discussion #13 disease
Maintaining Lasting Improvements: One-Year Follow-Up of Children With Severe Chronic Pain Undergoing Multimodal Inpatient Treatment
Gerrit Hirschfeld,1 PHD, Tanja Hechler,1 PHD, Michael Dobe,1 PHD, Julia Wager,1 MSC,
Pia von Lützau,1 MSCN, Markus Blankenburg,1,2 MD, Joachim Kosfelder,3 PHD, and
Boris Zernikow,1 MD, PHD 1German Peadiatric Pain Centre and Department of Children’s Pain Therapy and Paediatric Palliative Care,
Children’s and Adolescents’ Hospital, Datteln, Witten/Herdecke University, Faculty of Health, School of
Medicine, Germany, 2Center for Child Neurology, Clinic for Children and Adolescent Hospital, Witten/
Herdecke University, and 3Department of Social Sciences and Cultural Studies, University of Applied Sciences,
Düsseldorf, Germany
All correspondence concerning this article should be addressed to Tanja Hechler, PHD, German Paediatric
Pain Centre and Department of Children’s Pain Therapy and Paediatric Palliative Care, Children’s and
Adolescents’ Hospital, Datteln, Witten/Herdecke University, Dr-Friedrich-Steiner-Str. 5, Datteln 45711,
Germany. E-mail: T.Hechler@kinderklinik-datteln.de
Received April 4, 2012; revisions received October 7, 2012; accepted October 13, 2012
Objective To investigate the long-term effectiveness of a 3-week multimodal inpatient program for children
and adolescents with chronic pain. Methods 167 adolescents were evaluated at pretreatment baseline, 3-,
and 12-month follow-up. Long-term effectiveness was investigated for pain-related variables (pain-related
disability, school absence, pain intensity) and emotional distress. Results We found statistically and
clinically significant changes in all variables. After 1 year, the majority (56%) showed overall improvement as
indexed by decreased pain-related disability or school absence. 22% had an unsuccessful treatment
outcome. Those showing only short-term improvements had higher levels of emotional distress at baseline.
Conclusions 1 year after completing a multimodal inpatient program adolescents report less chronic pain,
disability, and emotional distress. Clinically significant changes remain stable. Adolescents with high levels of
emotional distress at admission may require special attention to maintain positive treatment outcomes.
Specialized inpatient therapy is effective for children with chronic pain.
Key words adolescents; clinical significance; headache; inpatient pain treatment; somatoform pain disorder.
Introduction
Chronic pain is a pervasive problem for the health care
system that may begin in childhood and adolescence
with �5% suffering from severe and disabling chronic
pain (Huguet & Miro, 2008).
Intensive multimodal pain treatment is considered as
an adequate treatment options for these severely disabled
children because then professionals from several discip-
lines can see the child daily in various situations, and a
systematic change in pain-related disability and distress
can be achieved. There are only few inpatient programs
or intensive day care programs to date, and their concep-
tualization and labeling is diverse (Eccleston, Bruce, &
Carter, 2006; Hechler, Dobe, & Zernikow, 2010b; Logan
et al., 2012a; Logan, Conroy, Sieberg, & Simons, 2012b).
Here, we follow the definition of a multimodal inpatient
treatment (MIT) set out by the German Pain Society
(Arnold et al., 2009) defined as a treatment provided
by a minimum of three health care disciplines including
medical, psychological, and physical therapy, which
Journal of Pediatric Psychology pp. 1–13, 2012 doi:10.1093/jpepsy/jss115
Journal of Pediatric Psychology � The Author 2012. Published by Oxford University Press on behalf of the Society of Pediatric Psychology. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com
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are provided concurrently, generally lasting for 3 weeks.
This treatment addresses underlying pain mechanisms,
adapts pain medication, treats specific symptoms such as
emotional distress, and teaches active coping skills
(Hechler et al., 2010c; Maynard, Amari, Wieczorek,
Christensen, & Slifer, 2009a).
Effectiveness of MIT has rarely been investigated and if
so, comparison is hampered by the diversity of the pro-
grams. The few studies are diverse in terms of setting (some
programs provide inpatient and outpatient treatment
[Chalkiadis, 2001]), length of stay (ranging from 3-8 days
to 3 weeks), and interventions provided (exercise therapy
[Sherry, Wallace, Kelley, Kidder, & Sapp, 1999] compared
with multimodal interventions (Eccleston, Malleson,
Clinch, Connell, & Sourbut, 2003). The program
described by Maynard and colleagues can be classified as
MIT. There are, however, other intensive multidisciplinary
rehabilitation programs, which can not be classified as MIT
as defined earlier but which share similarities in thera-
peutic approaches and program philosophy (e.g. [Logan
et al., 2012a]). In addition, long-term follow-up has
rarely been investigated. However, extended follow-ups
are necessary to investigate the stability of treatment effects
and factors that influence the stability of treatment effects
(Maynard et al., 2009a). The only study that investigated
long-term effects has been conducted by Sherry and col-
leagues (1999). This study followed 96 children for up to 7
years after a short exercise therapy and found high stability
of treatment results, i.e. after 2 years, 88% of the children
were still symptom free. However, their program was an
exercise-based program, focused on children with complex
regional pain syndrome (CRPS), and suffered from a large
amount of dropout (50% after 2 years).
