ORIGINAL ARTICLES

Physical and Mechanical Therapies for Lower-Limb

Problems in Juvenile Idiopathic Arthritis
A Systematic Review with Meta-Analysis
Antoni Fellas, BHSc(Hons)*
Andrea Coda, PhD*
Fiona Hawke, PhD*

Background: Juvenile idiopathic arthritis (JIA), a chronic, autoimmune, inflammatory

joint disease, is the most common arthritis affecting children younger than 16 years.
Children with JIA commonly experience lower-limb dysfunction and disability. We
systematically reviewed the effectiveness of physical and mechanical therapies for
lower-limb problems in JIA.
Methods: Randomized controlled trials of physical and mechanical interventions for
lower-limb problems in children with JIA were included. Primary outcome was pain.
Secondary outcomes included disability, functional ability, and health-related quality of
life. Several databases were searched for eligible studies. Authors of included studies
and researchers in the field were contacted to identify additional studies.
Results: Two studies evaluating the effectiveness of customized/custom foot orthoses
in treating foot and ankle pain in children with JIA (N ¼ 100) were included. One study
also evaluated simple cushioned inserts. Meta-analyses for comparisons between
custom/customized foot orthoses and a control intervention after 3 months were not
significant for the outcomes of pain (mean difference, –8.97; 95% confidence interval
[CI], –18.01 to 0.07), child-rated health-related quality of life (mean difference, 4.38;
95% CI, –3.68 to 12.44), and parent-rated health-related quality of life (mean difference,
1.77; 95% CI, –6.35 to 9.90). Meta-analyses were supported by sensitivity analyses.
Conclusions: There is a paucity of research evaluating physical and mechanical
therapies for lower-limb problems in JIA. No physical therapy has been evaluated in
randomized controlled trials, and mechanical therapy evaluation is limited to foot
orthoses and shoe inserts for foot and ankle pain. The existing research is hampered by
small sample sizes. Until further research is conducted, the effectiveness of mechanical
and physical therapies for lower-limb problems in JIA remains unclear. (J Am Podiatr
Med Assoc 107(5): 399-412, 2017)

Juvenile idiopathic arthritis (JIA) is the most
common form of arthritis in children and adoles-
cents.1 Research in developed countries reports
prevalence rates from 16 to 150 per 100,000.1
Typical signs and symptoms of active JIA are
swelling, inflammation, pain, and stiffness, most
commonly in hip, knee, and ankle joints and in small
joints in the hands and feet.1 If uncontrolled, JIA
can cause joint damage and fixed deformities.2 The
International League of Associations for Rheuma-
*School of Health Sciences, Faculty of Health and
Medicine, University of Newcastle, Ourimbah, Australia.
Corresponding author: Antoni Fellas BHSc(Hons), School
of Health Sciences, Faculty of Health and Medicine,
University of Newcastle, Central Coast Campus, Ourimbah,
NSW 2258, Australia. (E-mail: Antoni.Fellas@uon.edu.au)
tology identifies seven subgroups of JIA.3 Active JIA
of any type may cause premature epiphyseal closure
and subsequent local growth defects, typically of
the knee.4 Children with JIA may also present with
enthesitis at the plantar fascia or Achilles tendon,
flexion contractures, synovitis, and muscle atro-
phy.1
Juvenile idiopathic arthritis has a substantial
financial burden on the families of affected children
and the health-care system.5-7 In 2013 to 2014, the
Australian government subsidized more than A$7.5
million for biological drug agents alone for children
with JIA.8 Also, JIA has a psychological effect on
affected children, with increasing disability being
associated with increasing rates of depression.9,10

