Thursday, May 28, 2009

CFS in Children


TESTIMONY PRESENTED TO THE
U.S. DEPT. OF HEALTH AND HUMAN SERVICES
CHRONIC FATIGUE SYNDROME ADVISORY
COMMITTEE


Pediatric ME/CFS
CDC CFS Research Program

May 29, 2009

submitted by Jill McLaughlin


I have been involved in ME/ CFS advocacy for nearly a decade. First
and foremost as a parent and caregiver, but I have also worked
with patient organizations, participated in several Department of Health
and Human Services (DHHS) committee meetings, attended and
participated in related conferences, and currently I am on a committee
of the International Association for Chronic Fatigue
Syndrome/Myalgic Encephalomyelitis (IACFS/ME).


During that time, CDC has done virtually nothing to study or address
ME (or what is being called CFS, commonly referred to as ME/CFS)
in children, or to promote physician, school or public education
and awareness of the illness or to even acknowledge that it exists.
When the CDC whistleblower scandal regarding the CDC misuse
of funds broke several years ago, a large study on children was
summarily dropped, ostensibly due to lack of funds. When the
money was restored, the study on children was never resumed.


The medical community and the public look to the federal
health agencies for information and guidance, but there
has been very little leadership in this area.


CDC did launch an expensive national campaign for chronic
fatigue syndrome for education and awareness. The message
was "Get Informed, Get Diagnosed, and Get Help." The general
reaction to the slogan was: about what - fatigue and
unwellness, by whom, and how? The erroneous assumption
being that there are healthcare providers capable of diagnosing
and treating it. According to the CDC only about 15% of CFS
patients in the U.S. have been diagnosed. What was suggested
did not exist and was next to impossible with the current
physician mindset and available research.


This campaign was a waste of time and money (except for the
CFIDS Association as contract recipients). Marketing and PR
are no substitute for science and physician education.


The illness is housed and studied in the National Center
for Zoonotic, Vector-Borne, and Enteric Diseases (ZVED),
yet most of the CDC CFS research is stress related.


A review of the scientific literature on the relationship between
stress and disease, published in JAMA,  found that stress is
a contributing factor in a wide range of  human disease. It is
hardly unique to CFS, yet CDC studies do not consider these
findings or use adequate study controls.


CDC research focuses on a few narrow, select gene studies
involving HPA axis as it relates to PTSD or stress disorders,
giving the impression that the illness is psychosomatic.


Consequently, many doctors still do not believe in CFS
or that it is a physical illness or that it's serious or disabling.
Most doctors, particularly pediatricians, still do not know
how children present with this illness or how it is diagnosed
or  treated.


Even intelligent parents  who are told by medical professionals
that there is nothing wrong with these kids and they just need
tough love and to try harder may take this at face value. If the
experts say it's nothing then these kids must just be faking or
making it up.


While the name issue is sometimes dismissed as unimportant,
trivial or "political," research done by DePaul University has
shown that the name 'Chronic Fatigue Syndrome' can
influence how patients are perceived and ultimately
treated by medical personnel, family members, and the
general public, and that the negative stigma associated
with CFS may be partially due to the trivializing
name. The article, "How Science Can Stigmatize:
The Case of Chronic Fatigue Syndrome" discusses how
flawed scientific policies have contributed to the
stigma associated with CFS and have affected research
findings and clinical care.


Schools are often the most intractable, though school problems
are secondary to the lack of education and awareness and
sufficient backup from the medical community. Calling the
illness 'chronic fatigue syndrome' implies that the child is tired
rather than suffering from multiple disabling effects of brain,
CNS and immune system dysfunction (as typically found in the
illness that was previously known as Myalgic Encephalomyelitis
or ME). In general,  "fatigue" is no excuse to miss school (as in
"we have sick kids with real diseases who 'try' to come to
school so no excuse for the tired ones").


