randomized controlled trials of psychological therapies for management of chronic pain in children...
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Randomized controlled trials of psychological therapies for management ofchronic pain in children and adolescents: An updated meta-analytic review
Tonya M. Palermo a,*, Christopher Eccleston b, Amy S. Lewandowski a, Amanda C de C. Williams c,Stephen Morley d
a Department of Anesthesiology and Perioperative Medicine, Oregon Health & Science University, Portland, OR 97239, USAb University of Bath, Bath, UKc Research Department of Clinical, Educational & Health Psychology, University College London, London, UKd Leeds Institute of Health Services, University of Leeds, Leeds, UK
a r t i c l e i n f o
Article history:Received 26 May 2009Received in revised form 1 September 2009Accepted 5 October 2009
Keywords:Psychological therapiesRandomized controlled trialsChronic painPainChildrenMeta-analysisSystematic review
a b s t r a c t
The purpose of this meta-analytic review was to quantify the effects of psychological therapies for themanagement of chronic pain in youth. Specifically, in this review we updated previous systematicreviews of randomized controlled trials by including new trials, and by adding disability and emotionalfunctioning to pain as treatment outcomes. Electronic searches of the Cochrane Register of RandomisedControlled Trials, MEDLINE, PsycLIT, EMBASE, and the Social Sciences Citation Index were conducted frominception through August 2008. Methodological quality of the studies was assessed, and data extractedon the three primary outcomes of interest. Twenty-five trials including 1247 young people met inclusioncriteria and were included in the meta-analysis. Meta-analytic findings demonstrated a large positiveeffect of psychological intervention on pain reduction at immediate post-treatment and follow-up inyouth with headache, abdominal pain, and fibromyalgia. Small and non-significant effects were foundfor improvements in disability and emotional functioning, although there were limited data on these out-comes available in the included studies. Omnibus cognitive–behavioral therapy, relaxation therapy, andbiofeedback all produced significant and positive effects on pain reduction. Studies directly comparingthe effects of self-administered versus therapist-administered interventions found similar effects on painreduction. Psychological therapies result in improvement in pain relief across several different pain con-ditions in children. Future trials are needed that incorporate non-pain outcome domains, that focus sig-nificant therapeutic content on reductions in disability, and that include extended follow-up to betterunderstand maintenance of treatment effects.
� 2009 International Association for the Study of Pain. Published by Elsevier B.V. All rights reserved.
1. Introduction
Chronic pain affects 15–30% of children and adolescents[36,37,39], and results in a measurable decline in children’s overallquality of life [31]. Many children with chronic pain experience sig-nificant pain-related disability such as limited social and physicalactivities and frequent school absences [20,21]. Children may alsoreport higher levels of distress, anxiety and depression [9,31]. More-over, children are at risk for continuing into adulthood with chronicpain, physical symptoms, and psychological problems [7]. Conse-quently, effective treatment in childhood may also lessen the eco-nomic and social impact of chronic pain in adulthood.
Psychological interventions for children with chronic pain are apromising modality demonstrated to be effective in decreasing painintensity [5].
To date, the primary psychological interventions evaluated inyouth with chronic pain are behavioral, cognitive, and cognitive–behavioral interventions (CBT) using strategies such as relaxationtraining, biofeedback, and parental operant strategies [6]. Most ofthis treatment literature has focused on children with headache[16], although effective treatments have also been developed forchildren with abdominal, musculoskeletal, and disease-related pain[17,19,41].
Several meta-analytic reviews using data pooling methods havedocumented the efficacy of psychological therapies for childrenwith chronic pain. Eccleston et al. [5] found that psychologicaltreatments were effective in reducing pain in children with chronicpain (primarily headache). The odds ratio for clinically significantreduction in pain (defined at 50%) was 9.62 and the number-needed-to-treat was 2.32. In their review of pediatric migraine,
0304-3959/$36.00 � 2009 International Association for the Study of Pain. Published by Elsevier B.V. All rights reserved.doi:10.1016/j.pain.2009.10.004
* Corresponding author. Address: Department of Anesthesiology and Periopera-tive Medicine, Mail Code: UHN-2, Oregon Health & Science University, 3181 SWSam Jackson Park Rd., Portland, OR 97239-3098, USA. Tel.: +1 503 494 0848; fax: +1503 494 3092.
E-mail address: [email protected] (T.M. Palermo).
PAIN� 148 (2010) 387–397
w w w . e l se v i e r . c o m / l o c a t e / p a i n
Author's personal copy
Hermann and colleagues [13] found that biofeedback and relaxa-tion were more effective than placebo treatments and prophylacticdrug treatments in controlling headache. More recently, Traut-mann et al. [40] found that psychological interventions for childrenwith recurrent headache produced higher rates of clinically signif-icant reductions in pain compared to the control conditions.
However, there remain several gaps in the literature due toshared limitations among the reviews that have used data poolingtechniques. Treatment outcomes were confined to pain intensityand relevant non-pain outcomes such as mood or disability werenot examined. In addition, all three meta-analyses focused almostexclusively on youth with headache. While Eccleston et al.’s review[5] aimed to include a range of pain conditions, twelve of thirteenstudies included in the meta-analysis were interventions for chil-dren with head pain. Therefore, data pooling techniques have notbeen used for RCTs of psychological interventions for children withother chronic pain conditions.
In the last decade there has been an increase in psychologicalinterventions for youth with chronic pain, and an updated meta-analytic review of treatment studies is warranted. The aims of thisreview were to update and expand upon previous reviews byincluding new trials of psychological treatments for youth withchronic pain, and to examine the efficacy of psychological inter-ventions in not only decreasing pain intensity but also in improv-ing emotional and physical functioning in children. In addition, weexamined the relationship of various characteristics and methodo-logical factors (e.g., intervention type, chronic pain condition) oneffect sizes related to treatment outcomes. We also sought todetermine the overall quality of trials and its relationship to studyoutcomes.
