The Chalder Fatigue Questionnaire is a valid and reliable measure

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The Chalder Fatigue Questionnaire is a valid and reliable measure
of perceived fatigue severity in multiple sclerosis
Joseph Chilcot1, Sam Norton1, Maedhbh Etain Kelly2, Rona Moss-Morris1
1
Health Psychology Section, Psychology Department, Institute of Psychiatry, Psychology and
Neuroscience, King’s College London, UK
2
Department of Psychosis Studies, PO63, Institute of Psychiatry, Psychology and Neuroscience,
King's College London, UK
Correspondence: Rona Moss-Morris (rona.moss-morris@kcl.ac.uk)
Health Psychology Section, Institute of Psychiatry, Psychology and Neuroscience King’s
College London, 5th floor Bermondsey Wing, Guy’s Hospital Campus, London Bridge, London,
SE1 9RT
Key words: Chalder fatigue questionnaire; Chalder fatigue scale; fatigue; multiple sclerosis; MS;
psychometrics; measurement; confirmatory factor analysis
Conflict of interest: None declared
1
Abstract
Background: Fatigue is one of the most distressing symptoms of Multiple Sclerosis (MS).
Measuring MS fatigue poses a number of challenges. Many measures confound definitions of
severity and impact of fatigue and/or lack psychometric validation in MS.
Objective: To evaluate the psychometric properties of an 11 item fatigue severity measure, the
Chalder Fatigue Questionnaire (CFQ) in MS including validity of the factor structure, internal
reliability, discriminant validity, and sensitivity to change.
Methods: Data was pooled from four previous studies investigating MS-fatigue using the CFQ
(n=444). Data analysis included confirmatory factor analysis to determine the factor structure
and model fit, correlations to assess discriminant validity, and effects sizes to determine
sensitivity to change.
Results: A bi-factor model with one general fatigue factor, incorporating 2 smaller group factors
(mental and physical fatigue) had good model fit and appeared the most appropriate factor
structure underlying the CFQ scale. The CFQ had high internal consistency, showed small to
moderate correlations with impact of fatigue and mood, and was sensitive to change across low
and high intensity behavioural interventions.
Conclusions: The CFQ measuring a composite of physical and mental fatigue severity (i.e. a total
score) is a psychometrically sound measure of fatigue severity in MS.
2
Introduction
Fatigue is reported as one of the most common and disabling symptoms of multiple sclerosis
(MS)1, 2. MS fatigue has been defined as “a subjective lack of physical and/or mental energy that
is perceived by the individual or caregiver to interfere with usual and desired activities” 3, and
remains a complex and debilitating phenomenon. Fatigue can be distinguished from fatigability,
whereby fatigue is conceptualised as the subjective sensation and fatigability the objective
changes in mental or physical performance 4. Subjective reports of MS fatigue have a significant
impact upon quality of life, and are associated with negative psychosocial factors including
unemployment 5, 6. MS fatigue can be distinguished from fatigue occurring in healthy persons by
its rapid onset, heat sensitivity, and tendency to interfere with day-to-day activities7, 8.
Fatigue is clearly an important symptom, but the assessment of fatigue in MS poses a challenge
due to its subjective and multi-faceted nature. Although attempts at objective measures of
fatigability have been made, self-report questionnaires are the most common, and possibly the
most effective way of evaluating fatigue in both research and clinical settings 9,
10
. There are
numerous fatigue self-report scales. The most commonly used in MS are the Fatigue Severity
Scale (FSS)8 and Fatigue Impact Scale (FIS)11. The 11 item FSS was initially developed and
validated on people with MS. However, whether the FSS measures severity specifically is
questionable. The items represent a conglomerate of effects of fatigue on daily life (e.g. fatigue
interferes with my daily functioning), triggers of fatigue (exercise brings on fatigue) and
miscellaneous items (I am easily fatigued). Although the FSS shows good psychometric
properties in MS
12
it measures multi-facets of fatigue rather than severity of the symptom
experience specifically.