Journal of the American Podiatric Medical Association  Vol 107  No 5  September/October 2017 399
At present, there is no drug therapy capable of Life Inventory [PedsQL] measurement model)14;
causing complete remission in all children affected participant satisfaction with the intervention; and
by JIA. As a result, many children with JIA adverse events.
experience disabling lower-limb symptoms despite
gold-standard drug treatment. Although the evi- Searches
dence base for mechanical and physical therapies
for lower-limb problems in children with JIA is The MEDLINE (January 1966 to June 2015) search
growing, it is still uncoordinated and in some cases strategy is presented in Table 1. This search strategy
difficult for health professionals to access. For was adapted for Embase (January 1980 to June
children with JIA to achieve their best clinical 2015), Cochrane Central Register of Controlled
outcomes, health professionals need access to high- Trials (The Cochrane Library, latest issue), PubMed
quality summaries of evidence to guide their daily (January 1966 to June 2015), and Cumulative Index
clinical practice. The objective of this study was to to Nursing and Allied Health Literature (1982 to
systematically review the evidence for physical and June 2015). No language or publication restrictions
mechanical interventions for lower-limb problems were applied. Reference lists of all included studies
in JIA. were checked for other potentially eligible trials.
Corresponding authors of included studies and
Methods researchers in the field were contacted via e-mail
to identify other potentially eligible studies.
The protocol for this systematic review has been Two reviewers (A.F. and A.C.) independently
published elsewhere11 and is registered with the screened the titles and abstracts of all studies
international prospective register of systematic identified by the search. Full-text articles of
reviews (PROSPERO). potentially eligible studies were retrieved by one
of us (A.F.) and independently screened by two of
Studies and Participants us (A.F. and A.C.). Authorship and results were not
masked. Disagreements between the two authors
All randomized controlled trials (RCTs) and quasi-
regarding full-text inclusion were resolved by a
RCTs of mechanical and physical interventions for
third reviewer (F.H.). If disagreements were not
lower-limb problems in JIA were included. Physical
resolved successfully by the third reviewer, study
interventions include but are not limited to stretch-
authors were to be contacted, although this was
ing, strengthening, and massage. Mechanical inter-
never required.
ventions include but are not limited to footwear,
orthoses, and splints. Studies evaluating invasive One of us (A.F.) extracted data from included
(eg, acupuncture), pharmacological, and surgical studies using a standardized pilot-tested form, and a
therapies were excluded, as were studies evaluating
the effectiveness of interventions for the prevention Table 1. OvidSP MEDLINE Search Strategy
of lower-limb problems in JIA.
1. randomized controlled trial.pt.
Studies including children diagnosed as having 2. controlled clinical trial.pt.
JIA and one or more lower-limb problems were 3. randomized.ab.
eligible for inclusion. A lower-limb problem was 4. randomised.ab.
defined as any pathologic disorder of the lower 5. placebo.ab.
limb, extending from the trunk and including the 6. randomly.ab.
gluteal region, femoral region, knee, leg, ankle, and 7. trial.ab.
foot.12 Any setting, such as public and community 8. groups.ab.
9. 1 or 2 or 3 or 4 or 5 or 6 or 7 or 8
health services, private clinics, preschools, and
10. exp animals/not humans.sh.
schools, was included.
11. 9 not 10
12. Juvenile idiopathic arthritis
Outcomes 13. Juvenile chronic arthritis
14. Juvenile rheumatoid arthritis
The primary outcome was any validated quantifiable 15. JIA
measure of pain, for example, the Pediatric Pain 16. 12 or 13 or 14 or 15
Questionnaire.13 The secondary outcomes were 17. 11 and 16
disability, functional ability, or both; health-related Abbreviations: ab, abstract; JIA, juvenile idiopathic arthritis;
quality of life (HRQoL) (eg, the Pediatric Quality of pt, publication type; sh, subject heading.