2. Methods
2.1. Literature search strategy
A comprehensive literature search was conducted using theCochrane Register of Randomised Controlled Trials, MEDLINE,PsycLIT, EMBASE, PubMed, and the Social Sciences Citation Indexin order to identify randomized controlled trials (RCTs) of psy-chological treatments of chronic pain in children. Search dateswere from inception of these databases to August 2008. RCTswere also sought in references of all identified studies, meta-analyses and reviews. Two separate searches were conducted.The first search was undertaken prior to the previously pub-lished systematic review [5] from the inception of abstractingservices to the end of 1999. This search yielded 3715 abstractsof which 123 papers were read in full for a final set of 18 RCTs.The second search focused on an update, overlapping by 1 yearfrom 1999 to August 2008. This search yielded 1319 abstracts,of which 46 papers were read in full for a final set of 17 RCTs.Because the present meta-analysis focuses on outcomes thathad not been extracted from the previous trials, we combinedthe two searches. We chose to continue to focus exclusively onRCTs instead of a more exhaustive systematic review includingnon-randomized controlled trials in order to make direct com-parisons to the previously published systematic review [5] andto allow for combining the searches. In this way, we are ableto make conclusions about progress in RCTs of psychologicaltherapies for children with chronic pain over the past 10 years.
The central search strategy included key words to define painand pain conditions, specific psychological interventions, and thechild and adolescent population. Pain and pain condition terms in-cluded pain, chronic pain, headache disorders, abdominal, stomachache, back, fibromyalgia, toothache, etc. Search terms for interven-tions included psychology, psychotherapy, behavior therapy, cog-nitive–behavioral therapy, biofeedback, relaxation, hypnosis,
imagery, family therapy, computer, and internet intervention.Search terms used for the population included child, adolescent,youth, juvenile, and pediatric. We also used terms to filter for ran-domized controlled trials including randomized controlled trial,randomized, controlled clinical trial, placebo, and trial. All termswere expanded in specific databases. The complete strategy isavailable on request from the author.
2.2. Inclusion and exclusion criteria
Trials were included in the current review if they were avail-able as a published report of a RCT comparing a psychologicaltreatment with primary psychotherapeutic content with placebo,other active treatment, treatment as usual, or wait-list control.The target population was children and adolescents (<19 yearsof age) with persistent, recurrent, or episodic pain in any bodysite, not associated with cancer or other life threatening malig-nant disease. In addition, studies had to be published (or elec-tronically pre-published) in English, and have 10 or moreparticipants in each treatment arm at immediate post-treatmentassessment. Last, studies needed to include data suitable forcomputation of effect sizes on at least one of the three outcomes(pain, disability, or emotional functioning).
2.3. Data extraction
Each paper was read in full in order to extract and record dataon a standardized data extraction form. This form included the fol-lowing information: authors and year of the published report,methods (e.g., number of treatment arms, assessment time points),participant characteristics (e.g., number of participants, age, gen-der distribution, type of pain problem, source), intervention details(e.g., duration of treatment, type of intervention, parent involve-ment), outcomes including names of all measurement instrumentsreported, and notes about each trial. In addition, a table was usedto record raw outcome data (e.g., means, standard deviations) fromeach study. The most appropriate instruments for the three out-come domains: pain intensity, functional interference or disability,and emotional functioning were selected.
2.4. Measurement domains
Three outcome domains, pain intensity, functional interferenceor disability, and emotional functioning, were selected as dependentvariables. Because multiple measurement tools were used in sometrials, we selected one measure considered most appropriate foreach of the three outcome domains. To guide the choice of outcomemeasure, we applied two rules. First, if an outcome measure wasestablished and occurred frequently among studies it was selectedover more novel instruments. Second, given a choice between singleitem and multi-item self report tools, multi-item tools were chosenon the basis of inferred increased reliability. These decision ruleshave been previously applied in a meta-analytic review of psycho-logical treatments for adults with chronic pain [29].
Pain intensity outcomes were most commonly reported asbinary count data on a clinically significant improvement in pain(50% or greater) obtained from pain diaries. Continuous painintensity scores were also used in some trials from numericalpain rating scales and visual analogue scales. The disability out-come was assessed using measures of school absence, functionaldisability, headache-related disability, and physical health-re-lated quality of life. While these measures examine different do-mains of functioning, each assesses children’s abilities toparticipate in routine, daily activities. Emotional functioningwas assessed by measures of depressive symptoms, internalizingsymptoms, anxiety symptoms, and psychosocial health-related
388 T.M. Palermo et al. / PAIN� 148 (2010) 387–397
Author's personal copy
Tabl
e1
Char
acte
rist
ics
ofin
clud
edRC
Ts.
Au
thor
s(y
ear)
Pain
con
diti
onSa
mpl
ede
scri
ptio
n(M
age,
ran
ge,%
fem
ale)
Inte
rven
tion
type
(Gro
up
n’s)
Trea
tmen
tex
posu
rePa
inin
ten
sity
outc
ome
Emot
ion
alfu
nct
ion
ing
outc
ome
Phys
ical
fun
ctio
nin
gou
tcom
e
Qu
alit
yra
ting
s
Bar
ryan
dvo
nB
aeye
r(1
997)
Hea
dach
e9.
4,7–
12ye
ars;
66%
fem
ale
Bri
efC
BT
grou
ptr
eatm
ent
(n=
12);
wai
t-li
stco
ntr
ol(n
=17
)
Two
90-m
inse
ssio
ns
Hea
dach
edi
ary
usi
ng
0–10
NR
Sin
ten
sity
Dia
ry–
moo
dra
tin
gD
iary
–sc
hoo
lab
sen
ces
TQ=
3D
Q=
11To
tal=
14B
uss
one
etal
.(1
998)
Hea
dach
e11
.1/1
3.0;
11–1
5ye
ars,
50%
fem
ale
Bio
feed
back
-ass
iste
dre
laxa
tion
(n=
20);
rela
xati
onpl
aceb
oco
ntr
ol(n
=10
)
10Se
ssio
ns,
40m
inea
chH
eada
che
diar
yu
sin
g0–
4in
ten
sity
Stat
eTr
ait
An
xiet
yIn
ven
tory
for
Ch
ildr
enN
one
TQ=
5D
Q=
13To
tal=
18
Con
nel
lyet
al.
