master in de ergotherapeutische wetenschap...versterkt (buccino et al., 2012; kim, & ko, 2014;...
TRANSCRIPT
Faculteit Geneeskunde en Gezondheidswetenschappen
The efficacy of a novel upper limb intensive training model on participation and quality of
life in children with unilateral cerebral palsy.
Marieke VANHALST
Masterproef ingediend tot
het verkrijgen van de graad van
Master of Science in de ergotherapeutische wetenschap
Promotors: Prof. Dr. Hilde Feys
Dr. Katrijn Klingels
Dr. Dominique Van de Velde
Academiejaar: 2015-2016
MASTER IN DE ERGOTHERAPEUTISCHE WETENSCHAP
Interuniversitaire master in samenwerking met:
UGent, KU Leuven, UHasselt, UAntwerpen,
Vives, HoGent, Arteveldehogeschool, AP Hogeschool Antwerpen, HoWest, Odisee, PXL,
Thomas More
Faculteit Geneeskunde en Gezondheidswetenschappen
The efficacy of a novel upper limb intensive training model on participation and quality of
life in children with unilateral cerebral palsy.
Marieke VANHALST
Masterproef ingediend tot
het verkrijgen van de graad van
Master of Science in de ergotherapeutische wetenschap
Promotors: Prof. Dr. Hilde Feys
Dr. Katrijn Klingels
Dr. Dominique Van de Velde
Academiejaar: 2015-2016
MASTER IN DE ERGOTHERAPEUTISCHE WETENSCHAP
Interuniversitaire master in samenwerking met:
UGent, KU Leuven, UHasselt, UAntwerpen,
Vives, HoGent, Arteveldehogeschool, AP Hogeschool Antwerpen, HoWest, Odisee, PXL,
Thomas More
Abstract
Background. Therapy camps using modified constraint movement therapy (mCIMT)
have shown their efficacy in upper limb rehabilitation of children with unilateral cerebral
palsy (UCP). Recently, Action observation therapy (AOT) in children with UCP gained
emerging evidence targeting motor planning. However, it is unclear if AOT has an added
value to mCIMT. Further, the effect of these intervention models on participation and
quality of life (QOL) have only scarcely been studied.
Objective. To investigate the effects of a novel intensive training model and the additional
effects of AOT to mCIMT on participation and QOL in children with UCP.
Method. Twenty children (median age 10y 5mo [IQR= 9,5mo - 11,6mo]) with UCP were
randomly assigned to a combined group (mCIMT+ AOT) and a mCIMT group. All
children had to wear a constraint on the unaffected hand in a nine-day camp, six hours a
day, while performing unimanual exercises. Children in the combination group received
two sessions of AOT each day. Measurements of participation (LIFE-H) and quality of
life (CP-QOL) were performed before and six months after intervention. In addition,
qualitative interviews targeting the lived experience of the children were conducted after
the therapycamp.
Results. No meaningful between-group differences, as an outcome of the intervention,
could be found. Nevertheless, this study demonstrated significant changes measured on
the CP-QOL on the domain feelings about functioning (p=0.03) for the total group.
Qualitative data confirmed research results.
Conclusion. mCIMT yielded significant improvements on QOL. AOT provided no
additional effect on participation and QOL measures.
Number of words master thesis: 11 930 (excluding appendices and bibliography)
Abstract
Inleiding. Therapiekampen, die gebruik maken van gemodificeerde constraint-induced
movement therapy (mCIMT) zijn doeltreffend in de behandeling van arm- en handfunctie
bij kinderen met een unilaterale cerebrale parese (UCP). Recent won actie observatie
therapie (AOT), gericht op motorische planning, aan evidentie bij kinderen met UCP. Het
is niet duidelijk of AOT een toegevoegde waarde heeft op mCIMT. Daarnaast werd het
effect van deze interventies op het gebied van participatie en kwaliteit van leven (QOL)
nauwelijks bestudeerd.
Doel. Effect nagaan van een trainingsmodel en het additioneel effect van AOT op mCIMT
onderzoeken op het vlak van participatie en QOL bij kinderen met UCP.
Methode. Twintig kinderen (mediaan leeftijd 10j en 5m [IQR= 9,5 m – 11,6 m]) met UCP
werden gerandomiseerd naar een gecombineerde (AOT+mCIMT) en mCIMT groep. Alle
kinderen droegen tijdens een kamp van negen dagen, zes uur per dag een spalkje aan hun
niet-aangedane zijde en voerden unimanuele oefeningen uit. Kinderen in de
combinatiegroep kregen dagelijks twee sessies AOT. De LIFE-H en CP-QOL werden
afgenomen vóór en zes maand na de interventie. Daarnaast werden kwalitatieve
interviews over de kinderen hun beleving afgenomen na het kamp.
Resultaten. Tussen de groepen werden geen betekenisvolle verschillen, als een uitkomst
van de interventie, gevonden. Desalniettemin werden voor de totale groep significante
verschillen aangetoond op het domein ‘gevoelens omtrent functioneren’ (p=0.03) van de
CP-QOL. Kwalitatieve data bevestigde deze resultaten.
Conclusie. mCIMT toonde significante verbeteringen aan op het vlak van levenskwaliteit.
Er kon geen additioneel effect van AOT aangetoond worden op het vlak van participatie
en QOL.
Aantal woorden masterproef: 11 930 (exclusief bijlagen en bibliografie)
Table of contents
Woord vooraf
Situering
Introduction .................................................................................................................................................. 1
Method primary outcomes ............................................................................................................................ 7
Participants ............................................................................................................................................... 7
Design ....................................................................................................................................................... 7
Intervention .............................................................................................................................................. 8
Assessment ............................................................................................................................................. 10
Descriptive information and classification measures ......................................................................... 10
Primary outcomes intervention .......................................................................................................... 11
Statistical Analysis ................................................................................................................................. 13
Method Secondary outcomes ..................................................................................................................... 14
Theoretical basis ..................................................................................................................................... 14
Data collection ........................................................................................................................................ 14
Data analysis........................................................................................................................................... 14
Results ........................................................................................................................................................ 16
Participants ............................................................................................................................................. 16
Baseline comparisons between groups ................................................................................................... 18
Efficacy of the intervention .................................................................................................................... 20
Primary outcomes ............................................................................................................................... 20
Secondary outcomes ........................................................................................................................... 25
Discussion .................................................................................................................................................. 29
Conclusion .................................................................................................................................................. 36
References .................................................................................................................................................. 37
Appendix I, Day structure .......................................................................................................................... 45
Appendix II, List of Goal-directed actions of AOT (House Functional Classification 4-5) ....................... 46
Appendix III, List of Goal-directed actions of AOT (House Functional Classification 6-7) ..................... 47
Appendix IV, Psychometric properties LIFE-H & CP-QOL...................................................................... 48
Appendix V, LIFE-H .................................................................................................................................. 53
Appendix VI, CP-QOL ............................................................................................................................... 58
Appendix VII, Additional tables ................................................................................................................ 63
Appendix VIII, ‘We are in the newspaper!’ ............................................................................................... 64
Appendix IX, Toestemming tot consultatie ................................................................................................ 70
List of figures
Figure 1. Classification tree for subtypes of Cerebral palsy, SCPE Collaborative Group ................... 1
Figure 2. Flowchart: number of the children participating in the study ............................................. 17
Figure 3. Individual overall scores on the LIFE-H at pre and post measurement .............................. 20
Figure 4. Individual overall scores on the CP-QOL at pre and post measurement ............................. 22
Figure 5. Individual scores on the domain feelings about functioning on the CP-QOL
at pre and post measurement................................................................................................ 23
List of tables
Table 1. House classification scale ..................................................................................................... 10
Table 2. MACS ................................................................................................................................... 11
Table 3. Demographics and baseline characteristics of participatns in the mCIMT group and
mCIMT+AOT group ............................................................................................................ 20
Table 4. Descriptive statistics of the LIFE-H before and after intervention,
and statistical comparison .................................................................................................... 22
Table 5. Descriptive statistics of the CP-QOL before and after intervention,
and statistical comparison .................................................................................................... 23
Woord vooraf
Deze masterproef kwam tot stand door de inbreng van velen. In het bijzonder gaat mijn
dank uit naar mijn promotors professor Hilde Feys, professor Katrijn Klingels en Dra Lisa
Maillieux. Hun toewijding voor onderzoek en kinderen met een cerebrale parese gaf de
aanzet tot een boeiende studie en ontzettend leuke therapiekampen. Door hun intensieve
begeleiding was het uitwerken van deze thesis een verrijkende en fijne ervaring. Hun
betrokken manier van samenwerken, zowel als therapeut gedurende de kampen als
promotor, was voor mij zeer motiverend. Bijzondere dank gaat ook uit naar Dr.
Dominique Van de Velde voor zijn verhelderende gesprekken en waardevolle
ergotherapeutische beschouwingen in functie van deze thesis.