3
The FIS and Modified FIS11, 13 provide a clearer operational definition. Items measure impact of
fatigue on physical, cognitive, and psychosocial functioning in MS. The measure is
psychometrically sound
14
. However, a recent evaluation of the face validity of the FIS by MS
health professionals concluded that the items were non-specific to fatigue impact 14. In addition,
impact is not the same as severity of fatigue. Guidelines in the pain literature suggest outcomes
of clinical interventions for pain should include both measures of the impact of pain on daily life
and the severity or intensity of pain15. Although pain interference and impact are correlated they
are sufficiently distinct that interventions can show change in one but not the other16.
This may also be true for fatigue. Including measures of perceived severity and impact of
fatigue in MS will not only help us understand fatigue better but elucidate intervention effects on
fatigue. This is consistent with a recent review on measuring fatigue in neurological illness
which emphasised the need for measures which clearly define specified components of fatigue 4.
As both the FSS and FIS incorporate measurement of impact of fatigue, a validated measure
specific to fatigue severity in MS is warranted. The Chalder Fatigue Questionnaire (CFQ) was
originally developed for use amongst patients with Chronic Fatigue Syndrome17. It consists of
11 items loading onto two dimensions of fatigue severity – mental fatigue and physical fatigue
which map onto to the operational definition of MS fatigue presented in the opening paragraph.
The instrument has been found to have good clinical validity and internal consistency within this
population18, 19. Given its efficiency and easy utilization, the CFQ is a popular assessment of
fatigue within a range of illnesses. However, there has been no formal assessment of the validity
and consistency of this scale in the specific context of MS-related fatigue. Furthermore, the
multidimensionality of fatigue scales has been widely debated, with data suggesting that most
measures, including the CFQ, are in fact unidimensional20
4
The overarching objective of this study was to consider whether the CFQ is a suitable tool to be
used in the evaluation of fatigue severity in people with MS. Our aims were to evaluate the
psychometric properties of the CFQ with respect to its factor structure, internal reliability,
sensitivity to change following intervention and discriminant validity. To assess discriminant
validity we explored relationships between the CFQ and measures of both fatigue impact and
depression. Previous work suggests there is a relationship between fatigue and depression but
that depression can improve independently of fatigue and vice versa
4, 21
. We would therefore
only expect small to moderate correlations between fatigue and depression. Similarly, as we
argued that severity and impact should be considered independently, we expected only moderate
relationships between the CFQ and measures of impact of fatigue.
Methods
Participants and design
Participants were drawn from four recent studies (n=444), which either investigated correlates of
MS fatigue22, 23 or trialed CBT-based treatments for MS fatigue21, 24. The data included in the
main analysis was either cross-sectional or at baseline in the context of the two randomised
controlled trials (RCTs). Demographic and illness characteristics for each study are shown in
Table 1 and the ethical approvals for each cohort are described in the relevant publications.
INSERT TABLE 1 ABOUT HERE
Instruments
The Chalder Fatigue Questionnaire (CFQ)17, also referred to as the Chalder Fatigue Scale, is
an 11-item questionnaire measuring the severity of physical and mental fatigue on two separate
5
subscales. Seven items represent physical fatigue (items 1-7) and 4 represent mental fatigue
(items 8-11). The studies from which the pooled data was collected used a slightly updated
version of the CFQ25,
26
, which has been used widely including in the PACE trial27. In this
version the item “Do you have problems thinking clearly?” is replaced with “Do you find it
more difficult to find the correct word?”. Cella and Chalder25 state that this slight amendment
improves the scales reliability, although either item could be used without impacting on the
measures interpretation26. Within the context of MS the questions are asked with the following
stem ‘We would like to know more about any problems you have had with fatigue in the last
month. Please answer ALL the questions simply by ticking the answer, which you think most,
applies to you. We would like to know how you feel at the moment, or recently, compared to
when you were last well’. Each item is scored 0-3; less than usual (0), no more than usual (1),
more than usual (2) and much more than usual (3). The ratings of items are added together to
calculate the total score (range=0-33). High scores represent high levels of fatigue.
