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Disability and Rehabilitation

ISSN: 0963-8288 (Print) 1464-5165 (Online) Journal homepage: http://www.tandfonline.com/loi/idre20

Rationale for prescription, and effectiveness of,


upper limb orthotic intervention for children with
cerebral palsy: a systematic review

Simon Garbellini, Yvette Robert, Melinda Randall, Catherine Elliott &


Christine Imms

To cite this article: Simon Garbellini, Yvette Robert, Melinda Randall, Catherine Elliott &
Christine Imms (2017): Rationale for prescription, and effectiveness of, upper limb orthotic
intervention for children with cerebral palsy: a systematic review, Disability and Rehabilitation, DOI:
10.1080/09638288.2017.1297498

To link to this article: http://dx.doi.org/10.1080/09638288.2017.1297498

Published online: 12 Mar 2017.

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Download by: [The UC San Diego Library] Date: 14 March 2017, At: 02:15
DISABILITY AND REHABILITATION, 2017
http://dx.doi.org/10.1080/09638288.2017.1297498

REVIEW ARTICLE

Rationale for prescription, and effectiveness of, upper limb orthotic intervention
for children with cerebral palsy: a systematic review
Simon Garbellinia,b, Yvette Robertb, Melinda Randalla , Catherine Elliottb,c and Christine Immsa
a
Centre for Disability and Development Research, Australian Catholic University, Melbourne, VIC, Australia; bDepartment of Pediatric
Rehabilitation, Princess Margaret Hospital, Perth, WA, Australia; cSchool of Occupational Therapy and Social Work, Curtin University, Perth, WA,
Australia

ABSTRACT ARTICLE HISTORY


Purpose: To explore (i) reasons for upper limb orthosis prescription for children with cerebral palsy (CP), Received 4 November 2016
(ii) the link between reason and effect according to intended outcome and outcome measure utilized and Revised 6 February 2017
(iii) to classify the prescribed orthoses using standard terminology. Accepted 16 February 2017
Method: A prospectively registered (center for reviews and dissemination: 42015022067) systematic
review searched for experimental and observational studies investigating rigid/thermoplastic upper limb KEYWORDS
orthotic intervention for children aged 0–18 with CP. The Cochrane central register, MEDLINE, CINAHL, Upper limb; Orthoses;
Embase, SCOPUS and Web of Science databases were searched. Included studies were assessed for risk cerebral palsy; prescription;
of bias. children
Results: Sixteen studies met selection criteria. Two studies described a specific reason for orthosis pre-
scription, six prescribed orthoses to manage a clinical symptom and eight did not describe a reason. Eight
studies were analyzed for effect according to intended outcome with no clear connection found between
reasons for prescription, outcome measures utilized and effect reported.
Interpretation: The lack of evidence for upper limb orthotic intervention for children with CP leads to
uncertainty when considering this treatment modality. Future research is needed to evaluate the effect of
orthosis wear in relation to intended outcome utilizing robust methods and valid and reliable outcome
measures.

ä IMPLICATIONS FOR REHABILITATION:


 Insufficient evidence exists about the reason for prescription of upper limb orthoses.
 The connection between reason for orthosis prescription, intended outcome, outcome measure
utilized and observed effect is unclear.
 Recommend orthosis prescription to be accompanied by clear documentation of the aim of the
orthosis and description using orthosis classification system terminology.
 Outcome measures consistent with the reason for orthosis prescription and intended outcome of the
intervention are essential to measure effectiveness of the intervention.

Introduction tissues to achieve the reason for which it is prescribed [14,17,18].


Orthoses used to mobilize tissues apply gentle forces aimed at
Limitations in hand use due to musculoskeletal change impact
increasing passive range of joint motion [14]. The use of upper
people with cerebral palsy (CP) across their life span and
can result in significant disability and pain [1–5]. Musculoskeletal limb orthoses for children with upper limb impairment is not a
problems develop as a result of growth, ageing, spasticity and lim- stand-alone intervention within clinical practice. Prescription needs
itations of movement against gravity [1,6–11]. Secondary musculo- to be made in consideration of other therapeutic, pharmacological
skeletal deformities are, therefore, progressive [1,8,12,13] and and surgical interventions specifically related to the individual
important to control, if possible. [19]. Orthoses may be prescribed to manage the effect of hyper-
Orthoses are one intervention prescribed to children with CP tonicity, prevent deformity and contractures, manage pain, main-
to minimize secondary upper limb musculoskeletal deformities tain tissue and joint integrity and improve function and
[14]. An orthosis is defined as an externally applied device used to participation in activity [17–21]. This range of reasons for orthosis
modify the structural and functional characteristics of the neuro- prescription targets the body function and structure, activity and
muscular and skeletal systems by applying forces to the body. In participation domains of the international classification of func-
clinical practice orthosis, splint and brace are interchangeable tioning, disability and health [22].
terms [15]. Orthosis is the term preferred by the international Practice guidelines for the prescription of orthoses in the man-
organization for standardization (ISO) [16]. The primary purpose of agement of neurologically-based upper limb impairments are
an orthosis fitted to the upper limb is to immobilize or mobilize scarce and great variability in prescription exists. Reported factors

CONTACT Simon Garbellini simon.garbellini@health.wa.gov.au Department of Paediatric Rehabilitation, Princess Margaret Hospital, GPO Box D184, Perth, WA,
Australia
ß 2017 Informa UK Limited, trading as Taylor & Francis Group
2 S. GARBELLINI ET AL.

