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PHYSIOTHERAPY THEORY AND PRACTICE

2017, VOL. 33, NO. 8, 653–660


https://doi.org/10.1080/09593985.2017.1331481

DESCRIPTIVE REPORT

Kinesiophobia and maladaptive coping strategies prevent improvements in pain


catastrophizing following pain neuroscience education in fibromyalgia/chronic
fatigue syndrome: An explorative study
Anneleen Malfliet, PT, MSc a,b,c, Jessica Van Oosterwijck, PT, PhD a,b,d, Mira Meeus, PT, PhDa,d,e,
Barbara Cagnie, PT, PhDd, Lieven Danneels, PT, PhDd, Mieke Dolphens, PT, PhDd, Ronald Buyl, PT, PhDf,
and Jo Nijs, PT, PhDa,b,c
a
Pain in Motion Research Group, University of Antwerp, Antwerp, Belgium; bDepartment of Physiotherapy, Human Physiology and Anatomy,
Faculty of Physical Education & Physiotherapy, Vrije Universiteit Brussel, Brussels, Belgium; cDepartment of Physical Medicine and
Physiotherapy, University Hospital Brussels, Brussels, Belgium; dDepartment of Rehabilitation Sciences and Physiotherapy, Faculty of
Medicine and Health Sciences, Ghent University, Ghent, Belgium; eDepartment of Rehabilitation Sciences and Physiotherapy, Faculty of
Medicine and Health Sciences, University of Antwerp, Antwerp, Belgium; fDepartment of Biostatistics and Medical Informatics, Vrije
Universiteit Brussel, Brussels, Belgium

ABSTRACT ARTICLE HISTORY


Many patients with chronic fatigue syndrome(CFS) and/or fibromyalgia(FM) have little understand- Received 8 February 2016
ing of their condition, leading to maladaptive pain cognitions and coping strategies. These should Revised 28 June 2016
be tackled during therapy, for instance by pain neurophysiology education (PNE). Although positive Accepted 31 July 2016
effects of PNE are well-established, it remains unclear why some patients benefit more than others. KEYWORDS
This paper aims at exploring characteristics of patients responding poor to PNE to further improve Chronic fatigue syndrome;
its effectiveness. Data from two RCT’s were pooled to search for baseline predictors. Subjects (n = fibromyalgia; pain
39) suffering from CFS/FM, as defined by the American College of Rheumatology, underwent PNE neuroscience education
treatment. The Pain Catastrophizing Scale (PCS); Pain Coping Inventory (PCI); and Tampa Scale of
Kinesiophobia (TSK) were defined as outcome measures. There was a significant negative relation-
ship between baseline TSK and the change in both PCS total score (r = −0.584; p < 0.001) and PCS
rumination (r = −0.346; p < 0.05). There was a significant negative relationship between the change
in PCS total score and baseline PCI worrying (r = −0.795; p < 0.001) and retreating (r = −0.356;
p < 0.05). FM/CFS patients who tend to worry allot about their pain and with high levels of
kinesiophobia are likely to experience less reductions in catastrophizing following PNE. It seems
that PNE alone is insufficient to reduce catastrophic thinking regarding pain, and supplementary
treatment is needed.

Introduction Additionally, the lack of understanding the nature of their


widespread pain may lead to the development of negative
Chronic fatigue syndrome (CFS) and fibromyalgia (FM)
and maladaptive thoughts, emotions and cognitions, as
are two conditions that share key symptoms like wide-
well as behaviors like catastrophizing, hypervigilance,
spread pain and fatigue and that often occur together
avoidance behavior and somatization (Meeus and Nijs,
(Aaron, Burke, and Buchwald, 2000; Buchwald and
2007), further influencing their pain perception.
Garrity, 1994; Martinez-Martinez et al., 2014). Many
Therefore, it is necessary to tackle these cognitions during
patients diagnosed with one or both conditions suffer
therapy, for instance by providing education (Moseley,
from widespread pain without knowing its underlying
2004). Most of the more “traditional” educational (biome-
cause or mechanisms. Patients who have little or faulty
dical) models that are mainly focusing more on basic
understanding of pain mechanisms seem to define pain
anatomy, biomechanics, pathophysiology, etc. seem to
as more threatening or dangerous due to damage or injury
have limited efficacy in decreasing pain and disability in
(Jackson et al., 2005; Meeus et al., 2010; Van Oosterwijck
chronic pain patients (Cherkin et al., 1996; Parker et al.,
et al., 2011). This perception results in lower pain tolerance,
1984; Roland and Dixon, 1989; Sirles, Brown, and Hilyer,
more catastrophic thoughts regarding pain and the use of
1991). Meanwhile, a systematic review by Louw, Diener,
less adaptive pain coping strategies (Jackson et al., 2005).
Butler, and Puentedura (2011) provided compelling

