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S Y S T E M A T I C R E V I E W

Effectiveness of Stretch
Interventions for Children With
Neuromuscular Disabilities:
Evidence-Based Recommendations
Jason Craig, PT, MPT; Courtney Hilderman, PT, MSc; Geoffrey Wilson, PT, MPT; Robyn Misovic, PT, MScPT
Department of Physical Therapy, Queen Alexandra Centre for Children’s Health, Island Health, Victoria, British
Columbia, Canada (Mr Craig, Ms Misovic, and Mr Wilson); and Department of Physical Therapy, BC Centre for Ability,
and University of British Columbia, Vancouver, Canada (Ms Hilderman).

Purpose: To determine whether casting, orthoses, stretching, or supported standing programs are effective in
improving or maintaining body functions and structures, activity, or participation in children with neuromus-
cular disabilities. Methods: A systematic review was conducted using 6 electronic databases to identify Level
1 and 2 studies investigating stretch interventions for children aged 0 to 19 years with neuromuscular dis-
abilities. Interventions were coded using the International Classification of Function and rated with Grading
of Recommendation Assessment, Development and Evaluation, the Oxford Levels of Evidence, and the Evi-
dence Alert Traffic Light System. Results: Sixteen studies evaluated the effectiveness of stretch interventions.
Low-grade evidence supports casting temporarily increasing ankle range of motion, orthoses improving
gait parameters while they are worn, and supported standing programs improving bone mineral density.
Conclusion: There is limited evidence suggesting stretch interventions benefit body functions and structures.
There is inconclusive evidence to support or refute stretching interventions for preventing contractures or
impacting a child’s activity or participation. Trial Registration: Prospero CRD42014013807. (Pediatr Phys Ther
2016;28:262–275) Key words: activities and participation, bone mineral density, casting, children and youth,
contractures, gait, neuromuscular disabilities orthoses, positioning, quality of life, range of motion, stretching,
supported standing programs, systematic review

INTRODUCTION AND PURPOSE (CP), muscular dystrophies, and neural tube defects.
Contractures, hip pathologies, and spinal To address complications and promote independence in
malalignments1-3 are common complications for children these children, considerable therapeutic resources are
with neuromuscular disabilities, including cerebral palsy used such as orthoses, therapy equipment, and therapy
time.4-7 Therapists frequently prescribe and encourage
compliance to a variety of stretch interventions including
0898-5669/283-0262 (1) active stretching, (2) passive stretching, (3) prolonged
Pediatric Physical Therapy
Copyright C 2016 Wolters Kluwer Health, Inc. and Academy of
positioning through supported standing, or (4) prolonged
Pediatric Physical Therapy of the American Physical Therapy stretching through casting and orthoses.8,9 The clinical
Association rationale for using these interventions is to avoid or defer
surgery, decrease complications such as contractures, and
Correspondence: Jason Craig, PT, MPT, Queen Alexandra Centre for
Children’s Health, 2400 Arbutus Rd, Victoria, BC V8N 1V7, Canada
promote function.8,9 Proposed causes of contractures
([email protected]). that have been hypothesized include agonist-antagonist
Supplemental digital content is available for this article. Direct URL muscle imbalance, muscle fiber atrophy, spasticity,
citation appears in the printed text and is provided in the HTML and PDF static positioning, and structural changes to muscle
versions of this article on the journal’s Web site (www.pedpt.com).
tendon tissue (eg, the reduction of in-series or in-parallel
The authors declare no conflict of interest. No grant support was provided
for this research.
sarcomeres).8,10,11 Regardless of the cause, research shows
that contractures interfere with activities of daily living,
DOI: 10.1097/PEP.0000000000000269
cause pain, sleep disturbance, and increase the burden

262 Craig et al Pediatric Physical Therapy


Copyright © 2016 Wolters Kluwer Health, Inc. and the Academy of Pediatric Physical Therapy of the American Physical Therapy Association.
Unauthorized reproduction of this article is prohibited.
of care.10 Stretching, positioning, and active movement (OCEBM) levels of evidence;18 (b) study participants were
are proposed to prevent contractures and malalignment younger than 19 years and had a confirmed neuromuscular
by avoiding the reduction of the number of in-series disability; (c) studies contained a stretch intervention; and
sarcomeres that decreased movement causes.8 Despite (d) studies evaluated the effect of stretch interventions on
the common practice of prescribing stretch interventions, any body structure, body function, activity, or participa-
these clinical rationales have not been validated as there tion provided that there was a primary outcome measure
is limited and varied evidence about the actual causes of flexibility. Studies were included if cointerventions in-
of contractures, the proposed physiological theory of volved education and/or other exercise prescription (eg,
stretching, and the clinical effectiveness of stretching in aquatic therapy, aerobic training, and strength training)
the human model.8,10 as long as one of the interventions was a stretch interven-
For all therapeutic interventions, clinicians need to tion. These types of cointerventions were included to allow
consider potential benefits and harms to the child and for comprehensive programs that can be fully delegated to
family.12 This clinical decision is even more important members of the child’s team (as stretch interventions often
when there is limited evidence to guide practice. Although are) under the supervision, but not direct treatment, of a
physical therapists have clinical rationales for the possible physical therapist.
benefits of stretch interventions, the possible harmful ef- To address studies that included both pediatric and
fects also need to be examined. For example, continuous adult subjects, the following was determined a priori to
postural management can have a negative effect on sleep determine study eligibility: (a) individual clinical studies
hygiene,13,14 and assisted stretching is frequently reported must have 50% or more pediatric subjects, or a mean par-
as the most common daily activity that causes pain for ticipant age of less than 19 years; and (b) SRs must have
children with CP.15 Complying with an intervention that 50% or more studies that met the pediatric criteria, or they
compromises a child’s sleep or induces pain can place a must provide subanalyses of the pediatric population.
significant emotional burden on the child, caregivers, and Exclusion. Exclusion criteria of this review were (a)
parents.13,16 observational studies and surveys; (b) studies included
Considering the routine prescription of stretch inter- able-bodied youth or youth with disabilities not consid-
ventions and the burden and cost of implementation, a ered neuromuscular in nature; (c) studies involved concur-
systematic analysis of the efficacy of these interventions is rent treatment of other physiotherapeutic interventions di-
needed. The objective of this systematic review (SR) is to rectly provided by a physical therapist or other health care
determine whether casting, orthoses, stretching programs, provider (eg, acupuncture, Botox, electric modalities, man-
or supported standing programs are effective in improving ual therapy, massage, or neurodevelopmental treatment);
or maintaining body functions and structures, activity, or (d) where casting was used as part of constraint-induced
participation in children and youth with neuromuscular movement therapy; or (e) where recent surgery was done.
disabilities.