which influence therapists’ prescription of orthoses include therap- relate to the lack of systematic investigation as to the reason for
ist training, familiarity and expertise in the orthoses used and the orthosis prescription and whether any observed effects meet the
availability of resources such as time, equipment and materials intended outcomes of the prescribed orthoses. In addition there is
[23]. Other factors that influence prescription include client goals; inadequate use of standardized terminology and inconsistent
client engagement in the intervention; consideration of the thera- reporting of orthosis descriptions in clinical practice.
peutic relationship; use of other interventions; family and carer The Australian Hand Therapy Association (AHTA) and American
support of the intervention; and the client’s environment [19,24]. Society of Hand Therapists (ASHT) endorse the use of orthosis
Although these many factors have been identified, there is a pau- classification systems to provide standard nomenclature to clearly
city of information about, and studies investigating, these factors describe an orthosis [15,21,36]. Classification of orthoses can be
in the literature. made regardless of the condition for which the orthosis is pre-
Evidence of the effect of upper limb orthoses for children with scribed, theoretical approach or material used to fabricate the
CP has been reviewed in four publications between 1989 and orthosis. Defined language from the classification systems elimi-
2014 [25–28]. Langlois et al. [27] conducted a literature review of nates confusion caused by colloquial jargon, improving communi-
13 studies on the use of hand splints and cerebral spasticity, with cation between clinicians. The more defined the orthosis
three studies including children with CP. The orthoses applied in description the greater potential there is for advancing know-
the three studies included Snook’s spasticity reduction splint [29]; ledge, improving patient outcomes and research [21].
dorsal and volar resting hand splints [30]; orthokinetic cuff, short This study aimed to:
opponens thumb splint and MacKinnon splints [31]. Langlois et al. 1. Explore the reasons for upper limb orthosis prescription for
[27] reported that clinical observation and scientific investigation children with CP;
supported spasticity reduction using orthotic intervention. No con- 2. Explore the link between reason and effect of upper limb
clusion was reached regarding the most effective orthosis design. orthoses for children with CP according to the orthoses’
Teplicky et al. [28] conducted a critical review of the literature intended outcome and outcome measures utilized;
on the use of upper limb orthoses for children with CP and trau- 3. Classify the upper limb orthoses using the ICF-CY [22] frame-
matic brain injury. The review included four papers utilizing (i) work and common criteria of the AHTA and ASHT orthosis
orthokinetic cuff, short opponens thumb splint and MacKinnon classification systems [15,36].
splints [31]; (ii) MacKinnon splint [32]; (iii) hand positioning device
[33]; (iv) dynamic proximal stability (LycraV) splint [34]. The
R

Methods
reviewers concluded that hand splints improved grasp. Autti-
Ramo et al. [25] completed an overview of review articles to sum- Design
marize evidence on the effect of upper and lower limb casting or
A systematic review was undertaken using objective and transpar-
orthoses provided to children with CP. Only one review by
ent methods as per the PRISMA guidelines [37], to identify, evalu-
Teplicky et al. [28] was included that investigated the use of
ate and summarize all relevant research findings [38]. The
upper limb orthoses. Autti-Ramo et al. [25] found inconclusive evi-
protocol for this review was submitted to the Prospero register
dence about the management of upper extremity problems using
and accepted on the 16/07/2015 (CRD42015022067).
casting or splinting.
Jackman et al. [26] assessed evidence of the effectiveness of
orthotic intervention for improving hand function for children Eligibility
with CP using a systematic review with meta-analysis. Only
Inclusion criteria
randomized and quasi-randomized trials were included in the
review. Although the authors reported study limitations made it Studies investigating the effect of an upper limb orthosis interven-
difficult to draw clear conclusions, their findings suggested that tion, regardless of publication date, that met the following criteria
were included in the review:
upper limb orthoses may have small benefits for hand skills how-
ever improvements diminished after wearing ceased. The authors 1. Included participants were children aged 0–18 years. In
identified four studies as being sufficiently homogenous in regard instances where a paper also included participants older than
to participants, reason for orthosis, impairment-based outcome 18 years, the study was included if 50% or more of the partic-
measures and wearing regimes, to include in a meta-analysis. On ipants were between 0 and 18 years;
closer inspection of these factors, particularly the reason for the 2. Participants had a diagnosis of CP. In instances where a paper
orthosis and wearing regimes, they may not be sufficiently homo- also included participants with other neurological diagnoses,
genous for inclusion in a meta-analysis. The intervention of a non- such as acquired brain injury, the study was included if 50%
functional orthosis included those constructed of thermoplastic or more of the participants had a diagnosis of CP;
material, plaster and fiberglass and metal lockable hinges. The 3. The intervention included a rigid/thermoplastic orthosis
joints on which the orthoses acted were described but there was applied to the upper limb (regardless of duration or dosage).
no comment as to whether the orthoses were designed to immo- An orthosis was considered rigid if it did not allow dynamic
bilize joint movement or mobilize tissue. Regimen of wear varied movement of the joints it acted upon, even if it was fabri-
from nightly for six months, at least 4 h daily for six months and cated with a combination of thermoplastic and flexible mate-
two 30 min sessions daily for six months. rials; and
4. The research design was a randomized controlled,
quasi experimental study, or an observational study using a
Rationale and aims cohort or case-control or single case experimental study
Upper limb orthoses are used to treat upper limb impairment in design.
children with CP; however, there is little evidence about their
effect and no clear guidelines regarding when the intervention Exclusion criteria
should be considered, what orthosis to use, and timing and Studies were excluded based on the following criteria:
length of wear [20,26,35]. The lack of guidelines for practice may 1. The participants had neuro-degenerative conditions;
UPPER LIMB ORTHOSES FOR CHILDREN WITH CEREBRAL PALSY 3