CONTACT Jo Nijs, PT, PhD jo.nijs@vub.be Department of Physiotherapy, Human Physiology and Anatomy (KIMA), Faculty of Physical Education &
Physiotherapy, Vrije Universiteit Brussel, Medical Campus Jette, Building F-Kine, Laarbeeklaan 103, BE-1090 Brussels, Belgium.
© 2017 Taylor & Francis
654 A. MALFLIET ET AL.

evidence for the positive effects of pain neuroscience edu- Methods


cation (PNE) in reducing pain complaints, disability, cata-
Setting and patients
strophizing and improving physical performance in
chronic musculoskeletal disorders. PNE aims to educate The data used in this analysis were obtained by pooling
patients about the neurophysiology, neurobiology, proces- the data of two preexisting datasets by Van Oosterwijck
sing and representation of pain. In addition to the review et al. (2013) and Meeus et al. (2010), investigating FM and
by Louw, Diener, Butler, and Puentedura (2011), more CFS patients respectively. These datasets were pooled
recent trials and case reports support the effectiveness of because of the similarities in both protocols. Moreover,
PNE in different chronic pain populations (Anandkumar, selection criteria of both CFS and FM were similar in the
2015; Anandkumar and Manivasagam, 2014; Louw, 2014; two pooled studies, with all included patients suffering
Louw, Diener, Landers, and Puentedura, 2014; Louw, from chronic nonspecific widespread pain, as defined by
Puentedura, and Mintken, 2012; van Ittersum, van the American College of Rheumatology (Wolfe et al.,
Wilgen, Groothoff, and van der Schans, 2011; Van 1990). Nonspecific was defined as not resulting from an
Oosterwijck et al., 2013; Zimney, Louw, and Puentedura, established medical illness like rheumatoid arthritis nor
2014). These studies provide additional evidence that PNE any other medical condition. The widespread pain experi-
can also reduce the impact of pain and illness in daily life, enced by these patients is likely to result from central
and kinesiophobia (fear of movement); as well as improve sensitization (Meeus and Nijs, 2007; Nijs et al., 2012;
illness perceptions, anxiety, and health status. Staud, Robinson, and Price, 2007).
Although the effects of PNE in the chronic pain The initial analysis of both trials focused on the
population are well-established, it remains unclear effectiveness of PNE and are reported elsewhere
why some patients benefit more than others. (Meeus et al., 2010; Van Oosterwijck et al., 2013).
Exploring baseline (pretreatment) characteristics of Both original study protocols were approved by the
patients that respond poorly to PNE will be important ethical committee of the University Hospital Brussels/
to further improve this educational strategy and hence Vrije Universiteit Brussel.
to extend its effectiveness to a larger number of patients