Absent adequate research and information, it has become
too easy  to assume that the illness is psychological.
Patients and particularly children are being harmed by these
erroneous assumptions. The overwhelming experience as
well as some available research shows that pacing and living
within the energy envelope is the most effective way to manage
the illness and that it is harmful to push beyond the boundaries
and force recovery. Yet much of the advice on "CFS" from
uninformed physicians is to the contrary.



It has become impossible to separate the medical and
educational systems. School absences often become the
focus and trigger suspicions of truancy or educational neglect.
Yet children are often made worse by inappropriate or unrealistic
educational demands and pushing too hard. It is abuse to send
a child to school when they are ill but if the illness is not
recognized by pediatricians, parents have nowhere to turn
and may even be charged with neglect if they do not send
them to school.


Families of ME/CFS children can face their worst nightmare
at the hands of these misinformed doctors and school systems.
Parents are being falsely accused of Munchausen Syndrome
by Proxy, also known as Factious or Induced Illness
(MSBP/FII), simply because the child has a neurological
illness that is often stigmatized and misunderstood. False
accusations of MSBP/FII have frequently involved social
service agencies regarding ME/CFS children, with the result that
the child may be removed from the home.


The Tymes Trust children's charity in the UK produced
"The Forgotten Children" Dossier in 2003. A  survey was
conducted and featured on BBC Panorama, which  showed
very disturbing statistics regarding the accusation rate of
Munchausen Syndrome by Proxy (MSBP/FII) of parents with
ME/CFS children.


Statistics at that time showed that overall accusations
of Munchausen's Syndrome by Proxy affected
just one in 100,000 families.  Yet 7% of children from
families questioned had been subject to child protection
proceedings and court proceedings had either been
threatened or carried out. If this is representative, it implies
that seven out of every one hundred children with
ME/CFS will be threatened with being taken away from
their parents.


Cases of children with ME/CFS being taken into custody
due to false allegations and accusations of abuse, neglect
and MSBP/FII are by no means diminishing but are continuing
worldwide. 


Thus it was extremely disturbing that a press release was
issued on a CDC funded study which identified childhood trauma,
sexual abuse and emotional maltreatment as a major risk factor
for the development of CFS. Yet other studies on the relationship
of trauma to CFS have shown the opposite.


Apparently no consideration was given by CDC to an extensive
risk factor study ("Risk factors for chronic fatigue syndrome/myalgic
encephalomyelitis: a systematic scoping review of multiple predictor
studies") that reported on various potential risk factors for the
development of CFS.  It concluded that definitive evidence
that appears meaningful for clinicians is lacking.


Why was CDC studying something as vague as childhood
trauma and when CDC has not looked into more obvious
or compelling risk factors associated with physical
or biological rather than psychosocial aspects.


For example, there is  widespread agreement that a variety
of infections are capable of precipitating ME/CFS (which the
National Center for Zoonotic, Vector-Borne, and Enteric
Diseases (ZVED) should be able to properly study). Furthermore,
if an antigenic challenge by infection can precipitate ME/CFS,
then it would follow that vaccines could act similarly. Lloyd
et al reported that several patients linked the onset of ME/CFS
to receiving a vaccination in the absence of infection.
These associations have been reported by Dr. Byron Hyde
of Canada and by Dr. Charles Shepherd of the UK and were
included in the CMO report (Report to the Chief Medical Officer
of an independent working group to the UK National Health
Service).


CDC constantly focuses on the role of stress, yet it is very
stressful to have an illness that remains unrecognized
and unaccepted. Research has found that the majority of
CFS patients seeking medical treatment  reported feelings
of estrangement, and one study found that 66% of individuals
with CFS felt that they were made worse by the care they
received
(source: Prohealth Live Chat Q&A with ME/CFS
Research & Policy Leader Leonard A. Jason, PhD, August 14,
2007).


Dr. Nancy Klimas, renowned expert and past President of the
International Association for CFS/ME, was quoted in the Miami
Herald as saying  "I've had patients who met post-traumatic stress
disorder criteria.. . . where their trauma was their interaction with
their physician around this illness. They came to a doctor with
Chronic Fatigue Syndrome; they left the doctor with PTSD."