Operational Definitions
METHODS For the purpose of this review, the definition of a neu-
Search Strategies romuscular disability is any chronic disease or syndrome
that impairs the function of skeletal muscles. This impair-
English language titles were searched from the ear- ment can affect the muscle structure itself and/or the signal
liest date available until December 31, 2014, in the sent to the muscle. Examples of neuromuscular disabilities
following electronic databases: CINAHL, EMBASE/Ovid, that were considered for review include CP, muscular dys-
EBMR/Ovid, MEDLINE/PubMed, MEDLINE/EBSCO, and trophies, neural tube defects, spinal cord injuries, spinal
Physiotherapy Evidence Database. See the Appendix for muscular atrophies, traumatic brain injury, and other rare
the detailed electronic database search strategy. We did neuromuscular diseases. The definition of a stretch inter-
not use population-specific search terms (eg, CP and mus- vention is an intervention aimed at maintaining or increas-
cular dystrophy) to get comprehensive search results to ing joint mobility by influencing the extensibility of soft tis-
later limit by inclusion criteria. Preliminary searches did sues spanning joints.10 The following were preidentified as
not yield any articles with the same objective of this re- possible stretch interventions: bracing, casting, orthoses,
view. Details of the protocol for this SR were registered on positioning programs, self-administered stretches, splint-
September 19, 2014, on the International Prospective Reg- ing, stretches by caregivers, and yoga programs. Bracing,
ister of Systematic Reviews (PROSPERO) and can be ac- splinting, and orthoses were considered to be one treat-
cessed at: http://www.crd.york.ac.uk/PROSPERO/display ment category, herein “orthoses,” to improve clarity and
record.asp?ID=CRD42014013807.17 knowledge translation. Both active and passive range of
motion (ROM) and stretch programs were included. For
Eligibility Criteria
the purpose of this review, flexibility was defined as the
Inclusion. The inclusion criteria of this review were: ability to move a joint through its complete ROM19 and
(a) studies published in peer-reviewed journals appraised could have been measured with a goniometer, through
as Level 1 or 2 Oxford Centre of Evidence-Based Medicine gait analysis or with another valid instrument.

Pediatric Physical Therapy Systematic Review: Stretch Effectiveness 263


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Selection of Studies rates interventions based on the quality of evidence ac-
One reviewer (JC) screened the titles and abstracts of cording to the following criteria: green, go (ie, high-quality
found articles using the inclusion and exclusion criteria evidence supporting the effectiveness of this intervention,
stated earlier. Full-text copies of any study that appeared therefore use this approach); yellow, measure (ie, low-
to meet the inclusion criteria were obtained for further quality or conflicting evidence supporting the effective-
inspection. Two reviewers (JC and GW or RM) indepen- ness of this intervention, therefore measure the outcomes
dently read each article and recommended inclusion or of the intervention when using this approach to ensure
exclusion. In cases of disagreement, a third reviewer (CH) the patient’s goal is met); red, stop (ie, high-quality ev-
was consulted and discussion occurred until agreement idence demonstrating this intervention is unsafe or inef-
could be met. fective, therefore do not use this approach).25,26 Three re-
view authors (JC, GW, and CH) independently performed
a GRADE strength of recommendation and EATLS rating
Data Extraction for each identified outcome measure. A decision about the
Two review authors (JC and GW or RM) indepen- final grading and strength of recommendations assigned
dently performed data extraction of included studies using was reached through discussion and consensus. Reporting
internally made data extraction forms. For each study, we of SRs followed the Preferred Reporting Items for System-
collected information on the authors’ main conclusions, atic Reviews and Meta-analyses (PRISMA) statement.27
level of evidence,18 outcome measures coded by the
International Classification of Functioning, Disability RESULTS
and Health (ICF),20 participant baseline characteristics,
sample size, study design, study methods, and type of Results of the Search
intervention(s). Electronic database and hand searching yielded
24 930 references. After removing duplicates and screening
titles and abstracts, 81 studies were eligible for full-text re-
Assessment of Risk of Bias and Study Quality in
view. There were 10 studies that required a third reviewer
Included Studies
to determine eligibility. After inspecting the full reports,
For randomized controlled trials (RCTs), we as- 16 articles were included (See Figure 1).
sessed the risk of bias of individual studies by using a
domain-based evaluation recommended by the Cochrane Excluded Studies
Collaboration21 because the use of scales for assessing qual-
Figure 1 provides a summary of reasons for exclusion
ity or risk of bias is explicitly discouraged.22 The quality
of studies (See Supplemental Digital Content 1, available
of SRs was assessed using an OCEBM appraisal sheet23
at http://links.lww.com/PPT/A106, which lists all articles
and recorded on the data extraction form. OCEBM levels
excluded at the full-text level). The most common reasons
of evidence were also assigned during the data extraction
for exclusion were lower level of evidence (n = 26), adult
process and could be downgraded due to study bias or
population (n = 9), no stretch intervention (n = 8), coin-
upgraded because of large effect sizes.18
terventions (n = 7), and downgraded due to risk of bias
We used the Grading of Recommendation Assess-
(n = 6). All articles downgraded due to risk of bias were de-
ment, Development and Evaluation (GRADE) approach
termined to have unacceptable risk of attrition, detection,
to assess risk of bias across studies.24 For purposes of
performance, selection, and other biases. Other biases in-
SRs, GRADE defines the quality of a body of evidence as
cluded confounding cointerventions, poor compliance to
high, moderate, low, or very low for a particular outcome
intervention or poor reporting of compliance, lack of sta-
measure.24 Three review authors (JC, GW, and CH) first
tistical analyses to determine whether groups were similar
independently performed the quality of evidence grading
at baseline, and sample populations not representative of
for each identified outcome measure. A decision about the
exposed cohort.
final grade assigned was reached through discussion and
consensus.
Included Studies
Levels of Evidence and Risk of Bias in Included
Knowledge Translation Studies. Among the 16 included articles, 12 were SRs
The strength of clinical recommendations was made and 4 were randomized controlled or crossover trials
using the GRADE approach12 and Evidence Alert Traffic (Table 1).28-43 Two of the SRs were appraised as Level 1
Light System (EATLS)25,26 to facilitate knowledge transla- evidence as they had both high methodological qual-
tion. A GRADE strength of recommendation is the ex- ity and only included RCTs or SRs.28,29 The 10 other
tent to which one can be confident that the desirable SRs were appraised as Level 2 evidence due to inclu-
consequences of an intervention outweigh its undesirable sion of lower levels of evidence and/or poor methodol-
consequences.12 The recommendations are graded as ei- ogy. All of the included RCTs were graded as Level 2 evi-
ther strong or weak, and in support of or against an inter- dence. In general, the methodological quality of included
vention for a particular outcome measure.12 The EATLS articles was poor. Common methodological weaknesses