with chronic pain. However, studies examining whether
baseline characteristics predict the outcome following Outcome measures
PNE in chronic pain patients are currently lacking.
A battery of self-reported questionnaires regarding the
For the reasons outlined above, the present study
presence of maladaptive cognitions and coping strate-
aimed at exploring baseline factors predicting treat-
gies of pain was defined as the outcome measures.
ment outcome following PNE in patients with FM/
CFS. Therefore, data from two previously published
randomized controlled clinical trials (RCT’s) (Meeus Primary outcome measure
et al., 2010; Van Oosterwijck et al., 2013) were pooled The Pain Catastrophizing Scale (PCS) assesses the pre-
to search for such baseline predictors. Based on our sence and degree of pain catastrophizing. Pain catastro-
current knowledge regarding the effectiveness of PNE phizing was described by Sullivan and Bishop (1995) as
and the study of Tan, Teo, Anderson, and Jensen “an exaggerated negative mental set brought to bear
(2011), it was hypothesized that the presence of mala- during actual or anticipated painful experience.”
daptive pain cognitions and coping strategies may Persons who tend to catastrophize, experience pain as
predict the treatment outcome following PNE. More extremely threatening (Vlaeyen and Crombez, 1999).
specially, given the nature (Butler and Moseley, 2003) Pain catastrophizing may includes three dimensions,
and rationale (Nijs et al., 2011) of PNE, including being pain rumination, pain magnification and feeling
long-term reconceptualization of pain, alterations in helplessness (Sullivan and Bishop, 1995). Pain catastro-
illness beliefs and establishing adaptive pain cogni- phizing was chosen as primary outcome based on a
tions, it is hypothesized that patients with more severe theoretic approach, as previous research showed the
maladaptive pain cognitions and coping strategies pre- importance of pain catastrophizing (measured by the
treatment will have a poorer outcome after PNE, as it PCS) in both objectifying treatment effect and predict-
will be more challenging to address these maladaptive ing long-term pain and functioning (Meeus et al., 2012;
pain cognitions and coping strategies in such patients. Picavet, Vlaeyen, and Schouten, 2002; Severeijns,
Therefore, this paper investigates whether high kine- Vlaeyen, van den Hout, and Weber, 2001; Spinhoven
siophobia, high catastrophizing and/or maladaptive et al., 2004; Sullivan et al., 1998; Sullivan et al., 2001).
coping strategies at baseline are related to poorer The PCS consists of 13 items describing different
treatment outcome following PNE. thoughts and feelings that individuals may experience
PHYSIOTHERAPY THEORY AND PRACTICE 655

