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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 tology identifies seven subgroups of JIA.3 Active JIA
common form of arthritis in children and adoles- of any type may cause premature epiphyseal closure
cents.1 Research in developed countries reports and subsequent local growth defects, typically of
prevalence rates from 16 to 150 per 100,000.1 the knee.4 Children with JIA may also present with
Typical signs and symptoms of active JIA are enthesitis at the plantar fascia or Achilles tendon,
swelling, inflammation, pain, and stiffness, most flexion contractures, synovitis, and muscle atro-
commonly in hip, knee, and ankle joints and in small phy.1
joints in the hands and feet.1 If uncontrolled, JIA Juvenile idiopathic arthritis has a substantial
can cause joint damage and fixed deformities.2 The financial burden on the families of affected children
International League of Associations for Rheuma- and the health-care system.5-7 In 2013 to 2014, the
*School of Health Sciences, Faculty of Health and Australian government subsidized more than A$7.5
Medicine, University of Newcastle, Ourimbah, Australia. million for biological drug agents alone for children
Corresponding author: Antoni Fellas BHSc(Hons), School
with JIA.8 Also, JIA has a psychological effect on
of Health Sciences, Faculty of Health and Medicine,
University of Newcastle, Central Coast Campus, Ourimbah, affected children, with increasing disability being
NSW 2258, Australia. (E-mail: Antoni.Fellas@uon.edu.au) 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 Data Synthesis. One of us (F.H.) constructed
there was any absent or uncertain information, data analyses in The Cochrane Collaboration’s
study authors were contacted. Inconsistencies in statistical package Review Manager 5.16 Another
data extraction were discussed between A.F. and one of us (A.F.) entered data, which were checked
A.C. and, if needed, through arbitration by F.H. Risk by a third author (A.C.).
of bias of each included study was rated indepen- Subgroup Analyses. No subgroup analyses were
dently by two of us (A.F. and A.C.) using the planned as different types of interventions and
following criteria described in the Cochrane Hand- different presentations of lower-limb pathologies
book for Systematic Reviews of Interventions15: 1) were being analyzed separately in the primary
sequence generation; 2) allocation of concealment; analyses.
3) blinding of participants, personnel, and outcome Sensitivity Analysis. Sensitivity analyses were
assessor; 4) incomplete outcome data; 5) selective performed by eliminating trials that failed to blind
outcome reporting; and 6) other sources of data. For participants or conceal allocation. If outliers con-
each criterion, high indicates a high risk of bias, low tributed to heterogeneity and the reason for the
indicates a low risk of bias, and unclear identifies discrepancy was evident, analyses with and without
an ambiguous or unclear risk of bias. the outlying trials were to be performed.15