400 September/October 2017  Vol 107  No 5  Journal of the American Podiatric Medical Association
second author (A.C.) checked all extracted data. If
there was any absent or uncertain information,
study authors were contacted. Inconsistencies in
data extraction were discussed between A.F. and
A.C. and, if needed, through arbitration by F.H. Risk
of bias of each included study was rated indepen-
dently by two of us (A.F. and A.C.) using the
following criteria described in the Cochrane Hand-
book for Systematic Reviews of Interventions15: 1)
sequence generation; 2) allocation of concealment;
3) blinding of participants, personnel, and outcome
assessor; 4) incomplete outcome data; 5) selective
outcome reporting; and 6) other sources of data. For
each criterion, high indicates a high risk of bias, low
indicates a low risk of bias, and unclear identifies
an ambiguous or unclear risk of bias.
Statistics
Measures of Treatment Effects. Group means
and SDs were analyzed to produce mean differences
and 95% confidence intervals (CIs). Standardized
mean difference analyses were to be conducted
when different measurement scales were used.
Different follow-up periods were to be pooled after
adjustment if steady rates of change could be
demonstrated.
Assessment of Heterogeneity. Different types
of interventions for JIA (eg, foot orthoses [FOs]
versus splints) and different types of lower-limb
problems (eg, foot pain versus gait instability) were
analyzed separately. Studies that are consistently
clinically homogenous in terms of outcomes,
participants, and interventions were pooled in a
meta-analysis. Intertrial statistical inconsistency
was quantified using I2, which was calculated by
the following formula15: I2 ¼ 100% [(Q – df)/Q],
where Q is Cochran’s heterogeneity, v2 statistic, and
df represents degrees of freedom. Cochran’s Q was
acquired by summing the squared deviations of each
trial’s approximation from the overall meta-analytic
estimate and a P value attained by comparing the
statistic with a v2 distribution with k – 1 degree of
freedom (where k is the number of trials). The
subsequent guidelines were used to interpret the I2
values: 0% to 40% might not be important, 30% to
60% may represent moderate heterogeneity, 50% to
90% may represent substantial heterogeneity, and
75% to 100% may represent considerable heteroge-
neity.15 A random-effects model to incorporate
heterogeneous trials in a meta-analysis was to be
used when there was heterogeneity that could not
be explained.
Journal of the American Podiatric Medical Association  Vol 107  No 5  September/October 2017
Data Synthesis. One of us (F.H.) constructed
data analyses in The Cochrane Collaboration’s
statistical package Review Manager 5.16 Another
one of us (A.F.) entered data, which were checked
by a third author (A.C.).
Subgroup Analyses. No subgroup analyses were
planned as different types of interventions and
different presentations of lower-limb pathologies
were being analyzed separately in the primary
analyses.
Sensitivity Analysis. Sensitivity analyses were
performed by eliminating trials that failed to blind
participants or conceal allocation. If outliers con-
tributed to heterogeneity and the reason for the
discrepancy was evident, analyses with and without
the outlying trials were to be performed.15
Results
Studies
After duplicates were removed, 3,670 studies were
retrieved by electronic searches. Screening of titles
and abstracts identified 29 potentially eligible
studies, for which full texts were retrieved.17-45
When a potentially eligible paper did not present
data specific to lower-limb problems, the corre-
sponding author was contacted via e-mail. Five
papers were excluded following author advice that
lower-limb–specific follow-up data were not avail-
able19,26-28 or that the trial was not an RCT.23 Three
additional papers were excluded because authors
failed to reply to at least two e-mail reminders sent
over a 1-month period.22,24,29 Of the 29 potentially
eligible studies, 27 were excluded (Fig. 1). Recur-
sive checking of reference lists of included studies
and e-mail correspondence with known researchers
in the field and corresponding authors of the
included studies did not disclose any additional
potentially eligible studies.
Both included studies were parallel-designed
RCTs20,21 and were published in peer-reviewed
journals within the past 12 years (Table 2).
Participants
A total of 100 participants were included from two
trials. Coda et al20 included 60 participants and
Powell et al21 included 40 participants. All of the
participants in the two included trials were children
diagnosed as having JIA according to the Interna-
tional League of Associations for Rheumatology
criteria. The mean 6 SD ages of participants in
Coda et al’s study was 10.916 3.68 years, and in
401
Figure 1. Preferred Reporting Items for Systematic Reviews and Meta-Analyses flowchart depicting the
number of studies identified by the search and listed as potentially eligible. Reasons for excluding potentially
eligible studies can be seen with a final count of studies included in the systematic review. RCT, randomized
controlled trial.