De zomerkampen kwamen tot stand door verschillende medewerkers, therapeuten en
studenten. Hen wil ik bedanken voor de aangename samenwerking. In dit ‘Woordje
vooraf’ wil ik verder de ouders en de kinderen bijzonder bedanken. Hun medewerking
tijdens de kampen en hun doorzettingsvermogen tijdens de pre- en post- testen maakten
deze masterthesis mogelijk.
Tenslotte wil ik mijn vriend en mijn ouders bedanken voor hun jarenlange
onvoorwaardelijke steun tijdens mijn opleiding tot Master of Science in de
ergotherapeutische wetenschap.
De tekst die volgt is het eindresultaat van 2,5 jaar engagement in de studie naar het effect
van een intensief kampmodel voor kinderen met een halfzijdige verlamming en
jarenlange interesse in neurologische ontwikkelingsstoornissen bij kinderen. Ik ben mijn
promotors, de ondersteunende onderzoekers, de participerende kinderen, hun ouders en
medestudenten ontzettend dankbaar voor deze onvergetelijke ervaring.
Ik wens u, moedige lezer, veel leesplezier.
Marieke Vanhalst, 1 mei
Situering
Deze masterproef maakt deel uit van een onderzoekslijn over de evaluatie en behandeling
van arm- en handfunctie bij kinderen met een unilaterale Cerebrale Parese (UCP) binnen
de onderzoeksgroep neuromotorische revalidatie van het departement revalidatie-
wetenschappen aan de Katholieke Universiteit Leuven.
Een cerebrale parese (CP) is wereldwijd de meest voorkomende oorzaak van een fysieke
handicap bij kinderen en heeft een prevalentie van 2,11 op 1000 levende geboortes (Bax,
Goldstein, Rosenbaum, Leviton, & Paneth, 2005). CP ontstaat in de vroege ontwikkeling
van de hersenen tijdens de zwangerschap, rond de bevalling of kort na de geboorte. CP is
een verzamelnaam voor een groep blijvende, niet-progressieve letsels in de hersenen die
leiden tot beperkingen in het dagelijks leven (Rosenbaum et al., 2007). CP is een
heterogene groep en de ernst van de aandoening is afhankelijk van de leeftijd, het tijdstip,
de grootte en locatie van het hersenletsel (Bax et al., 2005). Zesendertig procent van de
kinderen met CP heeft een unilaterale cerebrale parese (UCP) (Stanley, Blair, &
Alberman, 2000).
UCP is een subtype waarbij de hersenbeschadiging voornamelijk gelokaliseerd is in de
linker of rechter hersenhelft. Het bovenste lidmaat is vaak meer aangedaan dan het
onderste lidmaat. Als gevolg van verminderde kracht, spasticiteit en beperkingen in
sensoriek is het effectief gebruik van de arm en hand om te reiken, grijpen, loslaten en
om objecten te manipuleren vaak aangetast (Sakzewski, Ziviani, & Boyd, 2009). Goed
functionerende bovenste ledematen zijn echter belangrijk voor functionele
onafhankelijkheid in vele activiteiten van het dagelijks leven (ADL) (Conell, Johnston,
& Kerr, 2012).
Een populaire therapiemethode in de behandeling van het aangedane bovenste lidmaat bij
kinderen met UCP is constraint induced movement therapy (CIMT). Doordat kinderen
met UCP vaak problemen hebben met het efficiënt gebruiken van hun aangedane hand en
arm, leren ze vaak effectieve strategieën om taken uit te voeren met één hand (Hoare,
Imms, Rawicki, & Carey, 2010).
CIMT is een therapiemethode, ondersteund door theorieën van motorisch leren en
neuroplasticiteit, die de aangedane hand en arm intensief gaat trainen door het gebruik
van de niet-aangedane zijde te verhinderen. Kinderen dragen hierbij een handspalkje en
voeren unimanuele repetitieve oefeningen uit met hun aangedane zijde (Hoare et al.,
2010; Sakzewski et al., 2009). mCIMT is een gemodificeerde vorm van CIMT waarbij
het soort spalkje kindvriendelijker en de dosis van de interventie minder intensief werden
gemaakt. Vele studies toonden reeds de effectiviteit aan van CIMT en mCIMT op
functie- en activiteitenniveau bij kinderen met UCP (Charles, Wolf, Schneider, &
Gordon, 2006; Eliasson, Krumlinde-Sundholm, Shaw, & Wang, 2005; Sakzewski et al.,
2009; Sakzewski et al., 2011; Sakzewski et al., 2012; Wallen, Ziviani, Herbert, Evans, &
Novak, 2008). Tot op heden werd het effect van deze therapieën op andere domeinen
zoals participatie en kwaliteit van leven slechts beperkt onderzocht. Verder werd het
meeste onderzoek bij kinderen met UCP verricht naar behandelingen, waaronder CIMT
en mCIMT, die gericht zijn op de motorische uitvoering van taken. Recente studies
hebben echter aangetoond dat tekorten in motorische planning even belemmerend kunnen
zijn bij de performantie van acties (Crajé, van der Kamp, & Steenbergen, 2009;
Steenbergen & Gordon, 2006; Steenbergen et al., 2007).
Actie observatie therapie (AOT) is gebaseerd op deze recente bevindingen. AOT heeft
als doel de motorische vaardigheden en motorische planning te verbeteren. In AOT
worden motorische acties, uitgevoerd door anderen, herhaaldelijk via videobeelden
geobserveerd en veelvuldig geïmiteerd. Dit proces wordt gefaciliteerd door het
spiegelneuronen-systeem dat wordt geactiveerd bij zowel het observeren als uitvoeren
van geobserveerde acties (Rizzolati, Fogassi, & Gaselle, 2001). De eerste resultaten van
AOT bij kinderen met CP en UCP zijn gunstig, maar er is verder onderzoek nodig om na
te gaan of AOT een toegevoegde waarde heeft en dus de behandeleffecten van mCIMT
versterkt (Buccino et al., 2012; Kim, & Ko, 2014; Sgandurra et al., 2013). Vorige
eindwerken van dit onderzoeksproject hebben zich gericht op de effecten mCIMT en
mCIMT in combinatie met AOT op het functie- en activiteitenniveau bij kinderen met
UCP. In dit eindwerk worden de effecten van mCIMT in een intensief trainingsmodel en
de toegevoegde waarde van AOT op mCIMT op de participatie en kwaliteit van leven bij
kinderen met UCP onderzocht.
1
Introduction
Cerebral palsy (CP) can be defined as a “group of permanent disorders of the development
of movement and posture, causing activity limitations that are attributed to non-
progressive disturbances that occurred in the developing fetal or infant brain. The motor
disorders of CP are often accompanied by disturbances of sensation, perception,
cognition, communication, behavior, by epilepsy and by secondary musculoskeletal
problems” (Rosenbaum et al., 2007).. It is the most common childhood physical disability
with a prevalence of 2,11 per 1000 live births (Bax et al., 2005). CP is a heterogeneous
group and the severity of the disorder depends on the timing of the brain insult, the size
and location of the lesions. CP is an early-onset condition but persists through the lifespan
of the child (Bax et al., 2005). The group for the Surveillance of Cerebral Palsy in Europe
(SCPE) developed a classification tree (shown in figure 1) to distinguish types of CP
based on the predominant neurological characteristics of a child (SCPE Collaborative
Group, 2004). Children with CP often experience neurological symptoms secondary to
the brain disturbance like spasticity, dystonia, athetosis and ataxia (Stanley, Blair, &
Alberman, 2000).
Figure 1. Classification tree for subtypes of cerebral palsy, SCPE Collaborative Group
2
Thirty-six percent of the general population of children with CP have unilateral cerebral
palsy (UCP) (Stanley et al., 2000). UCP is a subtype, based on the topography of the
limitations of the body, where brain damage is primarily localized in the left or right
hemisphere. Typically, the upper limb (UL) is more affected than the lower limb.
Effective use of the arm and hand to reach, grasp, release, and manipulate objects is often
compromised because of impairments of spasticity, sensation, and reduced strength
(Sakzewski et al., 2009). A well-functioning UL is important because it enables
functional independence in many activities of daily living (ADL) (MC Conell et al.,
2012). Although children with UCP generally have the intellectual capacity to attend
regular school, impaired arm and hand function decreases their chances to fully
participate in educational, leisure and later vocational roles (Sakzewski et al., 2009).
These reduced opportunities to participate in meaningful life situations could cause
limitations in daily independence and quality of life (Sakzewski, Ziviani, & Boyd, 2014).
Physiotherapists and occupational therapists are key members of the multidisciplinary
team for the management of the UL in children with UCP (MC Conell, Johnston, & Kerr,
2012). Numerous UL rehabilitation approaches have been reported in the literature such
as neurodevelopmental treatment (NDT), constraint-induced movement therapy (CIMT),
hand arm bimanual intensive training (HABIT), splinting and casting, intramuscular
injection of botulinum toxine, focal and generalized pharmacotherapy and surgery
(Sakzewski et al., 2014).
