Chronic Fatigue Syndrome at Age 16 Years

Instead of using diagnostic criteria designed, developed and Implememted by expert clinicians to identify the neurological disease chronic fatigue syndrome that is characterised by post exertional exacerbation of symptoms due to exertion- Crawly et al diagnose patients on the basis of parents saying that the teenagers feel tired.
A bit like getting the parents to diagnose cancer on the basis of their child having a mole or freckle or getting parents to diagnose diabetes based in their teenager being thirsty at times....
Crawley and her chronies don't diagnose chronic fatigue syndrome /myalgic encephalomyelitis in any of the study participants - so how can they justify reporting on the prevalence of this disease??? ME/CFS is quantifiable and is measurable - abnormalities on 2 day CPET tests, abnormal physiological responses to exertion, orthostatic intolerkxe .......
All Crawleys team can say is that 2% of parents with teens report they they are tired - some of these teens may have Me/CFS- but Crawley et al never investigated whether or not any of these kids had CFS/ME, depression, diabetes, lupus, cancer.... there are many diseases and or conditions that make teens tired and teens are notorious for needing more sleep than previously.

We agree with Medow et al that CFS/ME in a clinical setting should be diagnosed in accordance with established criteria, and we support their assertion that proper medical evaluation for any chronic condition is essential. Indeed, we use data from clinical settings (specialist adult and pediatric CFS/ME services) as the mainstay of our research into this debilitating illness.

Our ‘CFS at age 16’ study was based on data from a birth cohort. We believe that cohort studies add to the evidence base because they avoid the bias inherent in clinical studies, where only those who access health care are represented. They allow us to explore the prevalence of CFS using a population sample to provide the denominator. We used the ALSPAC cohort for our study because we wanted to investigate, and control for, variables recorded since before the child’s birth, and because it is broadly representative of the UK population. A major limitation common to most birth cohort studies is that ‘cases’ cannot be validated by a proper medical evaluation, as we stated in our discussion.

We do not share the concerns of Medow et al that the positive association of family adversity with chronic fatigue might support a behavioural foundation for CFS/ME. Instead, we believe that our replicated findings should dispel once and for all the unhelpful notion that CFS/ME is more likely to occur in middle class families.

We acknowledge in our limitations that we were unable to differentiate accurately between those with CFS and co-morbid depression and those with depression and fatigue. Given the high rate of co-morbid depression in children with CFS/ME, which we believe is due to the impact of CFS/ME (Bould et al 2013), we were reluctant to exclude all cases of depression. Instead, we reported the results of a sensitivity analysis in which adolescents with high levels of depressive symptoms were excluded.

The number of children in ALSPAC affected by traumatic events was too small to support analysis of childhood trauma. The ALSPAC family adversity index is a much broader measure of general socioeconomic adversity and, given the association that we found, would account for a larger population attributable fraction of CFS/ME. We did not investigate whether family adversity was differentially associated with CFS/ME according to presence/absence of depressive symptoms. This is something which we plan to investigate using ALSPAC data measurements at different ages.

We believe that research into this devastating condition should use a range of techniques to explore its prevalence, incidence and aetiology. Our prevalence estimates are not inconsistent with other studies, and we hope that our study serves to highlight that CFS/ME is a relatively common illness among adolescents. We trust that the Medow et al would agree with our concluding remarks that “Awareness needs to be raised to ensure that families of children affected by CFS can access specialist medical care, and that pediatricians and those looking after children are trained in the identification and management of CFS.”

Bould H, Collin SM, Lewis G, Rimes K, Crawley E. Depression in paediatric chronic fatigue syndrome. Arch Dis Child. 2013 Jun;98(6):425-8.

We have several concerns about the above-referenced study. While studying 16 year olds, the authors did not properly identify those with Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS) (they used “criteria similar to the definition of chronic disabling fatigue in children”), despite Chronic Fatigue Syndrome being part of their title. They ignored the stated caveat in the case definition which requires that the fatigue “has no other known cause” (ref 6). Not all with chronic fatigue have Chronic Fatigue Syndrome. ME/CFS comprises a subset of chronically fatigued individuals who fulfill specific diagnostic criteria, employment of which is critically important for the identification and treatment of diseases or conditions that can result in fatigue. An important component of the diagnosis is a physician's history and physical examination to exclude conditions that could explain the fatigue, including hypothyroidism, heart disease, cancer, liver failure, covert drug abuse, medication side effects, gastrointestinal/nutritional, infectious and psychiatric conditions.

