Health, Wellbeing and Prognosis of Australian Adolescents with(ME/CFS): A Case-Controlled Follow-up Study, 2021, Josev et al

[Sly Saint]

Health, Wellbeing, and Prognosis of Australian Adolescents with Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS): A Case-Controlled Follow-Up Study

Abstract
Background: The purpose of this study was to follow-up an Australian cohort of adolescents newly-diagnosed with ME/CFS at a tertiary paediatric ME/CFS clinic and healthy controls over a mean period of two years (range 1–5 years) from diagnosis.

Objectives were to
(a) examine changes over time in health and psychological wellbeing,
(b) track ME/CFS symptomatology and fulfillment of paediatric ME/CFS diagnostic criteria over time, and
(c) determine baseline predictors of ME/CFS criteria fulfilment at follow-up.

Methods: 34 participants aged 13–18 years (25 ME/CFS, 23 controls) completed standardised questionnaires at diagnosis (baseline) and follow-up assessing fatigue, sleep quality and hygiene, pain, anxiety, depression, and health-related quality of life. ME/CFS symptomatology and diagnostic criteria fulfilment was also recorded.

Results: ME/CFS patients showed significant improvement in most health and psychological wellbeing domains over time, compared with controls who remained relatively stable. However, fatigue, pain, and health-related quality of life remained significantly poorer amongst ME/CFS patients compared with controls at follow-up. Sixty-five percent of ME/CFS patients at baseline continued to fulfil ME/CFS diagnostic criteria at follow-up, with pain the most frequently experienced symptom. Eighty-two percent of patients at follow-up self-reported that they still had ME/CFS, with 79% of these patients fulfilling criteria. No significant baseline predictors of ME/CFS criteria fulfilment at follow-up were observed, although pain experienced at baseline was significantly associated with criteria fulfilment at follow-up (R = 0.6, p = 0.02).

Conclusions: The majority of Australian adolescents with ME/CFS continue to fulfil diagnostic criteria at follow-up, with fatigue, pain, and health-related quality of life representing domains particularly relevant to perpetuation of ME/CFS symptoms in the early years following diagnosis. This has direct clinical impact for treating clinicians in providing a more realistic prognosis and highlighting the need for intervention with young people with ME/CFS at the initial diagnosis and start of treatment. View Full-Text

https://www.mdpi.com/2077-0383/10/16/3603

 

[Wyva]

They used the Canadian Consensus Criteria.

This is what they said at the end:

"Given the observed persistence of ME/CFS symptoms, poorer health and reduced psychological well-being at follow-up compared with healthy controls (i.e., fatigue, pain, and health-related quality of life problems), the current study highlights the need for early identification and targeted and intensive treatment in these domains that continues at least two years post-diagnosis, but ideally longer.

The symptom domain of pain may be a particularly pertinent area of focus in the management of paediatric ME/CFS, given pain was most frequently endorsed at follow-up, and pain at baseline was significantly associated with fulfilment of ME/CFS criteria at follow-up. Clinical multidisciplinary strategies targeting pain relief and management including medication, physiotherapy, cognitive and behavioural techniques (i.e., meditation, mindfulness and acceptance and commitment therapy), and regular follow-up with the treating physician will be essential in this regard."​

 

[Hutan]

It's a shame these studies have variable follow up times (1 to 5 years in this case). It also matters how long the young people have had ME/CFS before being seen - some young people will have recovered before getting near the 'tertiary referral service'. Perhaps those things are addressed in the text, but it makes it hard to have any idea what the chance of recovery is at a specified time after onset.

It's not looking great for recovery though.

Sixty-five percent of ME/CFS patients at baseline continued to fulfil ME/CFS diagnostic criteria at follow-up, with pain the most frequently experienced symptom.

I thought that was interesting - pain was more frequently experienced than fatigue?

This has direct clinical impact for treating clinicians in providing a more realistic prognosis and highlighting the need for intervention with young people with ME/CFS at the initial diagnosis and start of treatment.

I'm not sure how they worked that out. Their sample was only of young people who got to the tertiary referral centre - and they don't seem to be fixing many. How can they know what happens to the young people that they don't see - how do they know that the young people who don't get to benefit from their interventions do worse?

Katherine Rowe isn't bad I think, but Lubitz was a fan of GET and CBT type approaches when I came across him. I'm sad to hear that he is still inflicting his ideas on vulnerable young people and their families in Melbourne.

 

[Peter T]

the current study highlights the need for early identification and targeted and intensive treatment in these domains that continues at least two years post-diagnosis, but ideally longer.

I see it as a positive that some 35% appear to be ‘recovered’, though this is much less than the general medical opinion of most children recovering, but how on Earth can they conclude from the fact that current service provision has failed to cure 65% that what is needed is yet more of the same?

These figures could be equally interpreted to indicate that current service provision is just plain wrong, and the third that ‘recovered’ did so despite current intervention rather than because of it.

