Jason 2012: Pediatric Myalgic Encephalomyelitis/Chronic Fatigue Syndrome

[Firestormm]

Reviews in Health Care: Full paper: http://ojs-seed.neagen.it/index.php/rhc/article/view/280/html

Corresponding author

Leonard A. Jason, Ph.D. Director, Center for Community Research, DePaul University, 990 W. Fullerton Ave., Chicago, IL 60614

Abstract

Research on pediatric Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS) is reviewed in this article. Many recent articles in this area highlight the existence of key differences between the adult and pediatric forms of the illness.

This review article provides an overview of pediatric ME/CFS, including epidemiology, diagnostic criteria, treatment, and prognosis. Challenges to the field are identified with the hope that in the future pediatric cases of ME/CFS can be more accurately diagnosed and successfully managed.

Pediatric Myalgic Encephalomyelitis/Chronic Fatigue Syndrome

Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS) is a protracted and persistent illness that produces a variety of debilitating effects in children and adolescents [1,2].

While a hallmark symptom of ME/CFS is profound fatigue, sufferers report a wide variety of concomitant symptoms including post-exertional malaise, body, myofascial and/or joint pain, cognitive difficulties, unrefreshing or disturbed sleep, headaches, and sore throat.

Some symptoms are more common than others; however, the condition is characterized by unique symptom patterns and unpredictable symptom severity, which further complicates both treatment and prognosis [3].

Children and adolescents often present with symptoms that differ from their adult counterparts [4]. For example, stomachaches and rash are often reported in cases of pediatric ME/CFS, but are far less common in adults [2,3].

According to Bell [5], the three most common symptoms (aside from fatigue) in children with ME/CFS were headaches, sleep disturbance, and cognitive difficulties.

Furthermore, children and adolescents may exhibit symptoms in response to their unique day-to-day routine (i.e., trouble in school, loss of friends, dropping out of extracurricular activities, etc.), and, due to their still-developing coping skills, are more apt to display irritability in response to the disease [2].

Dowsett and Colby [6] found ME/CFS to be the most common cause of prolonged medical leave from school among adolescents. In another study, Carter and colleagues [7] found that 55% of children with ME/CFS reported a decline in academic performance since illness onset and 80% indicated major reduction in extracurricular activities.

Smith and colleagues [8] found that one third of adolescents with ME/CFS report severe restrictions of all activities and marked drops in school performance; some missed up to 80 days in a six-month period.

I have yet to read it all thoroughly, though I generally look out for things from Jason, and I don't think kids receive much in the way of special focus - so this review looked appealing.

Especially liked the following - which is about as far as I have got thus far:

Epidemiology

Epidemiologic methods summarize information about the distribution of illnesses and allow investigators to make inferences about the importance of risk factors or even causal factors (e.g., smoking as a precursor to cancer).

However, even if the specific cause of an illness or social condition is not known, epidemiologists can uncover associations between risk factors that can lead to important ways of understanding the etiology and maintenance of an illness [9].

If biased sampling methods such as the utilization of physician gatekeepers to identify cases of myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) result in an over- or underestimate of the true prevalence rate, it may impede efforts to understand the nature of the condition.

This error occurred with the first generation of adult ME/CFS prevalence studies. Reyes and colleagues[10] published epidemiologic research that portrayed ME/CFS as being relatively rare, affecting about 20,000 adults, and characterized patients as being European-American and middle to upper-class women.

These findings supported the myth of ME/CFS as a “YUPPIE flu” disease. So as long as scientists and the public perceived ME/CFS as rare, those with this illness would be unlikely to receive the needed federal attention and community resources to develop a better understanding of this illness.

This CDC epidemiologic study was based on a method that relied on physician referral of patients with ME/CFS [10]. However, if the physicians did not believe the illness existed, or if patients did not have a physician, many people with ME/CFS would not be referred to researchers or counted in prevalence surveys.