Perhaps the finding of PTSD or stress related gene expressions
in ME/CFS is iatrogenic. CDC's focus on stress or related gene
abnormalities may be studying the result, not the cause.


And consider how stressful it must be, particularly for sick
children, who have lost much of their formative years and
identity, to be dismissed, blamed, viewed as lazy, malingering
or irresponsible, when nothing could be further from the truth.
When in fact they generally try much harder and push well
beyond what they are capable of physically and cognitively
to the detriment of their own health.


With any other illness these children would be heroes and
applauded for their tenacity and efforts, with all kinds of help
and support available to them and their families. Anyone facing
a crisis or illness always admits they they could not have
managed or survived without support and understanding, yet 
patients with ME/CFS often have none.


As difficult as it is for adults, think of what happens to children
who are not believed and forced to attend school and gym class
and are literally punished for being sick. Adults may be fully
disabled and do not have to work, yet children are required
to attend school.


Health should come first, and children should be given sufficient
time to recover  before returning to school. Sustainable,
manageable or even home-based education is important
to meet education requirements.


These families are not only trying to fight the illness itself to care
for their children but must constantly deal with all of the prejudice
and misinformation surrounding it. Affected children continue to
struggle for recognition of their needs, and often are dismissed
or even bullied by medical and educational professionals. It is
the children and families that are being abused and stressed
by the failure of the systems, agencies and institutions to
properly research the illness and educate those who should be
helping and caring for them.


With support and concern or even basic understanding lacking,
it can become overwhelming for families. Parents have to try to
assume the role of doctor, nurse, teacher, psychologist and
often best friend. Over time many children are never able to
work and contribute financially, many cannot drive, so the
burdens on families are enormous. It is not uncommon to
have mothers who have ME/CFS as well as the child, or to
have more than one child with it. It  becomes nearly a full
time job handling all of the medical, school and
social/emotional issues.


Children can miss a great deal of school, often years. A UK
study (Dowsett/Colby) found that ME/CFS constitutes the
biggest cause of long-term sickness leading to absence
from school, in both staff and pupils. It found that the scale
of the problem in children is substantial, and the pattern of
illness showed a clustering of cases.


Many children have not received an adequate education, have
had very limited social interactions  and normal childhood
and "life" experiences and become very isolated. They may
very well end up having emotional problems, depression or
would understandably be angry, resentful or distrustful
of authorities.


These children may get so far behind and frustrated that they
simply drop out of school. Many families have such traumatic
experiences with schools that they literally just try to stay beneath
the radar of suspicion and bide their time until the child is 16
and can drop out.


Under these circumstances, how may children with ME/CFS,
or those who are not properly diagnosed or treated, have
turned to drugs or alcohol, or even suicide? 


Studies have looked at the profound economic impact of CFS
in the adult population, but what of the future effect on these
children and society? Some studies have found that children
may recover in their late teens or early 20's, but even for those
lucky enough to recover, many have still missed their formative
years and large amounts of school, often years. These educational
opportunities  are completely lost with regard to public education
as most school districts do not provide services beyond age 21.


For those who get sick in childhood or in their teens and do not
recover, many get worse over time, unable to work or even become
bedridden. And aging parents struggle and worry about what will
happen to their children, as disabled adults,  when they can no
longer care for them.


How successful can a research program be when the real stakeholders
(i.e. patients) report thusly. In the 20 years since the inception of the
CDC "CFS" research program, there has been no real progress of
direct benefit to patients in terms of recognition, diagnosis, treatment
or basic care. The merit of the investment should be judged by the
final result.


This situation must change with. The real tragedy is that with proper
diagnosis and treatment, children can receive appropriate care and
support even if there is no cure, and with appropriate accommodations
they will be able to access the best educational opportunities possible. 


Jill McLaughlin



Input on CDC CFS Strategic Research Plan



Discontinue the use of the new "emperical" definition and
rebranding CFS as a "stress-related disorder."  This
definition is too non-specific, reduced to questionnaires based
on a Wichita 2-day hospital stay, and excludes the most
serious patients and includes patients who mainly
have depression.