Over the past decade, the interest in CIMT has increased exponentially (Eliasson et al.,
2013). Children with UCP often disregard or learn not to use the impaired UL during their
development of motor functions. Developmental disregard is a phenomenon in which
children, with a mild to severe impairment in the UL, learn effective strategies to execute
tasks with one hand and perceive these as the most efficient way to manage ADL (Hoare
et al., 2010). CIMT is a therapy method, underpinned by theories of motor learning and
neuroplasticity, that aims to reverse the behavioral suppression of movement in the
impaired UL by constraining the non-affected limb and providing extensive practice of
the affected hand and arm (Hoare et al., 2010; Sakzewski et al., 2009).
3
Until today, there are no international guidelines for the implementation of CIMT and
there has been a wide variation in content and intensity of CIMT (Eliasson et al., 2013).
CIMT has generally been reported with a total dosage ranging from 60 to 126 hours
(Sakzewski et al., 2009). The duration of CIMT varies greatly across studies, up to six
hours of daily training and from three to seven trainings a week. CIMT programmes
reported in literature varied in length from two to 10 weeks (Eliasson et al., 2013)..
Several studies indicated that CIMT in children with UCP improves the efficiency and
quality of UL movement, increases spontaneous use of the affected limb in ADL, and that
it has a large treatment effect for the development of new UL motor skills (Taub, Ramey,
DeLuca, & Echols, 2004; Sakzewski et al., 2009). CIMT showed improvements in
functions, structures, activities and a few studies showed domain-specific changes in
quality of life and participation (Sakzewski et al., 2011; Sakzewski et al., 2012; Wallen
et al., 2008).
Because CIMT is a highly intense intervention, modified constrained-induced movement
therapy (mCIMT) has been developed. In mCIMT, modifications in terms of therapy
delivery, dose of intervention and type of applied restraint (e.g. glove, mitt, sling) have
been made to make it more child friendly. mCIMT also showed modest to large effects
on improving efficiency and quality of movement of the impaired UL compared with
usual care. These results were reported in several studies and meta-analysis (Charles et
al., 2006; Eliasson et al., 2005; Sakzewski et al., 2009). The effect of mCIMT on
participation and QOL has not been studied.
Currently some studies suggest that disrupted action performances in children with UCP
are not solely due to impairments at the level of motor execution but are also related to
problems with action planning. Steenbergen et al. (2007) stated that most research on
motor control in UCP has focused on problems related to movement execution, while
deficits in action planning may be just as limiting for action performances (Crajé et al.,
2009; Steenbergen & Gordon, 2006; Steenbergen et al., 2007).
4
Mutsaarts et al. (2006) describe action planning as: “the ability to anticipate the
forthcoming perceptual-motor demands of the action goal when a first movement in a
sequence movement towards an object is made”. In sequential tasks and tasks that involve
the use of objects, this becomes especially apparent (Steenbergen, Meulenbroek, &
Rosenbaum, 2004).
Action- observation therapy (AOT) is a novel UL rehabilitation approach based on these
recent findings. AOT aims to enhance motor skills and motor planning by observing
motor actions conducted by others and repeated training of those actions by imitating
them. This process is facilitated by the mirror neuron system (MNS), which has been
shown through brain imaging studies (Rizzolati et al., 2001). By observing actions, the
same neural structures are activated as those involved in actual actions. Specifically, the
primary motor cortex contralateral to the viewed hand, which corresponds to the paretic
limb, were activated when children with CP observed actions (Kim et al., 2014). Positive
effects of AOT has been reported in adults with stroke, Parkinson’s disease, and during
recovery from orthopedic surgery (Belleli, Buccino, Bernardini, Padovani, Trabucchi,
2010; Bucciono, Gatti, & Giusti, 2010; Ertelt, Small, & Solodkin, 2007; Pelosin et al.,
2010).
AOT possibly recruits the same neural structures in the brain as motor imagery (Buccino
et al., 2012; Jeannerod, 2001). Motor imagery is a mental process by which an individual
simulates a given action without actually seeing or performing the action. This
intervention has been successfully used as a rehabilitative tool in neurological
rehabilitation (Mulder, 2007). Despite the fact that motor imagery may target the same
neural structures, AOT is easier to apply in children who have less capacity to imagine
themselves performing specific actions (Buccino et al., 2012). During the development
of motor skills in childhood, observing and imitating actions performed by another person
is a common way to learn new tasks. Kim et al. (2014) stated that children with cerebral
palsy who did not experience normal development due to damage before birth or
immediately after birth can be aided by observing normal movements and imitating those
movements (Kim et al., 2014). AOT may therefore be a promising tool in children with
uCP.
5
Since 2012 a few experimental studies have been conducted to investigate AOT in
children with UCP. Buccino et al. (2012) conducted a pilot randomized controlled trial
(RCT) in 15 children with CP aged between six and 11 years including both UCP and
tetraplegic CP. Children in the AOT group showed greater improvements on the
Melbourne Assessment Scale than the control group and these results were consistent in
all participants. Sgandurra et al. (2013) extended these results based on a RCT with more
strict inclusion criteria and in 24 children with UCP. The children performed both
unimanual as bimanual goal-directed actions during the AOT. The Assisting Hand
Assessment (AHA) was used as the primary outcome measure. According to their
findings, AOT has positive effects on enhancing the use of the assisting affected hand in
bimanual activities. Finally, in the last RCT conducted in 16 children with CP by Kim et
al. (2014), AOT also showed to have positive effects on the performance of activities of
daily living and upper extremity functions in comparison with simple physical training.
According to these researchers, AOT enables efficient learning and the development of
physical skills.
These studies show first evidence of the efficacy of AOT to improve motor planning and
execution in children with UCP. So far it remains unclear if AOT could have an additional
treatment effect on an intensive treatment approach focusing on motor execution, such as
mCIMT. Therefore we have set up a randomized controlled trial to investigate the
effectiveness of AOT with mCIMT in comparison with mCIMT alone. Other studies of
the same trial focus on the area of body functions, structures and activities based on the
framework of the International Classification of Functioning, Disability and Health (ICF)
(WHO, 2011). Measurement across broader domains, such as participation and quality of
life, is necessary to understand change in UL function in children with UCP (Vargus-
Adams & Martin, 2009). Some authors even consider participation and quality of life as
the key outcomes for disabled people in the evaluation of interventions (Colver,
Dickinson, & SPARCLE group, 2010).
6
Through the ICF, the World Health Organisation (WHO) challenges rehabilitation
providers to attend participation as the ultimate health and intervention outcome (WHO,
2001). Additionally, participation is considered as an important domain by parents,
medical professionals, and the young people themselves (Ramstad, Jahnsen, Skjeldal, &
Diseth, 2012). The WHO defines participation as “a person’s involvement in life
situations” (WHO, 2011). However, understanding of participation patterns of children
with congenital hemiplegia is limited. It is known, nevertheless, that the leisure and
recreation of children with physical disabilities is less varied, involves fewer social and
active physical opportunities, with greater engagement in quiet recreation compared to
nondisabled peers (Sakzewski et al., 2011). The ultimate goal of occupational therapists
(OT’s) is enabling participation in everyday activities that are meaningful to the children
and their environment (WFOT, 2012). Participation in daily life is a vital part of human
development and living experience and can improve the quality of life (QOL) (Keren-
Capelovitch, Jarus, & Fattal-Valevski, 2010).
QOL is a multidimensional construct and considered as an important outcome of
interventions as it is related to a person’s perception of wellbeing across a number of
domains including both health (i.e. physical, emotional, spiritual, social) and nonhealth
domains (i.e. finances, school, autonomy) (Bjornson & McLauglin, 2001). Children with
CP generally report lower QOL compared to nondisabled peers. However no clear
relationship has been found between the level of functioning and overall QOL
(Sakzewski et al., 2012). There is also little evidence of trials where interventions that
aim to improve upper limb function impact QOL.
In spite the importance of participation and QOL as an outcome, the effects of mCIMT
and AOT on participation and quality of life (QOL) have only scarcely been studied.
Therefore the aim of this study is to investigate the effects mCIMT and the additional
effects of AOT to mCIMT on the participation and quality of life of children with UCP.
7
Method primary outcomes
Participants
Twenty children between 7 and 12 years with congenital and acquired UCP participated
in this study. Children were included if they had a score of 4 or more on the House
Functional Classification test (= minimum ability to grasp), and if they were mentally
able to understand the test instructions. Children were excluded if they had a very well-
functioning hand and a score of 8 on the House Functional Classification test, if they had
undergone UL surgery over the past 2 years or received botulinum toxin injections in the
upper limb within six months prior to study entry. Children were recruited from the CP-
care program of the University Hospital Leuven and were included in the study between
June 2014 and August 2015. This study was approved by the Ethics Committee of the
University of Leuven and an informed consent was obtained from the parents.
Design
The design of the study was a randomized, controlled and evaluator-blinded trial.