264 Craig et al Pediatric Physical Therapy


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Fig. 1. PRISMA flow diagram.

in the studies included lack of reporting, or inadequate mation was well reported, comparison groups were often
randomization methods, allocation concealment, report- lacking because of study design (eg, case report) (Table 1).
ing of dropouts, and controlling for confounding coin- Intervention dosing parameters such as treatment du-
terventions (see Supplemental Digital Content 2, avail- ration, frequency, and intensity were well recorded in all of
able at http://links.lww.com/PPT/107, which summarizes the RCTs; however, these parameters were inconsistently
methodological quality of included studies). reported in the SRs. When recorded, casting intervention
lasted for 3 to 5 weeks with casting protocols not being
well documented.28,29,34-36 There was a lack of informa-
Participants tion about the specific orthotic intervention dosing proto-
Sample sizes of the included studies ranged from 14 to cols recorded by the SRs.28,29,37-39 RCTs investigating night
1110 (Table 1). The effectiveness of stretch interventions splinting required splints to be worn all night30,32,33 or
was investigated in the following populations: CP (n = 9), every other night31 for a duration of 4 weeks,32 6 weeks,33
mixed disabilities (n = 4), Charcot-Marie-Tooth (n = 2), 12 months,31 or 30 months.30 Passive stretching or posi-
and Duchenne muscular dystrophy (n = 1). The age of tioning dosing reported in the SRs noted that 30 minutes’
participants ranged from 20 months to 30 years, with all total stretch program was the most commonly chosen ses-
studies having a median age less than 19 years. sion time, with each stretch typically being held for 30
to 60 seconds and repeated for several repetitions.35,40,41
One SR noted an average duration of passive stretching or
Interventions positioning study length to be 8.2 weeks with a mean fre-
The included studies evaluated the effectiveness of quency of intervention to be to 4.5 times per week.40 Sup-
casting (n = 5), orthoses (n =10), passive stretching ported standing program dosing was well recorded in 1 SR
or positioning (n =5), and supported standing programs with the following evidence-based dosage recommended:
(n =6). A wide range of casting protocols, orthoses con- 5 days/wk positively affects bone mineral density (BMD)
figurations, prescription of stretching programs, and sup- (60-90 min/d), hip stability (60 min/d in 30◦ -60◦ degrees
ported standing equipment was evident from the literature. hip abduction), ROM of hip knee and ankle (45-60 min/d),
Not all research reports described this in adequate detail and spasticity (30-65 min/d).42 The majority of included
to be replicated. Comparison interventions were explicitly studies in the 2 other reviews noted 30 minutes as the com-
mentioned in all included RCTs30-33 ; however, SRs often mon duration of supported standing; however, there was
did not record comparison interventions. Where this infor- a large variation in study duration from just 1 session to

Pediatric Physical Therapy Systematic Review: Stretch Effectiveness 265


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266
TABLE 1
Characteristics of Included Studies

OCEBM

Craig et al
Study Level of Articles and Sample Body Structure and Function Activity and Participation
Author Design Evidence Population Intervention(s) Size Outcome Measures Outcome Measures

Autti-Rämö et al28 SR 1 CP I: Upper and lower limb casting 5 SR (n = 663)a EMG, energy expenditure, gait Balance, COPM, functional tasks
and orthoses analysis, muscle tone, muscle (sit to stand, stair use, walking),
C: Varied (eg barefoot condition, strength, quality of movement, GMFM, grasp, hand function/
no casting, within participant ROM use, parent perception, Peabody,
orthoses comparison) QUEST, visual motor
performance
Blackmore et al34 SR 2 CP I: Ankle serial casting 19 studies (n = 395) 3D gait analysis, passive ankle Noneb
C: No comparison interventions ROM
(eg, 10 of the 12 studies had no
controls); varied (eg, 2 of the
12 studies: NDT-based physical
therapy, physical therapy, and
home program)
Effgen et al35 SR 2 School-aged I: Lower extremity casting, 15 SR (n = not BMD, gait parameters, prevention Balance, functional task (eg, sit to
children with orthoses, splints; passive reported)c of contracture, ROM, spasticity stand), GMFM, hand function
disabilities stretching; weight-bearing
interventions
C: No comparison interventions
recorded
Figueiredo et al37 SR 2 CP I: Any type of AFO 20 studies (n = 446) EMG, energy expenditure, gait BOTMP, GMFM, GMPM, PEDI
C: Varied (eg, within-group kinematics, gait kinetics, ROM
barefoot condition, shoes only,
hinged or nonhinged AFO)
Franki et al40 SR 2 CP I: Passive stretching and 83 studies (n = 660)d Behavioral state, BMD, bowel ADL (feedback form), endurance
weight-bearing interventions activity (diary), gait analysis, (2-min walk test), CRIB, personal
C: No comparison interventions gait velocity, muscle tone, ROM feeling of improved daily

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recorded functioning, PEDI
Hyde et al30 RCT 2 DMD I: Passive stretching combined n = 27 Anthropometric measures, hip Gower’s maneuver, MA, timed
with use of night orthoses flexor and tendoachilles physical performance (eg, time
C: Passive stretching contracture, muscle strength taken to run)
Lannin et al36 SR 2 Children with I: Upper extremity casting 23 studies (n = 326) EMG, hypertonicity, ROM None
neurological C: No comparison interventions
conditions (eg, 13/23 studies); varied (eg,
10/23 studies: no casting,
passive stretching, postcasting
follow-up of either cast or
orthotic regime, shorter casting
duration, traditional therapy)
(continues)