The finding of a high threshold for depressive symptoms suggests an over-representation of those with depression, among whom increased Family Adversity might be an expected precursor. Perhaps individuals with chronic fatigue and a high rate of depression, as identified in this study, might have a higher rate of early childhood adversity, but the same cannot be said for those with ME/CFS. Samples from CFS clinics do not identify childhood trauma (sexual and physical) as a risk factor for pediatric CFS (Gjone and Wyller, 2009). In suggesting a high prevalence of Family Adversity, the Collins report may inappropriately provide support for a primarily behavioral foundation for ME/CFS.

The authors stated that participants “were not assessed by a physician, and our classification was not subject to clinical verification”, and it is possible that parents and children “reported significant levels of disabling fatigue that was caused by another disorder.” They did not however state that proper medical evaluation for this or any chronic condition is essential, particularly for children or whether follow-up care was recommended.

Not employing accepted criteria when identifying ME/CFS can result in unintended deleterious consequences, especially in the vulnerable pediatric population. Inappropriate diagnoses are potentially dangerous if “obtaining a diagnosis” confers complacence by the physician and dissuades parents from seeking medical evaluation of their child’s fatigue. And physicians may discount a diagnosis of ME/CFS and therefore employ incorrect treatments.

We therefore need to re-state the importance of using established criteria to identify ME/CFS to allow for the diagnosis, treatment, and research of the many aspects of this condition. Since the authors did not use established diagnostic criteria and employed self-evaluations of ME/CFS, their paper should be titled “Chronic Fatigue but not Chronic Fatigue Syndrome at Age 16 Years."

Gjone H, Wyller VB. Chronic fatigue in adolescence - autonomic dysregulation and mental health: An exploratory study. Acta Paediatrica, International Journal of Paediatrics 2009;98(8):1313-1318.

Heim C, Wagner D, Maloney E, Papanicolaou DA, Solomon L, Jones JF, Unger ER, Reeves WC: Early adverse experience and risk for chronic fatigue syndrome: results from a population-based study. Arch Gen Psychiatry. 2006 Nov;63(11):1258-66.

Publication of the paper by Collin et al on the prevalence of myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) in adolescents (1) led to Britain's best selling newspaper,The Sun, stating: New research reveals that yuppie flu hits one in 50 teenagers (2). Similar 'one in 50' coverage appeared in the international press and the BBC news.

Whilst welcoming the extensive media coverage being given to the problems facing adolescents with ME/CFS in relation to late or non diagnosis, lack of appropriate management, and difficulties relating to education, I have serious concerns about the methodology that was employed to arrive at a prevalence figure of almost 2%.

These concerns are based on the following:

1 Classification of ME/CFS was made on parental and adolescent completed questionnaires where the adolescent was reported to have persisting and disabling fatigue.

2 This self-reported ME/CFS classification was not then validated by a health professional, and there is no information in the paper to indicate whether any of these adolescents did already have a diagnosis of ME/CFS.

3 Even when health professionals in primary care make a provisional diagnosis of ME/CFS, a high level of misdiagnosis is found when the patients are fully re-assessed in a hospital based referral service - approximately 40% of consecutive referrals had another diagnosis in the study by Newton et al (4).

4 There is no indication as to whether following self-reported classification of ME/CFS, the parents were advised to see their GP and whether a diagnosis of ME/CFS was then confirmed.

5 A high proportion of those classified with ME/CFS were also found to have a significant level of depression, which can cause chronic fatigue. When this group was removed, the ME/CFS prevalence figure fell to 0.6%.

6 Sending out questionnaires to over 5,000 parents, the vast majority of whom have normal healthy adolescents, is likely to produce a biased response. Parents of healthy adolescents are less likely to respond whereas parents of children who have an undiagnosed health problem are more likely to be interested in responding.

7 Where the prevalence of adolescent ME/CFS was estimated in a Dutch study (5), using general practice questionnaires and prospective registration of new hospital patients, ME/CFS was found to be far less common (i.e. 0.11%).

With ME/CFS already being reported to be the most common cause of long-term sickness absence from school (5), reliable evidence on the epidemiology is clearly required.

But I do question whether this study is just helping to confirm the fact that teenage lifestyles can produce significant levels of tiredness and chronic fatigue - which is not the same as having ME/CFS.

References

1 Collin SM et al. Chronic fatigue syndrome at age 16 years. Pediatrics, 2016, Jan 25. pii: peds.2015-3434. [Epub ahead of print]

2 New research reveals that yuppie flu hits one in 50 teenagers. The Sun, 25 January 2016.

3 Newton JL et al. The Newcastle Chronic Fatigue Syndrome Service: not all fatigue is the same. Journal of the Royal College of Surgeons of Edinburgh, 2010, 40, 304 - 307

4 Sanne L et al. Adolescent chronic fatigue syndrome: Prevalence, Incidence and Morbidity. Pediatrics, 2011, 127, 169 - 175

5 Dowsett EG and Colby J. Long-term sickness absence due to ME/CFS in UK schools: an epidemiological study with medical and educational implications. Journal of Chronic Fatigue Syndrome, 1997, 3, 29 - 42.