Children were assigned using stratified random sampling. After baseline assessments
(T1), children were first stratified according to the House Functional Classification Scale
(4-5 vs 6-7), age (7-9y vs 10-12 y), and the type of cortical reorganization (contralateral,
ipsilateral and mixed reorganization) assessed by Transcranial Magnetic Stimulation
(TMS) to maximise homogeneity and minimise group differences at baseline. A permuted
block design of 2 was then used, created by a computer random number generator to
randomize the participants to the mCIMT+AOT or the mCIMT group within each
stratum. Randomization was performed by an independent person who was not involved
in the selection procedure and did not have access to clinical information about the
children.
Children were evaluated before (T1) and six months after (T2) the 9-day therapy
intervention period. The questionnaires were completed by parents and the other
assessments were performed in the clinical motion analysis laboratory of the University
Leuven, campus Pellenberg by one physiotherapist who was blinded for group allocation.
8
Intervention
The intervention was organized in a two- week day camp model. During the summer of
2014 and 2015, three camps were organized for children with unilateral cerebral palsy in
Leuven and Diepenbeek. Children received nine out of 11 consecutive days, six hours
intensive therapy per day, for a total of 54 hours of therapy. Each day, all children wore
an individually adjusted hand splint at the non-affected UL while performing exercises.
The splint was a rigid orthosis, tailor made and covering fingers, thumb, wrist and a volar
plastic was inserted to prevent grasp. During the nine day intervention, the children
received individual therapy, group therapy and AOT or placebo AOT on each day. A table
of the day structure is shown in appendix I.
The individual therapy was applied one hour each day. For the individual therapy,
therapists focused on individual treatment goals which were defined based on the baseline
assessments at T1. These assessments included body function measures and video-based
assessment of the Melbourne Assessment of Unilateral Upper Limb Function and the
Assisting Hand Assessment. Individual goals could focus on specific fine motor skills,
pinch or grip strength and improvement of active range of motion, supination etc.
Exercises were hereby specifically adapted to the motor severity of the child.
Group activities including crafting, painting, cooking, and several outdoor games were
based on shaping and repetitive practice. The group activities were specifically developed
and adjusted to train as intensively as possible the affected side. A 1:1 therapist-child
ratio was provided to secure individual guidance.
Further, the children received two hours of AOT or placebo therapy on a daily basis. The
children, assigned to the experimental group, watched video sequences showing unilateral
goal directed actions followed by execution of these actions. Fifteen tasks (one for each
session) consisted three sequential sub-activities of increasing complexity. Each sub-
activity was observed and executed twice. Each action was repeatedly shown for three
minutes.
9
After the three minutes of observation, the same objects shown on the video were placed
on the table and the children were requested to perform the observed action in the exact
order for three minutes with their hemiplegic UL.
The children in the control group watched videogames without biological movements and
without using their UL for three minutes. The therapist controlled the keypad and after
three minutes the control group also executed the tailored action, after verbal instruction
of the therapist, for three minutes synchronously and in the exact order performed by the
experimental group.
The unimanual goal-directed actions were adapted to the UL function level of the child.
Two sets of actions were compiled based on the house functional classification score of
the children: one series for children with a score 4 or 5 and one for children with a
classification score from 6 or 7. The goal and setting of the activities was the same. The
videos used in the action observation therapy were recorded from the first perspective to
avoid potential additional mental rotation and children were seated 50 cm in front of a
computer screen. Each child, regardless of their assigned group, was assisted by a
physiotherapy student or occupational therapist at the affected side. These therapists
ensured that the child continued to focus on the screen and encouraged the children in the
execution of the exercise without giving suggestive verbal instructions. In appendix 1I,
the list of the AOT-activities for House Functional Classification 4-5 is displayed. The
list of AOT-activities for children with House Functional Classification 6-8 is included
in appendix III. The protocol of AOT was based on the study of Sgandura et al., 2013.
10
Assessment
Descriptive information and classification measures
Before the start of the camps, descriptive information about the children was obtained
such as age, sex, affected side, aetiology of the brain lesion, type of reorganization,
educational level, House functional classification level and Manual Ability Classification
System (MACS) level.
The House functional classification scores the grip function in each hand separately in
nine subgroups or functional levels (see table 1) (Arner, Eliasson, Nicklasson,
Sommerstein, & Hägglund, 2008). Koman et al. (2008) reported excellent values in
terms of interrater (ICC=0.92), and intrarater reliability (ICC=0.94) in a sample of 65
children with CP (mean age 9y 2mo, SD 4y 1mo). McConnell and colleagues (2012)
reviewed 18 classification systems for the upper limb in children with cerebral palsy. The
House classification was described as reliable and clinically useful, and recommended,
together with the MACS to classify UL function.
Table 1. House classification scale (House, Gwathmey, & Fidler, 1981)
Class Designation Activity level
0
1
2
3
4
5
6
7
8
Does not use
Poor passive assist
Fair passive assist
Good passive assist
Poor active assist
Fair active assist
Good active assist
Spontaneous use
Spontaneous use hand
Does not use hand
Uses as stabilizing weight only
Can hold onto object placed in hand
Can hold onto objects and stabilize it for use by other hand
Can actively grasp object and hold it weakly
Can actively grasp object and stabilize it well
Can actively grasp object and then manipulate it against other hand
Can perform bimanual activities easily
Uses hand completely independently without reference to the other
hand
11
The MACS is a classification system judging how children with CP use their hands when
handling objects in daily life. These daily activities are for example eating, dressing,
writing, playing, etc. The classification system is designed to classify the typical manual
performance and not the maximal capacity of the child. An ordinal five point scale is used
to score the abilities of the child (see table 2). The interrater reliability of the MACS was
tested in a sample size of 168 children and found to be excellent (ICC = 0.97) (Eliasson
et al., 2006). The content validity of the MACS, according to Eliasson et al. (2007) is
good and research has shown that the five levels makes a distinction between different
clinical conditions.
Table 2. MACS (Eliasson et al., 2007)
MACS Level Description
I
II
III
IV
V
Handles objects easily and successfully.*
Handles most objects but with somewhat reduced quality or speed of
achievement.*
Handles objects with difficulty; needs help to prepare or modify activities.
Handles a limited selection of easily managed objects in adapted situations.
Does not handle objects and has severely limited ability to perform even simple
actions.
*Children in MACS levels I and II are considered to be independent in age-relevant daily activities.
Primary outcomes intervention
Participation was measured with the Assessment of Life Habits for children (LIFE-H)
and the quality of life with the CP QOL Child Questionnaire (CP-QOL). Children were
assessed at baseline (T1), and after six months (T2).
Participation
The Assessment of Life Habits for children assesses the participation of children from
five to 13 years with a disability. Regardless of the type of underlying impairment, the
LIFE-H is adjusted to potential physical limitations of a child (Noreau, Lepage, Boissiere,
Roger, & Fougeyrollas, 2007).
12
The short version of the LIFE-H contains 64 items and uses an item score ranging from
0 to 9 with 0 indicating total impairment (meaning that the activity or social role is not
accomplished or achieved) while a score of 9 indicates optimal participation (meaning
the activity or social role is performed without difficulty and without assistance).The
following domains are included to assess participation: communication, personal care,
housing mobility, nutrition, fitness and recreation, responsibilities, education, community
life and interpersonal relationships. A combination of scales are developed to score an
item. The first scale assesses performance over two dimensions, i.e.: the level of difficulty
when performing a life habit (without difficulty, with difficulty, performed by others, not
executed, not applicable) and the type of required assistance (assistive device, adaptation
and/or human assistance). The second scale is an ordinal scale that assesses the child’s or
caregivers satisfaction with the daily activity or social roles. The satisfaction can vary
from 1 (very unsatisfied) to 5 (very satisfied) (Noreau et al., 2007). The second scale was
not analyzed for the present study. In this study, we used the official Dutch translation
of the short form. To analyse the data, a transformation of the scores was calculated to
give similar weighting to each life habit. The LIFE-H has a good validity and a good
internal consistency for 10 of the 11 categories with a Cronbach’s alpha ranging from
0.73 to 0.90. For the domain interpersonal relationships, a Chronbach’s alpha of 0.58 was
reported. The test-retest reliability of the short version of the LIFE-H is moderate (ICC=
0.67) (Sakzewski, Boyd, & Ziviani, 2007). Further details of the psychometric properties
of the LIFE-H are provided in appendix IV. The LIFE-H assessment is added
in appendix V.
Quality of Life
The CPQOL-Child is a condition-specific measure, designed for children with CP, that
evaluates the well-being of children across seven areas of a child’s life: social well-being
and acceptance, functioning, participation and physical health, emotional well-being,
access to services, pain and impact of disability and family health (Waters, Davis, Boyd,
& Reddihough, 2013). In this study, the primary caregiver-proxy report version for
children aged four to 12 years was used which contains 66 items.
13
Almost all items start with 'How do you think your child feels about...' or 'How do you
feel about…’ and are scored on a nine-point rating scale (1 =very unhappy, 3 = unhappy,
5 = neither happy nor unhappy, 7=happy, and 9=very happy). Only the items on pain were
are assessed differently and begin with 'How does your child feel about the amount of
pain that they have and scored as 1=not upset at all to 9=very upset. All items of each
area were transformed to a score ranging from 0 to 100. The CPQOL-child has a high
internal consistency (0.70 – 0.95) and good test-retest reliability (0.76-0.89) (Waters et
al., 2013). The CP-QOL is based on the definition of QOL as described by Bjornson and
McLaughlin (2001) and assesses QOL over broad domains and measures well-being.