Copyright © 2016 Wolters Kluwer Health, Inc. and the Academy of Pediatric Physical Therapy of the American Physical Therapy Association.
Pediatric Physical Therapy
TABLE 1
Characteristics of Included Studies (Continued)

OCEBM
Study Level of Articles and Sample Body Structure and Function Activity and Participation
Author Design Evidence Population Intervention(s) Size Outcome Measures Outcome Measures

Pediatric Physical Therapy


Maas et al31 RCT 2 CP I: Use of a night KAFO n = 28 Ankle-foot dorsiflexion ROM, GMFM
C: No KAFO use ankle-foot and knee angle in
gait, complaints, orthosis
wearing time
Montero et al38 SR 2 Children with I: Technical devices including 27 studies (n = 664)e BMD, energy expenditure, gait Balance, BOTMP, changes in daily
motor orthoses and supported standing analysis, hip migration activities and posture
disabilities programs percentage, hip and spine (questionnaire), checklist of
C: No comparison interventions x-rays, hip subluxation and feeding problems, GMFCS,
recorded dislocation, joint ROM, muscle GMFM, GMPM, PEDI
alignment, muscle strength,
perceived exertion, spasticity,
transitional movement of sit to
stand
Neto et al39 SR 2 CP I: Articulated AFO 7 studies (n = 120) EMG, energy expenditure, gait None
C: Rigid AFO analysis: kinetics, kinematics,
and gait parameters
Novak et al29 SR 1 CP I: Casting; orthoses; stretching via 166 studies (n = not BMD, gait analysis, prevention of Upper and lower limb function
manual stretching, splinting or reported)f contracture, ROM, spasticity
positioning; weight-bearing via
standing frame
C: No comparison interventions
recorded
Paleg et al42 SR 2 Children with I: Supported standing program 30 studies (n = Functions of the bone as related Mobility and major life areas (ie,
atypical C: No comparison intervention 1110)g to BMD, functions of the bone speed of feeding, social
development, recorded or no comparison as related to hip stability, interaction, eased burden of care,

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with or without intervention (eg, 18/30 studies); functions of the digestive GMFM)
neuromuscular varied (eg, 12/30 studies: system, mental functions,
diagnosis addition of trochanteric girdle to muscle power functions,
including CP long leg braces set in abduction, muscle tone functions,
dynamic standing, no stander or neuromusculoskeletal and
positioning equipment, standing movement-related functions,
with hip abduction and skin and related functions
extension, straddled
weight-bearing, whole body
vibration, within-group
nonstanding phase)
(continues)

Systematic Review: Stretch Effectiveness


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267
268
TABLE 1

Craig et al
Characteristics of Included Studies (Continued)

OCEBM
Study Level of Articles and Sample Body Structure and Function Activity and Participation
Author Design Evidence Population Intervention(s) Size Outcome Measures Outcome Measures

Pin41 SR 2 CP I: Passive stretching programs 7 studies (n = 133) EMG, gait analysis, ROM, None
C: No detailed information about spasticity
comparison interventions (eg,
3/7 studies participants acted as
their own control with no
record of within-group
comparison intervention; 4/7
studies had a comparison
intervention but not recorded)
Pin43 SR 2 CP I: Static lower or upper body 10 studies (n = 122) Behavioral state, BMD, EMG, gait Bayley Scales of Infant and Toddler
weight-bearing analysis, hand posture, hand Development (mental scales),
C: No detailed information about surface area, muscle tone, ROM CRIB, grasp and release, Jebsen
comparison interventions (eg, Taylor Hand Function test,
7/10 studies had a comparison prehension, spontaneous use of
but not recorded) hand
Refshauge et al33 RCOT 2 Charcot-Marie- I: Night ankle orthoses n = 14 Isometric strength, passive ROM None
Tooth C: No night ankle orthoses
Rose et al32 RCT 2 Charcot-Marie- I: Serial night orthoses for 4 wk, n = 30 Ankle ROM (lunge test), foot Balance, falls, mobility (eg, standing
Tooth followed by 4 wk of stretching deformity up from chair, walking, and
C: No intervention stairs), self-reported activity
limitations

Abbreviations: ADL, activity of daily living; AFO, ankle-foot orthotic; BMD, bone mineral density; BOTMP, Bruininks-Oseretsky Test of Motor Proficiency; C, comparison; CP, cerebral palsy; COPM, Canadian

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Occupational Performance Measure; CRIB, Carolina Record of Individual Behavior; DMD, Duchenne muscular dystrophy; EMG, electromyography; GMFCS, Gross Motor Function Classification System;
GMFM, Gross Motor Function Measure; GMPM, Gross Motor Performance Measure; I, intervention; KAFO, knee-ankle-foot orthosis; MA, motor ability scale; OCEBM, Oxford Centre of Evidence-Based
Medicine; PROM, passive range of motion; QUEST, Quality of Upper Extremity Skills Test; PEDI, Pediatric Evaluation of Disability Inventory; RCOT, randomized crossover trial; RCT, randomized controlled
trial; ROM, range of motion, SR, systematic review.
a 1/5 studies did not report sample size.
b Only studies that had a cointervention of Botox had functional measures.
c 6/15 studies on stretch interventions.
d 12/83 studies on stretch interventions.
e 17/27 studies on stretch interventions.
f 11/166 studies on stretch interventions.
g 5/30 studies did not report sample size.