Dear Editor

Re: Chronic fatigue syndrome at 16 years

Publication of the paper by Collin et al on the prevalence of myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) in adolescents (1) was followed up in Britain's best selling newspaper, The Sun, with a news item headed: New research reveals that yuppie flu hits one in 50 teenagers (2). Similar 'one in 50' coverage appeared elsewhere in the international press and on the BBC news website (3).

Whilst welcoming the fact that extensive media coverage was being given to the undeniable problems facing adolescents with ME/CFS in relation to lack of recognition and resulting late or non diagnosis, lack of appropriate management, and difficulties relating to education, this patient support charity has serious concerns about the methodology that was employed to reach what appears to be a very firm conclusion that 1 child in 50 in the UK has ME/CFS.

We believe that a prevalence figure of almost 2% is a significant over-estimate of the true situation.

We base our concerns on the following 7 points:

1 Diagnosis/classification of ME/CFS was made on the basis of parent and adolescent completed questionnaires where the adolescent was reported to have some form of persisting and disabling tiredness or lack of energy.

2 This self-reported ME/CFS classification/diagnosis was not then checked or validated by a health professional, and there is no information in the paper to indicate whether any of these adolescents did already have a diagnosis of ME/CFS. Presumably it was possible for notification of a pre-existing diagnosis of ME/CFS to be made on the questionnaire, and it would be useful to know if this question had been asked and whether any did.

3 Even when health professionals in primary care are involved in making a provisional diagnosis of ME/CFS, a high level of misdiagnosis is found when they are fully re-assessed using Fukuda defined CFS in a hospital based referral service - approximately 40% had another diagnosis in the study by Newton et al (4) involving consecutive referrals to the Newcastle ME/CFS Service.

4 There is no information to state whether following self-reported classification/diagnosis of ME/CFS, the parents were advised to see their GP and whether a diagnosis of ME/CFS was then confirmed.

5 A high proportion of those who were classified/diagnosed with ME/CFS were also found to have a significant level of depression, which can cause chronic fatigue. When this group was removed, the ME/CFS prevalence figure fell to 0.6%.

6 Sending out questionnaires to over 5,000 parents, the vast majority of whom have normal healthy adolescents, seems almost bound to produce a biased response - in that parents of healthy adolescents are less likely to respond whereas parents of children who have an undiagnosed health problem are more likely to be interested in responding.

7 Where the prevalence of adolescent ME/CFS was estimated in a Dutch study (5), using GP questionnaires and prospective registration of new hospital patients, it was reported that ME/CFS is uncommon (i.e. 0.11%) and is not the same as having chronic fatigue.

With ME/CFS already being reported to be the most common cause of long-term sickness absence from school (6), reliable evidence on the epidemiology is clearly required.

But we do question whether this study is providing an accurate estimate of prevalence.

Or whether it is just helping to confirm the fact that teenage lifestyles can produce significant levels of tiredness and chronic fatigue - which is not the same as having ME/CFS.

Dr Charles Shepherd
Hon Medical Adviser, ME Association

7 Apollo Office Court
Radclive Road
Gawcott
Buckingham MK18 4DS
UK

Website: www.meassociation.org.uk

References

1 Collin SM et al. Chronic fatigue syndrome at age 16 years. Pediatrics, 2016, Jan 25. pii: peds.2015-3434. [Epub ahead of print]

2 New research reveals that yuppie flu hits one in 50 teenagers. The Sun, 25 January 2016. http://www.bbc.co.uk/news/health-35383832

3 Chronic fatigue syndrome on the rise among 16-year-olds. BBC News on line, 25 January 2016. http://www.bbc.co.uk/news/health-35383832

4 Newton JL et al. The Newcastle Chronic Fatigue Syndrome Service: not all fatigue is the same. Journal of the Royal College of Surgeons of Edinburgh, 2010, 40, 304 - 307

5 Sanne L et al. Adolescent chronic fatigue syndrome: Prevalence, Incidence and Morbidity. Pediatrics, 2011, 127, 169 - 175

6 Dowsett EG and Colby J. Long-term sickness absence due to ME/CFS in UK schools: an epidemiological study with medical and educational implications. Journal of Chronic Fatigue Syndrome, 1997, 3, 29 - 42.