A second generation of adult prevalence studies involved a randomly selected group of individuals being telephoned and screened for symptoms of ME/CFS [11]. Those who were identified in the telephone screen as having several ME/CFS symptoms underwent a complete medical and psychiatric examination to determine whether they actually had ME/CFS.

This study estimated that over 800,000 people had ME/CFS, and this illness was found to be more prevalent among people of color. This study [11] and others [12] that used community-based samples differed from the methods used in the earlier CDC study [10] whereby physicians determined who was referred as a possible ME/CFS case.

Edit: Y'all will LOVE the bit on CBT (well in part I suppose)...

 

[Firestormm]

Gonna post some extracts that caught my eye as I read through it - having had a rough night I need a distraction. Bear with me folks

Hypothalamic-Pituitary-Adrenal (HPA) Axis Dysfunction

Cortisol serves as the main signaling hormone of the HPA axis. Dysregulation of glucocorticoid function is associated with ME/CFS [30-33]. Patients with ME/CFS have lower cortisol secretion [34], a pattern of glucocorticoid resistance [35], and a disruption or dysregulation of the expected diurnal cortisol pattern [36].

The presence of HPA axis dysregulation in adolescence may serve as a predictor of this illness [37]. For example, Miike and colleagues [38] found that cortisol secretion was reduced in a pediatric ME/CFS population in comparison to controls.

In addition, prior adverse events can “prime” inflammatory responses, promoting excessive cytokine production in response to subsequent stressors [39]. [????]* Thus, prior life adversity may produce stable alterations in processes that regulate the inflammatory response to subsequent stress [40].

Attenuated decline of salivary cortisol concentrations across the day and increased IL-6 concentration, a key mediator of such inflammatory responses, have been associated with fatigue symptoms in ME/CFS [41]. Others have found higher levels of pro-inflammatory immune responses, including IL-8 [42] and TNF-alpha [43] for individuals with ME/CFS versus controls.

Variance in the expression of genes associated with HPA axis function has been found to be associated with ME/CFS [44-46]. For example, Rajeevan and colleagues [47] found that those with certain cortisol regulatory mechanisms (i.e., a single nucleotide polymorphism in the glucocorticoid receptor (GR) gene) were at increased risk for ME/CFS.

Others have also found differences in the expression of glucocorticoid receptor NR3C1 in individuals with ME/CFS as compared to controls [33]. This suggests that patients with ME/CFS may have decreased sensitivity to the effects of cortisol due to a down-regulation of this glucocorticoid receptor [35].

Those with ME/CFS demonstrated improvements in a clinical trial of hydrocortisone [48]. However, such therapies are not appropriate for long-term treatment due to adrenal suppression [49].

As other pharmacologic modalities have not been beneficial in providing consistent relief to those with ME/CFS, it is important to investigate non-pharmacologic methods of influencing hormonal pathways.

[* Can anyone offer me a simple explanation for that sentence? Thanks]

Ok. So now we enter the realm's of CBT etc. I guess that this first extract will be something most likely to be repeated, although to be honest I found the above cortisol explanation etc. very interesting, and the notion that CBT and other strategies could help to stabilise cortisol dysfunction (my word and understanding - please correct where necessary), is something new to me.

I have to repeat that I do like these reviews and the way in which Jason reviews (or tries to) all the evidence, is credible in my own tiny mind.

Viz:

Treatment
....

Negative patient reactions to health care services and providers might in part be explained by the types of interventions offered to individuals with this illness.

While CBT has been applied to many medical and psychiatric problems, from pain to fibromyalgia to depression, its application to ME/CFS has been more controversial.

This might be due to several of the components of CBT as practiced by some European investigators (e.g. [56,57]).

Typical of the psychogenic view of ME/CFS is a research group from the Netherlands [58] who believe that these individuals attribute their symptoms to physical causes, are overly preoccupied by their physical limitations, and do not maintain regular activity.