2. The orthotic intervention was designed to constrain the papers was completed between the two authors with no disagree-
unaffected upper limb for the purpose of improving move- ments regarding inclusion.
ment of the affected upper limb;
3. The orthotic intervention was a cast applied to the upper
Data extraction process
limb;
4. The orthosis used in the intervention was fully constructed of Two authors (Simon Garbellini, Yvette Robert) independently
soft, non-rigid materials for example neoprene or lycra; extracted data using a customized data extraction form developed
5. The study design was qualitative or a systematic review; and for the purpose of the review. The data extraction form was
6. Full papers not written in English requiring translation. piloted for use and tested for agreement with two studies. Once
agreement between the authors was reached regarding complete-
ness of the data extraction form, data was extracted from the
Information sources
remaining identified studies. Data extracted for each study were
Databases searched included the Cochrane central register, tabulated. Extracted data were compared and consensus was
MEDLINE, CINAHL, Embase, SCOPUS and Web of Science. reached via discussion between the two independent reviewers
Database searches were conducted in June 2015 and again in where differences in data extraction were evident.
May 2016. Hand searches of reference lists of included studies
were conducted to ensure additional relevant references were
Data synthesis
identified. Although systematic reviews were excluded, reference
lists were checked to ensure all primary research was located for A narrative synthesis with tabulation of the data for each study
inclusion. There was no limit placed on the publication dates of aim was completed. Comparison of the data relating to the reason
the included studies. Only full papers written in English were for upper limb orthosis prescription, the link between reason and
included. Full papers that required translation were excluded due intended outcome, outcome measures utilized and effect, and
to a lack of resources. Where multiple publications reporting on orthosis classification is presented. To meet the aim of classifying
the same study existed, data from the study were extracted and orthoses, the domain(s) of the ICF-CY [22] which the orthosis was
reviewed only once. prescribed to influence were identified and common criteria
adapted from the ASHT and AHTA orthoses classification systems
were used to describe the anatomical focus and primary purpose
Search
of each orthosis [15,18,21,36]. Figure 1 displays the common clas-
A full electronic advanced search strategy conducted in the sification criteria.
MEDLINE complete database, utilizing the EBSCOhost Research
Databases interface, is presented in Table 1. This strategy was
Risk of bias in individual studies
modified as needed and applied to the remaining databases.
All studies were assessed independently for risk of bias by two
authors (Simon Garbellini, Yvette Robert). The PEDro scale [40]
Study selection
was utilized to assess methodological quality of randomized con-
Study selection was conducted in two stages utilizing the trolled trials. Evaluation of methodological quality of intervention
Covidence systematic review software for data management [39]. studies using single-case methodology was completed utilizing
In stage one two authors (Simon Garbellini, Yvette Robert) inde- the risk of bias in N of 1 trials (RoBiNT) scale [41]. All non-random-
pendently screened titles and abstracts against the inclusion/ ized intervention studies were assessed using A cochrane risk of
exclusion criteria and identified relevant papers. In stage two, the bias assessment tool: for non-randomized studies of intervention
same two authors independently reviewed the full text studies (ACROBAT-NRSI) [42]. Using this tool, the highest assessment of
unable to be excluded by title and abstract alone. Comparison of risk of bias for an individual domain is used to determine the

Table 1. Electronic search strategy in MEDLINE complete database.


Search ID# Search terms
S1 (MH “Cerebral Palsy”) OR (MH “Hemiplegia”) OR (MH “Brain Injuries”) OR (MH “Craniocerebral Trauma”) OR (MH “Brain Damage, Chronic”) OR
(MH “Hypoxia, Brain”)
S2 (MH “Brain Hemorrhage, Traumatic”) OR (MH “Brain Stem Hemorrhage, Traumatic”) OR (MH “Cerebral Hemorrhage, Traumatic”)
S3 AB (“cerebral palsy” OR CP OR hemiplegi OR “brain injur” OR “head injur” OR “brain damage” OR hypoxi) OR TI (“cerebral palsy” OR CP OR
hemiplegi OR “brain injur” OR “head injur” OR “brain damage” OR hypoxi)
S4 S1 OR S2 OR S3
S5 (MH “Child”) OR (MH “Child, Preschool”) OR (MH “Adolescent”) OR (MH “Infant”) OR (MH “Infant, Newbornþ”)
S6 AB (child OR infant OR toddler OR babies OR baby OR adoles OR youth OR preschool) OR TI (child OR infant OR toddler OR babies OR
baby OR adoles OR youth OR preschool)
S7 S5 OR S6
S8 S4 AND S7
S9 (MH “Splints”) OR (MH “Orthotic Devices”) OR (MH “Braces”)
S10 AB (splint OR orthot OR orthos OR brace) OR TI (splint OR orthot OR orthos OR brace)
S11 S9 OR S10
S12 (MH “Hand”) OR (MH “Fingers”) OR (MH “Thumb”) OR (MH “Wrist”) OR (MH “Metacarpus”) OR (MH “Upper Extremity”) OR (MH “Elbow”) OR (MH
“Arm”) OR (MH “Hand Deformities”) OR (MH “Hand Deformities, Acquired”) OR (MH “Hand Deformities, Congenital”)
S13 AB (hand OR arm OR elbow OR wrist OR thumb OR “upper limb”) OR TI (hand OR arm OR elbow OR wrist OR thumb OR “upper
limb”)
S14 S12 OR S13
S15 S8 AND S11
S16 S14 AND S15
Terms were modified as needed and the search re-run in each of the Cochrane central register, MEDLINE, CINAHL, SCOPUS and Web of Science databases.
4 S. GARBELLINI ET AL.