Discriminant validity was assessed through:
1. Work and Social Adjustment Scale (WSAS) is a valid and reliable 5 item self-report
measure of impairment in relation to an identified disorder or symptom 28. In the context of
these studies items measured impact of fatigue on home management, work, social leisure
activities, private leisure activities and the ability to form and maintain close relationships.
2. The Modified Fatigue Impact Scale (MFIS) is a shortened version of the Fatigue Impact
Scale validated in people with MS 11.
6
3. Hospital Anxiety and Depression Scale (HADS) 29 is a commonly used self-report measure
of mood in patients with medical illnesses. 7 items relate to anxiety and 7 items relate to
depression.
Statistical Methods
The factor structure of the CFQ was examined using CFA in MPlus 7.1. Competing models were
estimated using Weighted Least-Squares with Mean and Variance adjustment (WLSMV)
estimation, testing one, two factor and bi-factor models of fatigue. In the bi-factor models, all 11
items were loaded onto a general fatigue factor. In addition, items were also loaded onto a
number of group factors, with correlations between each of these latent factors fixed to zero.
Assessment of goodness-of-fit based on standard structural equation modeling criteria: root mean
squared error of approximation (RMSEA) <.08, confirmatory fit index (CFI) >.95, and TuckerLewis index (TLI) >.95
30
. Reliability of the total and subscale scores was assessed using the
omega index, along with an indicator of the saturation of a multidimensional scale by a general
factor, omega-hierarchical, for the bifactor models31, 32. Discriminant validity between the fatigue
factors with other patient reported outcomes (depression, anxiety and disability) was evaluated
using Pearson’s correlation.
Sensitivity to change was assessed using the data from the two RCTs21, 24. Treatment effects, in
terms of post-treatment standardised mean differences (Cohen's d), on the CFQ were estimated
for CBT versus treatment as usual24 and for CBT versus relaxation21. Following the intention-totreat principle, missing post-treatment scores were imputed by carrying forward the baseline
score. In addition to the treatment effects, the proportion of individuals showing a reliable
improvement in fatigue following the method proposed by Jacobsen and Traux33 was calculated.
7
In order to assess if the measure remains relatively stable over a 10-week period without
treatment we calculated Pearson’s correlations between baseline and follow-up CFQ in the no
treatment control group.
Results
Confirmatory factor analysis of the Chalder fatigue Questionnaire
A series of CFA models were examined. The details of the five models are presented in the
technical appendix together with a table of the summary statistics for the fit of each model. The
first three models illustrated that items 6 (less strength in muscles) and 7 (feeling weak) of the
original CFQ negatively impacted the fit indices. Since these items appear to measure weakness
rather than fatigue, they were dropped in the final two models. The model with the best fit and
most satisfactory face validity was a 9-item bi-factor model with two group factors (see technical
appendix table 1; model 4b). Model estimates from this analysis are shown in Table 2. The
general factor explained 81.4% of the common variance between items. The mental (4 items)
and physical group (5 items) factors explained only a small amount of common variance – 12.4%
and 6.2% respectively. Omega hierarchical was .89, indicating that the total score across all
items included in the scale predominantly reflects a general fatigue factor. Considering the
mental and physical subscales separately, the reliability coefficients were both .96. However,
controlling for the part of the reliability attributable to the general factor the coefficients drop to
.20 and .10, respectively. Together this indicates that, even though the scale is multidimensional,
the total score for the scale is a reliable indicator for general fatigue. Total scores for the physical
and mental subscales are saturated by the general factor and thus reflect general fatigue rather
than separate constructs of physical and mental fatigue.
8
INSERT TABLE 2 ABOUT HERE
Discriminant validity: correlations between fatigue, depression, anxiety and disability.
The original total CFQ sum score (11-items), and shortened sum score (9-items) were correlated
with the HADS (depression and anxiety sum scores) and WSAS (see Table 3) to determine
discriminant validity. As hypothesised, depression, anxiety and impact of fatigue (WSAS) all
had significant but small to moderate positive associations with the total CFQ fatigue factor (bifactor model), original (11-item) and shortened sum (9-item) scores. The size of the correlations
were very similar for the 11 and 9 item versions. In a small subset of the total population, the 9item summed CFQ showed a small correlation with the Modified FIS supporting the argument
that severity and impact may be distinguishable (r=.22, p=.19 [n=39; data from Moss-Morris et
al., 2012]).