It is yet unclear which factors influence long-term
treatment outcome. Given that there is numerous empirical
support for the role of emotional variables on the mainten-
ance and exacerbation of pediatric chronic pain and dis-
ability (e.g., [Cohen, Vowles, & Eccleston, 2010; Kashikar-
Zuck, Goldschneider, Powers, Vaught, & Hershey, 2001;
Sieberg, Williams, & Simons, 2011; Vervoort et al., 2011]),
emotional distress may be an important factor. Vervoort
et al. (2011) demonstrated recently that pain catastro-
phizing in schoolchildren was related to pain and disability
6 months later. They also found that children with
increased anxiety at baseline were more inclined to
report catastrophizing at follow-up. Whether this pattern
holds true for children with chronic pain has not yet been
investigated.
In addition, very few of the previous treatment studies
analyzed the clinical significance (Jacobson & Truax, 1991)
of treatment results as recommended by the Initiative for
Methods, Measurement, and Pain Assessment in Clinical
Trials (IMMPACT) (Dworkin et al., 2009). Jacobson and
Truax (1991) proposed a method for defining clinically
significant change and suggest two criteria: (1) the magni-
tude of change between pre- and post-treatment scores
should be statistically reliable. (2) By the end of the ther-
apy, patients should move from a dysfunctional to a func-
tional level to render them indistinguishable from healthy
people. Thus far, only one study reported on the clinical
significance of their treatment results 3 months after inpa-
tient treatment (Hechler et al., 2009). They found that 3
months after treatment, 72% of the patients had clinically
significant changes in pain intensity, and 45% had clinic-
ally significant changes in pain-related disability.
The present article aims to add to our limited know-
ledge on the long-term effects of MIT by investigating
long-term treatment outcome 12 months after treatment
compared with the pre-treatment status at admission.
There were four objectives of the study: First, to assess
12 month outcomes of three primary outcomes (pain in-
tensity, pain-related disability, school absences), with a
focus on clinically significant changes. We expected signifi-
cant and clinically relevant decreases in primary outcomes
from admission to 12-month follow-up. Second, to classify
children into two groups based on the composite measure
of ‘‘overall improvement.’’ Third, to predict clinically rele-
vant levels of overall improvement. We hypothesized that
patient- and pain characteristics at admission would pre-
dict overall improvement at 12-month follow-up. Fourth,
to assess the stability of change over time. Therefore, we
defined the following groups based on overall improvement
status at 3- and 12-month follow-up: (1) ‘‘stable long-term
improvers’’ (improved on both assessments), (2)
‘‘short-term improvers’’ (improved at 3-month follow-up
only), (3) ‘‘long-term improvers’’ (improved at 12-month
follow-up only), and (4) ‘‘children with unsuccessful treat-
ment results’’ (not improved at both follow-ups). We also
aimed to investigate the influence of pain characteristics
and emotional distress at admission on stability of change.
Materials and Methods Participants
In the present study, we report data from extended
12-months follow-up of our earlier study (Hechler et al.,
2009). Data from this sample have been described previ-
ously (Dobe, Damschen, Reiffer-Wiesel, Sauer, & Zerni-
kow, 2006; Dobe, Hechler, Behlert, Kosfelder, &
Zernikow, 2011; Hechler et al., 2009; Hechler, Blanken-
burg, Dobe, Kosfelder, Hübner, & Zernikow, 2010a;
Hechler et al., 2010c), but this is the first time that the
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clinical significant long-term outcomes are described.
The study was a naturalistic longitudinal effectiveness
study, with assessment at admission, 3 and 12 months
after treatment. Between January 2004 and December
2006, adolescents that completed the 3-week inpatient
chronic-pain treatment program at the German Paediatric
Pain Centre were included. All children and their guardians
were informed and agreed to their data being anonymously
stored and processed for research purposes. The study was
approved by the Ethics Committee of the Witten/Herdecke
University.
During the study, 200 children and adolescents
completed the program. Thirty-three were excluded from
this analysis because they were aged <11 years. At admis-
sion, the 167 adolescents included in the final analysis
were between the ages 11 and 18.3 years, mostly female
(62%), and the majority (50%) had recurrent headache (see
Table I for complete demographic information, and Table II
for information about the level of functioning). In all, 141
(84%) provided data at the 3 months follow-up, and 136
(81%) provided data at the 12 months follow-up (see
Figure 1). Participants completed the 12 month follow-up
data on average 394 days after therapy.
Comparing those who dropped out of the study at
12-month follow-up to those who remained in the study,
we found that dropouts were significantly older than
completers (14 vs. 15 years; p < .01) and had higher
levels of school aversion compared to dropouts (51 vs.
58; p < .01). There were no differences in pain character-
istics at admission (all p > .1). At the 12-month follow-up,
data were collected via mail for n¼105 patients (63%), via
telephone for n¼13 patients (8%), and during a scheduled
appointment at the German Paediatric Pain Centre for
n¼18 (11%) patients. Chi-square tests found no differ-
ences in sample characteristics (age, sex, diagnosis) and
the way of responding (mail, phone, in-person, or
drop-out) at the 12-month follow-up. We found however
a difference between the way of responding and overall
improvement (w2¼12.91; p¼ .002); responses given via telephone indicated less improvement than those given
via post and in-person.