Figure 1. Common criteria of the AHTA and ASHT orthosis classification systems (derived from [15,18,21,36]).

overall risk of bias for the study [42]. Moderate risk indicates the for both studies. The risk of bias assessment of the 12
study is judged to be at low or moderate risk for all domains and non-randomized studies is presented in Table 2. Nine of the non-
is sound for a non-randomized study with regard to this domain randomized studies of intervention were assessed with an
but cannot be considered comparable to a well-performed overall moderate risk of bias and three with serious risk of bias
randomized trial. Serious risk of bias indicates the study is judged (see Table 2).
to be at serious risk of bias for at least one domain [42].
Reason for orthosis prescription
Results All children in the included studies who were prescribed an upper
A total of 16 studies were selected for inclusion in this review limb orthosis had a diagnosis of CP and all but one study [58]
after the primary search was completed (see flow diagram of reported that the children had spastic motor type. All children
results in Figure 2). Only one study [43] was selected for full text were seen in an outpatient setting. Table 3 outlines the reasons
review following the second database search. The study did not for orthosis prescription documented in the included studies.
meet inclusion criteria even though it utilized a rigid/thermoplas- Information regarding study design, purpose of the study and
tic wrist and hand orthosis, as the study aim was to evaluate and inclusion criteria is also included in Table 3.
compare the effectiveness of repeated Botulinum toxin-A injec- The reported reasons for orthosis prescription varied in the
tions plus occupational therapy versus occupational therapy alone included studies as seen in Table 3. Two of the 16 studies
[43]. The impact of orthosis use in isolation could not be deter- described a specific reason for orthosis prescription. Barroso et al.
mined by the study design [44]. [49] reported the orthosis was to improve wrist and thumb joint
Two randomized controlled trials [45,46], two single-case positions. Carmick [51] reported the orthosis was to stabilize the
experimental designs [47,48] and 12 non-randomized intervention wrist in a functional position and ’replace the therapist’s hands’,
studies [31,49–59] were included in the review and assessed for to maintain an extended wrist position to encourage motor learn-
risk of bias. The scores on the PEDro scale for the randomized ing. Six studies [45,50,53,54,58,59] prescribed orthoses to manage
controlled trials by Kanellopoulos et al. [45] and Ozer et al. [46] the presentation of spasticity and increased muscle tone, altered
were 4/10 (fair quality) and 7/10 (high quality), respectively. arm and hand position or primary contracture that limited active
Kanellopoulos et al. [45] did not conceal allocation, blind assessors and passive movement. The remaining eight studies (50%)
to the intervention or provide point measures and measures of [31,46–48,52,55–57] did not report the reason for orthosis
variability for outcomes. Ozer et al. [46] did not have a control prescription.
group. Neither study was able to blind therapists administering
therapy or blind subjects to their allocated group, as this is not
Link between reason for prescription and effect
possible in this intervention. The single-case experimental design
studies by Goodman and Bazyk [47] and Kinghorn and Roberts Prescribed reason
[48] assessed with the RoBiNT scale [41] scored 11/30 and 12/30, For the eight studies reporting a reason for orthosis prescription,
respectively. Internal validity ratings suggested high risk of bias data describing the effect of the upper limb orthosis according to
(3/14 and 4/14, respectively). It is difficult to attribute the reported its intended outcome are presented in Table 4. For each of the
improvement in hand function to use of the orthosis as neither eight studies listed, their analyses for orthosis effect found a posi-
study discussed random assignment of treatment times or blind- tive effect. In the remaining eight studies where the reason for
ing of assessors to the intervention [47,48]. The findings may not the orthosis was not reported, it was not possible to present
be generalizable as indicated by external validity scores of 8/16 results exploring the link between reason and effect.
UPPER LIMB ORTHOSES FOR CHILDREN WITH CEREBRAL PALSY 5

Figure 2. Study selection flow diagram.

