Chronic Fatigue Syndrome
Dr Kathy Rowe
Royal Children’s Hospital, Melbourne, Australia
December 15, 2014
Chronic Fatigue Syndrome
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Is it a new illness?
What are its features?
Is it all ‘in the mind’?
How common is it?
How long does it last?
What are the self reported outcomes?
What management strategies do young people
find helpful?
• Do we know what makes a difference different approaches?
Is it a new illness?
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Neuraesthenia 1869
DaCosta’s Syndrome 1871
Royal Free Disease 1955
Icelandic Disease 1958
Chronic EBV
Post viral fatigue syndrome
Myalgic Encephalomyelitis
What are its features?
• In Australia:
– Typically after infective process
• EBV, CMV, influenza, polio, Chicken pox, gastroenteritis, Ross
River, Q fever, Mycoplasma, Malaria, Dengue Fever, HHV6,
giardia
– Overlaps with ‘overtraining syndrome’ – trivial illness in
the context of heavy training schedule – same symptoms.
• In Japan:
– Chronic sleep deprivation and stress – very rare to be
post – infective – typical onset - end primary school
What are the typical symptoms?
• Symptoms are very consistently reported:
– Defined onset of an ‘ill’ fatigue.
– Feel much worse after any activity (ill not just tired) (postexertional malaise)
– Unrefreshing sleep : sleep disturbance – too much/ difficulty
settling/ frequent waking / sleep phase shift /vivid dreams
– Concentration difficulties, easily ‘overloaded’
– Constitutional symptoms: headache, aches and pains, sore
throats, sore glands, nausea, dizziness, abdominal pain –
generally ‘feeling dreadful’, emotional fragility
Demographics
• Common illness >1200 in 20 years at RCH
• M:F 1:3
• All socioeconomic groups represented (high achievers
tend to be believed)
• 95% of RCH sample - Scottish/Irish/ Northern
European descent – Asian / Southern European
descent under-represented
• Age 6-18 years mode 16 years (EBV)
• 30% rural (reflects population) 2% interstate
Does CFS occur in young people?
• Concerns that adolescents may be different from
adults
• May be ‘somatization’ because no clear test
available
• “Unhelpful to give the diagnosis to young people”
• “Don’t believe in the syndrome”
Confirmatory Factor analysis
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38 item symptom questionnaire used by adult service
Data from 189 young people
Control group adolescents
24 of 38 symptoms included in analysis
remainder - had:
-low occurrence and severity response frequencies
-non-significant relationship with the underlying scale
- marginal relevance to CFS symptomatology
Sample characteristics
• Duration: minimum 6 months
• defined onset over hours or days
• fatigue exacerbated by exercise and not relieved
by rest
• neurocognitive symptoms
• at least 3 of following: myalgia, arthralgia,
headaches, sleep disturbance, abdominal pain,
dizziness, nausea, pharyngitis & lymphadenopathy
Analysis of data
• 24 of the 38 symptom items were included for
analysis
• factor scores were calculated for the 5 component
factors using proportionally weighted regression
coefficients for each item
– 5 factors •
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‘pain/fatigue’ items
‘headache/abdominal pain’
‘neurocognitive symptoms’
‘neurophysiological’
‘immunological symptoms’.