(200
6)H
eada
che
10.0
,7–1
2ye
ars,
49%
fem
ale
CB
Tde
live
red
via
CD
-RO
M(n
=14
);w
ait-
list
con
trol
(n=
17)
Fou
r1-
hm
odu
les
Hea
dach
edi
ary
Non
ePe
diat
ric
Mig
rain
eD
isab
ility
Ass
essm
ent
TQ=
8D
Q=
16To
tal=
24Fi
chte
lan
dLa
rsso
n(2
001)
Hea
dach
e15
.4;
13–1
8ye
ars
69.4
%fe
mal
eR
elax
atio
ntr
ain
ing
(n=
20);
wai
t-li
stco
ntr
ol(n
=16
)8–
10Se
ssio
ns,
45-m
indu
rati
onH
eada
che
diar
yu
sin
g0–
5in
ten
sity
Non
eN
one
TQ=
4D
Q=
11To
tal=
15G
riffi
ths
and
Mar
tin
(199
6)H
eada
che
11.4
/11.
5/11
.1;
10–
12ye
ars;
50%
fem
ale
Cli
nic
base
dC
BT
(n=
17);
hom
e-ba
sed
CB
T(n
=17
);w
ait-
list
con
trol
(n=
17)
8Se
ssio
ns,
90m
in;
hom
e-ba
sed
trea
tmen
t:3
sess
ion
s+
wri
tten
man
ual
for
hom
eov
er9
wee
ks
Hea
dach
edi
ary
usi
ng
0–5
inte
nsi
tyC
hil
dren
’sM
anif
est
An
xiet
ySc
ale,
Ch
ildr
en’s
Dep
ress
ion
Scal
eb
Non
eTQ
=5
DQ
=13
Tota
l=18
Hic
kset
al.
(200
6)H
eada
che,
abdo
min
alpa
in
11.7
,9–1
7ye
ars;
64%
fem
ale
On
lin
eC
BT
(n=
21);
stan
dard
med
ical
care
wai
t-li
stco
ntr
ol(n
=16
)
7O
nli
ne
chap
ters
(1/w
eek)
Pain
diar
yu
sin
g0–
10N
RS
inte
nsi
tyPe
diat
ric
Qu
alit
yof
Life
Inve
nto
ry(P
sych
osoc
ial
Hea
lth
Sum
mar
y)
Pedi
atri
cQ
ual
ity
ofLi
feIn
ven
tory
(Ph
ysic
alH
ealt
hSu
mm
ary)
TQ=
8D
Q=
19To
tal=
27H
um
phre
yset
al.
(200
0)A
bdom
inal
pain
9.75
;4–
18ye
ars;
59%
fem
ale
Fibe
r+
biof
eedb
ack
(n=
15)a
,fi
ber
+bi
ofee
dbac
k+
CB
T(n
=16
)a ,fi
ber
+bi
ofee
dbac
k+
CB
T+
pare
nt
trai
nin
g(n
=15
)fi
ber
only
con
trol
grou
p(n
=15
)
8Se
ssio
ns
Pain
diar
yu
sin
gvi
sual
anal
ogu
esc
ale
Non
eSc
hoo
lab
sen
ces
TQ=
5D
Q=
9To
tal=
14
Kas
hik
ar-Z
uck
etal
.(20
05)
Juve
nil
efi
brom
yalg
ia15
.8;
13–1
7ye
ars,
100%
fem
ale
CB
Tco
pin
gsk
ills
trai
nin
g(n
=13
);se
lf-m
onit
orin
g(n
=14
)
6Se
ssio
ns,
2te
leph
one
chec
k-in
s(1
/wee
k)V
isu
alan
alog
ue
scal
eC
hil
dren
’sD
epre
ssio
nIn
ven
tory
Fun
ctio
nal
Dis
abili
tyIn
ven
tory
TQ=
7D
Q=
18To
tal=
25K
ron
er-H
erw
igan
dD
enec
ke(2
002)
Hea
dach
e12
.1;
10–1
4ye
ars;
53.3
%fe
mal
eTh
erap
ist
grou
pad
min
iste
red
CB
T(n
=29
);se
lf-h
elp
CB
T(n
=27
),w
ait-
list
con
trol
(19)
8Se
ssio
ns,
90m
indu
rati
onH
eada
che
diar
yN
one
Non
eTQ
=7
DQ
=12
Tota
l=19
Labb
e(1
995)
Hea
dach
e12
;8–
18ye
ars;
43.3
%fe
mal
eR
elax
atio
ntr
ain
ing
only
(n=
10)a ;
biof
eedb
ack
+re
laxa
tion
trai
nin
g(n
=10
);w
ait-
list
cont
rol
(n=
10)
10Se
ssio
ns,
45m
inea
chov
er7
wee
ksH
eada
che
inde
xN
one
Non
eTQ
=2
DQ
=9
Tota
l=11
Labb
ean
dW
illi
amso
n(1
984)
Hea
dach
e10
.8;7
–16
year
s;50
%fe
mal
eB
iofe
edba
ck+
rela
xati
ontr
aini
ng
(n=
14);
wai
t-li
stco
ntro
l(n
=14
)
10Se
ssio
ns,
40m
inea
chov
er7
wee
ksH
eada
che
diar
yu
sin
g0–
5in
ten
sity
Non
eN
one
TQ=
4D
Q=
12To
tal=
16La
rsso
nan
dC
arls
son
(199
6)
Hea
dach
eM
ean
not
repo
rted
;10
–15
year
s;96
.2%
fem
ale
Gro
up
rela
xati
ontr
ain
ing
(n=
13);
con
trol
(n=
13)
10Se
ssio
ns,
20-m
indu
rati
onfo
r5
wee
ksH
eada
che
diar
yu
sin
g0–
5in
ten
sity
Non
eN
one
TQ=
6D
Q=
14To
tal=
20La
rsso
net
al.