Outcome measures of effect describing orthoses. Table 5 presents the review author’s
Of the eight studies in which a reason for orthosis prescription classification of the orthoses prescribed in the studies
was provided, only three studies [53,58,59] had a direct link according to anatomical focus and primary purpose of the
between the reason for the orthosis, the outcome measure uti- orthoses.
lized and effect (see Table 4). For example, Yasukawa, Malas [59] In all studies, the orthoses were classified as having an impact
prescribed an elbow orthosis for children where spasticity was on the body function and structure domain of the ICF-CY as all
interfering with range of motion (ROM) at the elbow. The out- were applied directly to the child and positioned the joints of the
come measures utilized included the modified Ashworth scale arm and hand. Only three studies [56,58,59] were classified as hav-
(MAS) (a spasticity measure) and ROM measures. The conclusion ing potential to impact the activity domain of the ICF-CY as the
was a positive effect of improved ROM from baseline. authors of these three studies included assessment of the orthosis
In the five remaining studies there was no clear link between effect on task execution.
the documented reasons for orthosis prescription, intended out- Four of six elbow orthoses prescribed [55,57–59] were mobi-
come, outcome measures utilized and effect (see Table 4). For lizing orthoses aimed at increasing the passive or active ROM
example, the reason described for orthosis prescription by Burtner at the elbow joint. The dynamic spiral wrist splint [50] was a
et al. [50] was spasticity warranting orthotic intervention, but this restrictive orthosis as it allowed 30 of movement but limited
was not supported by any spasticity measure, only the conclusion active wrist flexion ROM. This reflects consideration of the
dynamic interplay between wrist and finger musculature aimed
that grip improved. Kanellopoulos et al. [45] used the quality of
at improving grasp, however the connection between this fac-
upper extremity skills test (QUEST) as the sole outcome measure to
tor and orthosis design is not described in the study. The
evaluate quality of upper extremity function in four domains: disso-
orthoses acting on the wrist and/or wrist and thumb joints
ciated movement, grasp, protective extension and weight bearing
[49–51,54] were classified as immobilizing orthoses aimed at
[60]. No goniometric ROM measures or spasticity measure were uti-
improving grip for task specific or bimanual use. Again there
lized to support the conclusion reported by Kanellopoulos et al.
was no connection between the movement of the wrist and
[45] of improvement in muscle tone, ROM and hand cosmesis.
fingers and orthosis design evident in the study authors’
descriptions. All of the orthoses acting on the thumb
Classification of orthoses [31,47,53] were hand based and immobilized the thumb in
a position out of the palm aimed at developing improved
No studies classified orthoses using the domains of the ICF-CY grasp.
[22] framework or referred to an orthosis schedule when
6 S. GARBELLINI ET AL.

Table 2. Risk of bias assessment of non-randomized studies (n ¼ 12).


Domain of risk of bias
Departure from Selection
Selection of Measurement of intended Measurement of of reported
Study/study design Confounding participants intervention intervention Missing data outcomes results Overall biasd
Barroso et al. [49] Moderateb Moderate Lowa No information Low Moderate Moderate Moderate
Pre/post test
Burtner et al. [50] Moderate Moderate Low No information Low Moderate Moderate Moderate
Pre/post test
Carmick [51] Moderate Seriousc Moderate No information Low Serious Moderate Serious
Case study
Chhawchhria [52] Moderate Moderate Low No information Low Moderate Moderate Moderate
Pre/post test
Currie and Mendiola Moderate Moderate Low No information Low Serious Moderate Serious
[53]
Pre/post test
Exner and Bonder Moderate Moderate Low No information Low Serious Serious Serious
[31]
Pre/post test
Louwers et al. [54] Moderate Moderate Low No information Low Moderate Moderate Moderate
Pre/post test
Postans et al. [55] Moderate Moderate Low No information Low Moderate Moderate Moderate
Case series
Scheker et al. [56] Moderate Moderate Low No information Low Moderate Moderate Moderate
Case series
Yasukawa and Cassar Moderate Moderate Low No information Low Moderate Moderate Moderate
[57]
Case study
Yasukawa et al. [58] Moderate Moderate Low No information Low Moderate Moderate Moderate
Case series
Yasukawa et al. [59] Moderate Moderate Low No information Low Moderate Moderate Moderate
Case series
NB: Studies assessed for risk of bias with the ACROBAT-NRSI [42] in alphabetical order.
a
Low risk of bias indicates the study is comparable to a well-performed randomized trial with regards to the domain.
b
Moderate risk of bias indicates the study is sound for a non-randomized study with regard to the domain but cannot be considered comparable to a well-
performed randomized trial.
c
Serious risk of bias indicates the study has some important problems with regards to the domain.
d
Overall risk of bias is judged at the highest level of risk in any one of the domains [42].

Discussion It is not possible to determine the effect of adjunct interventions


on the reported effectiveness of upper limb orthoses in the stud-
This review aimed to explore reasons for upper limb orthosis pre-
ies in this review.
scription, explore the link between reason and effect according to
The lack of clearly documented reasons for upper limb orthosis
the intended outcome and outcome measures utilized, and to
prescription and the effect of other interventions on upper limb
classify orthoses within the domains of the ICF-CY [22] and com- impairment increases assumptions made regarding the clinical
mon criteria of the ASHT and AHTA orthosis classification systems. decisions for orthosis use and decreases capacity to use the evi-
Our findings suggest that connection between the reported dence to inform clinical practice. The absence of a reason for orth-
rationale for orthosis prescription, the selection of outcome meas- osis prescription impacts the selection of appropriate outcome
ures utilized according to intended outcomes and the effect measure(s) utilized to determine effect, as it is not clear if the
described, is not transparent, nor well defined. All interventional intended outcome is effectively measured. Often there was no
study designs were included in this review to maximize the infor- clear link between the stated reason of the orthosis, the way the
mation available to explore the rationale for orthosis prescription. results were measured and the stated outcome of the study.
If only randomized controlled trials were included, only one study Manual ability and the influence of spasticity on the movement
would have met selection criteria. of the wrist and fingers of the affected limb(s) are important fac-
The systematic review undertaken by Jackman et al. [26] tors to consider in orthosis prescription [19], yet the connection
included randomized and quasi-randomized trials of upper limb between these factors and reason for prescription and description
orthoses. These authors reported there is no evidence supporting of orthoses is not well described in the included studies. Only
the use of orthoses for children with CP in isolation. In contrast three studies [46,49,54] included a description of either the child’s
three studies included in this review [49,50,54] assessed the effect manual ability and/or active hand movement of the affected
of upper limb orthoses while the child wore the orthoses only limb(s). A tool to classify manual ability for children with CP, the
during testing. No additional therapy or adjunct interventions manual ability classification system (MACS) [61], was published in
were reported. The frequency of outcome measures were with 2006 however only two of the seven studies published after this
and without the orthosis within 30 min [49]; twice with and with- date utilized the MACS to report the manual ability level of chil-
out the orthosis within a two week period [50]; at baseline, a dren in their study. Barroso et al. [49] included children at MACS
week later with orthosis wear and a week following without orth- levels I and II and Louwers et al. [54] included children at MACS
osis wear [54]. The reported effect was improved hand function levels I–III. The Zancolli classification [62] is another classification
[49], greater grip dexterity [50] and the ability to grasp objects of tool which reports the action of spasticity on the muscles of the
different shapes without assistance of the dominant hand [54]. wrist and fingers and classifies wrist and finger deformity
UPPER LIMB ORTHOSES FOR CHILDREN WITH CEREBRAL PALSY 7