when they are facing pain and provides a general score behaviour that can be described as an unusual, irra-
and three subscale scores (helplessness, magnification, tional and debilitating fear for physical movement and
rumination). The psychometric properties of the Dutch activity (Kori, Miller, and Todd, 1990). The TSK-Dutch
version of the PCS, as used here, are well-established. A version used in this study has been shown to have good
Cronbach’s alpha ranging between 0.85 and 0.91 repre- reliability (alpha ranging from 0.70 to 0.83) and mod-
sents a high reliability, and good validity is reflected in erate validity (significant Pearson correlations of TSK
the high correlation (r = 0.73; p < 0.001) of the PCS with PCS, Beck Depression Inventory and Fear Survey
with the subscale “catastrophizing” of the Pain Schedule; r(s) =0.33 to 0.59, p < 0.01) (Crombez,
Cognition List (Crombez, Vlaeyen, Heuts, and Lysens, Vlaeyen, Heuts, and Lysens, 1999; Goubert et al.,
1999; Spinhoven et al., 2004; Van Damme et al., 2000). 2004; Nederhand et al., 2004; Swinkels-Meewisse
et al., 2003; Vlaeyen, Kole-Snijders, Boeren, and van
Secondary outcome measures Eek, 1995).
The Pain Coping Inventory (PCI) contains 33 items Van Oosterwijck et al. (2013) obtained these data at
assessing pain coping strategies. Pain coping strategies two moments post-therapy (1 week and 3 months fol-
include cognitive and behavioral responses to pain and lowing completion of the PNE therapy). For the current
can influence pain, functional capacity and psychologi- analysis, the data obtained at 1 week post-therapy was
cal functioning. These reactions on pain are mostly used, in order to fit well with the data used in the study
grouped under the term “pain coping” and are defined by Meeus et al. (2010), which was obtained immediately
as attempts to tolerate and handle pain (Brown and following the completion of PNE.
Nicassio, 1987; Jensen, Turner, Romano, and Karoly,
1991). Pain coping strategies can be classified as active
Intervention
or passive (Kraaimaat and Evers, 2003). Active strate-
gies are used to lighten or control the pain, or to The PNE session comprised a one-on-one educational
function despite the pain. When using passive pain session about the neurophysiology of pain. This included
coping strategies, patients show negative thoughts education regarding the physiology of the nervous system
about pain, avoidance of interference and social with- in general and of the pain system in particular. PNE
drawal (Kraaimaat, Bakker, and Evers, 1997). focused on informing the patient about the difference
Furthermore, these coping strategies can have an adap- between “nociception” and “pain” and taught patients
tive or maladaptive influence on pain, physical func- that the central nervous system has the ability to increase
tioning and psychological functioning, depending on or decrease its sensitivity (neuroplasticity) (Nijs et al.,
their immediate or long-term consequences 2011). Attention was given to the fact that the human
(Kraaimaat, Bakker, and Evers, 1997). body can both amplify and mute pain (e.g., by either
The PCI assesses six specific pain-coping strategies focusing on pain or rather shifting attention from the
that represent two higher-order pain coping dimen- pain). This approach aims to help patients cope with
sions: 1) maladaptive (i.e., worrying, retreating, and persistent pain (Louw, Diener, Butler, and Peuntedura,
resting) and adaptive (i.e., transformation, distraction, 2011). The content of the education sessions was based on
and reducing demands); and 2) coping (Kraaimaat, the book “Explain Pain” by Butler and Moseley (2003).
Bakker, and Evers, 1997). Patients were asked to rate Further details regarding this intervention are presented
the frequency by which these strategies were used on a elsewhere (Meeus et al., 2010; Nijs et al., 2011; Van
4-point Likert scale ranging from 1 (hardly ever) to 4 Oosterwijck et al., 2011; Van Oosterwijck et al., 2013).
(very often). Results of the different subscales were In the study by Meeus et al. (2010), the intervention
obtained by taking the mean score of the items belong- was limited to one individual session of 30 minutes. In
ing to that subscale. A higher score indicated a more the study by Van Oosterwijck et al. (2013), the inter-
frequent application of that specific coping strategy. vention was spread out over 7 days and comprised one
The Dutch version of the PCI has been found to have individual session of 30 minutes, written information at
sufficient reliability (Crohnbach’s alpha ranging home, and one individual session conducted by tele-
between 0.42 and .060) (Kraaimaat, Bakker, and Evers, phone. During the latter study, patients were motivated
1997; Kraaimaat and Evers, 2003). to apply their new insights into their daily life. The
The Tampa Scale of Kinesiophobia (TSK) consists of telephone session was used to elucidate any remaining
17 items to evaluate the presence of kinesiophobia questions and misunderstandings which existed follow-
(Vlaeyen, Kole-Snijders, Boeren, and van Eek, 1995). ing the oral and written education. In both studies, the
Kinesiophobia is defined as the fear of movement and/ educational sessions were led by individuals with a
or (re-)injury and is a specific type of fear or avoidance Bachelor’s degree in Physiotherapy.
656 A. MALFLIET ET AL.