(198
7a)
Hea
dach
eM
ean
not
repo
rted
;16
–18
year
s;86
.9%
fem
ale
Self
-hel
pre
laxa
tion
(n=
16);
ther
apis
tas
sist
edre
laxa
tion
(n=
14);
self
-m
onit
orin
gco
ntr
ol(n
=11
)
9Se
ssio
ns,
45-m
indu
rati
onfo
r5
wee
ks=
6h
ther
apis
tas
sist
edgr
oup;
2h
self
-hel
pgr
oup
Hea
dach
edi
ary
Non
eN
one
TQ=
6D
Q=
15To
tal=
21
(con
tinu
edon
next
page
)
T.M. Palermo et al. / PAIN� 148 (2010) 387–397 389
Author's personal copyTa
ble
1(c
onti
nued
)
Au
thor
s(y
ear)
Pain
con
diti
onSa
mpl
ede
scri
ptio
n(M
age,
ran
ge,%
fem
ale)
Inte
rven
tion
type
(Gro
up
n’s)
Trea
tmen
tex
posu
rePa
inin
ten
sity
outc
ome
Emot
ion
alfu
nct
ion
ing
outc
ome
Phys
ical
fun
ctio
nin
gou
tcom
e
Qu
alit
yra
tin
gs
Lars
son
etal
.(1
987b
)H
eada
che
Mea
nn
otre
port
ed;
16–1
8ye
ars;
94,4
%fe
mal
e
Self
-hel
pre
laxa
tion
(n=
12);
prob
lem
-di
scu
ssio
n(n
=10
);se
lf-
mon
itor
ing
con
trol
(n=
12)
Self
hel
p:4
audi
otap
es(1
0–15
min
),3
hof
ther
apis
tti
me.
Prob
lem
disc
uss
ion
:7
hof
ther
apis
tti
me
Hea
dach
edi
ary
usi
ng
0–5
inte
nsi
tyN
one
Non
eTQ
=5
DQ
=11
Tota
l=16
Lars
son
,Mel
inan
dD
ober
l(1
990)
Hea
dach
e16
–18
year
s;89
.8%
fem
ale
Self
-hel
pre
laxa
tion
trai
nin
g(n
=31
);W
ait-
list
con
trol
(n=
17)
5A
udi
otap
esu
sed
over
5w
eeks
Hea
dach
edi
ary
usi
ng
0–5
inte
nsi
tyB
eck
Dep
ress
ion
Inve
nto
ryc ,C
hil
dren
’sM
anif
est
An
xiet
ySc
alec
Non
eTQ
=4
DQ
=8
Tota
l=12
McG
rath
etal
.(1
988)
Hea
dach
e13
.1;9
–17
year
s;69
.7%
fem
ale
Rel
axat
ion
trai
nin
g(n
=32
);at
ten
tion
plac
ebo
con
trol
(n=
37);
own
best
effo
rts
con
trol
(n=
30)
Six
1-h
/wee
kse
ssio
ns
Hea
dach
edi
ary
Non
eN
one
TQ=
7D
Q=
16To
tal=
23
McG
rath
etal
.(1
992)
Hea
dach
eM
ean
age
not
repo
rted
;11
–18
year
s;72
.4%
fem
ale
Self
adm
inis
tere
dC
BT
(n=
24),
ther
apis
t-ad
min
iste
red
CB
T(n
=23
),at
ten
tion
con
trol
(n=
26)
8W
eekl
yse
ssio
ns
(60
min
each
incl
inic
grou
p)H
eada
che
diar
yu
sin
g0–
5in
ten
sity
Pozn
ansk
iD
epre
ssio
nSc
alec
Non
eTQ
=2
DQ
=13
Tota
l=15
Ost
erh
aus
etal
.(1
997)
Hea
dach
e15
.2;1
2–22
year
s;74
.4%
fem
ale
Mu
ltic
ompo
nen
tC
BT
wit
hbi
ofee
dbac
k(n
=25
),w
ait-
list
con
trol
(n=
14)
8Se
ssio
ns,
4-gr
oup
90m
inan
d4-
indi
vidu
al50
min
each
Hea
dach
edi
ary
usi
ng
5po
int
inte
nsi
tysc
ale
TQ=
6D
Q=
12To
tal=
18Pa
ssch
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ffici
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data
.
390 T.M. Palermo et al. / PAIN� 148 (2010) 387–397
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quality of life. The outcomes used in each trial for each domainare shown in Table 1.
2.5. Quality ratings
All trials were rated using a scale developed specifically forassessing the quality of psychological treatments for chronic pain[43]. The Quality Rating Scale is comprised of an overall total qual-ity score (0–35) consisting of two subscales, a Treatment Qualitysubscale (0–9) covering stated rationale for treatment, manualiza-tion, therapist training, and patient engagement; and a Design andMethods Quality subscale (0–26) covering inclusion/exclusion cri-teria, attrition, sample description, minimization of bias (random-ization method, allocation bias, blinding of assessment, andequality of treatment expectations), selection of outcomes, lengthof follow-up, analyses, and choice of control. All papers were ratedby two of the review authors (TP and AL) and consensus reachedafter initial comparison of ratings. Interrater reliability was calcu-lated on the overall quality score and the two subscales.
2.6. Data collection and analysis
Data were extracted and coded on details relating to the designof the study, the participants, pain condition, method of treatment,outcome measurement tools used, and outcome data for computa-tion of effect sizes. When data were missing on primary outcomesof interest, authors were contacted via email to obtain data neces-sary for effect size calculations. Four authors provided additionaldata upon this request. Data suitable for pooling were analyzedusing RevMan v5.0 (Cochrane Collaboration).
2.7. Organization of selected studies
Selected studies were organized along several dimensionsincluding intervention type and pain condition. In addition, noveltreatments including comparison of different exposures and differ-ent modalities were examined separately. Table 1 presents detailsof the 25 studies along these dimensions.
Psychological interventions were grouped into one of threebroad categories, including omnibus cognitive–behavioral therapy(CBT), relaxation-based therapy, and biofeedback. Omnibus CBT in-cluded interventions with a behavioral and cognitive component,such as family CBT, parent operant strategies, multicomponentCBT, and pain coping skills. Relaxation-based therapy includedinterventions that focused exclusively on progressive musclerelaxation and similar strategies such as hypnosis. Biofeedback in-cluded interventions that were focused on biofeedback trainingwith or without relaxation training. Effects of two different expo-sures of the same intervention (e.g., self-guided CBT at home ver-sus therapist-administered CBT in clinic) were examinedseparately. In addition, novel treatments were identified, in thiscase, computer-based applications, to evaluate effects separatelyin these studies.
Type of chronic pain condition was categorized into threegroups, headache, abdominal pain, or fibromyalgia. One trial in-cluded children who had diagnoses of either headache or abdomi-nal pain and this study was included in both categories in thesubgroup analysis by pain condition [14], and the effects of inclu-sion of this study in each analysis were examined.