Table 3. Reason for orthosis prescription (n ¼ 16).


Study/study design Study purpose Inclusion criteria Reason for orthosis prescription
Barroso et al. [49] To investigate indicators of hand improve- 5 to 12 years; Spastic hemi CP; Limited To improve position of hypertonic hand
Pre/post test ment when a wrist extending/thumb wrist and thumb ROM; MACS levels I/ Indicated by treating OT or PT
abduction (WETA) orthosis is used II; Able to follows instructions
Carmick [51] To describe a task-specific physical therapy Not reported To reduce wrist flexion
Case study approach using NMES and a dorsal wrist “Replace therapist’s hands” for motor
splint to aid upper limb function learning
Burtner et al. [50] To compare grip, pinch, dexterity and EMG Not reported Moderate spasticity warranting splinting
Pre/post test activity of selected UE muscles while par-
ticipants with and without CP wore (i)
no splint or (ii) static or (iii) dynamic
splints
Currie and Mendiola [53] To note whether thumb position improved Not reported Mild to moderate hemi CP
Pre/post test while wearing the orthosis “Cortical” thumb position limiting use
To monitor any change in function of the
paretic hand while wearing the orthosis
Kanellopoulos et al. [45] To evaluate the necessity and effectiveness Not reported Primary contracture in the upper limb
RCT of a static night splint following out-
patient botulinum toxin A treatment in
children with upper limb spastic CP
Louwers et al. [54] To investigate immediate effects of a static 4–12 years; Spastic hemi CP; Zancolli Hemiplegic pattern of the upper limb
Pre/post test wrist and thumb brace on the spontan- classification of I, IIA, or IIB
eous use of the affected upper limb in
bimanual activities in children with
hemi CP
Yasukawa et al. [58] To determine the efficacy of an ultraflex CP; Limited ROM; Abnormal muscle tone Abnormal muscle tone interfering with
Case series orthosis with dynamic elbow and static on MAS range of motion or functional
wrist components in maintaining or movement
improving PROM of the elbow and wrist
Yasukawa et al. [59] To measure the efficacy of maintaining Limited ROM; Severe to moderate spasti- Severe to moderate spasticity interfering
Case series range of motion with the use of the city at the elbow with passive or active movements at
bivalve long arm cast and the ultraflex the elbow
orthosis following botulinim toxin type A
injection and serial casting when worn
for an eight month period
Chhawchhria [52] To determine the effect of and compare an 18 months to 8 years; Spastic CP any Not reported
Pre/post test above elbow wrist hand orthosis topography; MAS of 1þ or greater
(AEWHO) and elbow gutter with below
elbow wrist hand orthosis (BEWHO) on
spasticity and hand function
Exner and Bonder [31] To investigate the effects of three hand 2–16 years; Spastic type; at least 18 Not reported
Pre/post test splints on bilateral hand use, grasp skills, months developmental level; able to
and arm-hand posture in children with follow instructions
hemiplegia
Goodman and Bazyk [47] To measure the effects of a short opponens Not documented Not reported
Single case design splint on hand function and on the
underlying aspects of hand function in a
child with moderate spastic quadriparesis
Kinghorn and Roberts [48] To investigate the use of an upper-extrem- 20 month old; spastic quadriplegia (age Not reported
Single case design ity weight-bearing splint on muscle tone and motoric involvement resembling
and functional hand skills in a child with Smelt’s (1989) subject)
cerebral palsy
Ozer et al. [46] To investigate whether the improvement in 3 to 18 years; Spastic hemi; Zancolli clas- Not reported
RCT the hand function was due to the effect sification type II or III; follows direc-
of NMES, dynamic bracing or a combin- tions; Sensation to light touch
ation of the two methods
Postans et al. [55] To investigate the feasibility of applying a Spastic CP; Limited ROM; follows orders Not reported
Case series custom-made dynamic splint with NMES,
to the wrist or elbow joint (or both)
Scheker et al. [56] To assess the effects of dynamic orthotic 3–21 years; spastic hemi CP, mild to Not reported
Case series and static traction and NMES on upper moderate spasticity; follows direc-
limb spasticity in cerebral palsy patients tions; Good sensation
Yasukawa and Cassar [57] Not reported Not reported Not reported
Case study
Hemi: hemiplegic; CP: cerebral palsy; ROM: range of motion; MACS: manual ability classification system; OT: occupational therapy; PT: physical therapy; NMES: neuro-
muscular electrical stimulation; EMG: electromyography; UE: upper extremity; PROM: passive range of motion; MAS: modified Ashworth scale.
NB: The order of the studies reflects the progression from well documented specific reasons to absence of documented reasons for orthosis prescription.
8
S. GARBELLINI ET AL.