402 September/October 2017  Vol 107  No 5  Journal of the American Podiatric Medical Association
Table 2. Summarized Characteristics of the Two Included Studies
Characteristic Coda et al,20 2014
Lower-limb problem Pain in the foot and ankle
Intervention groups 1. Customized foot orthoses
2. Sham orthoses
Participants (No.) 60
Age range (years) 5–18
Recruitment source Two hospitals in Scotland
Follow-up (mo) 3 and 6
Outcomes 1. Pain: 100-mm VAS
2. HRQoL: PedsQL pediatric rheumatology scores
(published), Generic PedsQL scores, physical
functioning scale (unpublished)
Abbreviations: HRQoL, health-related quality of life; PedsQL, Pediatric Quality of Life Inventory; VAS, visual analog scale.
Powell et al’s it was 12.69 6 3.64 years. Overall, 75
of the participants were girls and 25 were boys. Of
the 100 children diagnosed as having JIA, 14 had
enthesitis-related JIA, 39 had polyarticular JIA, 39
had oligoarticular JIA, six had systemic JIA, none
had psoriatic arthritis, and two had undifferentiated
JIA.
Lower-Limb Problems
The lower-limb problem assessed in both trials was
foot and ankle pain resulting from active joint
disease.
Setting
Coda et al recruited children from a pediatric
rheumatology clinic and two hospitals in Scotland
between 2011 and 2012. Powell et al recruited from
pediatric rheumatology clinics via three southern
California children’s hospitals.
Types of Physical and Mechanical Therapies
Mechanical Therapies. Coda et al used a two-
arm RCT to compare customized prefabricated FOs
with sham orthoses. Powell et al used a three-arm
RCT in which all of the participants received
supportive footwear. Participants were randomized
to one of three groups: shoes alone, custom FOs and
shoes, or neoprene inserts and shoes.
Coda et al prescribed preformed semirigid FOs
(Slimflex Plus; Algeos, Liverpool, England) that
were customized on the day of the initial biome-
chanical assessment to accommodate the child’s
lower-limb functional need. Control group partici-
Journal of the American Podiatric Medical Association  Vol 107  No 5  September/October 2017
Powell et al,21 2005
Pain in the foot and ankle
1. Custom foot orthoses
2. Neoprene inserts
3. Supportive shoes alone
40
5–19
Three pediatric rheumatology clinics in southern
California
3
1. Pain: pediatric 0–10 VAS
2. Function: Foot Function Index
3. Ambulation speed: timed walking test
4. HRQoL: Generic PedsQL 4.0 scores, physical
functioning scale
pants were given sham FOs made from 1-mm
leather board without any modifications or correc-
tions. Both the FOs and the shams had a top cover
of black ethylene vinyl acetate of 0.75-mm thick-
ness.20 Coda et al gave instructions to gradually
wear the FOs in the first few days and then proceed
to wear them at all shod times, including when
exercising.
Powell et al evaluated custom-made FOs (Langer
Biomechanics Inc, Ronkonkoma, New York), in
which nonweightbearing casts were taken in a
subtalar joint neutral position. The FOs were
semirigid devices made from metal particle–rein-
forced polyolefin with additional shock-absorbing
functional posts. The study did not report details of
the technique of casting or the FO prescription. At
visit 1, Powell et al randomized participants to their
allocated interventions and casted participants who
were receiving custom FOs. Powell et al provided
the shoes and allocated interventions at visit 2
(baseline). The time between casting and fitting was
not specified. At the third and final visit, all follow-
up outcomes were assessed.
Physical Therapies. No physical therapies were
evaluated in either trial.
Risk of Bias
Risk of bias is summarized in Table 3.
Outcomes
Lower-extremity and foot pain was an outcome
assessed by both included trials. Coda et al used a
100-mm visual analog scale (VAS). Lower scores on
the Foot Function Index (FFI) (pain) and VAS
403
Table 3. Risk of Bias
Domain Coda et al,20 2014 Evidence Powell et al,21 2005 Evidence

Sequence generation Low risk ‘‘After obtaining informed
consent, children were
randomised in blocks of
10 each by an online
computer random
number generator
(http://www.randomiza
tion.com).
Allocation concealment Unclear No specific mention of
allocation concealment.
Insufficient information
to permit judgment of
high or low risk.
Blinding of participants Low risk ‘‘The control FO was
made with leather
board (1 mm) without
corrections. Both FOs
had the same black-
ethylene vinyl acetate
(EVA) top-cover (0.75
mm) to allow for
blinding and monitoring
the level of adherence
to wearing the FOs.’’
Blinding of personnel High risk Study did not blind
investigators to the
intervention, and the
outcome may be
influenced by lack of
blinding.
Blinding of outcome assessors Low risk Pain measured by
participants;
HRQoL measured by
participants and
parents/caregivers;
Participants and their
parents/caregivers are
blinded.
Low risk ‘‘Once accepted into the
study, each subject
was randomly placed
. . . ’’
Unclear ‘‘Sealed envelope
containing a
predetermined
numbered placement
card into one of the 3
intervention groups.’’ It
is not clear whether
envelopes are opaque
or consecutively placed
High risk No blinding, and the
outcome may be
influenced by lack of
blinding.
High risk Physical therapist 1, who
administered the
interventions, was not
blinded.
Physical therapist 2,
who performed
baseline and follow-up
measures, was blinded.
High risk Pain, level of disability,
and activity limitation
measured by
participants.
Speed of ambulation
influenced by participant
and measured by
personnel.
The outcome
measurement may be
influenced by lack of
blinding.

(pain) indicate lower-limb pain. Powell et al used
the foot pain domain of the FFI) and a pediatric
VAS, scored between 0 and 10. For meta-analysis, 0
to 10 VAS scores were multiplied by 10 to match the
100-mm VAS scores. The minimal important differ-
ence (MID) for the 100-mm VAS in children with
rheumatic disease is 8 mm.46
Function was assessed by Powell et al using the
activity limitations domain of the FFI and speed of
ambulation using the timed walking evaluation test.
Lower scores on the FFI (function) and for speed of