Outcome Measures
We assessed the following three measures as primary out-
come measures: (1) mean pain intensity in the past 7 days,
(2) pain-related disability comprising disability in daily life
assessed by the Pediatric Pain Disability Index (Hübner et
al., 2009), and (3) school absence assessed via parental
report through the German Pain Questionnaire for
Children and Adolescents (DSF-KJ) (Schroeder et al.,
2010). Emotional distress and use of analgesics were
secondary outcome measures. We thus cover three core
outcome domains (pain intensity, role functioning, and
emotional functioning) from the recent IMMPACT recom-
mendations for pediatric patients (PedIMMPACT)
(McGrath et al., 2008).
Pain Intensity
Adolescents’ pain-intensity was assessed using a 11-point
numeric rating scale (NRS) (Denecke & Hünseler, 2000)
ranging from 0 (no pain) to 10 (maximal pain). This scale
has been shown to be valid and sensitive to change in
pediatric samples (von Baeyer et al., 2009).
Pain-Related Disability
Pain-related disability was assessed using the P-PDI
(Hübner et al., 2009). The P-PDI assesses disability in
daily activities owing to pain on 12 items rated on a
5-point scale (1 [never] to 5 [always]). Daily activities com-
prise activities such as going to school, doing homework,
engaging in physical activity, and sleeping. The total score
is computed by summing all items and ranges from 12 to
60 (Cronbach’s alpha in this sample¼ .85). The 2-way
imputation method (Sijtsma & van der Ark, 2003) was
Table I. Demographic Characteristics at Admission
Variable Value
Age (mean�SD; [range]) 14.1�1.91 (11–18.3)
Sex (n [%])
Female 103 (62)
Diagnosis (n [%])a
Headache 84 (50)
Recurrent abdominal pain 22 (13)
Back pain 16 (10)
Diffuse idiopathic pain 15 (9)
Disease-related pain 11 (7)
Fibromyalgia 8 (5)
Localized idiopathic pain 6 (4)
CRPS 5 (3)
Pain duration in months
(mean�SD, range)
42�37
Patients on pain medication at
preassessment n (%)
70 (62)
Mean pain intensity (mean�SD, range) 6.7�1.7
Pain intensity categories (%)b
Mild pain 4
Moderate pain 42
Severe pain 55
Note. NRS¼Numeric Rating Scale. aCumulative percentage > 100% owing to rounding up. bThe NRS was categorized into four categories: no pain (NRS 0), mild-to-moderate
pain (NRS 1–3), severe pain (NRS 4–6), and very severe pain (NRS 7–10).
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used to impute missing values when at least 9 of 12 items
were responded to.
School Absence
Parents were asked how many days their child missed
school owing to ongoing pain within the past 4 weeks
(i.e., 20 school days excluding weekends and holidays)
as part of the DSF-KJ (Schroeder et al., 2010). Thus, for
children admitted short after public holidays, some parents
had to report school absence before the holidays, but the
number of expected school days was similar for all chil-
dren, i.e., 20 days. Logan and colleagues provided evidence
for a high correlation between parent report and official
school attendance records (Logan, Conroy, Sieberg, &
Simons, 2008b).
Emotional Distress
Emotional distress was assessed using the Anxiety
Questionnaire for Pupils (AFS; [Wieczerkowski, Nickel,
Janowksi, Fittkau, & Rauer, 1981]) and the Depression
Inventory for Children and Adolescents (DIKJ;
[Stiensmeier-Pelster, Schürmann, & Duda, 2000a]). Both
measures are frequently used in adolescent samples and
allow a norm-based comparison with non-clinical school
children. We used these norms to transform individual
raw-scores to T values with a mean of 50 and standard
deviation of 10. We investigated three subscales of the
AFS; (1) fear of exams, (2) generalized anxiety, and (3)
school aversion, which all have good internal consistency
(Cronbachs alpha between .75 and .85; Wieczerkowski
et al., 1981) The DIKJ consists of 26 items measuring de-
pression with good reliability (internal consistency: 0.84)
and validity (Stiensmeier-Pelster, Schürmann, & Duda,
2000b). Raw scores > 18 are indicative of depression
(Stiensmeier-Pelster et al., 2000b). Individual missing
items (up to two for the DIKJ and the scale school aversion,
and up to three for the scales fear of exams and generalized
anxiety) were replaced as suggested by the test authors.
Use of Analgesics
Parents indicated whether the children used analgesics
before and after treatment as part of the DSF-KJ. Use of
analgesics was then classified into five groups: (1) non-
steroidal anti-inflammatory drugs (NSAID), (2) other
non-opioid analgesics, (3) triptans, and (4) opiods.