Muscle Pain & Fatigue:
First-order items & standardized factor loadings
Muscle pain (not joint pain) after activity
Muscle pain (not joint pain)
even when doing nothing
MUSCLE
PAIN & FATIGUE
Excessive muscle fatigue with minor activity
Joint pain
Prolonged feeling of fatigue after physical
activity lasting for hours (or days)
Neurocognitive:
First-order items & standardized factor loadings
Loss of concentrating ability
NEUROCOGNITIVE
Difficulty with speech - ‘lost for the word’
Memory loss
Vivid dreams or nightmares
Abdominal, Head & Chest Pain:
First-order items & standardized factor loadings
Stomach pain
ABDOMINAL,
HEAD & CHEST PAIN
Nausea
Headache
Recurrent chest pain
Neurophysiological:
First-order items & standardized factor loadings
Recurrent chest pain
Feeling of disturbed balance
Difficulty in focussing vision
Disturbed sleep or disrupted sleep pattern
NEUROPHYSIOLOGICAL
Persistent dryness in the eyes or mouth
Shortness of breath with minor activity
Palpitations (feeling the heart racing)
Needing to sleep for long periods
Immunological:
First-order items & standardized factor loadings
Tender glands in the neck
Tender glands elsewhere
IMMUNOLOGICAL
Sore throat without common cold symptoms
Repeated fevers and sweats
Chronic Fatigue Syndrome:
Second-order CFA standardized solution
MUSCLE PAIN
& FATIGUE
.701
.636
.702
Model Goodness-of-fit Indices:
ABDOMINAL
HEAD & CHEST PAIN
.914
.710
NEUROPHYSIOLOGICAL
2 (246) = 33.9; p = 0.999
RMSEA = 0.035; SRMR = 0.01
GFI = 0.996; AGFI = 0.971
Repeat sample
• Second sample of 239 young people –
analysis repeated
• Identical factor structure and only one item
loaded more heavily on a different factor
• Therefore young people very consistently
report the same symptoms in the same
clusters.
Structural equation regression model:
Standardized solution
Model Goodness-of-fit Indices:
IMMUNOLOGICAL
2 (4) = 1.1; p = 0.893
RMSEA = 0.00; SRMR = 0.01
GFI = 0.998; AGFI = 0.991
.73
NEUROPHYSIOLOGICAL
.27
.52
ABDOMINAL, HEAD
& CHEST PAIN
.67
.17
.60
NEROCOGNITIVE
MUSCLE PAIN
& FATIGUE
.486
.488
Clinical groups within CFS
• K means cluster analysis (Statistica)
– 3 groups were identified
– group classification was confirmed by discriminant
function analysis.
– Correct classification into 3 groups occurred with 99.87%
accuracy (P<0.0001).
Factor score means for 3 groups
Plot of discriminant analysis for 3 groups
Root 1 vs. Root 2
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4
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Root 2
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0
-1
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-8
-6
-4
-2
0
Root 1
2
4
6
8
G_1:1
G_2:2
G_3:3
3 clinical groups
– most severe group experiencing severe symptoms on all
factors
– the intermediate group had relatively more severe
pain/fatigue, neurocognitive, and neurophysiological
symptoms
– least severe group had a similar profile to the severe
group except that neurocognitive symptoms were
relatively more severe than the others
3 Clinical groups
– 25% in most severe
– 50% in the intermediate group
– 25% in the least severe group
– no difference between the groups on duration of illness
– i.e., the difference in severity was not related to
improvement with time.
Is it ‘all in the mind’?
• No difference in responses between control group and CFS group in
parental bonding scale (Parker) ie parents were not more
‘protective’
• CFS group were mildly more anxious than controls (Speilberger)
• Baseline depression in controls 20% (Beck), 27% in CFS group – rare
to have anhedonia
– Commonly associated with ‘not being believed’, severity of symptoms,
‘schooling’ not working well
– ie not very different from controls and appropriate response to illness
How long does it last?
• To follow up consecutive patients referred to the CFS
clinic at the Royal Children’s Hospital over 18 years
(1991 – 2009)
– level of functioning
– self-reported perception of recovery
– duration of illness
– usefulness of management strategies.
788 young people with CFS
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age 6-18 years (mean 15 years)
M:F 1:3
30% rural (reflects population distribution), 2% interstate
Ethnic origins
– 85% with either parent with Scottish / Irish surname
– 10% northern European (Dutch, Scandinavian, German)
– No Middle Eastern, African, 1 Sri Lankan (with Scottish
Grandfather), 1 Taiwanese, 3 Chinese descent with only 1 with
both parents Chinese
• CFS patient ethnic origins not representative of Victorian
population or those that visit RCH
Telephone interview
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Work / school / tertiary attendance
Work / school / tertiary workload
Type of work or course undertaken
Social activity
Physical activity
Generally, how are they managing?