404 September/October 2017  Vol 107  No 5  Journal of the American Podiatric Medical Association
Table 3. extended
Domain
Incomplete outcome data
Selective outcome reporting
Other sources of bias
Coda et al,20 2014 Evidence Powell et al,21 2005
Low risk ‘‘Overall, 179 out of a
possible 180
assessments were
completed (99.4%) and
accounted for statistical
analysis.’’
Author: ‘‘the missing
data was related to a
control patient that was
no longer reachable
telephonically to
reschedule the 6-month
appointment’’
Unclear No reference to study
protocol.
Low risk FOs were provided as
part of a PhD
scholarship fund at
Queen Margaret
University.
Evidence
Low risk There were 8
withdrawals: 1 refused
to participate and 7
were lost to follow up.
There was no
relationship between
completing the trial and
intervention group (v2
¼ 0.76; P ¼ .682), and
no child withdrew
because of discomfort
or lack of efficacy.
Seven children did not
complete the study: 2
received FOs; 2
received flat neoprene
shoe inserts; and 3
were given new shoes
worn alone. They did
not differ from those
who completed the
study with respect to
parental educational
level, family income,
race/ethnicity, or child’s
age, sex, or type of
arthritis.
Unclear No reference to study
protocol.
Low risk Orthoses supplied by
companies, but authors
seem to be
independent of the
companies:
"Our thanks to Langer
Biomechanics Group,
Inc., for supplying all
custom-made orthotics,
and to the Spenco
Medical Corporation for
their generous supply
of prefabricated
neoprene shoe inserts."

Abbreviations: FO, foot orthoses; HRQoL, health-related quality of life.

ambulation indicate better function. Disability was
assessed by Powell et al using the disability domain
of the FFI. Lower scores on the FFI (disability)
indicate lower disability. The MID for the FFI has
not been established in children. The MIDs for FFI
domains for adults are 12 for pain, 7 for disability,
and 0.5 for activity limitation.47
The HRQoL was assessed in both trials using the
PedsQL questionnaire. Both trials assessed HRQoL
using the Generic PedsQL 4.0; however, Powell et al
used only the physical functioning scale of the
Generic PedsQL. Coda et al assessed HRQoL with
both the Generic PedsQL and the PedsQL rheuma-
tology questionnaire 3.0. The PedsQL rheumatology
score is designed to measure pediatric rheumatol-
ogy-specific HRQoL.48 Meta-analysis of HRQoL was
achieved by extrapolating only the physical func-
tioning scores of the Generic PedsQL from both
included studies. Both PedsQL questionnaires con-
tain child and parent reports subdivided according

Journal of the American Podiatric Medical Association  Vol 107  No 5  September/October 2017 405
to the child’s age range. A higher score on the
PedsQL is indicative of better HRQoL; the MID for
the PedsQL is 5 points.48 Neither trial reported
adverse effects or participant satisfaction with the
intervention.
Comparisons
Coda et al and Powell et al compared customized/
custom FOs with a control condition (sham
orthoses and no additional intervention, respective-
ly). Powell et al also compared 1) custom FOs with
flat neoprene inserts and 2) flat neoprene inserts
with no additional intervention (all of the children
in this trial received supportive shoes).
Effects of Interventions
Fifty-eight participants were allocated to receive a
trial mechanical intervention (either customized/
custom FOs or neoprene inserts) and 42 partici-
pants were allocated to receive a standardized or
sham therapy. Coda et al reported median and
interquartile range scores because data were non-
parametric, and Powell et al presented follow-up
data using mean 6 SD scores. To complete the
meta-analysis, Coda et al supplied the follow-up
mean 6 SD scores for all of the outcomes.
Data and Analysis
Figures 2 to 4 present all of the meta-analyses
performed. Because both studies concealed alloca-
tion, sensitivity analyses for meta-analyses were
performed by excluding data from the study21 that
did not blind participants to their intervention.
Results of sensitivity analyses for comparisons
between custom/customized FOs and a control
intervention after 3 months of intervention were
not significant for the outcomes of pain (mean
difference, –2.88; 95% CI, –15.70 to 9.94); child-rated
HRQoL (mean difference, 0.30; 95% CI, –9.66 to
Figure 2. Meta-analysis of the effectiveness of custom/customized foot orthoses versus a control (sham
orthoses or supportive footwear alone) in treating foot pain after 3 months in children with juvenile idiopathic
arthritis. CI, confidence interval; IV, inverse variance.
406 September/October 2017  Vol 107  No 5  Journal of the American Podiatric Medical Association
10.26); and parent-rated HRQoL (mean difference, –
2.76; 95% CI, –13.12 to 7.60). Table 4 presents
analyses each containing data from only one study.
Discussion
Findings and Implications
Pain. Customized/Custom FOs versus Control
Intervention. At 3-month follow-up, a meta-analysis
of both included trials found no significant differ-
ence between customized/custom FOs and a control
intervention (either supportive footwear or sham
orthoses). This was supported by a sensitivity
analysis at the 6-month time point. In all compar-
isons, however, the mean difference was larger than
the MID of 8 mm for the 100-mm VAS. This indicates
that customized/custom FOs may have a small,
clinically important effect, but at this time the
available evidence does not allow for a precise
estimate of effect to be made.
Custom FOs versus Neoprene Inserts. Similarly,
the difference between customized/custom FOs and
neoprene inserts after 3 months was not statistically
significant but may have clinical importance.
Foot Function Index. Custom FOs versus
Control Intervention. Between-group differences in
means for all of the domains after 3 months were
significant and clinically important in favor of
custom FOs.
Neoprene Inserts versus Control Intervention.
Similarly, between-group differences in means for
all of the domains after 3 months were statistically
significantly in favor of neoprene inserts, but
differences were too small to be clinically impor-
tant, except for activity limitation, which holds
potential clinical importance in favor of neoprene
inserts.
Custom FOs versus Neoprene Inserts. Between-
group differences in means for all of the domains
after 3 months were not statistically significant, but
Figure 3. Meta-analysis of the effectiveness of custom/customized foot orthoses versus a control (sham
orthoses or supportive footwear alone) in improving health-related quality of life in children with juvenile
idiopathic arthritis. This figure represents the child-rated scores of the physical functioning subscale of the
Pediatric Quality of Life Inventory. CI, confidence interval; IV, inverse variance.