Table 4. Effect of upper limb orthoses according to intended outcome (n ¼ 8).


Participants Orthosis Orthosis wear Outcome measures
Conclusion
Sample Topographical Joint(s) position in Duration
Study/study design size (M/F) Age range (Mean) distribution Reason Name of orthosis orthosis Hours of wear Timing of wear of wear Measure Frequency Study conclusion
Currie and Mendiola 5 (no 20–26 months (not Hemiplegia Mild to moderate hemi- The cortical thumb Thumb abduction During all waking hours During all waking One week Clinical observa- Pre orthosis and one Improved thumb
[53] report) reported) plegic CP, “Cortical” orthosis hours tions, photos, week later with position, hand
Pre/post test thumb position limit- prehension orthosis function and
ing use pattern prehension
Yasukawa et al. [58] 6 (4/2) 7–16 years (12 years) Not reported Abnormal muscle tone Ultraflex orthosis Not described Not reported As much as pos- 10 months MAS, Baseline, 2, 3, 6 and 10 Positive correlation
Case series interfering with ROM sible requested, PROM, months of wear and
or functional report of day Caregiver interview ROM
movement wear only
Yasukawa et al. [59] 3 (1/2) 7 years (7 years) Quadriplegia Severe to moderate Ultraflex orthosis Elbow extension, fore- Average of 4 or more Night wear Eight months MAS, Pre BoNT-A, 2, 6 and 8 Improved ROM
Case series spasticity interfering arm and wrist in hours each day PROM, months post from baseline
with passive/active neutral alignment Caregiver injection
ROM at the elbow 
questionnaire
Barroso et al.† [49] 32 (15/ 5–11 years (8.65 years) Hemiplegia To improve hypertonic WETA orthosis Wrist joint 20 exten- Not reported During data collec- Not reported Digital images for With and without orth- Improved hand
Pre/post test 17) hand position; sion; tion with the ROM, osis at data function
Indicated by OT or PT Thumb abduction and orthosis on Muscle strength, collection
extension

JTTHF
Burtner et al.† [50] 15 (6/9) 4–13 years (8.5 years) Hemiplegia Moderate spasticity war- 1. Volar wrist immobiliz- 15-20 wrist extension Not reported During testing only Not reported Grip and pinch With and without the Greater grip dexter-
Pre/post test ranting splinting ing splint strength, orthosis at data ity with
2. Dynamic spiral wrist Adapted nine hole collection dynamic spiral
hand splint 
peg test, EMG splint
Carmick† [51] 1 (1/0) 7 years 7 months (7 Hemiplegia To reduce wrist flexion; Dorsal wrist splint 10 wrist extension 6 hours/day at school During home pro- Nine months Mowery’s functional Pre and post Functional gains
Case study years 7 months) “Replace therapist’s gram at least classification
hands” three times a
week
Louwers et al.† [54] 25 (16/9) 4–11 years (8 years 4 Hemiplegia Hemiplegic pattern of Children's wrist and Neutral wrist, thumb During assessment only During the second During AHA Pre-test; Positive effect for 9
Pre/post test months) the upper limb thumb support by abduction and AHA only assessment One week later with of 22 AHA
Otto Bock opposition only brace; items
Post-test (one week
later)
Kanellopoulos et al.† 20 (13/7) 2.5–12 years (7 years) Hemiplegia Primary contracture in Static night splint Neutral wrist, thumb Not reported Night time Six months QUEST Baseline, 2 and 6 Improved muscle
[45] the upper limb abduction, fingers months post tone,
RCT extended injection ROM and cosmesis
(M/F): (Male/Female); ROM: range of motion; MAS: modified Ashworth scale; PROM: passive range of motion; BoNT-A: Botulinum toxin type-A; OT: occupational therapy; PT: physical therapy; WETA: wrist extending thumb abduc-
tion; JTTHF: Jebsen Taylor test of hand function; EMG: electromyography; CP: cerebral palsy; AHA: assisting hand assessment; QUEST: quality of upper extremity skills test.
NB: Studies have been presented in order of those  with a link between orthosis reason, outcome measure and effect and those † without a link.
UPPER LIMB ORTHOSES FOR CHILDREN WITH CEREBRAL PALSY 9