Statistics Results

Measures of Treatment Effects. Group means Studies


and SDs were analyzed to produce mean differences
and 95% confidence intervals (CIs). Standardized After duplicates were removed, 3,670 studies were
mean difference analyses were to be conducted retrieved by electronic searches. Screening of titles
when different measurement scales were used. and abstracts identified 29 potentially eligible
Different follow-up periods were to be pooled after studies, for which full texts were retrieved.17-45
adjustment if steady rates of change could be When a potentially eligible paper did not present
demonstrated. data specific to lower-limb problems, the corre-
sponding author was contacted via e-mail. Five
Assessment of Heterogeneity. Different types
papers were excluded following author advice that
of interventions for JIA (eg, foot orthoses [FOs]
lower-limb–specific follow-up data were not avail-
versus splints) and different types of lower-limb
able19,26-28 or that the trial was not an RCT.23 Three
problems (eg, foot pain versus gait instability) were
additional papers were excluded because authors
analyzed separately. Studies that are consistently
failed to reply to at least two e-mail reminders sent
clinically homogenous in terms of outcomes,
over a 1-month period.22,24,29 Of the 29 potentially
participants, and interventions were pooled in a
eligible studies, 27 were excluded (Fig. 1). Recur-
meta-analysis. Intertrial statistical inconsistency sive checking of reference lists of included studies
was quantified using I2, which was calculated by and e-mail correspondence with known researchers
the following formula15: I2 ¼ 100% [(Q – df)/Q], in the field and corresponding authors of the
where Q is Cochran’s heterogeneity, v2 statistic, and included studies did not disclose any additional
df represents degrees of freedom. Cochran’s Q was potentially eligible studies.
acquired by summing the squared deviations of each Both included studies were parallel-designed
trial’s approximation from the overall meta-analytic RCTs20,21 and were published in peer-reviewed
estimate and a P value attained by comparing the journals within the past 12 years (Table 2).
statistic with a v2 distribution with k – 1 degree of
freedom (where k is the number of trials). The Participants
subsequent guidelines were used to interpret the I2
values: 0% to 40% might not be important, 30% to A total of 100 participants were included from two
60% may represent moderate heterogeneity, 50% to trials. Coda et al20 included 60 participants and
90% may represent substantial heterogeneity, and Powell et al21 included 40 participants. All of the
75% to 100% may represent considerable heteroge- participants in the two included trials were children
neity.15 A random-effects model to incorporate diagnosed as having JIA according to the Interna-
heterogeneous trials in a meta-analysis was to be tional League of Associations for Rheumatology
used when there was heterogeneity that could not criteria. The mean 6 SD ages of participants in
be explained. Coda et al’s study was 10.916 3.68 years, and in

Journal of the American Podiatric Medical Association  Vol 107  No 5  September/October 2017 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 Powell et al,21 2005

Lower-limb problem Pain in the foot and ankle Pain in the foot and ankle
Intervention groups 1. Customized foot orthoses 1. Custom foot orthoses
2. Sham orthoses 2. Neoprene inserts
3. Supportive shoes alone
Participants (No.) 60 40
Age range (years) 5–18 5–19
Recruitment source Two hospitals in Scotland Three pediatric rheumatology clinics in southern
California
Follow-up (mo) 3 and 6 3
Outcomes 1. Pain: 100-mm VAS 1. Pain: pediatric 0–10 VAS
2. HRQoL: PedsQL pediatric rheumatology scores 2. Function: Foot Function Index
(published), Generic PedsQL scores, physical 3. Ambulation speed: timed walking test
functioning scale (unpublished) 4. HRQoL: Generic PedsQL 4.0 scores, physical
functioning scale

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 pants were given sham FOs made from 1-mm
of the participants were girls and 25 were boys. Of leather board without any modifications or correc-
the 100 children diagnosed as having JIA, 14 had tions. Both the FOs and the shams had a top cover
enthesitis-related JIA, 39 had polyarticular JIA, 39 of black ethylene vinyl acetate of 0.75-mm thick-
had oligoarticular JIA, six had systemic JIA, none ness.20 Coda et al gave instructions to gradually
had psoriatic arthritis, and two had undifferentiated wear the FOs in the first few days and then proceed
JIA. to wear them at all shod times, including when
exercising.
Lower-Limb Problems Powell et al evaluated custom-made FOs (Langer
Biomechanics Inc, Ronkonkoma, New York), in
The lower-limb problem assessed in both trials was which nonweightbearing casts were taken in a
foot and ankle pain resulting from active joint subtalar joint neutral position. The FOs were
disease. semirigid devices made from metal particle–rein-
forced polyolefin with additional shock-absorbing
Setting functional posts. The study did not report details of
the technique of casting or the FO prescription. At
Coda et al recruited children from a pediatric visit 1, Powell et al randomized participants to their
rheumatology clinic and two hospitals in Scotland allocated interventions and casted participants who
between 2011 and 2012. Powell et al recruited from were receiving custom FOs. Powell et al provided
pediatric rheumatology clinics via three southern the shoes and allocated interventions at visit 2
California children’s hospitals. (baseline). The time between casting and fitting was
not specified. At the third and final visit, all follow-
Types of Physical and Mechanical Therapies up outcomes were assessed.
Physical Therapies. No physical therapies were
Mechanical Therapies. Coda et al used a two- evaluated in either trial.
arm RCT to compare customized prefabricated FOs
with sham orthoses. Powell et al used a three-arm Risk of Bias
RCT in which all of the participants received
supportive footwear. Participants were randomized Risk of bias is summarized in Table 3.
to one of three groups: shoes alone, custom FOs and
shoes, or neoprene inserts and shoes. Outcomes
Coda et al prescribed preformed semirigid FOs
(Slimflex Plus; Algeos, Liverpool, England) that Lower-extremity and foot pain was an outcome
were customized on the day of the initial biome- assessed by both included trials. Coda et al used a
chanical assessment to accommodate the child’s 100-mm visual analog scale (VAS). Lower scores on
lower-limb functional need. Control group partici- the Foot Function Index (FFI) (pain) and VAS