The main focus of the meta-analysis was to examine the overalleffect of psychological interventions in RCTs on pain, disability, andemotional functioning outcomes, including testing for effects ofintervention type and pain condition in subgroup analyses. In theoverall analysis and the pain condition analysis, treatment groupswere pooled. In the analysis of the effects of intervention type, inthe case where studies used multiple active treatments, the follow-
ing approach was used. Analysis of multiple active treatmentswould require a modeling of the dependency between groups toyield accurate effect sizes across the three intervention categories[11] as each effect size from the same study involves the samesample in the control group. Because necessary information wasnot available in the obtained studies to model such dependencies,it was not possible to include all treatment conditions in the anal-ysis of intervention types. Thus, in the case of multiple active treat-ment arms, if the intervention was the same but with a differentexposure (such as therapist and home administered CBT), treat-ment arms were pooled (n = 8 trials). In a subgroup analysis, weexamine differences between therapist and self (home-based)administration of CBT; however, there were too few trials to allowfor any further comparisons by treatment exposure. If the treat-ment arms included intervention groups that fell into two catego-ries (such as biofeedback and relaxation therapy), the interventionthat was the focus of the study or the group hypothesized to havethe best outcome due to intervention was selected for the sub-group analysis (n = 2 trials). There were too few trials to allowfor any direct comparison between two intervention types. Table1 indicates those treatments within the individual studies thatwere omitted from the subgroup analysis of intervention type.
In the overall analysis, two assessment points were selected foranalysis: post-treatment and follow-up. Post-treatment refers tothe assessment point occurring soonest following treatment, andfollow-up refers to the assessment point occurring at least3 months post-treatment but not more than 12 months (the longertime point was selected if two follow-up assessments occurredduring this timeframe).
2.8. Data analysis
The primary pain outcome that was considered was binarycount data on clinically significant reduction in pain (50% or great-er pain reduction). These data are reported as odds ratios (OR). Thenumber-needed-to-treat (NNT) for benefit was also computed onthe clinically significant reduction in pain outcome. The NNT rep-resents the expected number of patients who must be treated withpsychological therapy in order to expect one additional beneficialoutcome compared to the expected event rates under the controlconditions. When these data were not available, continuous dataon mean pain intensity were used for computation of standardizedmean differences (SMD). If a study had both pain outcomes avail-able, we chose the binary count data on clinically significant reduc-tion in pain as this was the most commonly reported metric. Alldisability and emotional functioning outcomes were presented intrials using continuous data, and therefore we used the SMD. TheSMD is computed from the difference between the two group’smeans divided by their pooled standard deviation. In social scienceresearch, Cohen [2] has defined SMD effect sizes of 0.2, 0.5, and 0.8,as representing small, medium, and large effect sizes respectively.In the current review, larger effect sizes are associated with greaterdifferences between treatment and control groups on measures ofpain intensity, disability or emotional functioning. The negativedirection of the effect size indicates greater improvement for thetreatment group. The overall meta-analysis with pooled treatmentconditions examined outcomes at two assessment points, immedi-ately post-treatment and at 3-month or longer follow-up. How-ever, due to limited follow-up data, the remaining subgroupanalyses were performed using the immediate post-treatmentassessment only.
Heterogeneity between treatment studies was examined usingthe I2 statistic. The I2 statistic describes the percentage of the var-iability in effect estimates that is due to heterogeneity rather thansampling error. A value greater than 50% may be considered sub-stantial heterogeneity [15]. Both fixed-effect and random-effects
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models were used to pool data on the outcomes, depending uponwhether significant heterogeneity was detected. In cases whereno substantial heterogeneity was found (I2 < 50%), a fixed-effectsmodel was used. However, for the three continuous outcomes,mean pain intensity, emotional functioning, and disability, a ran-dom-effects model was chosen a priori. It has been recommendedthat random-effects models be used for continuous outcomes andwhen differing measurement instruments are used across trials[15]. Therefore we made an a priori decision to use this model. Arandom-effects model assumes measurement error beyond subjectsampling error is randomly distributed and not from systematicdifferences among studies. When there is no heterogeneity,fixed-effects and random-effects models produce the same results.In addition to the SMD, confidence intervals and pooled odds-ratiostatistics were calculated. Confidence intervals not including zerowere considered statistically significant.
Studies included in the meta-analysis were also weightedaccording to sample size using a least-squares approach. This ap-proach gives studies with larger numbers of participants greaterweight in the meta-analysis.
3. Results
3.1. Characteristics of included studies
Twenty-five studies involving a total of 1247 participants wereincluded. Details of these studies are summarized in Table 1. A to-tal of 169 studies were identified by our two searches and read infull, of which 134 were excluded because they were outside thescope of this review. The remaining 35 studies were consideredfor this review and coded in full. The trials used in the previoussystematic review and meta-analysis [5] were automatically eligi-ble. Of the 35 trials, 10 were excluded because: they no longer metthe eligibility criterion of having 10 or more participants in eachtreatment arm at immediate post-treatment assessment[8,22,24,38,42]; the active treatment lacked psychological content[30]; there were no outcomes within the dependent variables ofinterest [10]; or they had insufficient available data for computa-tion of effect sizes [1,4].
The 25 included studies were from six countries. Most studieshad two treatment arms (n = 14), but several studies had three(n = 10) and one had four arms. The mean number of participantsper treatment arm was 21. Study participants ranged in age from9 to 17 years with a mean age of 12.63 years (SD = 2.36) reportedin n = 21 studies. Sixty-five percent of all participants were female.Only 10 studies reported the mean duration of the pain problem,with an average duration of 3.6 years.
Participants were recruited from hospital or clinic settings (10studies), school (five studies), or community advertisements (fivestudies). Nineteen interventions were conducted with youth withheadache (including migraine). Of the remaining interventions,four were for children with abdominal pain, one was for fibromy-
algia, and one study included children with either headache orabdominal pain.
The psychological treatments included 12 studies of omnibusCBT interventions, nine of relaxation-based therapy, and four ofbiofeedback. Four studies compared two different exposures, spe-cifically self-guided home exposure versus therapist-administeredclinic exposure, of the same intervention. Two studies of home-based interventions used computer applications to deliver treat-ment (i.e. CD-ROM, Internet). Treatments were typically of shortduration and the average number of hours of treatment contact re-ported in 20 studies was 6.4 h. The format and duration of theseinterventions differed, with treatments ranging from 1 to 12 ses-sions. Sessions were conducted individually (n = 14) and in groups(n = 8) in the included studies. In seven studies, parents were in-cluded in the intervention.