the effect size was clinically important in favor of
custom FOs.
Caution should be taken when applying these
findings because the FFI has not been validated in
children with rheumatic disease.
Generic PedsQL Physical Functioning Do-
main. Customized/Custom FOs versus Control
Intervention: At 3-month follow-up, a meta-analysis
of both included trials found no significant differ-
ence between customized/custom FOs and a control
intervention (either supportive footwear or sham
orthoses) for child- or parent-rated physical func-
tioning as measured with the PedsQL. This was
supported by a sensitivity analysis at the 6-month
time point. In addition, mean differences for both
parent and child scores did not reach a MID of 5
points after 3 months. A small clinically important
mean difference in child-rated HRQoL after 6
months is noted.
Neoprene Inserts versus Control Intervention.
Between-group differences in means after 3 months
were not significant.
Custom FOs versus Neoprene Inserts. Between-
group differences in means after 3 months were
significant for child-rated HRQoL in favor of custom
FOs. For parent-rated HRQoL, difference in means
at 3 months was clinically important but not
statistically significant.
PedsQL Pediatric Rheumatology Question-
naire. Customized FOs versus Sham Orthoses.
Between-group differences in means after 3 and 6
months were not statistically significant, but at both
time points, mean differences were clinically
important for child-rated HRQoL in favor of
customized FOs.
Timed Walking. At 3-month follow-up, mean
walking speed was significantly faster in children
with custom FOs than in those with neoprene
inserts. The MID is unknown, and it is possible that
the difference is too small to be clinically important.
No significant differences in walking speed were
found between children with 1) custom FOs and
shoes versus shoes alone or 2) neoprene inserts and
shoes versus shoes alone.
Summary of Comparisons
This review detected many between-group differ-
ences in means that are not statistically significant
but that are larger than the clinically important
difference for the scales used. Because the

Figure 4. Meta-analysis of the effectiveness of custom/customized foot orthoses versus a control (sham
orthoses or supportive footwear alone) in improving health-related quality of life in children with juvenile
idiopathic arthritis. This figure represents the parent-rated scores of the physical functioning subscale of the
Pediatric Quality of Life Inventory. CI, confidence interval; IV, inverse variance.

Journal of the American Podiatric Medical Association  Vol 107  No 5  September/October 2017 407
408
Table 4. Analyses Containing Data from One Study: Coda et al,20 2014, and Powell et al,21 2005
Effect Estimate Mean
Outcome and Follow-up Period Study Group (No.) Mean 6 SD Group (No.) Mean 6 SD Difference (95% CI)