Journal of the American Podiatric Medical Association  Vol 107  No 5  September/October 2017 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 Low risk ‘‘Once accepted into the
consent, children were study, each subject
randomised in blocks of was randomly placed
10 each by an online . . . ’’
computer random
number generator
(http://www.randomiza
tion.com).
Allocation concealment Unclear No specific mention of Unclear ‘‘Sealed envelope
allocation concealment. containing a
Insufficient information predetermined
to permit judgment of numbered placement
high or low risk. card into one of the 3
intervention groups.’’ It
is not clear whether
envelopes are opaque
or consecutively placed
Blinding of participants Low risk ‘‘The control FO was High risk No blinding, and the
made with leather outcome may be
board (1 mm) without influenced by lack of
corrections. Both FOs blinding.
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 High risk Physical therapist 1, who
investigators to the administered the
intervention, and the interventions, was not
outcome may be blinded.
influenced by lack of Physical therapist 2,
blinding. who performed
baseline and follow-up
measures, was blinded.
Blinding of outcome assessors Low risk Pain measured by High risk Pain, level of disability,
participants; and activity limitation
HRQoL measured by measured by
participants and participants.
parents/caregivers; Speed of ambulation
Participants and their influenced by participant
parents/caregivers are and measured by
blinded. personnel.
The outcome
measurement may be
influenced by lack of
blinding.

(pain) indicate lower-limb pain. Powell et al used ence (MID) for the 100-mm VAS in children with
the foot pain domain of the FFI) and a pediatric rheumatic disease is 8 mm.46
Function was assessed by Powell et al using the
VAS, scored between 0 and 10. For meta-analysis, 0
activity limitations domain of the FFI and speed of
to 10 VAS scores were multiplied by 10 to match the ambulation using the timed walking evaluation test.
100-mm VAS scores. The minimal important differ- 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 Coda et al,20 2014 Evidence Powell et al,21 2005 Evidence
Incomplete outcome data Low risk ‘‘Overall, 179 out of a Low risk There were 8
possible 180 withdrawals: 1 refused
assessments were to participate and 7
completed (99.4%) and were lost to follow up.
accounted for statistical There was no
analysis.’’ relationship between
Author: ‘‘the missing completing the trial and
data was related to a intervention group (v2
control patient that was ¼ 0.76; P ¼ .682), and
no longer reachable no child withdrew
telephonically to because of discomfort
reschedule the 6-month or lack of efficacy.
appointment’’ 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.
Selective outcome reporting Unclear No reference to study Unclear No reference to study
protocol. protocol.
Other sources of bias Low risk FOs were provided as Low risk Orthoses supplied by
part of a PhD companies, but authors
scholarship fund at seem to be
Queen Margaret independent of the
University. 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 used only the physical functioning scale of the
assessed by Powell et al using the disability domain Generic PedsQL. Coda et al assessed HRQoL with
of the FFI. Lower scores on the FFI (disability) both the Generic PedsQL and the PedsQL rheuma-
indicate lower disability. The MID for the FFI has tology questionnaire 3.0. The PedsQL rheumatology
not been established in children. The MIDs for FFI score is designed to measure pediatric rheumatol-
domains for adults are 12 for pain, 7 for disability, ogy-specific HRQoL.48 Meta-analysis of HRQoL was
and 0.5 for activity limitation.47 achieved by extrapolating only the physical func-
The HRQoL was assessed in both trials using the tioning scores of the Generic PedsQL from both
PedsQL questionnaire. Both trials assessed HRQoL included studies. Both PedsQL questionnaires con-
using the Generic PedsQL 4.0; however, Powell et al 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 10.26); and parent-rated HRQoL (mean difference, –
PedsQL is indicative of better HRQoL; the MID for 2.76; 95% CI, –13.12 to 7.60). Table 4 presents
the PedsQL is 5 points.48 Neither trial reported analyses each containing data from only one study.
adverse effects or participant satisfaction with the
intervention. Discussion
Comparisons Findings and Implications