3.2. Quality ratings of trials
Two independent raters scored the quality of each of the RCTs.The intra-class correlation co-efficient using absolute agreementfor the two raters was .90 for the Total Quality score, .88 for theTreatment Quality subscale, and .84 for the Design Quality subscale.Quality scores for each trial are shown in Table 1. For the 25 includedstudies, the mean overall quality score was 18.76 (SD = 4.41), themean design quality score was 13.48 (SD = 3.18), and the meantreatment quality score was 5.28 (SD = 1.65). The original scale didnot include published cut-off scores for levels of treatment quality,however average scores for excellent, average, and poor trials ob-tained in the validation sample were 22.7(1.95), 18.71(2.25),12.10(3.17) respectively [43]. Thus, included studies would be con-sidered average on total quality when referenced to a subset of RCTsfor psychological interventions in adults.
Spearman’s correlation was conducted to examine the associa-tion among year of publication and quality ratings. These correla-tions were significant, indicating that as year of publicationincreased so did total quality, trial quality, and design quality rat-ings of the trials (rho’s = .45 to .48, p’s < .05). Pearson’s correlationwas conducted to examine the association among quality ratingsand trial effect sizes, specifically, odds ratios for pain reduction.Among the 18 trials included in the analysis of clinically significantpain reduction, the correlations with quality ratings were not sig-nificant (r’s = �.09 to �.12), indicating no relationship between ef-fect sizes for pain reduction and quality ratings of the trials.
3.3. Meta-analysis results
3.3.1. Overall meta-analysisTable 2 provides a summary of the results of the overall meta-
analysis for each of the outcomes at two assessment points (post-treatment and follow-up). Odds ratios and 95% CIs are presentedfor the binary event data on clinically significant reduction(>50%) in pain, and standardized mean differences (SMDs) and
Table 2Summary of meta-analytic findings: pooled treatment conditions at post-treatment and follow-up.
Outcome k Total N OR 95% CI Z p I2 (%)
Post-treatment: clinically significant improvement in pain 18 714 5.92 4.07, 8.61 9.31 .0001 17Follow-up: clinically significant improvement in pain 7 239 9.88 5.25, 18.58 7.11 .0001 24
SMD
Post-treatment: mean pain intensity 7 287 �0.37 �0.82, 0.09 1.59 .11 80Follow-up: mean pain intensity 3 137 �0.43 �1.04, 0.17 1.41 .16 62Post-treatment: disability 6 220 �0.24 �0.51, 0.03 1.73 .08 0Post-treatment: emotional functioning 6 204 �0.12 �0.40, 0.17 0.80 .42 0
Note. k = number of studies, N = number of subjects; OR = odds ratios, SMD = standardized mean difference, CI = confidence interval, I2 = heterogeneity of studies.
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95% CIs are presented for continuous outcomes. A negative direc-tion for SMDs indicates that the treatment condition had moreimprovement on outcomes than control conditions.
Of the included studies, all 25 trials had outcome data on painintensity, six on disability, and six on emotional functioning thatcould be used in effect size calculations and incorporated intothe meta-analytic analyses. Ten studies had available follow-updata of 3 months or more on pain reduction and are includedin the overall meta-analysis with pooled treatment conditions.Because fewer than three studies presented data on disabilityor emotional functioning at follow-up, these effects were notestimated.
Eighteen studies were entered into the analysis of effects oftreatment on clinically significant reduction in pain. I2 analysesdid not reveal heterogeneity across studies (I2 = 17%), and thereforea fixed-effects model was used. Psychological therapies, consideredas a group, reduced pain intensity by at least 50% in significantlymore children and adolescents, as compared to control conditionsat post-treatment (OR 5.92; 95% CI 4.07–8.61), and this effect wassignificant (Z = 9.31, p < .0001). At 3-month follow-up, similar ef-
fects on pain reduction were found (OR 9.88; 95% CI 5.25–18.58,Z = 7.11, p < 0.0001). The number-needed-to-treat for benefit basedon these results is 2.64 (CI 2.27–3.21) at post-treatment and 1.99(CI 1.66–2.60) at follow-up. Forest plots in Figs. 1 and 2 show oddsratios and CIs for individual studies at the post-treatment and fol-low-up assessments.
The remaining seven studies were entered into the analysis ofpain reduction using continuous data on pain intensity. Psycholog-ical therapies had a small effect on reduction of mean pain inten-sity compared to control conditions at post-treatment (SMD�0.37, 95% CI �0.82 to 0.09), and at follow-up (SMD �0.43, 95%CI �1.04 to 0.17) and these were not significant. I2 analyses re-vealed substantial heterogeneity across studies (I2 = 80%). We re-moved each study individually from this analysis to identifypotential outliers that may account for this heterogeneity. The trialby Humphreys [18] has a much larger effect size in comparison tothe other trials (SMD �1.66, 95% CI �2.32 to �1.00), and when re-moved from the analysis drops the I2 value to 44%.
Six studies met criteria for the calculation of effect size for dis-ability. Psychological therapies had a small effect on disability in
Fig. 2. Clinically significant reduction in pain at follow-up by study and overall.
Fig. 1. Clinically significant reduction in pain at post-treatment by study and overall.
T.M. Palermo et al. / PAIN� 148 (2010) 387–397 393
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comparison to control conditions at post-treatment (SMD �0.24,95% CI �0.51 to 0.03) and this was not significant. I2 analyses didnot reveal heterogeneity across studies (I2 = 0%).
Six studies were entered into the analysis of effects of treatmenton emotional functioning. Psychological therapies did not improveemotional functioning in youth compared to no treatment or pla-cebo at post-treatment (SMD �0.12, 95% CI �0.40 to 0.17). I2 anal-yses did not reveal heterogeneity across studies (I2 = 0%).
3.3.2. Analysis by intervention typeTable 3 displays the results for the effects of different interven-
tion types (CBT, relaxation, and biofeedback) on pain, disability,and emotional functioning at post-treatment. As shown in Table3, all three interventions yielded positive effects on clinically sig-nificant pain reduction, with odds ratios of 4.13 (CBT), 9.93 (Relax-ation), and 23.34 (Biofeedback). Disability as an outcome was onlyavailable in studies of CBT, with a small and non-significant effect(SMD = �0.24, 95% CI �0.51, 0.03). Emotional functioning was in-cluded in 5 CBT trials (SMD = �0.09, 95% CI �0.40, 0.21) and oneBiofeedback trial (SMD = �0.15, 95% CI �0.91, 0.61), producingsimilar small and non-significant results.