Statistical analysis demonstrate less reduction in pain catastrophizing (PCS


total) and ruminating about pain (PCS rumination) in
All data were analyzed using IBM SPSS Statistics 22.0 for
response to PNE. Second, the use of negative, maladaptive
Windows (IBM Corporation, Somers, NY). Normality of
coping strategies such as avoiding negative environmental
the variables was tested with the Kolmogorov–Smirnov
stimuli (PCI retreating) and more worrying about pain
test. The original pooled data consisted of a control group
(PCI worrying), appears to be related to less reduction in
and an experimental group. As this study aims at exploring
catastrophizing following PNE. Last, these results seem to
the baseline predictors of PNE treatment, only the experi-
indicate that the older the patients with FM and CFS, the
mental (PNE) group was used in this analysis. Correlations
more they experience reductions in magnification of the
were performed between the change in the primary out-
obnoxious character of pain (PCS magnification) by PNE
come factor (PCS) and the baseline measurements of all
treatment. In summary, it appears that FM/CFS patients
other factors, being age, sex, PCI and TSK. Pearson’s cor-
who display higher levels of kinesiophobia and worrying
relation analysis was used for data with normal distribu-
about pain experience less reduction in catastrophizing and
tion, Spearman’s correlation analysis for the data that were
ruminating following PNE. The present results seem to
not normally distributed.
imply that in patients with CFS/FM with high levels of
kinesiophobia and worrying, PNE on its own seems insuf-
Results ficient to reduce pain catastrophizing compared to those
with lower levels of kinesiophobia and worrying.
This study included 39 patients with CFS or FM, with an Supplementary therapy may be necessary to address these
age ranging from 18 to 65. Detailed information on the elements (e.g., graded exposure/activity techniques, cogni-
included subjects can be found in Table 1. The study by tion-targeted exercises, commitment and acceptance ther-
Van Oosterwijck et al. (2013) made use of the intention-to- apy, or other cognitive behavioral techniques). However, as
treat principle, using the last-observation-carried-forward this comprises an explorative study, it needs to be stressed
approach for two subjects. The study by Meeus et al. (2010) that these results are preliminary and that no harsh con-
did not use this principle, which resulted in losing two clusions should be drawn.
subjects to follow-up. Currently, studies regarding pain cognitions and coping
There was a significant negative relationship between strategies as possible predicting factors for PNE therapy in
baseline TSK and the change in both PCS total score (r = FM and CFS are essentially lacking, being an argument for
−0.584; p < 0.001) and PCS rumination (r = −0.346; p < the necessity of this explorative study. Nevertheless, studies
0.05). There was also a significant negative relationship investigating other predictors in prospective studies are
between the change in PCS total score and baseline PCI fairly well presented. Recently, de Rooij et al. (2013) pub-
worrying (r = −0.795; p < 0.001) and retreating (r = −0.356; lished a review on predictors of treatment response to
p < 0.05). Last, age was related to the change in PCS multidisciplinary therapy in FM. Many factors were deter-
magnification (r = 0.395; p < 0.05). All correlations can be mined to predict poorer outcome, like pain, depression,
found in Table 2, inclusive of their significance levels and negative demographic and clinical factors, maladaptive
confidence intervals. cognitive and emotional factors and physical functioning.
Additionally, Huibers et al. (2004) determined older age,
exhaustion, female sex, low education level, number of
Discussion visits to the general practitioner, low self-perceived activity
This exploratory study aimed at searching possible predict- level, higher anxious level and bad self-rated health as
ing baseline factors of treatment outcome when using PNE general predictors of poorer outcome. Ray, Jefferies, and
in FM/CFS patients. Based on the findings of this additional Weir (1997) added longer illness duration, subjective cog-
analysis of the pooled data from two previously published nitive difficulty and somatic symptoms to this list. On the
RCT’s regarding PNE in patients with FM/CFS, several other hand, they found no predictive influence of anxiety,
preliminary assumptions can be made. First, it seems likely depression or general emotional distress (Ray, Jefferies, and
that patients with higher levels of kinesiophobia Weir, 1997). Other studies found prediction of poor

Table 1. Subjects’ characteristics.


Characteristics Meeus et al. (2010) Van Oosterwijck et al. (2013) Pooled data
Subjects n = 24 n = 15 n = 39
Sex 2 men; 22 women 3 men; 12 women 5 men; 34 women
Age (mean ± SD) 38.3 ± 10.6 45.8 ± 9.5 41.2 ± 10.7
PHYSIOTHERAPY THEORY AND PRACTICE 657

Table 2. Predictors of treatment outcome following PNE.