Procedure
Data on the initial status were collected by sending a bat-
tery of questionnaires via mail before admission. Data for
the follow-up were collected at the 3-month follow-up,
which was regularly scheduled with the patients. Data for
the 12-month follow-up were collected in three different
ways: (a) either during a scheduled appointment at the
German Paediatric Pain Centre for those participants who
were still in treatment, (b) via mail, or (c) via structured
telephone interview for those participants who did not
return their questionnaires. Children still in treatment
were seen for follow-up visits scheduled 3, 6, or 12
months after inpatient treatment (see also Hechler et al.,
Table II. Functioning
Variable Value
P-PDI (n¼160; mean�SD, range)a 40�9
School absence of >2 days (10%) within the
preceding 4 weeks (n [%])b 122 (73)
School absence because of pain (n [%])
<15% (3 days) school absence 45 (27)
15–50% (3–10 days) school absence 57 (34)
No school attendance at all 42 (25)
Significant emotional distress (n [%])
Fear of examination (n¼167)c 35 (21)
General anxiety (n¼167)c 61 (36)
School aversion (n¼167)c 34 (26)
Depression (n¼161)d 51 (31)
Note. P-PDI¼Pediatric Pain Disability Index. aCumulative percentage >100% owing to rounding up. P-PDI values ranged
between 12 and 60; the higher the value, the more disabled the child. bSchool absence was assessed as days absent from school within the past 4 weeks
(parental report). cSignificant values in the anxiety questionnaire were assigned if the patient
demonstrated T > 60 compared with a non-clinical population. dSignificant values in the depression questionnaire were assigned if the patient
demonstrated raw values > 18.
Figure 1. Flow of patients through the stages of the study.
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2011a). Responses were included if they were given max-
imum 18 months post discharge. The structured interview
was conducted by a research assistant not involved in
treatment.
Pain intensity and pain-related disability were assessed
via self-report. School absence and use of analgesics were
assessed by the parents. The sample size differs between
the variables (see Table II) owing to >2 missing items on
the questionnaires.
MIT at the German Paediatric Pain Centre
The MIT at the German Paediatric Pain Centre lasts 3
weeks. Children are referred to our specialized treatment
center by primary physicians due to persisting pain. The
treatment is based on operant and cognitive behavioral
principles throughout all modules. Parents are actively
engaged in the treatment as part of weekly family sessions
and coaching sessions during which parents were taught to
actively support their child to engage in healthy daily
activities. Furthermore, reintegration into the child’s daily
life is initiated from the second week onwards comprising
visits home and to their home school on one appointed
day. Patients complete various treatment modules such as
education, individual sessions on pain-related coping and
related problems, family sessions on parental cognitions
and reactions towards the child’s pain, and group sessions
on peer relationships that sum up to �5–8 hr per day. The
treatment is provided by a multiprofessional team
including clinical psychologists, pediatricians, children
and adolescent psychiatrists, pediatric nurses, physiother-
apists, occupational therapists, and social workers. A
scheduled appointment is arranged for each child and its
family 3 months after discharge. More information can be
taken from our earlier study (Hechler et al., 2009).
Statistical Analysis
Descriptive statistics (means, SD, and frequencies) of pa-
tients’ characteristics at admission were computed. Group
differences between the different ways of responding at the
12 months follow-up were tested with chi-square tests.
Treatment Effects at 12-Month Follow-Up—Statistical Significance
Statistically significant changes between admission and
12-month follow-up in all continuous variables (pain in-
tensity, pain-related disability, school absence, fear of
exam, generalized anxiety, school aversion, and depres-
sion) were assessed using repeated-measures analysis of
variance (ANOVA). Effect-sizes were calculated according
to Grawe and colleagues by dividing the difference between
12-month follow-up scores from prescores by the SD of the
scores at admission (Grawe, Bernauer, & Donati, 1994).
Effect sizes were interpreted according to established
standards (Cohen, 1988). Significant changes in the use
of analgesics (yes or no) were assessed using McNemar’s
test for paired proportions (McNemar, 1947).
Treatment Effects at 12-Month Follow-Up—Clinical Significance
The clinically significant changes in the core variables were
defined using the method developed by Jacobson and
Truax (1991). This procedure entails two steps; (1)
Computation of the reliable change index (RCI) to estimate
whether the change for an individual patient is due to
chance fluctuations or real change; (2) Calculation of
cut-off points that need to be crossed to count as clinically
relevant. The RCI was calculated using Jacobsen and
Truax’s formular and resulted in three outcome groups:
‘‘no reliable change,’’ ‘‘reliable deterioration,’’ and ‘‘reli-
able improvement’’ for each patient and test. Cut-off
points were defined using different methods, depending
on whether normative data for a test existed.
1. If no normative data were available, a cut-off point
at 90% of the patient population (mean before
treatment þ/� 1.6 SD) was used.
2. If a cut-off point already existed that indicates a
clinically relevant point, (e.g., a t-value, which in-
dicates that a score lies outside of the distribution
of the healthy population), this was used.
3. If no cut-off score existed, but distribution of the
healthy population was known, a cut-off point was
chosen, at which a value is more likely to belong
to a healthy than a clinical population.