3.3.2.1. Therapist vs self-administered treatment. Four trials con-ducted with youth with headache [12,23,25,26] directly comparedtwo exposures of psychological therapy, therapist-administeredversus self-administered (home-based). There was no significantdifference in the number of patients for whom pain intensity re-duced by 50% or more (OR 0.97; 95% CI 0.52–1.79; z = .11,p = .91) suggesting equal effectiveness of therapist-administeredand self-administered treatment.
3.3.2.2. Computer-based applications. Two trials were conductedusing a comparison of computer-based applications to wait-listcontrol conditions [3,14]. Odds ratio for clinically significant reduc-tion in pain (OR 7.99; 95% CI 2.65–24.08; z = .3.69, p = .0002) is
comparable to trials using other treatment formats to deliver psy-chological therapy.
3.3.3. Analysis by pain conditionTable 4 displays the results for the effects of psychological ther-
apy for children presenting with different pain conditions (head-ache, abdominal pain, and fibromyalgia) on their pain, disability,and emotional functioning at post-treatment. Clinically significantpain reduction data were available for headache and abdominalpain conditions, and similar, significant effects for pain reductionwere found in both groups, with odds ratios of 6.10 (headache)and 7.52 (abdominal pain). Because the trial by Hicks and col-leagues [14] included children with either abdominal pain or head-ache, we examined the difference in effects when this study wasexcluded from either the headache or abdominal pain analysis.For clinically significant reduction in pain, the analyses were notdifferent. However, for the analysis of disability, the effect size in-creased and became significant when the Hicks study was removedfrom the analysis (SMD = �0.43, 95%CI �0.86, �0.00, z = 1.96,p = .05) for the abdominal pain condition (no difference in effectswas found for the headache condition analysis). Emotional func-tioning was unchanged in trials of children with headache, abdom-inal pain, or fibromyalgia, with similar small and non-significanteffects. There was no difference in effects when the Hicks studywas excluded from either the headache or abdominal pain condi-tion analysis.
4. Discussion
Psychological treatments can significantly reduce pain intensityreported by children and adolescents with headache, abdominalpain, and fibromyalgia. These findings emerge from RCTs primarilycomparing psychological treatments to no-treatment control con-ditions. Our findings were similar to previous reviews by Eccleston
Table 3Meta-analytic results for intervention type at post-treatment assessment.
Outcome Intervention k Total N OR 95% CI Z p I2 (%)
Clinically significant pain reduction CBT 9 406 4.13 2.61, 6.54 6.05 .0001 0Relaxation 6 225 9.93 4.38, 22.54 5.49 .0001 0Biofeedback 3 82 23.34 5.87, 92.72 4.48 .0001 34
SMD
Disability CBT 6 220 �0.24 �0.51, 0.03 1.73 .08 0Relaxation 0 0 – – – – –Biofeedback 0 0 – – – – –
Emotional functioning CBT 5 174 �0.09 �0.40, 0.21 0.59 .55 0Relaxation 0 0 – – – – –Biofeedback 1 30 �0.15 �0.91, 0.61 0.40 .69 n/a
Note. CBT = omnibus cognitive–behavioral therapy, k = number of studies, N = number of subjects; OR = odds ratios, SMD = standardized mean difference, CI = confidenceinterval, I2 = heterogeneity of studies.
Table 4Meta-analytic results for type of pain condition at post-treatment assessment.
Outcome Condition k Total N OR 95% CI Z p I2 (%)
Clinically significantpain reduction
Headache 16 632 6.10 4.06, 9.15 8.72 .0001 23Abdominal 3 128 7.52 3.29, 17.16 4.79 .0001 0Fibromyalgia 0 0 – – – – –
SMD
Disability Headache 3 97 �0.08 �0.48, 0.33 0.38 .71 0Abdominal 3 133 �0.33 �0.68, 0.02 1.85 .06 0Fibromyalgia 1 27 �0.18 �0.93, 0.58 0.45 .65 n/a
Emotional functioning Headache 4 138 �0.16 �0.51, 0.19 0.88 .38 0Abdominal 2 76 �0.10 �0.55, 0.35 0.43 .67 0Fibromyalgia 1 27 .07 �0.68, 0.83 0.18 .18 n/a
Note. k = number of studies, N = number of subjects; OR = odds ratios, SMD = standardized mean difference, CI = confidence interval, I2 = heterogeneity of studies.
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et al. [5] and Trautmann et al. [40] who also found that psycholog-ical interventions in RCT designs are effective in reducing painintensity in children with headache. Importantly, however, thisstudy extends the findings to effects of psychological treatmentof children with non-headache conditions, specifically abdominalpain and fibromyalgia.
A primary goal of the meta-analysis was also to examine theefficacy of psychological treatments for reducing disability relatedto pain and improving emotional functioning, outcomes not previ-ously examined in meta-analytic reviews of RCTs for children withchronic pain. The effect size calculations for disability and emo-tional functioning outcomes revealed small and non-significanteffects of psychological treatments. The minimal impact of psycho-logical treatment on children’s pain-related disability and emo-tional functioning may be explained by several factors includingthe small number of studies included in the current review, differ-ing measures used across the included studies, and limitations ofavailable measures.
Another contribution of this meta-analysis is that we were ableto include subgroup analyses of intervention type and pain condi-tion. These analyses demonstrated that across interventions (CBT,relaxation, and biofeedback) positive effects on pain reductionwere found. Too few studies directly compared CBT, biofeedback,and relaxation to make conclusions about the superiority of oneintervention over the others. In the analysis of different exposuresto psychological treatment, self-administered treatment at homeperformed similarly to therapist-administered treatment in clinic,suggesting equivalent positive effects on pain reduction. Novelcomputer-based applications also produced significant pain reduc-tion in youth compared to control conditions, and effects were ofsimilar magnitude in comparison to face-to-face treatment deliv-ery. It will be important in future studies to directly compare activetreatments (e.g., CBT versus biofeedback) and delivery methods(e.g., computer-based versus face-to-face).