INSERT TABLE 3 ABOUT HERE
Sensitivity to change: Sensitivity to change was comparable for the 11 and 9-item CFQ versions
in terms of the treatment effects of CBT versus treatment as usual24 and versus relaxation21.
Compared to treatment as usual, the post intervention between group effect size for CBT using
the 11 and 9 item versions summed fatigue score was d=1.19 and 1.15, respectively. Compared
to relaxation, the post intervention between group effect size for CBT was d=0.76 and 0.81,
respectively.
For both the 11 and 9 item versions a reliable change was estimated to be a 3-point difference.
There was no difference between versions in the number of people in the intervention group that
9
exhibited a reliable improvement in fatigue between the baseline and post-treatment assessments.
In Moss-Morris et al 2416 of 23 (69.5%) and in van Kessel et al21 34 of 35 (97.1%) patients in the
CBT group exhibited a reliable improvement. For the treatment as usual group, the correlation
between CFQ at baseline and follow-up (10 weeks later) was r=.58; p=02 suggesting without
treatment scores remain moderately stable.
Discussion
The primary purpose of this study was to evaluate whether the Chalder Fatigue Questionnaire, is
a valid and reliable measure of fatigue severity in MS patients. In terms of factor structure, early
development of the CFQ with patients with chronic fatigue syndrome and healthy controls
revealed two-factors, measuring physical and mental symptoms of fatigue17. However, our
findings failed to support a two-factor model underlying the CFQ, as evidenced by poor model
fit and two highly correlated factors. Given this, we tested bi-factor models, which allows the
separation of variance into components related to a general factor, group factors and unique
variance. This modeling approach is increasingly used to test whether multidimensional
measures can be considered sufficiently unidimensional to allow for the use of a total score,
measuring one general construct34, 35.
A bi-factor model, containing a general fatigue factor, and two smaller group factors (physical
and mental) most appropriately fitted the data. Items 6 and 7 correlated highly and appeared to
measure something specific to weakness, rather than physical fatigue per se. Following
examination of models with these items correlated, loaded onto a third group factor (weakness)
or removed, model fit appeared most satisfactory when these items were removed. The two
10
group-factors explained relatively low variance, whereas the general factor, with all 9-items
loaded upon it, explained approximately 80% of the common variance. Therefore, whilst the
CFQ includes two dimensions of fatigue, it remains sufficiently unidimensional for the total
score (i.e. the sum-score) to be used as a reliable measure of general fatigue severity. The group
factors remain only fragile indicators of separate constructs, namely, mental and physical fatigue.
The saturation of total subscale scores by the general factor means the subscales would be
unreliable indicators of the unique constructs, thus we recommend using the total score as a
general fatigue measure in future studies. These findings support those of others20, 36, and casts
significant doubt over the practical distinction between physical and mental constructs of fatigue
in MS patients. Further support for this assertion regards the poor criterion validity of the
physical and mental subfactors in relation to other patient reported outcomes (depression, anxiety
and disability). That is, all of the association with these measures is due to common variance
accounted for by the general component of fatigue. The unidimensional nature of the CFQ is also
supported in the general population37, thus we encourage future research to use the measure as a
total score measuring fatigue severity, rather than subscales of mental and physical fatigue.
A secondary aim was to assess whether this measure of fatigue severity could be discriminated
from measures of impact of fatigue (WSAS and MFIS) and measures of mood. Whilst fatigue
severity was correlated with negative mood and the impact of fatigue on the ability to carry out
day-to-day tasks, the overlap between these constructs was small to moderate in size (accounting
for between 5-16% of the shared variance). These data suggest that it is worth including separate
measures of fatigue severity and impact, although it should be noted with respect to MFIS, the
available sample size was small. The data also suggest that fatigue severity can be discriminated
to some extent from negative mood since the correlations between fatigue and distress were
11
moderate in size. Correlations were very similar in size for both the 11 and 9 items versions of
the CFQ. The CFQ showed excellent sensitivity to change and large effect sizes in relation to
CBT designed specifically to reduce fatigue in MS, both when the therapy was delivered by a
therapist and through a website with some minimal support.