Foot pain 6 mo Coda Customized orthoses (31) 11.38 6 15.37 Sham insole (29) 21.97 6 27.59 –10.59 (–22.00 to 0.82)
FFI activity limitation 3 mo Powell Custom orthoses and shoes (15) 8.54 6 11.06 Shoes (13) 27.92 6 27.89 –19.38 (–35.54 to –3.22)
FFI foot pain 3 mo Powell Custom orthoses and shoes (15) 18.35 6 17.05 Shoes (13) 37.54 6 25.47 –19.19 (–35.50 to –2.88)
FFI disability 3 mo Powell Custom orthoses and shoes (15) 15.6 6 13.51 Shoes (13) 34.15 6 26.35 –18.55 (–34.42 to –2.68)
Timed walking 3 mo Powell Custom orthoses and shoes (15) 7.03 6 1.12 Shoes (13) 8.36 6 2.44 –1.33 (–2.77 to 0.11)
PedsQL (PF) child 6 mo Coda Customized orthoses (31) 83.46 6 16.61 Sham insole (29) 77.9 6 26.1 5.56 (–5.59 to 16.71)
PedsQL (PF) parent 6 mo Coda Customized orthoses (31) 78.78 6 20.94 Sham insole (29) 76.79 6 24.65 1.99 (–9.62 to 13.60)
PedsQL (RS) child 3 mo Coda Customized orthoses (31) 80.58 6 13.83 Sham insole (29) 74.66 6 22.41 5.92 (–3.58 to 15.42)
PedsQL (RS) parent 3 mo Coda Customized orthoses (31) 76.37 6 17.27 Sham insole (29) 75.77 6 22.72 0.60 (–9.66 to 10.86)
PedsQL (RS) child 6 mo Coda Customized orthoses (31) 85.73 6 12.26 Sham insole (29) 77.48 6 21.22 8.25 (–0.60 to 17.10)
PedsQL (RS) parent 6 mo Coda Customized orthoses (31) 80.14 6 15.35 Sham insole (29) 76.7 6 23.36 3.44 (–6.63 to 13.51)
Foot pain 3 mo Powell Neoprene inserts and shoes (12) 28.4 6 28.8 Shoes (13) 28.2 6 20.1 0.20 (–19.42 to 19.82)
FFI activity limitation 3 mo Powell Neoprene inserts and shoes (12) 19.96 6 19.73 Shoes (13) 27.92 6 27.89 –7.96 (–26.79 to 10.87)
FFI foot pain 3 mo Powell Neoprene inserts and shoes (12) 30.46 6 25.56 Shoes (13) 37.54 6 25.47 –7.08 (–27.10 to 12.94)
FFI disability 3 mo Powell Neoprene inserts and shoes (12) 29.98 6 25.26 Shoes (13) 34.15 6 26.35 –4.17 (–24.40 to 16.06)
Timed walking 3 mo Powell Neoprene inserts and shoes (12) 7.98 6 1.3 Shoes (13) 8.36 6 2.44 –0.38 (–1.90 to 1.14)
PedsQL (PF) child 3 mo Powell Neoprene inserts and shoes (10) 55.94 6 17.46 Shoes (12) 59.78 6 18.8 –3.84 (–19.01 to 11.33)
PedsQL (PF) parent 3 mo Powell Neoprene inserts and shoes (10) 55.31 6 15.8 Shoes (12) 55.95 6 13.97 –0.64 (–13.22 to 11.94)
Foot pain 3 mo Powell Custom orthoses and shoes (15) 13.2 6 13 Neoprene insert and shoes (12) 28.4 6 28.8 –15.20 (–32.77 to 2.37)
FFI activity limitation 3 mo Powell Custom orthoses and shoes (15) 8.54 6 11.06 Neoprene insert and shoes (12) 19.96 6 19.73 –11.42 (–23.91 to 1.07)
FFI foot pain 3 mo Powell Custom orthoses and shoes (15) 18.35 6 17.05 Neoprene insert and shoes (12) 30.46 6 25.56 –12.11 (–28.95 to 4.73)
FFI disability 3 mo Powell Custom orthoses and shoes (15) 15.6 6 13.51 Neoprene insert and shoes (12) 29.98 6 25.26 –14.38 (–30.22 to 1.46)
Timed walking 3 mo Powell Custom orthoses and shoes (15) 7.03 6 1.12 Neoprene insert and shoes (12) 7.98 6 1.3 –0.95 (–1.88 to –0.02)
PedsQL (PF) child 3 mo Powell Custom orthoses and shoes (13) 71.88 6 15.88 Neoprene insert and shoes (10) 55.94 6 17.46 15.94 (2.10 to 29.78)
PedsQL (PF) parent 3 mo Powell Custom orthoses and shoes (14) 64.96 6 19.92 Neoprene insert and shoes (12) 55.31 6 15.8 9.65 (–4.09 to 23.39)

Note: Lower scores on an outcome measure represent a better result, except for PedsQL.
Abbreviations: CI, confidence interval; FFI, Foot Function Index; PedsQL, Pediatric Quality of Life Inventory; PF, physical functioning; RS, rheumatology score.