According to the review of Carlon et al. (2010), the CP QOL-Child demonstrated the
strongest psychometric properties and clinical utility for school aged children with
cerebral palsy. The scale was also reported to measure, in contrast to other measures,
well-being rather than ill-being as questions in other outcome measures are often
negatively phrased. Further details of the psychometric properties of the CP-QOL are
added in appendix IV. The CP-QOL assessment is added in appendix VI.
Statistical Analysis
Due to the sample size (n=20), non-parametric statistics were applied. Baseline
characteristics and outcome measures were analysed to check for differences between
groups using Mann-Whitney U tests and chi-square tests. The LIFE-H and CP-QOL were
compared between pre- and post- measurements for the total group and between both
intervention groups. To compare the differences in pre- and post- measurements between
both intervention groups, Mann-Whitney U tests were used. To determine differences
between pre- and post-measurements for the total group, Wilcoxon signed-rank tests were
applied. All statistical analyses were conducted using SPSS version 23.0 (IBM corp.,
2015). The significance level was set at p < 0.05.
14
Method Secondary outcomes
As a complement to the quantitative and inductive research within this study, a qualitative
part was added to the study. Because the CP-QOL and LIFE-H questionnaires were
completed by the parents, the perspective and experience of the children were not
represented by these assessment. In-depth interviews were conducted between one month
and six months after the camp in order to increase the knowledge about the intensive
camp model.
Theoretical basis
A qualitative research design with a phenomenological hermeneutical method was used
to collect information on the lived experiences of the children about the camp.
Data collection
The children were asked to honestly tell their experiences through an open-ended initial
question: ‘Can you please tell me something about the therapy camp last summer?’ Then
they were encouraged to go deeper into their narratives through follow-up probes, such
as: ‘What did you feel when you were performing this activity?’, ‘Why did you like this
activity so much?, ‘What exactly do you mean by?’ and so forth. The responses of the
children were used to alter the questions. An iterative process was used in which obtained
information in previous interviews was used to revise the later interviews. The interviews
were disc-recorded and transcribed verbatim.
Data analysis
The transcripts were analysed based on the guidelines proposed by Lindseth and Norberg
(2004). In the first phase, naïve understanding, the data material was read trough for
several times in order to achieve a sense of entirety of the interviews. This phase is called
naïve understanding because a very preliminary interpretation of the meaning of the
whole occurs while reading narratives from the 20 children in relation to each other.
15
This phase is a circular rather than a static process of going back and forth until a first
naïve understanding was conceived. To validate the understanding, gained from the naïve
phase, a structural analysis was performed. In this analytical process, connections and
patterns were uncovered and divided into different themes. In the last phase, findings
were recontextualized in a comprehensive understanding.
16
Results
Participants
Twenty children with UCP participated in the camps: six girls and fourteen boys, eight
children with left-sided and twelve children with right-sided UCP. The median age was
10 years and 5 months (IQR= 9,5mo - 11,6mo). Five children were classified as level 1
on the Manual Ability Classification System (MACS)-level, 15 children as level 2.
Fifteen children followed mainstream school, five were attending special education. The
etiology was a periventricular lesion in 9 children, a deep grey matter lesion in 9 children
and in 2 children the cause of the brain lesion was unknown. All children received
physiotherapy or occupational therapy on a regular basis. Figure 2 shows the flowchart
of the subjects participating in the study. Eighteen children completed the LIFE-H and
CPQOL-Child parent proxy report at baseline. Sixteen children completed the
postquestionnaires at six months. Of the 20 children, the questionnaires were not filled in
by the parents of one child at the pre- and follow up testing and caregivers of another
child did not completed the questionnaires because they spoke a foreign language. There
were two additional drop-outs on the posttesting. One child withdrew and one child
received Botulinum toxin injections in the upper limbs after the intervention and was
excluded for the post testing for potential bias.
17
Recruitment of subjects
(n = 20)
Missing data
T1
(n = 2)
Pre-testing
LIFE-H & CP-QOL
(n = 18)
Concealed random allocation (n = 20)
mCIMT GROUP m-CIMT + AOT GROUP
(n = 9) (n = 11)
Therapy camp Therapy Camp
9 days 9 days
Drop-out Drop-out
T2 T2
(n = 2) (n = 2)
Post-testing Post-testing
LIFE-H & CP-QOL LIFE-H & CP-QOL
(n = 7) (n = 9)
Figure 2. Flowchart: number of the children participating in the study
Abbreviations: n = number, mCIMT = modified constraint-induced movement therapy group, mCIMT +AOT = modified constraint-induced
movement therapy and action observation group group, T1 = baseline, T2 = six months follow-up, LIFE-H = assessment of Life Habits for children,
CP-QOL= CP QOL Child Questionnaire
18
Baseline comparisons between groups
Demographics and baseline characteristics of participants in the mCIMT group and
mCIMT + AOT group are shown in table 3. There were no significant differences between
groups on demographic characteristics. Only, the item education reached borderline
significance (p= 0.07), showing a tendency that more children in the control group were
attending special education schools compared to the experimental group. There were no
significant differences between groups for most of the domains or areas of the outcome
measures except for education (p = 0.04) in the LIFE-H assessment and the areas feelings
about functioning (p = 0.04) and pain and impact of disability (p = 0.01) of the CP-QOL
questionnaire.
19
Table 3. Demographics and baseline characteristics of participants in the mCIMT group and
mCIMT+ AOT group
Characteristic m-CIMT Group
(n = 9)
m-CIMT + AOT Group
(n = 11)
p-value
Demographic
Age, mean (IQR) 10 years 9 months
(9Y 1Mo – 12Y 0Mo)
10 years 4 Months
(9Y 9Mo – 11Y 6Mo)
0.97 (a)
Sex, n (%)
Male 6 (67) 8 (73) 0.77 (b)
Female 3 (33) 3 (27)
Affected side, n (%)
Left 3 (33) 5 (45) 0.58 (b)
Right 6 (67) 6 (55)
Education, n (%)
Mainstream school 5 (56) 10 (91) 0.07 (b)
Special school 4 (44) 1 (9)
Classification
MACS, n (%)
Level I 2 (22) 3 (27) 0.80 (b)
Level II 7 (78) 8 (73)
House, n (%)
House 4-5 7 (78) 8 (73) 0.80 (b)
House 6-8 2 (22) 3 (27)
LIFE-H, n
Activities of regular living
Nutrition, Me (IQR)
Fitness, Me (IQR)
Personal Care, Me (IQR)
Communication, Me (IQR)
Housing, Me (IQR)
Mobility, Me (IQR)
Social roles
Responsibility, Me (IQR)
Interpersonal Relationships, Me (IQR)
Community life, Me (IQR)
Education, Me (IQR)
Employment, Me (IQR)
Recreation, Me (IQR)
9
6.1 (5.0-8.1)
9.4 (8.9 - 9.4)
5.6 (4.6-6.3)
8.9 (7.8 - 9.4)
9.6 ( 9.6 -10)
8.3 (7.5 - 9.4)
7.8 (6.0 - 7.8)
10 (9.1 -10.0)
10 (10 - 10)
8.0 (7.4 -9.3)
N/A
9.2 (6.9 - 9.3)
9
5.0 (4.4 - 7.8)
9.4 (9.4 - 10.0)
6.8 (5.2 - 8.9)
10 (9.7 - 10.0)
10 (10 - 10)
9.7 (7.5 - 10)
10 (9.6 - 10)
10 (9.6 - 10)
10 (5.6 - 10)
9.6 (9.4 - 10)
N/A
9.7 (9.0 - 9.8)
0.67 (a)
0.08 (a)
0.19 (a)
0.11 (a)
0.39 (a)
0.34 (a)
0.11 (a)
0.73 (a)
0.18 (a)
0.04 (a)
0.67 (a)
CP-QOL,n
Social well-being and acceptance, Me (IQR)
Feelings about functioning, Me (IQR)
Participation and physical health, Me (IQR)
Emotional well-being and self-esteem, Me (IQR)
Pain and impact of disability, Me (IQR)
Access to services, Me (IQR)
Family Health, Me (IQR)
9
77.3 (70.5 -79.5)
61.5 (58.3 - 69.8)
65.9 (54.5 - 73.9)
72.9 (68.8 - 77.1)
62.5 (60.9 - 67.9)
75 ( 71.9 - 77.5)
75.0 (75.0 - 84.4)
9
83.0 (75.0 - 84.1)
75.0 (65.6 - 77.1)
72.7 (62.5-75.0)
81.3 (75.0 - 83.3)
76.6 (71.9 - 84.4)
75.0 (70.0 - 80.0)
78.1 (75.0 - 90.6)
0.11 (a)
0.04 (a)
0.39 (a)
0.08 (a)
0.01 (a)
0.67 (a)
0.55 (a)
Abbreviations: n= number, mCIMT group = modified constraint-induced movement therapy group, mCIMT +AOT group = modified constraint-induced
movement therapy and action observation group, Y = years & MO = months, MACS = Manual Ability Classification System, N/A = not applicable; Me
= median, IQR = inter quartile range, LIFE-H = assessment of Life Habits for children, CP-QOL = CP QOL Child Questionnaire, (a) = Mann-Whitney
U test, (b) = Chi-square test, Significance level 0.05
20
Efficacy of the intervention
Primary outcomes
Participation
The median (IQR) for the domain scores and overall scores of the LIFE-H assessment at
baseline and at six months follow up are shown in table 4. The individual overall scores
of the LIFE-H at both measurement points are illustrated in figure 3. Comparison of the
difference scores of the pre and post measurement between the mCIMT and the
combination group (mCIMT + AOT group) showed no significant difference for the total
score (p= 0.21). Only for the domain interpersonal relationships, a significant difference
between the groups was found (p = 0.01). In the mCIMT group, the median scores
decreased from a median score of 10 to 8.7 while median scores in the combination group
(mCIMT +AOT group) remained stable. Comparison of the pre and post measurements
for the total group revealed no significant differences for the overall score (p= 0.50) and
for all domains of the LIFE-H assessment.