In the present article, criterion 1 was used for
pain-related disability, resulting in a cut-off score of
25.58. Criterion 2 was used for pain intensity, school ab-
sence, and the three anxiety scales. Specifically, we used
the same the cut-off point for pain-intensity that has been
identified in adult research, where a raw change of �1.74
on the 11-point NRS was found to be clinically significant
(Farrar, Young, LaMoreaux, Werth, & Poole, 2001). This
resulted in a cut-off point of 5 (6.82–1.74) for our sample.
The cut-off point for school absence could not be
based on a clear definition, as there are no official data
on school absence in Germany, and was thus based on
our previous definition that school absence of >10%
(i.e, 2 days) during a 4-week period would be considered
as unacceptable by the teaching staff. The cut-off point for
the three anxiety scales (60) was taken from published
research with these scales (Wieczerkowski et al., 1981).
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Criterion 3 was used for depression where data on the
distribution of a healthy sample were available, and a raw
score of >18 has been indicative of depression
(Stiensmeier-Pelster et al., 2000b). Using these cut-offs to-
gether with the RCI, five outcome groups were defined for
all variables;
1. ‘‘Clinically significant change’’: Patients who reli-
ably improved and whose post-scores were below
the cut-off.
2. ‘‘Significant but not clinically relevant’’: Patients
who reliably improved but did not cross the
cut-off.
3. ‘‘No change unproblematic’’: Patients who showed
no reliable change and had prescores below or
equal to the cut-off.
4. ‘‘No change problematic’’: Patients who showed
no reliable change and had prescore above the
cut-off.
5. ‘‘Reliable deterioration’’: Patients who showed
change in the undesired direction that was larger
than the RCI.
To make our results comparable with other reports
using only individual reductions in pain ratings as index
for clinically significant change, we also calculated the
number of patients who reduced their pain rating by at
least 2 points on the NRS and at least 50% of their initial
score.
Treatment Effects at 12-Month Follow-Up—Overall Improvement
We also computed an overall improvement score based on
the clinically significant changes between admission and
12-month follow-up in the primary outcome variables
(pain intensity, pain-related disability, school absence
[Hechler et al., 2009]). Patients were classified as showing
overall improvement if they did not show deterioration in
any pain-related variable (i.e. in pain intensity, pain-related
disability, and school absence) and had clinically signifi-
cant improvements in one or both disability-related
variables (pain-related disability or school absence). All
other patients were classified as showing no overall
improvement.
Furthermore, we compared the levels of overall im-
provement for patient and pain characteristics.
Chi-square tests were used to compare diagnostic groups.
Owing to the low group size, a mixed group of adolescents
with fibromyalgia, localized idiopathic pain, and CRPS
(n¼19) was formed in addition to the other five groups
(headache, recurrent abdominal pain, back pain, diffuse
idiopathic pain, disease-related pain). A logistic regression
was used to predict overall improvement with pain inten-
sity, pain-related disability, and emotional distress at ad-
mission, as well as sociodemographic variables such as age
and sex.
Maintenance Groups Over the Two Time Points (3- and 12-Month Follow-Up)
First, we investigated whether there was a systematic
change in the overall improvement status of the adoles-
cents from 3- to 12-month follow-up using McNemar’s
test. Specifically, we investigated whether more adolescents
changed from no overall improvement at 3-month
follow-up to overall improvement at 12-month follow-up
or vice versa.
Second, we subdivided the children into four mainten-
ance groups according to the stability of their overall im-
provement status at 3- and 12-months follow-up:
1. ‘‘stable long-term improvers’’: Patients who
showed stable overall improvement both at 3- and
12-month follow-up.
2. ‘‘long-term improvers’’: Patients who showed no
overall improvement at 3-month follow-up, but
overall improvement at 12-month follow-up.
3. ‘‘short-term improvers’’: Patients who showed
overall improvement at 3-month follow-up, but no
overall improvement at 12-month follow-up.
4. ‘‘children with unsuccessful treatment results’’:
Patients who showed stable no overall improve-
ment both at 3- and 12-month follow-up.
To investigate pre-treatment influences on mainten-
ance status, univariate ANOVA were used to compare the
four maintenance groups in terms of pain-related and
demographic variables; post-hoc tests (Tukey’s honestly
significant difference [HSD]) were computed to test
which of the four groups differed. Data were analyzed
using R. We defined p < .05 as significant.
Results Treatment Effects at 12-Month Follow-Up— Statistical Significance
Participants showed statistically significant reductions in
all variables 12 months after treatment (see Table III).
Participants showed statistically significant reductions in
all variables 12 months after treatment such that they re-
ported less pain, disability, and fewer school absences.
Similarly, adolescents reported less emotional distress
with lower levels of fear of exams, generalized anxiety,
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school aversion, and depression. Effect-sizes for primary
outcome measures were large. Effect-sizes for secondary
outcome measures were of medium size.
We found a significant decrease in the number of ado-
lescents taking pain medication (McNemar’s w2¼10.26; p¼ .001; Table IV).
Treatment Effects at 12-Month Follow-Up—Clinical Significance
In terms of the clinical significance of the changes, 74% of
patients showed clinically significant changes in pain in-
tensity, 53% in pain-related disability, and 39% in school
absence (see Figure 1). For the secondary outcome vari-
ables, ‘‘unchanged unproblematic’’ was the most frequent
category. Specifically, there were 27% who improved clin-
ically significant in depression and 24% in generalized anx-
iety. Across all measures, between 1% (for fear of exams)
and 7% (for depression), showed reliable deterioration.