According to this model, these factors cause individuals with ME/CFS to be functionally impaired, implying that the central problem with patients experiencing this condition is a psychosomatic preoccupation with one’s fatigue.

Song and Jason [59] tested this model with individuals with ME/CFS, and found that it was not valid for those individuals. In addition, Price, Mitchell, Tidy, and Hunot [60] reviewed 15 studies of CBT with a total of 1,043 ME/CFS participants. At treatment’s end, 40% of people in the CBT group showed clinical improvement in contrast to only 26% in usual care, but changes were not maintained at a one- to seven-month follow-up when including people who had dropped out.

Patient reactions to CBT and graded exercise have been mixed. A survey of 2,338 ME and ME/CFS respondents [61] found that 26% felt their condition worsened after trying CBT, and graded exercise was felt to be the treatment that negatively impacted the most people.

Results of other surveys conducted by the ME Association showed that, of those that had received graded exercise therapy, 33.1% felt “much worse” and 23.4% judged themselves to be “slightly worse” [62]. Similarly, a survey of treated patients with ME/CFS found that 34% of those who tried graded exercise therapy perceived themselves to be worse [63].

Anyway, this was what I found of interest to me personally, as I had not heard of it before:

The ability to employ an intervention that enables youth with ME/CFS to have more control over their environment may help normalize cortisol levels.

There is evidence that this has occurred following a CBT intervention for adults with ME/CFS [64]. Unfortunately, in the Roberts et al. study [64], there was no control or comparison group, and only a pre- and post-intervention (CBT therapy) cortisol level test.

[Important Point]

Treatment studies in the ME/CFS area have rarely examined neuroendocrine and immune dysregulation. It is important to examine critical neuroendocrine pathways in adolescents with ME/CFS at a time where other confounding conditions are minimized (e.g., long term use of multiple pharmacologic interventions, other medical illnesses).

So. A possible biological reaction, a stabilisation, of cortisol from a 'CBT' intervention. Buggar-me! Who would have thought. Joking aside, I had not heard it analysed like that before. Or that cortisol potentially contributed to some aspects of potentially my own symptoms. Funny that. Probably the old memory cell misfiring again. Or maybe I'm simply too thick these days

 

[Firestormm]

Prognosis

Given the lack of effective treatments available, the prognosis for children and adolescents with ME/CFS tends to be poor [72]. While previous research suggests that young sufferers of ME/CFS have a better prognosis than their adult counterparts [75], a recent study disputes this idea.

Brown and colleagues’ [72] research examined long-term health outcomes of 25 people who were diagnosed with ME/CFS while they were adolescents, approximately 25 years ago.

Researchers assessed whether participants maintained their ME/CFS diagnosis, as well as current levels of health and functioning. Of the 25 participants diagnosed with ME/CFS 25 years ago, only five self-reported maintaining a diagnosis, while 20 reported remission from the disease.

In spite of their self-reported remission, those 20 participants showed significantly more impairment compared to controls, demonstrating that, while adolescents diagnosed with the disorder may show some improvements over time, they still suffer some level of impairment and may not return to their premorbid level of functioning.

Further support for Brown and colleagues’ [72] findings is seen in Joyce, Hotopf, and Wessely’s [74] review of longitudinal ME/CFS studies.

They found that fewer than 10% of adult participants across studies reported returning to premorbid functioning.

A more recent review found a median ME/CFS recovery rate of 5%, although there was variability in recovery rates across studies [73]. These findings highlight both the vastly debilitating nature of the disease, as well as the need for further longitudinal research in this area [72].

Now I do wonder how much that contribution from Wessely will be re-posted... Still, I have to say that I was ignorant of the view in relation to Prognosis for younger people. I had understood it to be better than for adults over the longer term. Just goes to show...!!

Night all

 

[Firestormm]

Discussion:

...

Non-pharmacologic rehabilitative interventions are used for people with cancer and heart disease, but they are only one component of the treatment plan, and, when used by themselves, are not curative.