 

[ME/CFS Skeptic]

Strange that none of the variables measured (fatigue, sleep quality, pain, depression and anxiety, quality of life) predicted having ME/CFS at follow-up.

 

[ME/CFS Skeptic]

The abstract writes:

"34 participants aged 13–18 years (25 ME/CFS, 23 controls)"
​

Which is probably an error. 48 patients were invited to participate but 10 could not be contacted and 4 withdrew which gives 34 participants (17 with ME/CFS and 17 healthy controls).

 

[Art Vandelay]

Katherine Rowe isn't bad I think, but Lubitz was a fan of GET and CBT type approaches when I came across him. I'm sad to hear that he is still inflicting his ideas on vulnerable young people and their families in Melbourne.

From what I've heard, all of the hospital-based ME/CFS clinics in Australia are terrible. If I had a child with ME/CFS, I wouldn't let them anywhere near them.

 

[rvallee]

ME/CFS patients showed significant improvement in most health and psychological wellbeing domains over time, compared with controls who remained relatively stable

There is obviously a self-selection process at play here, with those who deteriorated obviously unable to participate in such studies. And I doubt most of the patients would find the improvements to be significant. The overall trend is probably roughly that most slightly improve over time but this is missing out entirely on the worse off, the trend may even be a simple reflection that people simply adapt and lower their expectations anyway.

How does junk like that get published?

Clinical multidisciplinary strategies targeting pain relief and management including medication, physiotherapy, cognitive and behavioural techniques (i.e., meditation, mindfulness and acceptance and commitment therapy), and regular follow-up with the treating physician will be essential in this regard.

Says who? Based on what? This is not a serious statement to make in medical literature. And given the mediocrity of pain management, this is definitely not something people should pin any hopes on, we know it's terrible all-around, medicine seriously has huge problems detaching the romantic ideal-case version they have of medicine and what it is actually capable of.

This is slightly better than usual but frankly still falls very short of minimal acceptable quality.

 

[Snowdrop]

I wonder how many of these trials will be needed going forward? One hundred? Two thousand?

Given that we know some people remain ill and CBT/GET have been debunked I wish gov't and other funding bodies would put money aside to research aetiology and cure or at least a treatment that actually benefits PwME enough to get back to their lives.

This may be a reasonable study. I think generally though that all fall short in some way. When these studies are so cheap by comparison why not do a thorough job of it?

I confess that even if we come across papers that are sound methodologically (although usually not) and tell us something there is a limit to my ability to read about this rather than that money was given to confirm a biological finding using a correct cohort or some other biological lead that could use some scrutiny.

A bit ill tempered of me I know. But this is how I feel every time now I see a new study going over the same ground covered ever so many times before. Even when it's different this time.

* I also seem to have lost the ability to add since there were 34 participants (25 ME / 23 controls) So a cure would really help me sort that problem.

 

[Mithriel]

If they asked people with ME they would discover that many of us have almost constant pain but it is not chronic pain but a deep burning ache that feels like a build up of lactic acid when you have done too much exercise. Basically the activities of daily life make us feel as if we have just played football for the first time in twenty years or dug up the garden.

No amount of meditation is going to cure that

 

[Peter T]

If they asked people with ME they would discover that many of us have almost constant pain but it is not chronic pain but a deep burning ache that feels like a build up of lactic acid when you have done too much exercise. Basically the activities of daily life make us feel as if we have just played football for the first time in twenty years or dug up the garden.

No amount of meditation is going to cure that

An important point, in that this study purports to be descriptive of the experience of children with ME in the context of a specific service, but does not first seek to develop meaningful measures of ME, rather it primarily relies on rating scales reflecting an ideological understanding of the condition based on a BPS approach (ie in reality a psychological approach) potentially ignoring key symptoms and then draws conclusions about appropriateness of specific treatment approaches, which the study is not designed to evaluate and which their results suggests are of at best very limited effectiveness.

[added - This is not to say that this study is totally without interest, but that we desperately need quantifiable descriptors that reflect current understandings/definitions of the condition before we can address issues like incidence, disease profile, prognosis and treatment outcomes.]

 

[Snow Leopard]

Overall this suggests that the often-quoted/suggested high recovery rate in children must be in the first year or so, before they are referred to tertiary care.

 

[Hutan]

Overall this suggests that the often-quoted/suggested high recovery rate in children must be in the first year or so, before they are referred to tertiary care.

Yes, as it probably is in adults (going by the Dubbo study and studies of Long Covid).

I wonder if the idea that children and young people are particularly likely to recover is in part due to the fact that most people getting EBV are young people, and EBV is a particularly obvious and well-known cause of post-viral fatigue syndrome/ME/CFS. So, people notice that a lot of young people are in bed for months after EBV, and then most recover.

Perhaps also it is partly wishful/hopeful thinking.