There were 62% of the children who showed a de-
crease of at least 50% from the initial NRS score and
80% of the children who showed reductions of �1.74
from the initial NRS score.
Treatment Effects at 12-Month Follow-Up—Overall Improvement
Twelve months after treatment, 74 patients (56%)
demonstrated ‘‘overall improvement.’’ Of these, only four
(5%) showed unchanged problematic pain-related disabil-
ity while displaying a clinically significant change in school
absence, and 11 (15%) showed unchanged problematic
school absence while displaying clinically significant
changes in pain-related disability. There were no differ-
ences between the diagnostic groups (w2¼5.5; p¼ .36). The logistic regression (method:enter) identified school ab-
sence at admission as the sole significant predictor for
overall improvement (Table V). Specifically, adolescents
with higher school absence at admission were more likely
to demonstrate overall improvement after 12 months
(OR¼1.073, p < .05).
Maintenance Groups Over 3- and 12-Month Follow-Up
Over the two time points, 53 (46%) of the adolescents were
‘‘stable long-term improvers,’’ 16 (14%) were ‘‘long-term
improvers,’’ 22 (19%) were ‘‘short-term improvers,’’ 25
(22%) had an ‘‘unsuccessful treatment outcome.’’ There
was no systematic change in the overall improvement
status (either decrease or increase) between 3 and 12
months follow-up (McNemar’s w2¼ .66; p¼ .4). Specifically, there were as many short-term improvers, as
there were long-term improvers.
There were no differences between the maintenance
groups in terms of diagnosis (w2¼16.76; p¼ .33) and those who participated versus those who did not partici-
pate in the 3-month follow-up (w2¼0.56; p¼ .45). ANOVAs comparing the four improvement groups in
terms of pretreatment psychosocial variables found that
the four groups significantly differed in school absence
(F¼3.03; p < .05), generalized anxiety (F¼2.90;
p < .05), and depression (F¼2.95; p < .05). Post-hoc
tests revealed that ‘‘stable long-term improvers’’ adoles-
cents had significantly higher rates of school absence at
admission than ‘‘unsuccessful’’ adolescents. Furthermore,
‘‘short-term improvers’’ had higher levels of generalized
anxiety and depression than ‘‘stable long-term improvers’’
adolescents (both p < .05).
Discussion
The present study presents data on the statistical and clin-
ical significance of treatment effects 1 year after an inter-
disciplinary MIT (Hechler et al., 2009). Our results show
that the majority of children exhibit statistically and clin-
ically significant changes in core outcome measures 12
months after treatment. Changes in secondary outcomes
were also observable albeit smaller. Importantly, the ma-
jority of adolescents reported no significant emotional dis-
tress at admission. The majority of children showed overall
Table III. Statistically Significant Changes in Outcomes From Admission to One-Year Follow-Up
Admission 3 months follow-up 12 months follow-up
M (SD) M (SD) p ES M (SD) p ES
Pain intensity (n¼120) 6.83 (1.78) 2.83 (2.66) <.001 2.25 2.49 (2.74) <.001 2.44
Pain-related disability (n¼109) 39.19 (8.51) 22.82 (9.06) <.001 1.92 24.11 (10.45) <.001 1.77
School absence (n¼120) 9.25 (7.76) 1.8 (3.92) <.001 .96 1.39 (3.08) <.001 1.01
Fear of exams (n¼72) 47.43 (12.37) 43.18 (11.05) <.001 .34 41.46 (1.32) <.001 .48
Generalized anxiety (n¼72) 52.96 (12.73) 46.6 (11.67) <.001 .5 45.72 (12.09) <.001 .57
School aversion (n¼72) 51.94 (10.87) 47.92 (9.95) <.001 .37 48.54 (10.13) <.001 .31
Depression (n¼73) 13.19 (7.89) 8.59 (6.92) <.001 .58 8.92 (8.69) <.001 .54
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improvement 12 months after treatment. During 3- and
12-month follow-up, approximately half of the adolescents
were classified as stable long-term improvers; 30% showed
either short-term or long-term improvements, and �20%
had an overall unsuccessful treatment outcome. The stable
long-term improvers reported more days absent from
school at admission; the short-term improvers reported
more emotional distress at admission.
Treatment Outcomes After 12 Months
The treatment outcomes at 12-month follow-up were re-
markably similar to the outcomes 3 months after treatment
(Hechler et al., 2009). We found statistically significant
changes in primary outcomes with large effect sizes and a
majority of patients showing clinically significant improve-
ments. The secondary outcomes changed to a lesser
degree. This is in line with the results at 3-month follow-up
(Hechler et al., 2009) but contrasts with other studies
(McCracken, MacKichan, & Eccleston, 2007) that found
large improvements in emotional distress in adults.
The most likely explanation for this is that only a minority
of our sample exhibited clinically relevant levels of depres-
sion and anxiety at admission and thus could not show
clinical improvements. Also, the program may have pre-
vented an aggravation of emotional distress so that most
children could maintain a low level of emotional distress.