Sensitivity to change was
comparable for both the 11 item total score and the reduced 9 item version. This suggests
removing the two items relating to weakness did not impact on the properties of the total score.
That is, internal reliability, and thus precision, was not affected. This along with the other
analysis suggests that, in practice, the use of the either the 11 or 9 item version to assess fatigue
is supported in the MS population. The two items removed in the bi-factor model related to
weakness and it is conceivable that responses are confounded by disease symptoms in MS. These
items may be stronger indicators of fatigue in other populations. However, as there is no
evidence that the original 11-item version biases the validity of the instrument, we recommend
the continued use of the 11-item version as it allows comparisons with non-MS samples. There
appears to be little utility for using the 9-item version over the 11-item version.
Whilst our study has a number of strengths including the sample size and representative nature of
the MS patient sample, a few limitations are worthy to note when interpreting these data. Firstly,
our results are specific to the MS population and thus may not generalise to other populations.
Second, English speakers only completed the measure, therefore these data may not be
generalised to other languages or cultures. Specifically, the measurement models of fatigue
tested here may not be robust in other cultures, due to possible differences in the representation
and expression of fatigue symptoms. Furthermore, the available follow-up data from the two
pilot RCTs reported here21, 24 had insufficient sample sizes to determine model invariance over
time, using multiple group confirmatory factor analysis. The test-retest reliability yielded a
12
moderate coefficient (0.58). This is likely because the retest data was taken from a control arm of
a fatigue intervention study. Therefore the retest period was 10 weeks, which is, not a typical
time frame employed when evaluating retest reliability. Other studies show that the CFQ has
good retest reliability38, however within individuals with MS this needs further evaluation.
Finally, it is possible that some of the CFQ items overlap with muscle weakness and perception
of cognitive dysfunction such as problems with memory.
In conclusion, the CFQ appears to be a valid and internally reliable measure of fatigue severity in
people with MS which is sensitive to change. We discourage the separation of physical and
mental fatigue by means of two factor scores; rather suggest that a total sum score provides an
appropriate and internally reliable measure of general MS-fatigue symptoms. Although the CFQ
was associated with measures of impact of fatigue the size of these correlations were small to
moderate suggesting that when measuring fatigue in MS including measures of both fatigue
severity and impact are warranted. Future studies should also explore the relationships between
the CFQ (measuring severity of fatigue), and measures of performance fatigability including
central factors relating to cognitive networks and peripheral factors such as loss of muscle force.
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Table 1: Baseline demographic and illness characteristics of MS participants across 4 studies
related to the treatment of fatigue.
Skerrett et al.,
Van Kessel et
Witt, 2005
Moss-Morris
2006
al., 2008
(unpublished)
et al., 2012
149
72
183
40
Cross-sectional
RCT
Cross-sectional
RCT
44.5 (11.8)
45 (9.6)
50.2 (12.9)
40.9 (15.1)
9.5 (7.4)
6 (5.4)
10.6 (9.5)
8.3 (7.0)
127 (85.2%)
54 (75%)
145 (79.2%)
32 (80%)
149 (100%)
41 (57%)
101 (55.2%)
22 (55%)
Secondary progressive
-
22 (30.6%)
37 (20.2%)
9 (22.5%)
Primary progressive
-
9 (12.5%)
44 (24%)
2 (5%)
Single/divorced/separated
42 (28.2%)
10 (13.9%)
44 (24%)
16 (40%)
Living with Partner/married
102 (68.5%)
62 (86.1%)
126 (68.9%)
22 (55%)
Working less
43 (28.9%)
26 (36.1%)
45 (24.6%)
3 (7.5%)
