarding management in pediatric/adolescent myalgic encepahalomyelitis/chronic fatigue syndrome (ME/CFS).

Methods: A cohort observational study of 784 young people, mean age 14.6 (6–18) years, with ME/CFS diagnosed at a specialist pediatric hospital and receiving regular care, was conducted with follow-up for a mean 8 (range 1–21) years after onset. Baseline symptoms, history, depression and anxiety questionnaires were available from 418. The remaining 366, did not have similar standardized baseline information. Questionnaires requested functional rating, persistent symptoms, duration of illness if “recovered,” social engagement and school/work attendance. Feedback was sought regarding management, support services, useful information, helpful interventions or personnel and use of alternative therapies. Reported recovery and function were compared with baseline information and between the two groups.

Results: Follow-up data were returned from 81.8%. There was no significant difference in functional score (if reported recovery) or illness duration related to provision of baseline data. The mean duration of illness was 5 (range 1–15) years in the 50% who reported recovery. By 5 years 38% and by 10 years 68% reported recovery. At 10 years the mean functional score was 8/10 (range 2–10) with 5% scoring <6. Depression, anxiety or severity of illness at diagnosis was not predictive of non-recovery. Designing and monitoring their own management plan that included educational, social, physical and enjoyable activities, as well as having symptom management and understanding professionals were highly valued. However, remaining engaged in an education system that flexibly accommodated their illness and aspirations was consistently reported as crucial for long term functioning.

Conclusions: ME/CFS in young people has a mean duration of 5 years (1–15) with 68% reporting recovery by 10 years. All improved functionally with 5% remaining very unwell and a further 20% significantly unwell. There were no obvious baseline predictors for recovery. However, depression, anxiety, orthostatic intolerance and to a lesser extent pain at follow up were identified as hampering recovery or function. Supportive professionals, remaining engaged in education and management strategies were identified as helpful.
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Open access at https://www.frontiersin.org/articles/10.3389/fped.2019.00021/full
 
My personal experience, which of course is only n=1, would be that, between 11 to 15 years after what I believe to be the start of my ME, I would have said that I was recovered. But looking back, that was only because I had improved so much compared to how I was, and, obviously, I subsequently relapsed.

The author does seem to cover that point though
The range of ME/CFS Functional score for those who reported that they had recovered was compared with those who reported they had not recovered. (Figures 5 and 6) There was a significant overlap. Some scored low due to other illnesses that they differentiated from CFS. Others reported that they did not know what was “normal” as it had been so long since they were well. They reported that “how a 22 year old spent their energy was different from 15 year, but they were not sure what that should be.” Others, who reported themselves as “well,” felt that they were managing well, but their parents made the comment that they “did not think that they had the stamina that they had demonstrated as an adolescent.” Some needed to be “perfect” to describe themselves as “well” whereas others compared themselves with how they were when they were first unwell and were very grateful to be able to do what they were currently able to do. Some who felt they had recovered also scored lower, as they were caring for small children, were working part time and felt sleep deprived. Others admitted that they were dealing with depression as an additional issue. Hence many factors influenced whether recovery was reported, as well as whether the level of functioning was solely related to ME/CFS.
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Interesting to see what is said about alternative therapies.
Use of Alternative Therapies
Seventy per cent of the young people used alternative therapies and these included 40 different types, some trying up to 10 different ones. The therapies ranged from naturopathy, chiropractics, homeopathy, Chinese herbs, intravenous vitamins, Reiki, Qi Gong, Tai Chi, Yoga, Myotherapy, Bowen therapy, massage, hypnosis, cupping, aromatherapy, color therapy, meditation etc. There were very few therapies that were considered of any value by the young people. The only ones that approached a 30% positive response involved massage (under a variety of different guises) for those with muscle pain and “good dietary advice” often in young people with associated abdominal discomfort. The most common comment was that they “wished their parents had not wasted their money or their energy by taking them to people who had promised to cure them but didn't.”
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On initial reading the abstract seems to include a contradiction. It says that 50% reported recovery. But then it says 68% were recovered at 10 years. Moreover, although it says that the mean duration was 5 years (1-15) for those reporting recovery in the results in the conclusion it says mean duration was 5 years without reference to recovery. And of course these figures only apply to the 81% that replied.

On re-reading it is clear that the figures do not apply quite as simply as it might seem but to be honest i cannot quite work out how they do apply and there remain inconsistencies. This manuscript should have been sent back for clarification.
 