Comprehensive approaches are also needed for patients with ME/CFS, and helping patients manage their illness non-pharmacologically is only one aspect of rehabilitation.




Historically, researchers have focused on the potential benefits of cognitive behavioral interventions. As discussed, the long-term outcomes of this type of intervention are still unclear, but interventions that challenge basic patient illness beliefs might solidify already negative attitudes of medical personnel toward people with ME/CFS.

The Energy Envelope Theory represents an alternative approach for helping patients, as it helps patients better monitor energy levels, stay within their energy envelopes, sustain lifestyle changes that involve reprioritizing activities, and possibly rebalance their lifestyle between work and leisure.

...

 

[SilverbladeTE]

A doc who refused a MS patient treatments except CBT, and then that CBT not for coping with distress, but as a "cure/treatment" would soon be out on his ass and maybe worse.

*SAME DAMN THING*

You can see the Weasels "weaselling" their way into "get out of jail clauses" with all this stuff as I've been saying for some time.
"Oh but officer! We did mention HPA!" etc.

anyway the recovery rate stuff is important and notes what many have experienced: damn few folk EVER recover from ME and many of those relapse years on.
What usually happens is that after some years, usually when you make yourself worse trying to fight it, putting up with bullshit or harmful treatment, you eventually figure out a better coping strategy for *yourself*
ie, all most docs are any damn use for with this illness is for PAINKILLERS, and that is one damnably digusting expose' of the reality of this, sigh.
Screw the CBT zealots, they are slowly admitting what many of us figured out for ourselves years ago: Pacing/managing stress etc.

 

[Dolphin]

Anyway, this was what I found of interest to me personally, as I had not heard of it before:

The ability to employ an intervention that enables youth with ME/CFS to have more control over their environment may help normalize cortisol levels.

There is evidence that this has occurred following a CBT intervention for adults with ME/CFS [64]. Unfortunately, in the Roberts et al. study [64], there was no control or comparison group, and only a pre- and post-intervention (CBT therapy) cortisol level test.

There was also another paper by a very similar team:

Psychol Med. 2010 Mar;40(3):515-22. Epub 2009 Jul 17.
Does hypocortisolism predict a poor response to cognitive behavioural therapy in chronic fatigue syndrome?
Roberts AD, Charler ML, Papadopoulos A, Wessely S, Chalder T, Cleare AJ.

CONCLUSIONS:
Low cortisol is of clinical relevance in CFS, as it is associated with a poorer response to CBT. Hypocortisolism could be one of several maintaining factors that interact in the persistence of CFS.

It's perhaps a bit like the claims that CBT can be useful for pain; except

2 studies found that the presence of severe pain at baseline predicts a poor response to CBT in adult patients with CFS (69,70).

 

[Sparrow]

"As other pharmacologic modalities have not been beneficial in providing consistent relief to those with ME/CFS, it is important to investigate non-pharmacologic methods of influencing hormonal pathways."

[* Can anyone offer me a simple explanation for that sentence? Thanks]

As far as I can see, they're saying that prescription drugs don't tend to help consistently with this illness, but that lifestyle-type things can also influence hormones (when you sleep, what you eat, what you do, how much stress you're under, etc.). So they're saying if drugs don't seem to be doing the trick, it might be worth a try to affect them with those other things too.


For what it's worth, I don't mind what was said here. It seems like this guy is on our side. From what I read (and I will admit that I skimmed large sections), they seem to be acknowledging us as a debilitating and physical illness, critiquing GET and "false illness beliefs" CBT, and providing a plausible explanation for why CBT seems to have occasionally helped certain people a little. And they've been great about explicitly saying that we need to combine any lifestyle changes with other kinds of treatment (just like they do for cancer, etc.).

Lots of autoimmune conditions are made worse by stress. That doesn't mean that stress-reduction is the only treatment that should be given, but it also doesn't mean that it can't have an impact.