Outcome measurement Baseline measurement Correlation coefficient Confidence interval
ΔPCS Total Age 0.208 [−.119; .513]
Sex −0.010 [11.36; 16.59]
TSK −0.584*** [−.776; −.323]
PCI Transformation −0.249 [−.527; .080]
PCI Distraction 0.035 [−.285; .397]
PCI Reducing demands −0.119 [−.39; .226]
PCI Retreating −0.356* [−.620; −.055]
PCI Worrying −0.795 *** [−.913; −.607]
PCI Resting −0.167 [−.490; .204]
ΔPCS Helplessness Age 0.261 [−.070; .541]
Sex −0.010 [2.14; 4.16]
TSK 0.026 [−.313; .313]
PCI Transformation −0.038 [*.337; .286]
PCI Distraction −0.250 [−.470; −.011]
PCI Reducing demands −0.245 [−.510; .094]
PCI Retreating −0.297 [−.568; .025]
PCI Worrying −0.066 [−.372; .267]
PCI Resting −0.153 [−.463; .162]
ΔPCS Magnification Age 0.395* [.040; .679]
Sex −0.302 [.970; 2.11]
TSK −0.107 [−.503; .238]
PCI Transformation −0.242 [−.550; .059]
PCI Distraction 0.098 [−.227; .429]
PCI Reducing demands −0.289 [−.523; .047]
PCI Retreating −0.133 [−.426; .170]
PCI Worrying −0.213 [−.515; .104]
PCI Resting 0.114 [−.230; .398]
ΔPCS Rumination Age 0.235 [−.094; .545]
Sex 0.003 [1.51; 3.00]
TSK −0.346* [−.649; .019]
PCI Transformation 0.090 [−.256; .442]
PCI Distraction −0.039 [−.330; .260]
PCI Reducing demands −0.054 [−.378; .269]
PCI Retreating −0.279 [−.615; .074]
PCI Worrying −0.231 [−.541; .111]
PCI Resting −0.092 [−.412; .249]
TSK = Tampa Scale for Kinesiophobia; PCS = Pain Catastrophizing Scale; PCI = Pain Coping Inventory;
ΔPCS = difference in PCS between post-treatment and baseline measurement.
* p < 0.05; ** p < 0.01; *** p < 0.001.

outcome in CFS by being member of a self-help group, more appropriate study protocols, are warranted to
being in receipt of sickness benefit, passive activity pattern confirm this assumption.
and higher level of depression (Bentall, Powell, Nye, and The current study findings are important as they
Edwards, 2002; Prins et al., 2002). provide the initial step in further exploring predictive
Although there are some general predictors investi- factors of treatment outcome after PNE and offer
gated in the studies presented above, there has been no important directions for further improving PNE for
attention the role pain cognitions and coping strategies patients with FM/CFS. However, the results should
in this area of research, while this might be pertinent be interpreted in the light of some study limitations.
for optimizing treatment outcomes after PNE, given the These include the limited sample size, despite the
established importance of pain cognitions and coping pooling of data from two trials, and the limited num-
strategies in patients with CFS/FM (Garcia-Campayo ber of identical outcome measures used in the original
et al., 2009; Giesecke et al., 2003; Nielson and Jensen, RCT’s. The limited sample size made it impossible to
2004). The present study tries to address this short- use a regression analysis. This could have been
coming in a first explorative way, and it would be addressed by implementing the study by van
interesting to have a further look on these and other Ittersum, van Wilgen, Groothoff and van der Schans
factors in subsequent studies, especially given the (2011), if this study had not been conceptually differ-
important suggestive findings reported here. Based on ent by using only written education. As we wanted to
our findings, it seems plausible that pain cognitions and focus on PNE delivered by a therapist, this data could
coping strategies have a role in treatment outcome (e.g., not be used. Additionally, as described above, there are
catastrophizing) following PNE in patients with FM/ many predictors identified for treatment types other
CFS, and further studies using larger numbers of sub- than PNE. It can be listed as a limitation of this study
jects, using more and different measurements and using that we did not included these predictors here, but, as
658 A. MALFLIET ET AL.

this paper, was intended purely as explorative the data ORCID


were limited to this extend. It is also necessary to point Anneleen Malfliet http://orcid.org/0000-0003-0598-7038
out that the pain coping strategy worrying (PCI wor- Jessica Van Oosterwijck http://orcid.org/0000-0002-8946-
rying) and the pain catastrophizing dimension rumi- 4383
nation (PCS rumination) are based on an overlapping
concept, which might explain the observed association.
Besides this, also the differences between the two
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