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Pediatric Physical Therapy
9 months of intervention.35,43 No studies commented on term effects to either support or abandon upper extremity
the intensity of the intervention, besides 1 RCT that men- casting.36 No studies identified in this review included the
tioned that if night splinting interfered with sleeping, direct measurement of the effect of casting on activity or
participants were to use them during day rest periods participation of children with neuromuscular disabilities.
instead.31 It is important to note that although several of Clinical Recommendation From the Evidence. Us-
the SRs did not record specific dosing parameters, they ing the EATLS, serial casting for short-term improvement
noted a lack of long-term follow-up in their included of ankle ROM is rated as a green intervention supported
studies.28,29,35,37,40 by very low evidence. All other outcome measures have
insufficient evidence, thus casting is rated as a yellow in-
tervention for these measures.
Outcomes
ROM, prevention of contractures, BMD, gait analy-
ses, and spasticity were the most studied body function Orthoses
and structure measures (Table 1). Functional mobility as- Evidence. The most consistent finding among studies
sessment (eg, sit to stand) and the Gross Motor Function identified in this review is that there is very low evidence
Measure were the most common activity measures. The that ankle-foot orthotic (AFO) devices that restrict plan-
Canadian Occupational Performance Measure was the only tarflexion improve gait kinematics and kinetics while the
participation measure identified in any study. device is worn (Table 2).28,29,35,37,38 One SR that compared
articulated and rigid AFOs for children with CP found
significant differences in peak dorsiflexion, reduction in
Analysis of the Evidence double-support time, increase in gait speed, and reduc-
The effectiveness of all of the interventions as coded tion in energy expenditure with the use of an articulated
by ICF levels, GRADE quality of evidence, and by the pre- orthosis.39 There is both conflicting and insufficient evi-
viously mentioned knowledge translation tools is summa- dence on the effectiveness of orthoses for the prevention
rized in Table 2. Because of the large heterogeneity and of contractures, either by the use of AFOs or by wearing
lack of reporting of interventions and outcome measures night splints.28-33,35,37,39 Two randomized trials showed
used in individual studies and SRs, effect-size estimation that night ankle splints or KAFOs do not improve ROM in
and meta-analysis of the data were not performed. children with CP or Charcot-Marie-Tooth, whereas 1 study
found that the expected annual change in tendoachilles
contracture for boys with Duchenne muscular dystrophy
Adverse Events was 23% less in the night splint and passive stretch group
Two RCTs,31,32 and 2 SRs4,36 on casting and orthoses compared with the passive stretch-only group.30,31,33 One
reported on adverse events. Adverse events such as bruis- RCT found that at 4 weeks of postserial night casting, the
ing and blistering were seen in 13% of subjects who had experimental group had significant but small increase in
serial casting,32 whereas the majority of participants in a ankle dorsiflexion; however, these effects were not main-
study on knee-ankle-foot-orthoses (KAFO) reported fre- tained with stretching at 8 weeks.32 There is also conflict-
quent pain because of muscle strain and pressure spots, ing and insufficient evidence in the studies identified to
as well as sleep disturbance.31 Additional complaints of support the use of orthotics for promoting activity or par-
night-time use of KAFOs included hot or sweating legs, ticipation while the device is worn. Two studies reported
itching, cramping, and bed-wetting.31 The most common that wearing a lower extremity device might make func-
adverse events of casting cited from the SRs included skin tional activities, such as rising up from the floor, more
irritation, skin breakdown, and pain.34,36 difficult,28,35 whereas another study showed that orthoses
have a positive effect on functional activities related to
mobility.37 Most SRs reported that there is insufficient ev-
Casting idence to support or refute the use of orthoses in improv-
Evidence. Consistent but very low evidence sup- ing function,28,29,35 whereas the majority of randomized
ports the use of 3 to 5 weeks of ankle casting for the trials30-32 showed no functional difference between exper-
positive short-term effects that it has on passive ankle imental and control groups or did not include a functional
dorsiflexion.28,29,34,35 Short-term improvements in gait pa- measure.33 Several authors mention that the wide variety of
rameters such as self-selected pace and stride length fol- lower limb orthoses investigated as well as different terms
lowing ankle casting have also been noted29,34 ; however, used for the same orthoses made a systematic evaluation
this review did not identify any Level 1 or 2 evidence sup- difficult.28,37,38 Poor compliance and tolerance of night or-
porting or refuting long-term benefits on gait and ROM. thoses has also been cited as a limitation in determining
There is insufficient research on the effectiveness of cast- the effectiveness of this intervention.31,32 No high-quality
ing for other lower extremity joints. One SR that assessed studies that assessed the effectiveness of upper extremity
the effectiveness of upper extremity casting for children orthoses were found.
with neurological conditions concluded that there is insuf- Clinical Recommendation From the Evidence. Ac-
ficient high-quality evidence regarding the effect or long- cording to the EATLS, orthoses that restrict plantarflexion

Pediatric Physical Therapy Systematic Review: Stretch Effectiveness 269


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TABLE 2
Summary of Findings: Stretch Interventions for Children With Neuromuscular Disabilities

GRADE
Quality of GRADE Strength of Traffic Light
Intervention Outcomea Studies Evidenceb Recommendationsc Actiond Comments

Intervention: casting
Body function: PROM of Autti-Rämö et al28 ⊕ Strong for Green: go Effective for increasing ankle
lower limbs Blackmore et al34 Very low range in the short term. No
Effgen et al35 evidence on the long-term
Novak et al29 effects for different joints
Body function: PROM of Autti-Rämö et al28 ⊕ Weak for Yellow: measure Insufficient evidence to support
upper limbs Lannin et al36 Very low or refute the use of casting
Novak et al29
Body function: gait kinetics Blackmore et al34 ⊕ Weak for Yellow: measure Immediate gains in gait
and kinematics Effgen et al35 Very low parameters (ie, stride length
Novak et al29 and walking speed) are likely
secondary to improvements in
ROM; however, the long-term
benefits on gait are unknown
Body function: spasticity Blackmore et al34 ⊕ Weak against Yellow: measure Insufficient evidence to support
Lannin et al36 Very low or refute the use of casting
Novak et al29
Activity and participation: Autti-Rämö et al29 ⊕ Weak against Yellow: measure Insufficient evidence to support
functional abilities Blackmore et al34 Very low or refute the use of casting
Effgen et al35
Lannin et al36
Novak et al29
Intervention: orthoses
Body function: PROM and Autti-Rämö et al28 ⊕ Weak for Yellow: measure Effective for increasing ankle
prevention of contracture Effgen et al35 Very low ROM while wearing the
of lower limbs Figueiredo et al37 device. There is no evidence
Hyde et al30 to support or refute the
Maas et al31 long-term benefit of wearing
Neto et al39 orthoses on ROM.
Novak et al29 Compliance has been noted
Refshauge et al33 as an important factor
Rose et al32
Body function: gait kinetics Autti-Rämö et al28 ⊕ Strong for Green: go AFO devices that restrict
and kinematics Effgen et al35 Very low plantarflexion are effective for
Figueiredo et al37 improving gait parameters
Montero et al38 while wearing the device
Neto et al38
Novak et al29
Activity and participation: Autti-Rämö et al28 ⊕ Weak for Yellow: measure Insufficient evidence to support
lower limb functional Effgen et al35 Very low or refute the use of lower
abilities Figueiredo et al37 limb orthoses.
Hyde et al30
Maas et al31
Montero et al38
Neto et al39
Novak et al29
Refshauge et al33
Rose et al32
Intervention: positioning, range of motion, stretching
Body function: PROM and Effgen et al35 ⊕ Weak for Yellow: measure Insufficient evidence to support
prevention of contracture Franki et al40 Very low or refute the use of stretching
Novak et al29 programs. Although there is
Pin41 insufficient evidence,
Rose et al32 generally studies showed an
increase in ROM
poststretching or a loss of
ROM after stretching stopped
Body function: spasticity Franki et al40 ⊕ Weak for Yellow: measure Insufficient evidence to support
Pin41 Very low or refute the use of stretching
Rose et al32 programs
(continues)