Table 5. Review author’s classification of orthoses according to anatomical focus and primary purpose.
Reviewer classification
Orthosis classification system
Study Orthosis ICF domain(s) Articular joint/anatomical focus Primary purpose
Postans et al. [55] Dynamic splint (Elbow) BF & S and activity Elbow Mobilizing
Yasukawa and Cassar [57] The forearm rotation elbow orthosis (FREO) BF & S and activity Elbow and forearm Mobilizing
Yasukawa et al. [58] Ultraflex orthosis BF & S Elbow and wrist Mobilizing
Yasukawa et al. [59] Ultraflex orthosis BF & S Elbow and wrist Mobilizing
Ozer et al. [46] Ultraflex orthotic device BF & S and activity Elbow, forearm, wrist and fingers Immobilizing
Chhawchhria [52] Above elbow wrist hand orthosis (AEWHO) BF & S and activity Elbow, wrist, fingers and thumb Immobilizing
Burtner et al. [50] Volar wrist immobilization splint BF & S and activity Wrist Immobilizing
Dynamic spiral wrist hand splint BF & S and activity Wrist Restrictive
Carmick [51] Dorsal wrist splint BF & S and activity Wrist Immobilizing
Postans et al. [55] Dynamic splint (Wrist) BF & S and activity Wrist Mobilizing
Chhawchhria [52] Below elbow wrist hand orthosis (BEWHO) BF & S and activity Wrist, fingers and thumb Immobilizing
Kanellopoulos et al. [45] Static night splint BF & S and activity Wrist, fingers and thumb Immobilizing
Barroso et al. [49] Wrist extending/thumb abduction (WETA) BF & S and activity Wrist and thumb Immobilizing
Louwers et al. [54] Children's wrist and thumb support BF & S and activity Wrist and thumb Immobilizing
Scheker et al. [56] Dynamic orthotic traction BF & S Fingers (MCP joints) Mobilizing
Kinghorn and Roberts [48] Inhibitive weight-bearing splint BF & S and activity Fingers and thumb Immobilizing
Currie and Mendiola [53] The cortical thumb orthosis BF & S and activity Thumb Immobilizing
Exner and Bonder [31] Short opponens thumb splint BF & S and activity Thumb Immobilizing
Goodman and Bazyk [47] Volar-type short thumb opponens splint BF & S and activity Thumb Immobilizing
NB: Studies have been organized by the classification of joint(s) addressed by the orthosis. Exner and Bonder [31] compared the effects of three different orthoses
on hand use, grasp and posture. The short opponens thumb splint was the only orthosis of Exner and Bonder’s study that met the rigid/thermoplastic inclusion cri-
teria of this review and therefore was the only orthosis in the study classified by the reviewers.
BF & S: body function and structure; Body functions are physiological functions of body systems; Body structures are anatomical parts of the body; Activity is an exe-
cution of a task or action by an individual [22]; MCP: metacarpophalangeal joints. Mobilizing orthoses aimed to gain specific passive and active range of motion;
Immobilizing orthoses prevent movement at joints; Restrictive orthoses restrict ranges of movement [15,21,36].

according to observed movements. Only two studies, Ozer et al. heterogeneity and lower level of evidence within the included
[46] and Louwers et al. [54] classified children using this classifica- studies.
tion. All children in these two studies had voluntary extension of
their wrist and/or fingers. Understanding how manual ability and
Recommendations for practice and research
movement limitations of the wrist and finger musculature of the
affected limb(s) inform the reason for and choice of orthosis is Evidence to inform the use of upper limb orthoses for children
critical. with CP in clinical practice is limited. This may be because it is dif-
All studies included in the review were evaluated to have ficult to research a complex multi-factorial intervention rather
some risk of bias. Therefore, the assessment of the effect of the than because the intervention lacks effect. Under these circum-
orthoses should be taken in context of the quality of the individ- stances it is strongly recommended that continued clinical use of
ual studies. Caution is required in generalizing the reported effect upper limb orthotic intervention should only occur with the inclu-
of orthotic intervention in this review. The positive effects sion of the following practice recommendations:
reported in the studies did not match the initial reason for the  A description of the dynamic interplay of the wrist and fin-
orthosis. This consistent mismatch between reason for prescription ger musculature as part of a comprehensive upper limb
and measured effect significantly limits our understanding of the assessment;
utility of orthoses for children with CP. It is critical in any interven-  Clear documentation of the use of the orthosis as an inter-
tion research (or intervention prescription) that the mechanism by vention including the aim(s) of the orthosis based upon
which the intervention is intended to effect change is demon- assessment findings;
strated. If associated, or additional, effects are anticipated, for  A description of the orthosis using consistent terminology
example, improved use of the hand following orthosis wearing, it applying an orthosis classification schedule such as those
is certainly appropriate to also measure those effects. However, in endorsed by the AHTA or ASHT; and
the absence of knowing whether the primary mechanism of the  Application (pre, during and post) of intervention outcome
orthosis is supported, it is difficult to interpret subsequent poten- measures that are consistent with the reasons for the orth-
tial impacts. osis when evaluating the effect of the orthotic
intervention.
Future clinical research is required to understand the factors
Limitations of review
that influence clinicians’ rationale for prescribing orthoses as well
There were limitations of this review related to the search criteria, as establishing the effect of upper limb orthotic intervention
study design selection and evaluation of effect of intervention. through connecting the reason for prescribing the orthosis,
Because the search was restricted to studies written in English, intended outcomes and effect utilizing valid and reliable outcome
four studies were excluded that may have provided additional evi- measures.
dence. Study design selection included observational studies to
determine the reason for the use of upper limb orthoses but,
Conclusions
given the lower level of evidence this limited the evaluation and
generalization of effectiveness of the intervention. It was not pos- The lack of clear evidence about the effect of upper limb orthoses
sible to synthesize the data using statistical analyses given the leads to many challenges for clinicians who are considering
10 S. GARBELLINI ET AL.

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