Follow up questionnaire
• Demographics
• Functional outcomes
– Academic level
– Nature of work
– Use of social security support
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Duration of illness if reported recovered
Additional illnesses
Use of alternative health practitioners
Reported usefulness of management strategies
Questionnaire
• Duration of illness - if well
• Persistence or exacerbation of symptoms
• If symptoms have recurred - was there a
trigger?
• Presence of other serious illness
• Strategies/treatments that were helpful
• Ways management could be improved
Month of onset of illness
16
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12
10
8
6
4
2
0
Jan
Feb
Mar
Apr
May June July Aug Sept Oct
Months
Nov
Dec
Management principles
• Symptom management
– Sleep
– Headache
– Dizziness (POTS/NMH –postural orthostatic tachycardia/neurally
mediated hypotension)
– Depression
– Pain
• ‘Lifestyle’ management - balance
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Social
Academic
Physical activity
Commitment to attend ‘something’ regularly
Review each month
Follow up
• 1st group
– At least one return from 342 of 398 (86%)
– 6 occasions between 1998 and 2008 provided 804 returns
• 50% reported recovery
• 2nd group
– 78% contacted and provided information
• 33% reported recovery
• Both groups follow up 1.7-21 years
Functional ratings (mean 7.6, sd 1.7) at follow up
mean 7.8 years sd 4.3 (range 1-21) n=570
Distribution of duration of illness
mean 5 years (sd 2.8) n=240
• Of those who report recovery
– 60% are well by 5 years
• Average duration is 5 years in young people
(range 1-13)
• For those followed more than 12 years 68%
reported recovery (n=67)
Functional status
• Of those who reported recovery ~1/3 indicated that
they were conscious of monitoring their workload.
• Less than 5% were not either studying or working
part or full time, often due to other factors than CFS.
• Many had married (n=38) and those with children
(n=15) reported being well.
• 90% completed or intended to complete postsecondary school training.
Use of alternative practitioners
• 70% used alternative therapists
• 40 different therapies described
• <10% ‘helpful’
– relief for muscle pain with massage
– good dietary advice.
– restrictive diets and supplements did not reach placebo levels
of response.
• Common comment:
– “I was grateful when my parents stopped carting me around
to people who said they could cure me”
What was useful?
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Management strategies
Assistance with school
Being believed
Feeling supported
Having some control
Having information
Symptom management
Contact with others with the illness (not alone)
What was a problem?
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Not being believed
Delay in diagnosis
School not being helpful
Family and friends offering advice/’cures’
“People telling me that exercise would ‘fix it’ – I love
sport, if I could do it, I would”
• “Being too frightened to try things sometimes”
• Ignoring the emotional impact on me
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Could anything be managed better?
Earlier diagnosis
Education authorities being more flexible
Not telling me that I would be well ‘soon’
Learning to pace myself better – too much ‘boom and
bust’
More access to support groups
Less visits to doctors/ ‘quacks’ before RCH
“I should have been more receptive to emotional issues
(probably wasn’t ready anyway)”
60% volunteered ‘happy with management’
Any predictors for outcomes?
• Best predictor for functional outcome (whether
recovered or not) was continued engagement with
education
• Attention to social connectedness very important for
the social learning of adolescence
• For many young people sporting activity was their social
network – for the less severe this contact was more
important than full school attendance
• Most effort was involved in negotiating with
school/educational authorities
Conclusion
• Average duration 5 years until ‘recovered’ (1-16 years)
• At 12 years 68% reported ‘recovery’
• Overlap in functional score between those recovered
and those ‘not yet recovered’
• Management strategies well received
• Engaging in education the best predictor of functional
and emotional well being
• Flexibility in education program essential