I think the following was an interesting observation.
The range of ME/CFS Functional score for those who reported that they had recovered was compared with those who reported they had not recovered. (Figures 5 and 6) There was a significant overlap.

This coincides with what professor Kristian Sommerfelt (paediatrician and neurologist with over 20 years experience with children and adolescents with ME at Haukeland university, Norway) said in a recent lecture. That his patients have a tendency to overestimate their level of function. If he asks a patient how often he goes to school, he might say once a week, but when he asks the parents they might say it's much more seldom.

If he asks how they're doing, they might answer "good", but that might just mean "good compared to how it was at its worse".

ETA: @Andy had already noticed and quoted the section about the overlap above.
 
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Shocked to see these results of 68% recovery for young people against the 5-8% for adults with mean scores 8/10 functionality. @Andy I accept that there is some remitting/relapsing in a portion of people, but 60% remitted and didn't relapse after 10 years? I find that strange.

All I can see is that there are different conditions and one improves over time is more common in children. Or, ME/CFS is a common diagnostic end point for various conditions all of which are more likely to improve over time. Or, whatever ME/CFS is, is also a fairly common state of being in childhood.

I always go back and forth if ME/CFS is one very strange condition or many different diseases. I suppose I won't know until I know.
 
Kalliope said:
I think the following was an interesting observation.
The range of ME/CFS Functional score for those who reported that they had recovered was compared with those who reported they had not recovered. (Figures 5 and 6) There was a significant overlap.

This coincides with what professor Kristian Sommerfelt (paediatrician and neurologist with over 20 years experience with children and adolescents with ME at Haukeland university, Norway) said in a recent lecture. That his patients have a tendency to overestimate their level of function. If he asks a patient how often he goes to school, he might say once a week, but when he asks the parents they might say it's much more seldom.

If he asks how they're doing, they might answer "good", but that might just mean "good compared to how it was at its worse".
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Both groups look like they have improved, but the recovered group looks like they average a little above a 9 and the unrecovered looks like they average around a 6.5. That's a pretty big difference.
 
Some background: this is not Peter Rowe from John Hopkins but Kathy Rowe from Australia. She is currently on the NHMRC committee.

In my comment on the NHMRC draft report, I criticized the following sentence about adolescents with ME/CFS: "By 5 years, 60% reported recovery and by 12 years 88% reported recovery." I could track the reference for these figures to an oral presentation by Kathy Rowe at the IACFS/ME Biennial International Conference Ottawa, Ontario, Canada in 2011. I criticized that these results "have not been published in a peer review article where the trials methodology can be checked and criticized by other scientists."

I'm pleased that Professor Rowe responded to my concerns so swiftly... :)

No seriously, it seems that she managed to publish her results, after all, 8 years later. The recovery rates have dropped a little bit but are still very high.
 
dreampop said:
Both groups look like they have improved, but the recovered group looks like they average a little above a 9 and the unrecovered looks like they average around a 6.5. That's a pretty big difference.
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I agree the averages look significantly different, but the thing that puzzles me from looking at the graphs is that some people classed as recovered have scores lower than some people classed as not recovered. There seems to be considerable overlap. I have not read the full paper, so I don't know how recovery was defined.
 
dreampop said:
All I can see is that there are different conditions and one improves over time is more common in children. Or, ME/CFS is a common diagnostic end point for various conditions all of which are more likely to improve over time. Or, whatever ME/CFS is, is also a fairly common state of being in childhood.
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An alternative possibility is that ME is a pattern of breakdown of signalling in a complex control system (maybe neurological or immunological or both). That reversibility of that pattern may depend on the state of maturation or development of the system. Early on there may be ways to re-route around the problem but once the system is fully in place that may be much more difficult.
 
Jonathan Edwards said:
An alternative possibility is that ME is a pattern of breakdown of signalling in a complex control system (maybe neurological or immunological or both). That reversibility of that pattern may depend on the state of maturation or development of the system. Early on there may be ways to re-route around the problem but once the system is fully in place that may be much more difficult.
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Are there any examples of better understood illnesses with that characteristic, and if so could there be any clues in that?
 