We extend previous studies (Hechler et al., 2009;
Eccleston et al., 2003; Lanzi et al., 2007; Maynard et al.,
2009a; Palermo & Scher, 2001) by showing that adoles-
cents are able to maintain these positive treatment effects
over a period of 1 year. Here, positive long-term outcomes
for a 12-month period could be replicated with a lower
drop-out rate (32% compared with 50% by Sherry et al.,
1999) and a stricter definition of overall improvement
(based on the concept of clinical significance of Jacobson
and Truax) compared with previous studies (Sherry et al.,
1999). Hence, in line with existing studies, the present
study highlights the importance of MITs for severely
disabled children. Several possible explanations accounting
for the effectiveness have previously been discussed: The
treatment intensity of the inpatient setting, changes in
coping strategies, the ability to engage in activities with
peers, and experiences of success (Eccleston et al., 2003;
Hechler et al., 2009; Hechler et al., 2010c; Lanzi et al.,
2007; Maynard, Amari, Wieczorek, Christensen, & Slifer,
2009b; Palermo & Scher, 2001; Sherry et al., 1999;).
Future studies are warranted to examine the exact mech-
anisms of change, i.e., which changes for instance in cog-
nitive or emotional variables such as pain catastrophising
(Vervoort et al., 2011), or treatment adherence (Simons,
Logan, Chastain, & Cerullo, 2010) may moderate changes
in primary outcomes (Keefe, Buffington, Studts, &
Rumble, 2002).
In addition, future research should address two prac-
tical issues (Jensen & Foster, 2010): First, the cost-benefit
ratio needs to be assessed in a large group of patients to
make possible financial incentives more obvious (Maynard
Table IV. Changes in Medication Between Pretreatment and 12
Months Follow-Up
Admission
(n¼90)
12 months
follow-up
(n¼90)
Number of patients who
reported using
n (%a) na % p¼ .001
Any pain medication (yes, no)a 56 (62) 35 (39)
NSAID 29 (32) 25 (28)
Other non-opioid analgesics 21 (23) 5 (6)
Triptanes 3 (3) 10 (11)
Opioid 3 (3) 3 (3)
Anti-depressants
Note. aThe cumulative number of patients taking a specific medication is larger
than the number of patients taking medications overall owing to intake of mul-
tiple medications.
Table V. Predictors of Overall Amelioration at 12 Months Follow-Up
Predictors Coefficient SE OR (standardized OR) 95% CI lower 95% CI upper p
School absence at admission 0.07 0.03 1.07 (1.73) 1.02 1.14 <.05
Pain intensity �0.1 0.13 0.91 (.84) 0.7 1.16 n.s.
Pain-related disability 0.03 0.03 1.03 (1.34) 0.98 1.09 n.s.
Fear of examinations 0 0.02 1 (.99) 0.95 1.05 n.s.
General anxiety �0.01 0.03 0.99 (.86) 0.93 1.04 n.s.
School aversion �0.03 0.03 0.97 (.74) 0.92 1.02 n.s.
Depression �0.02 0.04 0.98 (.84) 0.89 1.07 n.s.
Age �0.22 0.12 0.8 (.67) 0.63 1.02 n.s.
Sex �0.08 0.5 0.92 (.96) 0.34 2.47 n.s.
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et al., 2009a). Second, it is important to investigate inpa-
tient treatment in the context of other treatment options.
Most participants proceed through the health care system
in various steps of treatment intensity, such as starting with
primary care, and proceeding to specialized treatment if the
pain persists. Here, they may be referred to outpatient or
intensive inpatient treatment dependent on their clinical
status (Hechler et al., 2011a; Von Korff & Moore, 2001).
Further research should investigate the feasibility of a
stepped-care approach in which treatment can be
intensified over time (Hechler et al., 2011a). Within such
a framework, screening tools that detect children at risk of
developing chronic pain and explicit decision criteria for
treatment allocation can be developed (Vowles, Jordan, &
Eccleston, 2009).
Maintenance of Improvement
We found that 50% showed improvement at both
follow-ups, 20% were not significantly improved at any
of the two follow-ups, and �30% of the participants
showed improvement 3 months after treatment, but not
12 months after treatment or vice versa. Our analysis of
pre-admission differences between these groups revealed
important differences. First, stable long-term improvers
had the highest levels of school absence at admission.
This is in accordance with the finding that adolescents
with high school absence at admission were more likely
to display overall improvement at 3-month follow-up
(Hechler et al., 2009). These children may have been en-
trenched in the vicious cycle of pain, disability, and school
absence at admission. As demonstrated by Logan et al.
(2008b), their academic achievement may have decreased
and contributed to long-term disability. These children
may, however, be highly academically competent (Logan
et al., 2008b). Once these children have surmounted
their disability and pain, they may therefore be able to
quickly reintegrate into school functioning. This may lead
to experiences of success, positive contact with peers, and
teachers, which may prevent deterioration and relapse over
time. These suggested mechanisms need to be further
explored in future studies.