Figure 3: Individual overall scores on the LIFE-H at pre and post measurement
0
2
4
6
8
10
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16
OVERALL SCORES LIFE-H
PRE LIFE-H POST LIFE-H
21
Table 4. Descriptive statistics of the LIFE-H before and after intervention,
and statistical comparison
Abbreviations. (1) : p-values between groups, (2): p-values pre-post total group, n= number, mCIMT group = modified constraint-induced movement
therapy group, mCIMT +AOT group = modified constraint-induced movement therapy and action observation group, Y = years & MO = months,
MACS = Manual Ability Classification System , N/A = not applicable; Me = median, IQR = inter quartile range, LIFE-H = assessment of Life Habits
for children, Significance level 0.05
Participation,
LIFE-H
Pre Post
p-value
Activities of regular
living
Nutrition
Fitness
Personal Care
Communication
Housing
Mobility
Social roles
Responsibility
Interpersonal
Relationships
Community life
Education
Employment
Recreation
TOTAL
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR
Me(IQR)
Me(IQR)
Me(IQR
Me(IQR)
Me(IQR)
Me(IQR
N/A
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
mCIMT-group
mCIMT-group + AOT group
Total group
mCIMT-group
mCIMT-group + AOT group
Total group
mCIMT-group
mCIMT-group + AOT group
Total group
mCIMT-group
mCIMT-group + AOT group
Total group
mCIMT-group
mCIMT-group + AOT group
Total group
mCIMT-group
mCIMT-group + AOT group
Total group
mCIMT-group
mCIMT-group + AOT group
Total group
mCIMT-group
mCIMT-group + AOT group
Total group
mCIMT-group
mCIMT-group + AOT group
Total group
mCIMT-group
mCIMT-group + AOT group
Total group
mCIMT-group
mCIMT-group + AOT group
Total group
mCIMT-group
mCIMT-group + AOT group
Total group
6.7 (6.0 - 8.3)
5.0 (4.4 - 7.8)
6.4 (4.9 - 8.2)
9.4 (9.2 - 9.4)
9.4 (9.4 - 10)
9.4 (9.3 - 10)
5.6 (4.8 - 6.2)
6.8 (5.2 - 8.9)
5.9 (4.9 - 8.8)
8.5 (7.0 - 9.3)
10 (9.7 - 10)
9.6 (7.7 - 10)
9.6 (9.6 -10)
10 (10 - 10)
10 (9.6 - 10)
8.3 (7.5 - 9.5)
9.7 (7.5 - 10)
9.2 (7.5 - 10)
7.8 (5.6 - 7.8)
10 (9.6 - 10)
8.7 (6.5 - 10)
10 (6.7 - 10)
10 (9.6 - 10)
10 (9.0 - 10)
10 (10 - 10)
10 (5.6 - 10)
10 (8.7 - 10)
8.0 (7.5 - 9.1)
9.6 (9.4 - 10)
9.4 (8.0 - 9.7)
9.2 (7.5 - 9.7)
9.7 (9.0 - 9.8)
9.3 (7.8 - 9.9)
8.1 (7.1 - 8.5)
9.1 (7.9 - 9.6)
8.3 (7.4 - 9.2)
8.3 (6.0 - 9.3)
6.1 (5.0 - 8.5)
7.8 (5.0 - 9.2)
9.4 (8.9 - 9.6)
9.4 (9.4 - 10)
9.4 (8.9 - 10)
6.7 (5.4 - 8.1)
6.7 (5.6 - 9.3)
6.7 (5.4 - 8.5)
9.2 (9.2 - 9.7)
10 (9.0 - 10)
9.7 (9.2 - 10)
9.6 (7.8 - 9.7)
9.6 (8.7 - 10)
9.6 (7.8 - 9.9)
8.9 (7.5 - 9.7)
9.4 (8.9 -10)
9.4 (7.5 - 10)
7.2 (5.8 -7.7)
8.9 (7.2 -9.6)
7.7 (5.9 - 9.4)
8.7 (7.2 - 8.9)
10 (10 - 10)
9.8 (8.7 - 10)
10 (9.2 - 10)
10 (10 - 10)
10 (9.5 - 10)
8.5 (8.0 - 9.1)
9.3 (7.8 - 10)
8.7 (7.8 - 9.6)
9.2 (8.1 - 9.4)
9.6 (9.0 -9.9)
9.3 (8.5 - 9.8)
8.4 (8.0 - 8.7)
8.9 (8.5 - 9.5)
8.6 (8.1 - 9.3)
0.61 (1)
0.21 (2)
0.35 (1)
0.64 (2)
0.76 (1)
0.35 (2)
0.30 (1)
0.19 (2)
0.61 (1)
0.05 (2)
0.54 (1)
0.89 (2)
0.25 (1)
0.41 (2)
0.01 (1)
0.40 (2)
0.96 (1)
0.86 (2)
0.41 (1)
0.23 (2)
0.41 (1)
0.75 (2)
0.21 (1)
0.50 (2)
22
Quality of life
Median domain scores and overall scores of the CP-QOL are shown in table 5. Individual
data of overall scores of the CP-QOL are illustrated in figure 4.
Comparison of the difference scores of the pre and post measurement between the
mCIMT and the combination group (mCIMT + AOT group) showed no significant
difference for the total score (p = 0.25). Significant differences were found between
groups in the domains regarding social well-being and acceptance (p= 0.02) and pain and
impact of disability (p = 0.03). On the domain social well-being and acceptance, the
median score in de mCIMT group decreased from 77.3 at baseline to 71.6 at the post
measurement while the median score in the combination group remained stable. In the
area pain and impact of disability, the median score in de mCIMT group improved from
60.9 at baseline to 65.5 at the post measurement while the median score in the
combination group improved less with 76.6 at baseline and 78.1 at the post measurement.
No significant differences between the the mCIMT and the mCIMT+AOT could be found
on the other QOL domains.
Comparison of the pre and post measurements for the total group revealed no significant
differences for the overall score (p= 0.50) on the CP-QOL assessment.
Figure 4: Individual overall scores on the CP-QOL at pre and post measurement
0
20
40
60
80
100
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16
OVERALL SCORES CP-QOL
PRE CP-QOL POST CP-QOL
23
Statistical analysis of the domains revealed a significant improvement six months after
the intervention for feelings about functioning of the CP-QOL assessment with a p-value
of 0.03. In this domain, the median scores improved from 67.2 to 72.4 meaning that
children felt better about their functioning following the intervention. Individual data of
the changes in feelings about functioning between T1 and T2 are shown in figure 5. The
total group also demonstrated significant changes in the domain family health (p= 0.04)
in which the median score for the domain decreased from 79.7 at baseline to 76.6 at the
post measurement.