270 Craig et al Pediatric Physical Therapy


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TABLE 2
Summary of Findings: Stretch Interventions for Children with Neuromuscular Disabilities (Continued)

GRADE
Quality of GRADE Strength of Traffic Light
Intervention Outcomea Studies Evidenceb Recommendationsc Actiond Comments

Activity and participation: Effgen et al35 ⊕ Weak against Yellow: measure Insufficient evidence to support
functional abilities Franki et al40 Very low or refute the use of stretching
Novak et al29 programs
Pin41
Rose et al32
Intervention: supported standing
Body structure: BMD Effgen et al35 ⊕ Strong for Green: go Effective to increase lower limb
Franki et al40 Very low bone mineral density;
Montero et al38 however unclear whether this
Novak et al29 prevents pathological
Paleg et al42 fractures
Pin43
Body function: PROM and Effgen et al35 ⊕ Weak for Yellow: measure Insufficient evidence to support
prevention of contracture Franki et al40 Very low or refute; however, several
of lower limbs Montero et al38 studies showed a positive
Paleg et al42 effect on hip range of motion
Pin43 or migration percentage
Body function: spasticity Paleg et al42 ⊕ Weak for Yellow: measure Effective in the temporary
Pin43 Very low reduction of lower limb
spasticity
Activity and participation: Effgen et al35 ⊕ Weak for Yellow: measure Insufficient evidence to support
functional abilities Franki et al40 Very low or refute the use of casting
Montero et al38
Paleg et al42
Pin43

Abbreviations: AFO, ankle-foot orthosis; BMD, bone mineral density; GRADE, Grading of Recommendations Assessment, Development, and Evaluation;
PROM, passive range of motion; ROM, range of motion.
a Coded with the International Classification of Functioning, Disability and Health.20
b GRADE specifies 4 quality of evidence ratings (high, moderate, low, and very low) that are applied to a body of evidence. The GRADE quality of evidence

rating reflects the confidence that the estimates of the effect are correct.24
c The GRADE strength of a recommendation is separated into strong and weak. It is defined as the extent to which one can be confident that the desirable

effects of an intervention outweigh its undesirable effects.12


d The Evidence Alert Traffic Light System rates interventions according to the following criteria: Green, go (ie, high-quality evidence supporting the

effectiveness of this intervention, therefore use this approach); yellow, measure (ie, low-quality or conflicting evidence supporting the effectiveness of this
intervention, therefore measure the outcomes of the intervention when using this approach to ensure the patient’s goal is met; red, stop (ie, high-quality
evidence demonstrating this intervention is unsafe or ineffective, therefore do not use this approach)”.25,26

to improve a child’s gait while the device is worn is a passive stretching for improving ROM and spasticity.40,41
green intervention. Orthotic use for the prevention of con- Although there was limited/weak evidence, generally stud-
tractures and promotion of activity and participation are ies showed an increase in ROM poststretching or a loss
yellow interventions. of ROM after stretching stopped.40,41 One author con-
cluded that it appeared that sustained stretching of longer
Positioning and Stretching duration was preferable to improve range of movements
Evidence. The most commonly reported outcome and to reduce spasticity of muscles around the targeted
measures for positioning and stretching programs were joints.41 No studies identified by this review reported the
prevention of contractures and ROM (Table 1). Two SRs effect of positioning, ROM, or stretching programs on the
reported that there is insufficient evidence to support or activity or participation of children with neuromuscular
refute the use of passive ROM35 or positioning29 to pre- disabilities.
vent contractures. One review29 concluded that manual Clinical Recommendation From the Evidence. There
stretching is ineffective for contracture prevention in the is insufficient evidence to make any recommendations in
short to medium term (<7 mo) based on a comprehen- regard to the use of positioning or stretching programs;
sive and robust meta-analysis; however, this conclusion therefore, all are yellow interventions in accordance with
was based mainly on one review10 that included mostly the EATLS.
adults, only looked at static stretches, and was not able
to define or standardize the control condition of usual Supported Standing Programs
care. Two SRs identified in this study noted that there is Evidence. Six SRs evaluated the effectiveness of sup-
limited or weak evidence to support the effectiveness of ported standing programs.29,35,38,40,42,43 In this review,