Second, short-term compared with stable long-term
improvers had higher levels of emotional distress at admis-
sion. Possibly, deterioration in their emotional distress over
time, especially from 3- to 12-month follow-up, may have
lead to increases in pain and disability at 12-month
follow-up. The relationship between increased emotional
distress and pain and disability has been extensively
studied in children (Cohen et al., 2010; Kashikar-Zuck
et al., 2001). Our findings suggest that children with
increased emotional distress at admission benefit from
the intensive inpatient treatment as suggested by
Figure 2. Percentage of adolescents demonstrating clinically significant changes in the seven outcome variables at 12-month follow-up. Displayed
are the percentages of children for the five outcome groups: Clinically significant change; Positive change, but not clinically significant; No
change—unproblematic; No change—problematic; Reliable deterioration.
Effectiveness of Multimodal Chronic Pain Treatment 9
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improvements in primary outcomes. They deteriorate,
however, with time. The patterns of these deteriorations
need to be further investigated. On the basis of these
findings, additional treatments for these children may
need to be conceptualized specifically addressing the
aspect of emotional distress to prevent relapse and deteri-
oration such as how to cope with pain when emotional
distress increases again.
Finally, children with unsuccessful outcome did not
show particularly high levels of emotional distress or high
levels in school absence. Different to children high in
school absence at admission, they did not experience suc-
cess when being able to attend school again. Whether these
children really do not experience emotional distress, or
whether these children are high in social desirability and
deny emotional distress (Logan, Claar, & Scharff, 2008a)
needs to be investigated in future studies. In addition,
other potential candidates that may account for unsuccess-
ful treatment results such as the child’s pain-specific emo-
tions (fear of pain [Simons, Sieberg, Carpino, Logan, &
Berde, 2011]) or pain-specific cognitions (Vervoort et al.,
2011) and parental reactions such as adherence to treat-
ment (Simons et al., 2010) or parental cognitions and re-
actions (Hechler et al., 2011b; Vervoort et al., 2011)
warrant further investigation.
Limitations
There are some limitations inherent of the present study.
First, the study is limited by the lack of a control group.
However, there is little reason to suspect spontaneous
changes in the participants we studied (Perquin et al.,
2003), and the inclusion of a waiting-list control group
would have been highly unethical, given the known effect-
iveness of our program. Second, we found unexpected dif-
ferences in the overall improvement rates depending on the
way in which participants responded. Improvement rates
were lower for those who responded by phone, compared
with those who responded in person at a scheduled
follow-up visit. The latter group had contacted the tertiary
institute for a follow-up visit. During this follow-up visit,
the current status of the child is evaluated. Dependent on
the child’s status, further pain management strategies are
adjusted to the needs of the child (Hechler et al., 2011b).
At this stage, we do not if the difference in improvement
rates is due to the additional treatment or a social desir-
ability effect. Possibly, families who were interviewed by a
research assistant were more willing to report negative as-
pects than those faced with the clinicians in person. In
light of the small sample size, only 13 responded by tele-
phone; this pattern needs to be replicated before any
strong conclusions can be drawn. Third, our outcome
measures relied on self-report. More objective measures
of pain-related disability have only recently been used in
samples with chronic pain (Wilson & Palermo, 2012).
Similarly, this study did not include a measure of the eco-
nomic benefit. Fourth, we dichotomized our outcome vari-
ables. This greatly enhances the communication of results.
On the other hand, this clearly leads to a drop in power. At
present, there is no statistical method that maintains max-
imal power, and can be easily interpreted for binary out-
comes and predictors; thus the use of the Jacobson and
Truax method is recommended (Lambert & Ogles, 2009).
It will be most important to develop methods that classify
individuals’ change over time, and at the same time retain
the comparability across studies. On a similar note, cut-off
points for clinically relevant relative changes in pain inten-
sity need to be determined for children and adolescents
(Farrar et al., 2001). Also absolute levels of clinical signifi-
cant pain that are required according to Jacobsen and
Truax to differentiate the ‘‘no change problematic’’ from
the ‘‘no change unproblematic’’ group are not established
for children and adolescents. At present, it is not clear
whether methods based on relative or absolute cut-offs
are more relevant to patients. Although relative cutpoints
may overestimate small changes in almost healthy patients,
absolute cutpoints may overestimate small changes near
the border between healthy and diseased. The application
of the adult cut-off score may have hampered results. For
better study comparison, we therefore integrated report on
children displaying a 50% pain reduction. Finally, we as-
sessed general emotional distress and no pain-specific emo-
tions such as fear of pain (Simons et al., 2011). Future
treatment studies should implement assessment of
pain-specific emotions to investigate its potential influence
on treatment results.
The clinical implications of our results can be
summarized as follows: (1) the MIT is effective in improv-
ing the overall status of children over long periods. (2)
However, there were 20% with an overall unsuccessful
treatment result and 30% who showed only short-term
or long-term improvement. (3) Emotional distress seems
to play a crucial role for long-term improvement, given that
adolescents with high levels of emotional distress displayed
a risk to relapse after short-term improvements. These ado-
lescents may require special attention and interventions
that are useful in enhancing maintenance of positive out-
comes for this particular group of patients.
Acknowledgments
The authors, H.G. and H.T., contributed equally to the study.
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Conflicts of interest: None declared.
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