Others reported that they did not know what was “normal” as it had been so long since they were well. They reported that “how a 22 year old spent their energy was different from 15 year, but they were not sure what that should be.” Others, who reported themselves as “well,” felt that they were managing well, but their parents made the comment that they “did not think that they had the stamina that they had demonstrated as an adolescent.” Some needed to be “perfect” to describe themselves as “well” whereas others compared themselves with how they were when they were first unwell and were very grateful to be able to do what they were currently able to do. Some who felt they had recovered also scored lower, as they were caring for small children, were working part time and felt sleep deprived. Others admitted that they were dealing with depression as an additional issue. Hence many factors influenced whether recovery was reported, as well as whether the level of functioning was solely related to ME/CFS.
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The concept of "recovery" is always sketchy when you have been ill for so long and no real point of comparison to know what that is.
 
strategist said:
Reading this makes me think I have some other illness than these patients. Why am I not getting better? One could say that in some aspects I have improved, but there is clearly no general improvement, more a trend to decline further.
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It wasn't obvious for me which criteria had been used when the participants were diagnosed. I found this quote:

- The clinic has been functioning since 1989. In the early years of the clinic, the Holmes definition and Fukuda criteria for CFS were available
 
The authors do not give a precise definition of recovery. It seems like they simply asked patients, and because the clinicians and parents sort of made the same conclusion and school attendance increased, this was deemed reliable.

They say they had regular feedback concerning management and documentation of any relapses, but do not explain if this information was used to define recovery. For example, whether a patient had to report recovery at multiple follow-up assessments.

Several passages in the paper seem to suggest that relying on the patients account of being recovered is doubtful. For example:
As some young people found the concept of recovery difficult, they were asked to describe how they thought they were functioning with a rating of 10 being “very well” or “back to normal” and 1 being “bedridden.”
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Or:
The range of ME/CFS Functional score for those who reported that they had recovered was compared with those who reported they had not recovered. (Figures 5 and 6) There was a significant overlap. Some scored low due to other illnesses that they differentiated from CFS. Others reported that they did not know what was “normal” as it had been so long since they were well.
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And:
Others, who reported themselves as “well,” felt that they were managing well, but their parents made the comment that they “did not think that they had the stamina that they had demonstrated as an adolescent.”
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Might have been better to speak of remission or return to normal functioning instead of recovery, because that suggests that the illness is gone. I've heard from several ME/CFS patients that they once thought they were recovered and then relapsed several years later.
 
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Jonathan Edwards said:
Yes, on re-reading it becomes clearer what the sentences mean. And I guess that reading the full paper would help. However, as it stands the abstract is pretty opaque and I am still not convinced that the way the data are presented is always legitimate.
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I would think that still leaves the possibility that the work around to parts of a system can also then experience problems in time too eventually.
 
Barry said:
Are there any examples of better understood illnesses with that characteristic, and if so could there be any clues in that?
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Immunological problems in childhood or adolescence that peter out in adulthood are reasonably common. Juvenile inflammatory arthritis often peters out, whereas adult rheumatoid tends not to. Childhood allergies and asthma may peter out whereas adult asthma probably less often does.

Neurological damage in children can be followed by major recovery of a sort less likely in the adult, but it depends a lot on the structural basis of the damage. I suspect the neurological context shows fewer convincing examples.
 
Kalliope said:
This coincides with what professor Kristian Sommerfelt (paediatrician and neurologist with over 20 years experience with children and adolescents with ME at Haukeland university, Norway) said in a recent lecture. That his patients have a tendency to overestimate their level of function.
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There is this famous study by David Bell who followed up on the adolescents who got sick in the Lyndonville outbreak. More than a third considered themselves recovered after 13 years. They had improved, but when Bell dug deeper, he found that they still had significant health problems:

"Some of the young adults rated their health as ‘good’, while the amount of activity they could perform was minimal. They had become so used to their performance level, they accepted it as their new normal. Unfortunately, many of them, while they described their health as good, they were unable to work full time or carry on other duties."​

Source: https://www.omf.ngo/2016/08/01/prognosis-of-mecfs/
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Bell wrote a paper on this, together with Leonard Jason. They showed that those who were deemed recovered still reported many symptoms and looked more like those who retained the CFS diagnosis than healthy controls.

Source: https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3940158/


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Does medicine have a definition of recovery for other diseases where objective measures may not be applicable?

I don't know, but one hears about blood counts for some diseases as a measure of at least remission, if not recovery.

The recovery numbers in this study do seem very high, given what we know about ME.

And, alternative medicine - maybe I'm being too cynical, but I would agree with the note in this study that many of these therapies are a waste of time and money.
 
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