Figure 5. Individual scores on the domain feelings about functioning on the CP-QOL at pre and post
measurement
0
10
20
30
40
50
60
70
80
90
100
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16
CP-QOL: FEELINGS ABOUT FUNCTIONING
PRE FEELINGS ABOUT FUNCTIONING POST FEELINGS ABOUT FUNCTIONING
24
Table 5. Descriptive statistics of the CP-QOL before and after intervention,
and statistical comparison
Quality of life,
CP-QOL
Pre Post p-value
Social well-being and
acceptance
Feelings about
functioning
Participation and physical health
Emotional well-being
and self-esteem
Access to services
Pain and impact of disability
Family Health
TOTAL
Me(IQR)
Me(IQR) Me(IQR)
Me(IQR) Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR) Me(IQR)
Me(IQR) Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR) Me(IQR)
Me(IQR) Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
Me(IQR)
mCIMT-group
mCIMT-group + AOT group Total group
mCIMT-group mCIMT-group + AOT group
Total group
mCIMT-group
mCIMT-group + AOT group Total group
mCIMT-group mCIMT-group + AOT group
Total group
mCIMT-group
mCIMT-group + AOT group
Total group
mCIMT-group
mCIMT-group + AOT group Total group
mCIMT-group mCIMT-group + AOT group
Total group
mCIMT-group
mCIMT-group + AOT group
Total group
77.3 (73.3 - 79.0)
83.0 (75.0 - 84.1) 79.0 (74.4 - 83.2)
61.5 (58.9 - 67.7) 75.0 (65.6 - 77.1)
67.2 (60.9 - 76.0)
65.9 (58.0 - 72.7)
72.7 (62.5-75.0) 66.5 (58.8 - 74.1)
72.9 (64.6 - 75.0) 81.3 (75.0 - 83.3)
76.0 (71.9 - 83.3)
75.0 (64.7 - 77.5)
75.0 (70.0 - 80.0)
75.0 (68.8 - 80.0)
60.9 (59.6 - 69.5)
76.6 (71.9 - 84.4) 73.4 (62.5 - 77.1)
81.3 (75.0 - 84.4) 78.1 (75.0 - 90.6)
79.7 (75.0 - 85.2)
71.0 (68.1 - 73.7)
81.0 (71.4 - 84.1)
72.7 (69.4 - 79.2)
71.6 (71.0 -74.5)
83.0 (81.5 - 87.5) 76.8 (72.4 - 86.6)
71.9 (62.5 - 76.6) 72.9 (65.6 - 83.3)
72.4 (63.0 - 81.0)
65.9 (59.7 - 74.1)
72.7 (68.2 - 79.5) 71.0 (61.1 - 74.8)
77.1 (66.7 - 77.1) 79,2 (75.0 - 87.5)
77.1 (68.8 - 81.8)
67,5 (60.0 - 81.3)
75.0 (72.5- 82.5)
73.8 (65.6 - 83.1)
65.6 (64.3 - 73.4)
78.1 (73.4 - 91.1) 73.4 (69.9 - 81.3)
71.9 (70.3 - 89.1) 78.1 (68.8 - 81.3)
76.6 (68.8 - 87.5)
75.2 (65.2 - 77.4)
79.2 (69.8 - 84.2)
76.2 (65.6 - 79.5)
0.02 (1) 0.82 (2)
0.79 (1)
0.03 (2)
0.35 (1) 0.41 (2)
0.35 (1)
0.97 (2)
0.47 (1)
0.97 (2)
0.03 (1) 0.22 (2)
0.68 (1)
0.04 (2)
0.25 (1)
0.50 (2)
Abbreviations. (1): p-values between groups, (2): p-values pre-post total group, n= number, mCIMT group = modified constraint-induced movement
therapy group, mCIMT +AOT group = modified constraint-induced movement therapy and action observation group, Y = years & MO = months, MACS
= Manual Ability Classification System , N/A = not applicable; Me = median, IQR = inter quartile range, CP-QOL = CP QOL Child Questionnaire
Significance level 0.05
25
Secondary outcomes
Every child (n = 20) was interviewed by a participating practitioner. Of the 20 completed
interviews, 16 interviews took place in the home environment of the children. The
findings and interpretations of the interviews are reported and supported by several quotes
from the children or parents.
Experience of a robot camp
Almost all children could remember numerous activities and fun moments. Some children
found the camp challenging, others children claimed that they had to work really hard.
All children had the feeling that they had learned something and 19 of the 20 children
would recommend the camp to other children with unilateral cerebral palsy. Some of the
children had never met another person with unilateral cerebral palsy and told about the
unique experience of the camp to meet and play with other children with hemiplegia.
Seventeen children perceived this positively, three children had no opinion on this topic.
These experiences are reflected by the following quotes:
K.V.: "It was nice not to be the only one with an impairment, I was not always the last or the
slowest one and I felt less alone during these weeks.”
L.B.: "I did not know there were so many children who had the same as me! It was fun to do things
together because I felt that we understood each other and everyone struggled with something.”
B.V: "It was amazing to see other children with limitations and to experience how they dealt with
them. It was good to feel that I was not alone and that there are children just like me.”
Some caregivers who attended the interview also spontaneously shared their experience
about the camp. Several parents expressed about how the camp positively influenced the
self-image of their child and that it was nice to meet other parents of children with
hemiplegia.
R.T.: “For us, besides the improvement in hand function, the experience of the camp was very
valuable. The camp certainly created a different self-image for my daughter. My daughter goes to
a regular school and she is clever but just a bit slower and sometimes it is hard for her to deal
with this. Yes, this was at least as important as the initial goal of the therapy camp.”
26
Through the camp, parents came in touch with other parents of children with unilateral
CP and often shared some time together after a camp day and during the last afternoon of
the camp when the parents were invited. One father organizes CP football and ever since
a few children from the camps play soccer in a team together and see each other every
week. So the camp has indirectly contributed to these valuable connections between
children and parents.
The camp theme was robots and the mascot Zora was a great motivator for the children.
Zora is a small robot with a high ‘feel-good factor’, she is very mobile and gives verbal
(programmable) instructions.
T.L.: “Zora was very intelligent. She even knew who I was!”
I.D.: “Zora told what we needed to do that day, but I could also tell her a lot.”
L.V.: “Every day I looked forward to see Zora again. She was such a good dancer.”
Experience of wearing a splint, mCIMT
Opinions about wearing a splint were very diverse. The children repeatedly stated that the
duration of wearing the splint was long and some children experienced eating with a splint
as very frustrating (after the first camp this assignment was no longer expected from the
children). The kids liked the fact that their splint was personalized with their name and
that “everyone had to keep going”. Thus an important aspect in tolerating the splint was
the group dynamics. Some kids did not like that the therapists were not wearing splints.
This might be a potential idea for the following camps.
M.L.: “The splint made it easier not to use my good side. But the splint made everything harder to
execute and that was sometimes frustrating. Especially when we needed to ask help to others.”
J.K.: “Sometimes it was funny because sometimes we had to do weird things in order to accomplish
some tasks. In the beginning it was difficult but after two days we became used to it.”
A.P.: “It was nice that everyone had to wear a glove, If I had to do this alone, I would have found
it much less fun.”
27
Only four children have worn their splint after the camp at home, during occupational
therapy or physical therapy.
Experience action observation therapy, AOT
To the question “Were there things you did not like during the camp?”, almost every
child answered “the therapy on the computers”. Especially young children found AOT
boring and tedious. Although, older children indicated to be able to concentrate on the
videos and six out of nine children in the experimental group were convinced that viewing
the videos helped them to execute their exercises. More than half of the children in the
experimental group would never perform AOT again, which raises the question if AOT
can be made more child friendly.
S.C.: “After watching the videos once, they were predictable and it became hard to concentrate.”
T.G.: “In AOT we had to work hard but the exercises were challenging.”
L.D.: “Sometimes it was frustrating that you had to do an annoying task again and again.”
Experience effects camp
Seventeen out of 20 children perceived functional improvement in their affected side after
the camp. The children could give numerous examples. Many children noticed
improvement in ADL activities. Some children experienced that they had more strength
and others noticed less tension in their affected hand. Several children experienced that
they used their affected hand more to try out something new. They explained that they
have more confidence in using their affected side in everyday activities.
B.S. (Mother): “In the beginning the advancement in his affected side was crazy, I never expected
such a spectacular result! Now it has subsided, but it is still improved. I think he knows that he
can actually achieve things with his affected hand. He is also unconsciously more aware of his
hand.”
28
Children reported improvement in several areas including self-care. Almost all children
could eat more easily with knife and fork after the camp. A few children find it easier to
prepare a sandwich. Some children noticed improvement in opening a bottle or drinking
from a bottle. Others told that it is easier to open a door or a refrigerator. Some children
noticed change when they tried to close a zipper or when they put on trousers with two
hands. One boy indicated that he put on his orthoses independently since the camp.
Many children reported improved academic skills including cutting or handling writing
materials. Apparently, for many children it became easier to pick up small things and to
operate electronics such as a mobile phone and tablet.
S.V.: “I noticed a big difference during gymnastics, I find it easier to do handstand.”
One mother mentioned that through the camp she knows better how to stimulate the
affected upper limb of her child.
I.H. (Mother): “Through the camp I realized that I also can practice at home with my daughter in
everyday activities. Now, when we cook together, she has to pay attention to her difficult hand. Or
when we play with the cards, she has to take her cards with her affected side.”
29
Discussion
This study investigated the efficacy of an intensive therapy camp and the additional
effects of AOT on the participation and quality of life of children with UCP. Twenty
children with UCP were wearing a constraint on the unaffected hand for nine out of 11
consecutive days, six hours a day, performing unimanual exercises. Children who were
assigned to the experimental group received AOT for two hours each day, whereas the
other children received placebo AOT. Measurements of participation and quality of life
revealed no meaningful significant results, as an outcome of the intervention, between the
two groups. Nevertheless, this study demonstrated that a upper limb intensive training
model can lead to domain specific changes measured on a condition-specific QOL
outcome measure and that these changes last for six months.
Children with CP generally report lower QOL than their typically developing peers.