Coda et al and Powell et al compared customized/ Pain. Customized/Custom FOs versus Control
custom FOs with a control condition (sham Intervention. At 3-month follow-up, a meta-analysis
orthoses and no additional intervention, respective- of both included trials found no significant differ-
ly). Powell et al also compared 1) custom FOs with ence between customized/custom FOs and a control
flat neoprene inserts and 2) flat neoprene inserts intervention (either supportive footwear or sham
with no additional intervention (all of the children orthoses). This was supported by a sensitivity
in this trial received supportive shoes). analysis at the 6-month time point. In all compar-
isons, however, the mean difference was larger than
Effects of Interventions the MID of 8 mm for the 100-mm VAS. This indicates
that customized/custom FOs may have a small,
Fifty-eight participants were allocated to receive a clinically important effect, but at this time the
trial mechanical intervention (either customized/ available evidence does not allow for a precise
custom FOs or neoprene inserts) and 42 partici- estimate of effect to be made.
pants were allocated to receive a standardized or Custom FOs versus Neoprene Inserts. Similarly,
sham therapy. Coda et al reported median and the difference between customized/custom FOs and
interquartile range scores because data were non- neoprene inserts after 3 months was not statistically
parametric, and Powell et al presented follow-up significant but may have clinical importance.
data using mean 6 SD scores. To complete the
meta-analysis, Coda et al supplied the follow-up Foot Function Index. Custom FOs versus
mean 6 SD scores for all of the outcomes. Control Intervention. Between-group differences in
means for all of the domains after 3 months were
Data and Analysis significant and clinically important in favor of
custom FOs.
Figures 2 to 4 present all of the meta-analyses Neoprene Inserts versus Control Intervention.
performed. Because both studies concealed alloca- Similarly, between-group differences in means for
tion, sensitivity analyses for meta-analyses were all of the domains after 3 months were statistically
performed by excluding data from the study21 that significantly in favor of neoprene inserts, but
did not blind participants to their intervention. differences were too small to be clinically impor-
Results of sensitivity analyses for comparisons tant, except for activity limitation, which holds
between custom/customized FOs and a control potential clinical importance in favor of neoprene
intervention after 3 months of intervention were inserts.
not significant for the outcomes of pain (mean Custom FOs versus Neoprene Inserts. Between-
difference, –2.88; 95% CI, –15.70 to 9.94); child-rated group differences in means for all of the domains
HRQoL (mean difference, 0.30; 95% CI, –9.66 to after 3 months were not statistically significant, but