September/October 2017  Vol 107  No 5  Journal of the American Podiatric Medical Association
estimate of effect is imprecise (with broad CIs), no
final judgment can be made regarding whether
potential clinically important differences between
interventions exist. Table 5 summarizes outcome
significance for customized/custom FOs versus
control or alternative interventions.
Limitations
This systematic review is limited to RCTs and quasi-
RCTs of physical and mechanical therapies for
lower-limb problems in children with JIA and to
only journals indexed in the databases searched or
known by the researchers we contacted.
Limitations of the Included Trials
Only two RCTs were included in this systematic
review, in which only one lower-limb problem (foot
and ankle pain) was explored. In addition, both
included studies evaluated the same mechanical
intervention (FOs).
Powell et al had a small sample size. It is unclear
whether power calculations were performed. Com-
pounding the issue of the small sample, the attrition
rate was high (eight of 48 patients). This may have
affected the ability of analyses to detect significant
and clinically important effects. In addition, be-
cause nonparametric data from Coda et al could not
be pooled with data from Powell et al in Review
Table 5. Summary of Outcome Significance for Customized/Custom Foot Orthoses versus Control or Alternative
Interventions
Outcome and Time Point
Customized/custom foot orthoses versus control interventions
Pain
3 mo
6 mo
HRQoL: Generic PedsQL 4.0
3 mo
6 mo
HRQoL: PedsQL 3.0
3 mo
6 mo
Foot Function Index at 3 months
Custom foot orthoses versus neoprene inserts
Pain at 3 mo
HRQoL: Generic PedsQL 4.0 at 3 mo
Foot Function Index at 3 mo
Abbreviations: HRQoL, health-related quality of life; PedsQL, Pediatric Quality of Life Inventory.
a
None represents no statistical significance or clinical importance.
b
Possible represents possible or incomplete statistical significance or clinical importance.
Journal of the American Podiatric Medical Association  Vol 107  No 5  September/October 2017
Manager, parametric data were obtained and used
in the analyses. This may have contributed to broad
CIs.
Powell et al assessed HRQoL with a generic
pediatric quality of life questionnaire, which is not
specific to pediatric rheumatology. The PedsQL
pediatric rheumatology 3.0 questionnaire was
already available at the time of the study, which
may have better reflected the HRQoL of this
specific pediatric population. Custom FOs, as
evaluated by Powell et al, require a manufacture
period, which delays provision of the intervention,
sometimes by weeks. Where research evidence is
inconclusive and when no clear clinical benefit
exists for prescribing custom-made over custom-
ized prefabricated devices, clinicians with their
patients may select to use prefabricated devices to
achieve earlier intervention, which is the gold
standard in pediatric rheumatology.49,50 Clinicians
may also consider the cost of the interventions. At
the time of the study, the cost of the customized
(prefabricated) FOs used by Coda et al was
estimated to be £10 to £15 (A$21–A$32, October
2015 currency rate); and the cost of custom-made
FOs used by Powell et al were $200 to $350 per pair
(approximately A$275–A$481, October 2015 cur-
rency rate). Although customized prefabricated
orthoses are less expensive than custom-made
devices, both are considerably cheaper compared
with drug therapy for JIA.
Statistically Significant Clinically Important
nonea possibleb
none possible
none none
none possible
none possible
none possible
possible possible
none possible
none possible
none possible
409
Recommendations for Clinical Practice
Until more conclusive research of the effectiveness
of FOs is available, health professionals may use
FOs with care when managing foot and ankle pain
in children with JIA. Health professionals should
consider FOs as part of a holistic therapeutic
approach and consider the potential risks and
benefits of alternative interventions.
Recommendations for Future Research
At present, only two RCTs of the effectiveness of
mechanical therapies for lower-limb problems in
JIA were found.20,21 We did not find an RCT that
evaluates the effectiveness of physical therapies for
lower-limb problems in children with JIA. Physical
therapies that may be effective for specific lower-
limb problems in JIA are massage, specific muscle
and stretching exercise programs, and hydrothera-
py. Methodologically rigorous RCTs of long dura-
tion with adequate sample sizes determined by a
priori power calculations are required.
Conclusions
The effectiveness of FOs for foot and ankle pain in
children with JIA is unclear. Existing research
demonstrates inconsistent and nonsignificant differ-
ences that potentially hold clinical benefit. Current-
ly, there are no RCTs that evaluate the effectiveness
of physical therapies for lower-limb problems in
children with JIA, and evaluation of mechanical
therapies is limited to FOs and shoe inserts for foot
and ankle pain. Future research should focus on
methodologically rigorous RCTs that explore phys-
ical and mechanical therapies for a range of specific
lower-limb problems in JIA.
Financial Disclosure: None reported.
Conflict of Interest: Dr. Coda is an author of a
trial included in this review.
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