Pediatric Physical Therapy Systematic Review: Stretch Effectiveness 271


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1 SR was identified that evaluated the effectiveness of static this increases the child’s quality of life (Table 2). Treat-
weight-bearing in children with CP. It found that other ment interventions varied immensely with a wide range
than the findings of increased BMD and temporary re- of the following interventions being recorded: casting pro-
duction in spasticity, there is limited evidence supporting tocols, orthoses configurations, prescription of stretching
the intervention because of weak research methodology.43 programs, and supported standing program equipment.
Another study, we identified assessed the effectiveness Adverse events were rarely mentioned and when recorded
of standing programs for children with atypical develop- they included skin irritation and pain from casting and
ment, concluded that there is evidence that the interven- orthoses.31,32,34,36 Only one review35 has looked at the
tion positively affects BMD, hip stability, ROM of the hip same breadth of interventions for multiple pediatric dis-
knee and ankle and spasticity.42 The authors concluded abilities as this current research has presented. The com-
that to see these positive results, standing program dosage parable review35 included lower levels of evidence and no
should be between 30 and 90 min/d.42 The most consis- quality assessment was conducted. All other studies have
tent finding reported in all studies we identified is that focused on 1 population28-34,37,39,40,41,43 and/or on 1 or
there is very low evidence that supported standing pro- 2 stretch interventions.28,30-34,36,37,39,41-43 To date, this is
grams using an external device increase lower limb and the most comprehensive review on all stretch interven-
vertebral BMD.29,35,38,40,42,43 There is no evidence that tions for a large population treated by pediatric physi-
improved BMD prevents pathological fractures.35,43 Al- cal therapists. In addition, this is only the second review
though there is insufficient evidence to refute or support article29 on pediatric stretch interventions that has used
the effectiveness of this intervention for promoting ROM the GRADE approach and the EATLS to categorize treat-
and/or preventing contractures, 2 studies showed a posi- ments based on the quality and strength of evidence. These
tive effect on hip ROM or hip migration percentage.38,42 knowledge translation tools allow clinicians to quickly im-
Two SRs we identified also found that there is evidence plement research into practice. This is valuable, as previ-
that supported standing programs temporarily decrease ous research has shown that although pediatric physical
spasticity.42,43 This review identified no quantitative re- therapists have a positive attitude toward evidence-based
search on the effect of supported standing programs on practice, they routinely self-report that they are unable to
the activity or participation of children with neuromuscu- implement this information into practice.44 Although this
lar disabilities. review confirms the effectiveness of some stretch inter-
Clinical Recommendation From the Evidence. Using ventions, it is clear that there is still a large gap between
the EATLS, supported standing programs using an external clinical practice, treatment rationales, and the available evi-
device are rated as green interventions for increasing lower dence. More specifically, there is no high-quality evidence
extremity BMD. Supported standing programs are yellow to support or refute that stretch interventions can avoid
interventions for all other outcome measures. or defer surgery, decrease complications such as contrac-
tures, or promote function as reported by pediatric physi-
DISCUSSION cal therapists.8,9 The yellow rating of many interventions
identified in this review highlights the importance of clin-
Summary of Main Findings icians using not only the best-available research regarding
The primary outcome measure for this SR was flex- function and basic science muscle/tendon physiology but
ibility. For this outcome measure, the strongest evidence also the 2 other tenets of evidence-based practice: clinician
found in this review was for the use of casting to increase experience and patient values.45,46 For example, clinicians
passive ankle dorsiflexion in the short-term. Conflicting should consider their historical knowledge about how dis-
evidence was found for the use of orthotics to prevent use, muscle imbalances, and immobility affect flexibility
contractures in populations of children with CP, Charcot- and function.45 When the evidence in the literature is un-
Marie-Tooth, and Duchenne muscular dystrophy. Insuffi- clear, clinicians are encouraged to use outcome measures
cient evidence was found to support or refute stretching/ specific and meaningful to their clients to track the effec-
positioning programs or supported standing programs for tiveness of and to modify treatment as necessary.46
the increase of ROM or prevention of contractures.
In addition to the flexibility measure, the strongest
evidence identified in this review supports the use of or- Limitations
thoses for improving gait kinetics and kinematics while Although this review only included Levels 1 and
worn, and for supported standing programs for improving 2 evidence, 10 of the 12 identified SRs did incorporate
lower extremity BMD. The majority of research has focused lower levels of evidence. There is a possibility that some
on assessing short-term outcomes of the lower extremity relevant studies of lower levels of evidence may have
at the body structure and function level. Of the limited been missed were they not included in these reviews,
reporting of activity and participation measures, no treat- and could have provided additional insight into this re-
ment effect was found when compared with controls.30-32 search question. However, although case studies or series
For example, although AFOs that restrict plantarflexion may have demonstrated more polarized results for the in-
have a favorable effect on improving gait kinetics and kine- dividuals (larger positive or negative effects) compared
matics, we identified no consistent research to indicate with groups (because of regression to the mean), it is

272 Craig et al Pediatric Physical Therapy


Copyright © 2016 Wolters Kluwer Health, Inc. and the Academy of Pediatric Physical Therapy of the American Physical Therapy Association.
Unauthorized reproduction of this article is prohibited.
unlikely that their results could have been generalized to and top-down interventions is important as a recent SR
the population investigated in this review. Inclusion crite- found that the majority of green interventions for children
ria requiring 1 primary outcome measure of flexibility may with CP were top-down therapy approaches, aimed at im-
have also excluded articles that only looked at other mea- proving activities performance.29 Thus, not only should
sures of body functions and structures or primarily on ac- clinicians and researchers assess functional and quality of
tivity or participation measures. This may mean that other life measures with sensitive tools, but functional interven-
benefits of these interventions, apart from those on flex- tions should be considered in conjunction with interven-
ibility measures, were missed. The large umbrella search tions targeted at the body structure and function level.
strategy, however, should have still found these articles, For the clinician, assessment and treatment should be or-
and only 3 studies were excluded only on the premise of ganized according to findings and hypotheses about im-
not having a flexibility measure (see Table, Supplemental pairments and limitations of the specific individual client
Digital Content 1, which lists all articles excluded at the in the context of best evidence, clinician experience, and
full-text level). client values.
Research Considerations. Future studies also need
Implications for Research and Clinical Practice to address study design and rigor to improve the quality of
Assessment. Future research is needed to verify the research and thus the applicability of the knowledge
whether stretch interventions have a clinically significant into clinical practice. More specifically, studies need
effect on the activity, participation, and quality of life of to account for dropouts, blind assessors and report on
children with neuromuscular disabilities. Several of the concurrent activities and therapies. Researchers also
included studies note the need to use outcome measures need to look at long-term follow-up (eg, >1 y) and
of activity and participation that are meaningful for the report on adverse events and safety considerations, as
child.28,29,32,35,37,38,43 This is significant in that both clini- the majority of the included studies did not address
cians and parents identify quality of life as the most impor- these events. This is paramount because the majority of
tant domain to assess in this population.47 The majority of stretch interventions are considered yellow interventions,
stretch intervention studies have focused at the body func- meaning that there is low-quality or conflicting evidence
tion and structure level, and it is rare to see a study that supporting the effectiveness of the interventions.25,26 For
assessed all ICF levels at once. When functional measure- all interventions, especially where there is insufficient
ments were assessed, no difference between experimental evidence to support or refute its effectiveness, the possible
and control groups were detected.30-32 The intervention desirable effects of an intervention have to be weighed
itself might be ineffective for functional changes; how- against its undesirable effects, such as adverse events and
ever, an alternative explanation is that the activity and burden of care.12 Possible negative effects of a stretch
participation measures currently designed may not be reli- intervention may include increased emotional burden on
able, sensitive, or valid enough to detect change during the caregivers,13,16 pain,15,31,34,36 sleep disturbances,13,14,31
course of intervention(s). For example, in SRs of activity or even a negative physiological effect.51 Research on other
and participation measures in children with CP, many of pediatric populations has demonstrated better coreporting
the tools did not have sound psychometric properties.48,49 of primary outcome physiological measures along with
Thus, it is evident from the literature that even if clini- secondary activity and participation outcome measures
cians and researchers start to implement more activity and including adverse events. For example, extensive research
participation assessment, further development of outcome has been conducted on the clinical effectiveness of
measures is needed to address the reliability, sensitivity, orthoses for adolescent idiopathic scoliosis along with the
and validity of these tools. coreporting of pain, quality of life, psychological issues,
Treatment. Another observation when functional and self-image.52,53 Similar research should be conducted
measures were assessed was that improvements at the for children with neuromuscular disabilities. In order to
body function level did not correlate with improvements perform high-quality clinical trials, researchers should
in activity or participation.29,40 A probable explanation conduct sample size calculations a priori to provide
for the lack of carryover between ICF levels uses the mo- adequate power. This was only mentioned in one of the
tor learning principles of bottom-up and top-down in- included studies of this review.31 The reporting of sample
terventions. Bottom-up treatment interventions focus on size calculation in physical medicine and rehabilitation
remediating an underlying impairment or motor deficit, research has been identified as being inadequate given cur-
whereas top-down treatment interventions typically use rent publication guidelines.54 Where RCTs are unethical
a problem-solving approach to motor skill development or impossible to conduct,10 long-term prospective cohorts
or task-specific interventions focused on the direct teach- that address the previously identified methodological
ing of a skill.50 Employing an intervention at the bottom weaknesses may be useful. Multicenter trials organized by
(eg, body structure and function level) and assuming that using databases, such as CP registries, can acquire the large
there is overflow40 or translation upstream to the activities sample sizes needed for this type of research. Following
level29 is not as logical as implementing an intervention a large group of children over a long period, while using
aimed at improving activity and participation and then as- sensitive outcome measures and employing top-down in-
sessing for change at this level. Distinguishing bottom-up terventions, will likely yield the most meaningful evidence.