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
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 at 3 months was clinically important but not
custom FOs. statistically significant.
Caution should be taken when applying these
findings because the FFI has not been validated in PedsQL Pediatric Rheumatology Question-
children with rheumatic disease. naire. Customized FOs versus Sham Orthoses.
Between-group differences in means after 3 and 6
Generic PedsQL Physical Functioning Do- months were not statistically significant, but at both
main. Customized/Custom FOs versus Control time points, mean differences were clinically
Intervention: At 3-month follow-up, a meta-analysis important for child-rated HRQoL in favor of
of both included trials found no significant differ- customized FOs.
ence between customized/custom FOs and a control
intervention (either supportive footwear or sham Timed Walking. At 3-month follow-up, mean
orthoses) for child- or parent-rated physical func- walking speed was significantly faster in children
tioning as measured with the PedsQL. This was with custom FOs than in those with neoprene
supported by a sensitivity analysis at the 6-month inserts. The MID is unknown, and it is possible that
time point. In addition, mean differences for both the difference is too small to be clinically important.
parent and child scores did not reach a MID of 5 No significant differences in walking speed were
points after 3 months. A small clinically important found between children with 1) custom FOs and
mean difference in child-rated HRQoL after 6 shoes versus shoes alone or 2) neoprene inserts and
months is noted. shoes versus shoes alone.
Neoprene Inserts versus Control Intervention.
Between-group differences in means after 3 months Summary of Comparisons
were not significant.
Custom FOs versus Neoprene Inserts. Between- This review detected many between-group differ-
group differences in means after 3 months were ences in means that are not statistically significant
significant for child-rated HRQoL in favor of custom but that are larger than the clinically important
FOs. For parent-rated HRQoL, difference in means 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 Manager, parametric data were obtained and used
final judgment can be made regarding whether in the analyses. This may have contributed to broad
potential clinically important differences between CIs.
interventions exist. Table 5 summarizes outcome Powell et al assessed HRQoL with a generic
significance for customized/custom FOs versus pediatric quality of life questionnaire, which is not
control or alternative interventions. specific to pediatric rheumatology. The PedsQL
pediatric rheumatology 3.0 questionnaire was
Limitations already available at the time of the study, which
may have better reflected the HRQoL of this
This systematic review is limited to RCTs and quasi- specific pediatric population. Custom FOs, as
RCTs of physical and mechanical therapies for evaluated by Powell et al, require a manufacture
lower-limb problems in children with JIA and to period, which delays provision of the intervention,
only journals indexed in the databases searched or sometimes by weeks. Where research evidence is
known by the researchers we contacted. inconclusive and when no clear clinical benefit
exists for prescribing custom-made over custom-
Limitations of the Included Trials ized prefabricated devices, clinicians with their
patients may select to use prefabricated devices to
Only two RCTs were included in this systematic achieve earlier intervention, which is the gold
review, in which only one lower-limb problem (foot standard in pediatric rheumatology.49,50 Clinicians
and ankle pain) was explored. In addition, both may also consider the cost of the interventions. At
included studies evaluated the same mechanical the time of the study, the cost of the customized
intervention (FOs). (prefabricated) FOs used by Coda et al was
Powell et al had a small sample size. It is unclear estimated to be £10 to £15 (A$21–A$32, October
whether power calculations were performed. Com- 2015 currency rate); and the cost of custom-made
pounding the issue of the small sample, the attrition FOs used by Powell et al were $200 to $350 per pair
rate was high (eight of 48 patients). This may have (approximately A$275–A$481, October 2015 cur-
affected the ability of analyses to detect significant rency rate). Although customized prefabricated
and clinically important effects. In addition, be- orthoses are less expensive than custom-made
cause nonparametric data from Coda et al could not devices, both are considerably cheaper compared
be pooled with data from Powell et al in Review with drug therapy for JIA.

Table 5. Summary of Outcome Significance for Customized/Custom Foot Orthoses versus Control or Alternative
Interventions
Outcome and Time Point Statistically Significant Clinically Important

Customized/custom foot orthoses versus control interventions


Pain
3 mo nonea possibleb
6 mo none possible
HRQoL: Generic PedsQL 4.0
3 mo none none
6 mo none possible
HRQoL: PedsQL 3.0
3 mo none possible
6 mo none possible
Foot Function Index at 3 months possible possible
Custom foot orthoses versus neoprene inserts
Pain at 3 mo none possible
HRQoL: Generic PedsQL 4.0 at 3 mo none possible
Foot Function Index at 3 mo none possible

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 409
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