 

[DokaGirl]

What "intensive treatment", there is none at all. Unless you count the potentially harmful GET and CBT. I find it puzzling, the things that some call treatments.

 

[Milo]

fatigue, pain, and health-related quality of life representing domains particularly relevant to perpetuation of ME/CFS symptoms in the early years following diagnosis

this veers on blaming patients for not getting better

Edit to add: let’s change ME/cfs for MS and see how that rings…

fatigue, pain, and health-related quality of life representing domains particularly relevant to perpetuation of MS symptoms in the early years following diagnosis

I don’t think so.

 

[Amw66]

Not any coffee in the house this morning
I can't make sense of this
Sixty-five percent of ME/CFS patients at baseline continued to fulfil ME/CFS diagnostic criteria at follow-up, with pain the most frequently experienced symptom. Eighty-two percent of patients at follow-up self-reported that they still had ME/CFS, with 79% of these patients fulfilling criteria.

65 % of one group ( 11)
79% overall ( 27) fulfilling criteria - does this represent the development over time?
Not an insignificant effect.

 

[Trish]

79% of 82% would be about 65%.

I haven't read the paper. These percentages are certainly confusion.

 

[Hutan]

On psychological factors as risks for not recovering, and depression rates

3.4. Predictors of ME/CFS Criteria Fulfilment at Follow-Up​

None of the baseline variables of health and psychological wellbeing were significant predictors of fulfilment of ME/CFS diagnostic criteria at follow-up (all p > 0.05, d range = 0.02–0.33, OR range = 0.83–2.54). Pain experienced at baseline was close to reaching significance (b = 0.05, p = 0.053) and was associated with a moderate effect size (d = 0.33), but a small OR (OR = 1.05; SE(OR) = 0.02). For the Pearson correlation analysis, only pain experienced at baseline was significantly associated with ME/CFS criteria fulfilment at follow-up, with moderate effect (R = 0.6, p = 0.02). The Pearson correlation matrix for this analysis can be found in Table S4.

Unlike previous studies [6,31,62,66], we did not find higher rates of depression in adolescents with ME/CFS compared with healthy controls, nor an increase in depression over time. However, our findings do support two recent paediatric ME/CFS follow-up studies showing stable depression levels over time [67,68]. Although Loades et al. [68] observed consistently higher levels of depression in the paediatric ME/CFS group than healthy controls, baseline ME/CFS depression was found to explain most of the variance in follow-up ME/CFS depression, which appears to suggest stable depression levels across time in paediatric ME/CFS. Of note, Loades et al.’s sample included a higher proportion of adolescents with depression compared with previous studies [6,66]. It may be that there is a subtype of ME/CFS that is particularly associated with comorbid depression [69], which was represented in Loades et al.’s [68] sample but not in our study. Major psychiatric illness that could adequately explain fatigue symptoms was an exclusionary criteria in our study, which would have played an additional role.

The main finding from the third aim of our study was that no aspects of baseline health and psychological wellbeing were found to significantly predict ME/CFS criteria fulfilment at follow-up, with any great effect. However, despite not reaching significance as a predictor (with moderate effect), pain at baseline was significantly and positively associated with meeting criteria at follow-up, and was also the most commonly endorsed symptom by patients at follow-up. This would suggest that the experience of pain early in illness course may be relevant to later diagnostic status in the wider paediatric ME/CFS population, which is supported by previous research [3,75].

Although many reports imply that the presence of poor health at ME/CFS onset influences future recovery [1,63,76], empirical support is lacking. In fact, investigations focused on anxiety and depression suggest otherwise. For example, Rowe [11] reported no association between baseline depression/anxiety and recovery, and whilst Rimes et al. [15] found an association between baseline anxiety/depression and new onset chronic fatigue, they found no association between baseline anxiety/depression and persistent chronic fatigue. Rowe [11] and Rimes et al. [15] findings are consistent with the present study’s results. It is worth noting that previous follow-up studies have tended to identify demographic predictors (rather than health/psychological predictors) of future clinical status, namely: older age, female gender, higher IQ, higher BMI, and school absenteeism [12,15,24,76]. Those studies that have found associations between baseline health/psychological wellbeing—e.g., sleep quality [77], depression [28,78], anxiety and fatigue [32]—and follow-up recovery status have relied on a proxy for ME/CFS (e.g., “CFS-like symptoms”, “Chronic Disabling Fatigue”, etc.). Therefore, it remains to be seen whether baseline aspects of health and psychological wellbeing are useful prognostic indicators of future diagnostic status or recovery in adolescents who have been diagnosed by a paediatric ME/CFS specialist.

 

[Utsikt]

Therefore, it remains to be seen whether baseline aspects of health and psychological wellbeing are useful prognostic indicators of future diagnostic status or recovery in adolescents who have been diagnosed by a paediatric ME/CFS specialist.

I think we can be fairly certain that most factors will not be «useful» for anything. It’s a waste to keep looking for them when they won’t have any utility at all.