Adopt the Canadian Myalgic Encephalomyelitis/ Chronic Fatigue
Syndrome: Clinical Working Case Definition, Diagnostic and
Treatment Protocols. A DePaul study ( "Comparing the Fukuda
et al. Criteria and the Canadian Case Definition for Chronic
Fatigue Syndrome") found that the Canadian criteria group, in
contrast to the CFS Fukuda group, had more variables that
statistically significantly differentiated them from the psychiatric
comparison group. The Canadian criteria selected cases with
less psychiatric co-morbidity, more physical functional impairment,
and more fatigue/weakness, neuropsychiatric, and neurological
symptoms.


Recognize and focus research direction on known abnormalities of
reduction in grey matter of the brain, mitochondrial abnormalities,
channelopathies and aberrant ion transport, low natural killer cell
cytotoxicity, cytokine shift from Th1 to Th2, sympathetic nervous
system hyperactivity, cortisol deficiency, left ventricular dysfunction
in the heart and other cardiovascular abnormalities that can have
serious clinical implications
.  A variety of theories have been proposed
to explain these findings and offer insight into the pathophysiology,
including infectious agents, viruses, bacteria, tick borne infections,
immune dysregulation, neuroendocrine problems, as well as
neurologic abnormalities, oxidative stress and kindling.
We need clinical and laboratory-based studies of homogeneous
groups of patients to produce meaningful data that can be replicated
and used to provide insight into the nature and pathophysiology,
not  questionnaires  about fatigue and "unwellness."


Expand the CDC CFS research program.  One person should not
have full control of any research program or budget. Then fund and
award contracts and grants to outside investigators who have
proven abilities and accomplishments in targeted areas.


Expand the genetic research beyond narrow focus of stress related
genes and HPA axis abnormalities.  Genomics and proteomics have
proven to be useful in the detection of diseases and development of
therapeutic modalities.  Use genomic and proteomic tools that are
available for the diagnosis of infectious diseases.


Extend investigations and collaboration with others in the field doing
similar work and share data.


Remove references and links to the thoroughly flawed and discredited
NICE (UK NHS National Institute for Health and Clinical Excellence)
guidelines  for diagnosis and management. These guidelines do not
follow the neurological WHO classification and  promote CBT and GET
(graded exercise therapy), which has been shown to make patients
worse. Pacing involves the conservation of energy, not expending more to force recovery.


Discontinue the "Awareness" campaign PR Marketing strategy. Millions of
dollars have been wasted that could have been put toward research
and provider education, which is much more important than having
photo exhibits at Malls. PR and Marketing are no substitute for science
and research. Establish broader consideration of the awarding of
contracts and collaborations and have measurable outcomes
by which progress can be evaluated. Have content of all material
developed and approved by a wide range of experts, not exclusively
by CDC or under the control of one organization. Establish transparency
of the use of funds and keep track of and report on progress of all
contracts through benchmarks and outcomes, which are made public. 
Too much money and time have been wasted on superficial, ill-conceived,
non-productive endeavors that have had little effect.


The International Association for Chronic Fatigue Syndrome/Myalgic
Encephalomyelitis (IACFS/ME) has developed a case definition for
Pediatric ME/CFS.  The existing case definitions have been developed
for adults and may not be appropriate for children. The IACFS/ME
convened a working group of experts who determined that there
is sufficient evidence to put forward a case definition for children
and adolescents. Having a consistent and reliable case definition
is of utmost importance to properly diagnose and classify patients
for research in order to determine the pathophysiology, identify
biomarkers and develop effective treatments.


The CDC should recognize the IACFS/ME Pediatric Case
Definition and use it as the basis to develop a research program
on pediatric ME/CFS with focus on determining the cause, risk
factors, biomarkers and effective treatments. Since children
with ME/CFS can become adults with the illness over time,
the natural history must also be considered. 