Unemployed
52 (35%)
22 (30.6%)
63 (34.4%)
11 (27.5%)
n
Design
Age (mean, SD)
Years with MS (mean, SD)
Gender (female, n %)
MS type
Relapsing remitting
Marital status
Employment related to MS
RCT: randomised control trial
16
Table 2: 9-item bi-factor model for the Chalder Fatigue Questionnaire (model 4b, see technical
appendix)
Residual
Factor
Original Item Description
General Physical
Mental
Variance
Item No
1
Tiredness
.77**
.58**
.07
2
Need to rest more
.79**
.51**
.11
3
Sleepy/drowsy
.80**
.39**
.22
4
Problems starting things
.84**
.19**
.26
5
Lack energy
.83**
.42**
.13
8
Difficulty concentrating
.93**
.14
.11
9
Slips of the tongue when speaking
.84**
.43**
.11
10
Difficulty finding correct word
.84**
.50**
.05
11
Memory
.88**
.17**
.19
Standardised estimates shown; **p<.01
17
Table 3: Correlates of the Chalder Fatigue Questionnaire (CFQ) general factor scores
Total CFQ sum score
Shortened CFQ sum score
Original 11-items
9-items
HADS-depression
.40**
.40**
HADS -anxiety
.37**
.38**
WSAS – impact of fatigue
.36**
.34**
.24
.22
n=444
MFISa
HADS: Hospital Anxiety and Depression Scale
WSAS: Work and Social Adjustment Scale
MFIS: The Modified Fatigue Impact Scale
a
n=39
**p<.01 *p<.05
18
Technical appendix
Appendix table 1: Summary of model fit
Model
Description
1
2a
2b
3
4a
4b
5
1-factor
2-factor
2-factor with residual correlation
3-factor
Bi-factor with 2 group factors
Modified Bi-factor with 2 group factorsa
Bi-factor with 3 group factors
No of free
parameters
44
45
46
47
55
45
54
Chi-square (df)
CFI
TLI
RMSEA
692.5 (44) p<.01
395.4 (43) p<.01
208.0 (42) p<.01
222.1 (41) p<.01
145.3 (33) p<.01
33.4 (18) p=.01
89.9 (34) p<.01
.98
.99
.99
.99
.99
.99
.99
.97
.99
.99
.99
.99
.99
.99
.18
.14
.10
.10
.09
.04
.06
a
items 6 and 7 removed (weakness); root mean squared error of approximation (RMSEA) confirmatory fit index
(CFI); Tucker-Lewis index (TLI)
Model 1: A one factor model with all 11-items loaded onto a single fatigue factor had poor model fit as
indicated by a RMSEA>.08.
Model 2: A two-factor model, specifying correlated physical (items 1-7) and mental fatigue factors (items
8-11) also had poor fit.
Model 2b: Inspection of Mplus derived modification indices suggested improved model fit for model 2 if
a residual correlation was added between items 6 (less strength in muscles) and 7 (feeling weak). Since
these items appear to measure “weakness” it was deemed reasonable to add this residual correlation to the
model. This two-factor model (model 2b), including the residual correlation, had improved fit, although
this was still marginal as evidence by a RMSEA of .10. The correlation between the physical and mental
fatigue factors was high, r=.80 (p<.01), suggesting a considerable amount of shared variance.
Model 3: Given the high correlation between mental and physical fatigue, a 3-factor model was also
examined, which included a weakness factor (model 3) and was shown to have unsatisfactory fit given the
RMSEA.
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Model 4: Given the high correlation between the physical and mental fatigue factors, a bi-factor model
was tested. In the bi-factor model, all 11 items were loaded onto a general fatigue factor. In addition items
1-7 were loaded onto a physical fatigue factor and items 8-11 on a mental fatigue factor. Correlations
between each of these three factors were fixed to be zero. The bi-factor model yielded a lower RMSEA
(.09) and a substantial reduction in the chi-square statistic, albeit this was still significant (p<.01). Since
items 6 and 7 appear to tap into the concept of weakness, two alternative bi-factor models were tested,
one in which these two items were dropped (model 4b) and another with them loaded onto a third group
factor (“weakness”; model 5). The 9-item bi-factor model (model 4b) demonstrated the best fit in terms
of the lowest Chi-square and RMSEA values (RMSEA=.04) and CFI and TFI values of .99.
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