In the subgroup analysis by pain condition, similar positive ef-fects of pain reduction were found in children with abdominal painand headache. However, interventions delivered to children withabdominal pain produced greater changes in disability outcomescompared to interventions delivered to children with headacheor fibromyalgia. Due to the limited number of studies, it was notpossible to examine systematic differences that may explain whychildren with abdominal pain had greater reductions in disabilityin these trials. Differences in intervention content, treatment expo-sure, therapist training, or individual characteristics of these chil-dren may be responsible for these effects.
Taken together, our findings lend support for the flexibility andutility of psychological treatments for managing chronic pain inyouth. In particular, they appear to produce positive effects on painreduction when delivered in different ways, using different cogni-tive and behavioral strategies, and with children with various painconditions.
Our review also demonstrates progress in RCT trial quality andquality reporting over the past 10 years. Analyses indicated that asyear of publication increased so did total quality, trial quality, anddesign quality ratings. Quality ratings were not, however, associ-ated with trial outcomes. Likely, this improvement in quality re-flects changes in standards for quality reporting after thepublication of the CONSORT statement [28] along with increasedawareness and attention to these standards by authors, reviewers,and journal editors. Nonetheless, several aspects of trial design areworth highlighting as they represent areas where design qualitycould be enhanced. Most of the included RCTs used treatment asusual or wait-list control conditions, making it difficult to separatetreatment from placebo effects. Therefore the comparator in mosttrials was no treatment. Use of attention control comparison con-ditions would significantly enhance the quality of trial design. In
addition, very limited information was available on extended fol-low-up to allow for testing of the maintenance of treatment effects.Previous recommendations have been made to conduct follow-upassessments for pain intervention studies at a minimum of6 months following the completion of treatment and few studiesin the current review provided these data. Intent-to-treat analysesand study flow (e.g., drop outs) were additional areas that were re-ported inconsistently in these trials.
There are several limitations of this meta-analysis that shouldbe considered in interpreting the findings. First, our review is lim-ited to studies that attempt to control for bias by the use ofrandomized controlled trial designs and does not include uncon-trolled trials, case studies, or observations. The focus on RCTs aloneallowed us to increase the precision of our effect size estimates.However, the ecological validity of the findings in terms of theirrelevance to everyday clinical practice remains to be investigated.Second, although progress has been made in applying interven-tions in youth with pain conditions other than headache incontrast to Eccleston et al.’s [5] systematic review and meta-anal-ysis, additional studies with samples of children with a variety ofother pain conditions are clearly needed. Third, our ability to sum-marize data for the meta-analysis along disability and emotionalfunctioning outcomes was extremely limited due to the smallnumber of studies incorporating these measurements. Fourth,there was substantial variability in measurements, particularlyfor disability and emotional functioning outcomes, which mayhave influenced effect size estimates. Given recent recommenda-tions for specific outcome domains (PedIMMPACT) [27], andsuggestions for measurement instruments, future trials are likelyto have greater consistency in using well-validated outcomemeasures.
This study highlights several gaps in the scientific study of psy-chological interventions to reduce chronic pain and improve func-tional outcomes in a pediatric population that are important areasfor future research. First, the sample size of many of the includedstudies was quite small with an average treatment arm of 21 sub-jects. Multi-site recruitment will be necessary to produce large en-ough sample sizes that allow for stronger tests of treatmentefficacy and for examination of individual differences in treatmentresponse. Second, virtually no information is available on childindividual differences such as age, gender, or pain duration thatmay influence treatment response. Third, the evidence base con-tains studies with participants recruited from various sites, includ-ing patients attending clinics, people attending schools, and thoseresponding to advertisement. Future comparisons between recruit-ment sites and between clinic attendees and non-clinic attendeeswill be instructive. Fourth, social desirability effects were largelyuncontrolled in these studies. Given the absence of attention con-trol comparisons it is possible that treatment effects are due to ahost of patient–therapist factors non-specific to treatment. Finally,improvements in measurement technology and practice are neces-sary. Measurement over a broader array of outcome domains isnecessary [27]. However, further use and validation of existingmeasures would be helpful.
In particular, it will be important to establish additional valida-tion of commonly used measures of children’s functioning to ad-dress the significant gaps in knowledge of how these measuresperform [32]. For example, there are no established cut-off scoreson most disability measures, and therefore interpretation of theclinical relevance of scores is not possible. Moreover, althoughthe domain of functioning is by definition multi-dimensional, fac-tor analyses have not been performed on most available measuresof disability [33], limiting sensitivity in detecting changes in spe-cific areas of functioning. While treatment goals for the child withchronic pain often center around accomplishing improvement inthe child’s ability to participate in activities of daily living includ-
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ing school attendance and taking part in social or physical activi-ties, further research will be necessary to ensure that such changesin function can be adequately measured [34].
In conclusion, although findings from this meta-analysis arepromising, suggesting that psychological treatments produce asubstantial improvement in children’s pain intensity across severalchronic pain conditions, there remain important challenges to ad-dress in future work. Although the evaluation of specific treatmentcomponents is beyond the scope of this review, it is important toconsider how psychological therapies developed for children andadolescents with chronic pain address core mechanisms expectedto lead to functional improvements. Such consideration may leadto further treatment development, enhancing current behavioraland cognitive–behavioral treatments to address mechanisms ofchange. Although research has highlighted the lack of direct corre-spondence between children’s pain intensity and level of disability[31], most psychological interventions for pediatric pain weredeveloped for the stated goal of reducing pain. Theory of how psy-chological intervention strategies lead to change in physical andemotional functioning related to children’s pain is needed. Last,as we move into an era where technology is being used increas-ingly in the delivery of healthcare, including to children withchronic pain [3,14,35], a challenge will be to identify the most rel-evant and effective components of psychological interventions, andto consider how they can be adapted for delivery via technologicalmedia.
Conflict of interest
There are no conflicts of interests.
Acknowledgements
This work was partially supported by an International Associa-tion for the Study of Pain Collaborative Research Grant awarded toT.P. and C.E. and by NIH K24HD060068 (T.P.). We would like tothank Hannah Somhegyi and Simona Patange for their assistancewith data extraction and coding. In addition, we thank the follow-ing authors, Drs. Carrie Hicks, Paul Robins, Lisa Scharff, and Carlvon Baeyer who provided additional data from their studies forthe meta-analysis.
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