Jill McLaughlin


------

References:

The Miami Herald Friday, 05.01.09 HEALTH Q&A "Seeking answers
for chronic fatigue syndrome" by Gigi Lehman

The Energy Envelope Theory and Myalgic Encephalomyelitis/Chronic
Fatigue Syndrome," Leonard Jason, PhD  AAOHN Journal Vol. 56  
No. 5   May 2008   

Jason, L.A., Holbert, C., Torres-Harding, S., & Taylor, R.R.
(2004).  Stigma and chronic fatigue syndrome: Surveying a
name change. Journal of Disability Policy Studies, 14, 222-228.

Reeves WC, Wagner D, Nisenbaum R, Jones JF, Gurbaxani B,
Solomon L, Papanicolaou DA, Unger ER, Vernon SD, Heim C,
Chronic fatigue syndrome - a clinically empirical approach to its
definition and study  BMC Medicine 3:19, 2005

"Problems with the New CDC CFS Prevalence Estimates"
Leonard Jason, Ph.D., DePaul University, IACFS/MEwebsite
Case Definitions, CDC Criteria: http://www.iacfsme.org/IssueswithCDCEmpiricalCaseDefinitionandPrev/tabid/105/Default.aspx

Carruthers BM, Jain AK, De Meirleir KL, et al. Myalgic encephalomyelitis/chronic
fatigue syndrome: clinical working case definition, diagnostic and treatment
protocols. Journal of Chronic Fatigue Syndrome. 2003;11:7-116.

Cohen S, Janicki-Deverts D, Miller GE. Psychological stress and disease.
JAMA. 2007 Oct 10;298(14):1685-7.

De Lange, FP, Kalkman, JS, Bleijenberg, G, Hagoort, P, van der Meer JWM,
& Toni, I. Gray matter volume reduction in the chronic fatigue syndrome.
NeuroImage, 2005;26:777-781.

Patarca-Montero, R, Mark, T, Fletcher, MA, & Klimas, NG. Immunology of
chronic fatigue syndrome. Journal of Chronic Fatigue Syndrome,
2000;6:69-107.

Jason, L.A., Taylor, R.R., Stepanek, Z., &  Plioplys, S. (2001). Attitudes
regarding chronic fatigue syndrome: The importance of a name. Journal
of Health Psychology, 6, 61-71.

Hyde BG, JA; Levine, P. The clinical and scientific basis of myalgic
encephalomyelitis/chronic fatigue syndrome. Ottowa, Ontario: The
Nightingale Research Foundation; 1992.

Kennedy G, Spence VA, McLaren M, Hill A, Underwood C, Belch JJ.
Oxidative stress levels are raised in chronic fatigue syndrome and
are associated with clinical symptoms. Free Radical Biology and
Medicine. 2005;39(5):584-9.

Jammes, Y, Steinberg, JG, Mambrini, O, Bregeon, F, &
Delliaux, S. Chronic fatigue syndrome: assessment of increased
oxidative stress and altered muscle excitability in response to
incremental exercise. Journal of Internal Medicine.
2005;257:299-310.

The Forgotten Children - A Dossier of Shame, Tymes Trust
Publication. Presented to the Prime Minister on the 12th of
May 2003. It details shocking experiences and statistics that
the organization has collated from information supplied by
families of children with ME.

University of North Carolina at Chapel Hill (2008, August 20). '
Chilling' HardshipRates Among Families Raising Disabled
Children. ScienceDaily.

Lloyd A et al. What is myalgic encephalomyelitis? Lancet. l988; l:
1286-7.

Weir W. The post-viral fatigue syndrome. Current Medical Literature:
Infect Dis. l992; 6: 3-8.

Hyde B. The clinical investigation of acute onset ME/CFS and
MS following recombinant hepatitis B immunisation. Second World
Congress on CFS and Related Disorders, Brussels. 1999; September
9-12.

Report of the working group on the possible relationship between
hepatitis B vaccination and the chronic fatigue syndrome. Canadian
Med Assoc J. l993; 149: -9.

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