Pediatric Physical Therapy Systematic Review: Stretch Effectiveness 273


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Unauthorized reproduction of this article is prohibited.
CONCLUSION 11. Skalsky AJ, McDonald CM. Prevention and management of limb con-
tractures in neuromuscular diseases. Phys Med Rehabil Clin N Am.
Three green interventions were found: ankle casting 2012;23(3):675-687.
for improving passive ankle dorsiflexion, orthoses for im- 12. Andrews J, Guyatt G, Oxman AD, et al. GRADE guidelines: 14. Go-
proving gait kinetics and kinematics, and supported stand- ing from evidence to recommendations: the significance and presen-
ing programs for improvement of lower extremity BMD. tation of recommendations. J Clin Epidemiol. 2013;66(7):719-725.
doi:10.1016/j.jclinepi.2012.03.013.
All other stretch interventions are yellow interventions for 13. Gough M. Continuous postural management and the preven-
a particular outcome measure, meaning that clinicians can tion of deformity in children with cerebral palsy: an appraisal.
continue to implement these interventions but need to use Dev Med Child Neurol. 2009;51(2):105-110. doi:10.1111/j.1469-
sensitive outcome measures to see whether these interven- 8749.2008.03160.x.
tions have helped the child reach their goal. No red stretch 14. Lloyd C, Logan S, McHugh C, et al. Sleep positioning for children
with cerebral palsy [Protocol]. Cochrane Database Syst Rev. 2012;10.
interventions were found, indicating that, at this time, no doi:10.1002/14651858.CD009257.
interventions need to be discontinued on the basis of inef- 15. Swiggum M, Hamilton ML, Gleeson P, Roddey T. Pain in
fectiveness or detrimental effects. Further investigation of children with cerebral palsy: implications for pediatric physi-
stretch interventions is warranted because the gap between cal therapy. Pediatr Phys Ther. 2010;22(1):86-92. doi:10.1097/PEP
clinical practice and the lack of clear scientific evidence can .0b013e3181cd18a7.
16. Hutton E, Coxon K. Posture for learning: meeting the postural care
have implications that could influence the future allocation needs in mainstream primary schools in England a research into prac-
and use of pediatric physical therapy services. tice exploratory study. Disabil Rehabil. 2011;33(19-20):1912-1924.
doi:10.3109/09638288.2010.544837.
17. Craig J, Hilderman C, Misovic R, Wilson G. The effectiveness of
stretching programs, bracing, casting and orthoses for improving or
ACKNOWLEDGMENTS maintaining body functions and structure, activities, or participation
in children or youth with neuromuscular disabilities: a systematic
The authors thank the BC Centre of Ability, Queen
review. PROSPERO: International Prospective Register of System-
Alexandra Centre for Children’s Health, and the University atic Reviews. http://www.crd.york.ac.uk/PROSPERO/display record.
of British Columbia for supporting this research, and Sarah asp?ID=CRD42014013807. Updated March 13, 2015. Accessed May
Craig, MEd, for her assistance with editing. 20, 2015.
18. OCEBM Levels of Evidence Working Group. The oxford 2011
levels of evidence. Oxford Centre for Evidence-Based Medicine.
http://www.cebm.net/index.aspx?o=5653. Updated 2011. Accessed
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APPENDIX

Search Strategy

The following search strategy was employed along with relevant MESH terms for each database: “stretches” or “stretch” or
“stretching” or “range of motion” or “passive range of motion” or “active range of motion” or “positioning” or “casting”
or “casts” or “cast” or “splints” or “splinting” or “splint” or “bracing” or “braces” or “brace” or “yoga” or “orthotics” or
“orthoses” or “orthotic” (and) “passive range of motion” or “active range of motion” or “joint mobility” or “flexibility” or
“flexible” or “pain” or “quality of life” or “spastic” or “spasticity” or “activities of daily living” or “activity of daily living”
or “participation” or “contracture” or “contractures.”

Pediatric Physical Therapy Systematic Review: Stretch Effectiveness 275


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