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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
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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
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
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
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 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.
offering upper limb orthotic intervention for children with CP. The [10] Rosenbaum P, Paneth N, Leviton A, et al. A report: the def-
variability in design, materials and presumed mechanism of effect inition and classification of cerebral palsy April 2006. Dev
of upper limb orthoses further increases the difficulty clinicians Med Child Neurol Suppl. 2007;109:8–14.
have in determining whether or not the intervention has value. [11] Vaz DV, Cotta M, Fonseca ST, et al. Muscle stiffness and
Variability in terminology and stated rationale of orthoses used strength and their relation to hand function in children
creates a communication barrier and introduces considerable diffi- with hemiplegic cerebral palsy. Dev Med Child Neurol.
culty in synthesizing research evidence to determine the out- 2006;48:728–733.
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Damiano D, Mayston M, editors. Management of the motor
disorders of children with cerebral palsy. Clinics in develop-
Acknowledgements mental medicine no. 161. 2nd ed. London: Mac Keith Press;
Simon Garbellini’s doctoral studies are nested within two national 2004. p. 105–129.
multicentre randomized controlled trials of upper limb orthoses [13] Georgiades M, Elliott C, Wilton J, et al. The neurological
for children with CP led by Professor Christine Imms. The trials are hand deformity classification for children with cerebral
funded by the Australian Catholic University (REF 2013000413) palsy. Aust Occup Ther J. 2014;61:394–402.
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Melinda Randall http://orcid.org/0000-0002-1908-5070 of neurological dysfunction. Hand splinting orthotic inter-
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