Therefore, QOL should be considered as an important outcome of interventions
(Sakzewski et al., 2012). The total group showed a significant improvement after the
intervention in the domain feelings about functioning on the CP-QOL. Questions in this
domain address the ability of the children to play on their own, the way the children use
their arms and hands, their ability to keep up academically with their peers, their
opportunities in life, their ability to dress, eat, drink independently and their ability to use
the toilet by themselves. Both groups clearly improved in arm and hand skills which
directly reflect the aim of the intervention camp and thus can be considered as meaningful.
These improvements, perceived by the parents, were confirmed by the children through
in-depth interviews. Seventeen out of 20 children perceived functional improvement in
their affected side after the camp, this mainly in ADL activities including self-care and
academic skills. Some children experienced that they had more strength and others
noticed less tension in their affected hand. Several children experienced that they used
their affected hand more to try out something new. They explained that after the therapy
camp they had more confidence in using their affected side in everyday activities.
30
Sakzewski et al. had similar results in 2012 on the CP-QOL after they organized a circus
camp for children with UCP (n= 64) who were randomized in a CIMT group and a
bimanual therapy group during a 10 day training camp (Sakzewski et al., 2012). Parents
from the CIMT group also reported changes in feelings about functioning, which
maintained until 26 weeks, but not up to 52 weeks. In contrast, several trials and also a
Cochrane systematic review reported no effects of intramuscular upper limb Botox
injections and occupational therapy in well-being (Hoare et al., 2010; Sakzewski et al.,
2012). The total group also demonstrated significant changes in the domain family health
(p= 0.04) in which the median score for the domain decreased from 79.7 at baseline to
76.6 at the post measurement. This is an unexpected finding. However, this area questions
the physical health, the work situation, the financial situation and the degree of happiness
of the caregiver. A change in this area is likely to be due to other factors than a
consequence of the intervention because this domain aims at factors related to the
caregiver.
Comparison of the difference scores of the pre and post measurement between the
mCIMT and the combination group (mCIMT + AOT group) showed no significant
difference for the total score (p = 0.25) on the CP-QOL. However, significant differences
were found between groups regarding social well-being and acceptance (p=0.02) and pain
and impact of disability (p = 0.03). Though, the baseline comparison indicated that the
groups on the domain pain and impact of disability scored significantly different from the
outset (p=0.01). One possible explanation for the differences between the groups in this
domain is that more severely disabled children were randomized to the control group.
This is also reflected in the baseline comparison which shows a tendency that children in
the control group went more to a special school (p=0.07) and scored significantly lower
on the item education of the LIFE-H (p=0.04). The changes in this domain are possibly
related to these baseline differences rather than as an outcome of the intervention.
31
On the domain social well-being and acceptance, the scores in de mCIMT group
decreased while the median score in the combination group remained stable. This domain
assesses how the child gets along with other children, family, adults and how the child is
accepted by these persons. We would assume that these changes are possibly due to
factors other than the intervention camp. Furthermore, there were no other statistical
differences on the other QOL domains. Although both groups clearly improved
significantly on the area feelings about function, it remains unclear whether these
statistically significant changes in the area feelings about function are clinically
meaningful. Still in our study, 15 out of 20 children improved on this area. According to
Sakzewski et al. (2012) the maintenance of changes in the UL over a 52-week period may
suggest clinical significance.
Nevertheless, we could have expected more benefits on other areas of this scale or in the
total score. According to Carlon et al. (2010), the CP-QOL is designed to detect small
changes but information about the responsiveness of the instrument is lacking. A notable
drawback is that the instrument is designed for children with severe cerebral palsy (Carlon
et al., 2010). Children in this study had MACS levels I and II, and children who have
these levels are considered to be independent in age-relevant daily activities. In present
study some items often received the maximum score which possibly could have affected
the results.
Unfortunately, there were no significant differences for the total group on the overall
score (p= 0.50) or on any domain of the LIFE-H assessment. For the LIFE-H, a significant
difference between groups was revealed for the the domain interpersonal relationships (p
= 0.01). This domain assesses how the child maintains social relationships with parents,
friends, family and other adults. However, special attention should be given to the domain
interpersonal relationships of the LIFE-H because ceiling effects have been reported in
the literature and the internal consistency of the domain scored weak (ICC= 0.40)
(Sakzewski, Boyd, & Ziviani, 2007). The weak psychometric properties of this domain
may also have contributed to this outcome.
32
Nonetheless, Sakzewski et al. (2011) found significant gains in the domain personal care
of the LIFE-H in the CIMT group at 26 weeks, after the execution of previously
mentioned circus camp for children with UCP. However, this result has been obtained in
a larger sample size (n=64). However, similar to previously reported participation studies,
no significant differences between training approaches could be found (Sakzewski et al.,
2011, 2012). According to Sakzewski et al. (2012), the understanding of participation
patterns of children with congenital hemiplegia is limited.
Nonetheless, three RCT’s showed positive effects of AOT on upper limb outcomes in
children with UCP. We have not investigated the UL outcomes of AOT within the scope
of this study. Therefore, we cannot directly compare our results to previous studies.
Nevertheless, we found no meaningful additional effect of AOT on participation and
quality of life outcomes. Various aspects could have contributed to these findings. Both
groups (mCIMT and mCIMT + AOT group) received equal doses of 54 hours mCIMT
which possibly caused a ceiling effect making it more difficult to prove the added value
of AOT. In addition, the duration of the intervention varied. Even though the total number
of AOT sessions was equal to the trial of Sgandurra et al. (2013), the duration of the latter
study was three weeks while the therapy camp in present study only lasted 11 days
(Sgandurra et al., 2013). It is therefore possible that AOT in isolation is an effective
intervention, but that it has no added value on an intensive therapy approach such as
mCIMT. A second possible reason is that some children who participated in the study did
have behavioural, attentional and executive dysfunctions which could have affected the
effects of AOT. Watching repeated tasks during AOT demanded attention and
concentration across all sessions.
Another interesting finding, through observations in AOT sessions, is that during the
execution of subtasks, some children did not repeat the identical action showed on the
video screen. Children often focused on the goal of the tasks, regardless of how it was
shown. The children often tried out a way more conform with their capabilities of their
affected hand, which had more chances to succeed. Thus the children were often more
focused to achieve the goal of the task than to imitate the movement as accurate as
possible.
33
However, the majority of children in the experimental group indicated during the
interviews that watching the video really helped them to accomplish a task. They also
indicated that a second observation after the first performance helped them to adapt their
approach to reach their goal. Consequently, children could complete a task relying on
their adapted anticipatory planning, which was not always identical, but still based on the
AOT video. It is therefore possible that AOT can help in the reduction of trial and error
and enables efficient learning in a goal oriented way.
Although the AOT interventions in present study were based on the latest evidence,
following suggestions made in the literature may be valuable. While most action
observation interventions described in the literature have focused on visual perception of
movement, the effect of multimodal action-related stimuli on the motor system have been
tested in studies on adults (Aglioti & Pazzaglia, 2010; Bassolino, Sandini, & Pozzo,
2010). Initially this was demonstrated in studies with primates, where neurons responded
to the acoustic effect produced by the motion and not only to the visual presentation of
the action (Kohler et al., 2002). More research is needed to investigate the added value of
action-related sounds to AOT videos in typically developing children and children with
CP. Another note, made by Bassolino et al. (2010) is that AOT may be a promising
alternative in the early stages of stroke patients when intensive practice with the UL is
not yet possible. According to Bassolino, even very young children may benefit from the
early application of AOT and gain in motor and developmental skills. However, AOT
requires a maximum of concentration to succeed, therefore the application of AOT in
younger children could be very challenging (De Bie & Dela Ruelle, 2015). Through the
interviews in this study it became clear that especially young children found AOT tedious.
In addition, more than half of the children in the experimental group would never perform
AOT again. These findings suggest that adaptation to AOT should be made to make it
more child-friendly and suitable for young children.
This study also has some limitations including a relatively small sample size, in particular
to detect differences between training approaches. Additionally, little is known about the
responsiveness of instruments used and psychometric properties of the Dutch version of
these assessment.
34
Further research is needed in order to assess more accurately change in quality of life and
participation in the future. In addition, several therapists and students assisted the AOT
intervention in this study and this may have induced variability. Another limitation of the
study is that the data was collected only twice and the length of time between the two
measurements. Measurements were performed before and six months after the
intervention. An intermediate measurement shortly after intervention could have provided
more information about the short-term effects of the intervention on participation and
quality of life and these effects would have been less influenced by other factors and
potential impacts due to the maturation of the child. A control group that received no
intervention would provide more information on longitudinal changes in QOL owing to
immature or cognitive development and changing expectations at school, home and with
peer relationships (Sakzewski et al., 2012).
However, this study was set-up as a evaluator-blinded, controlled randomized trial.
Through stringent inclusion criteria and a highly homogeneous sample, a blinded assessor
and triangulation of data (the use of both quantitative and qualitative data), the data was
obtained in an accurate way. The therapists were carefully instructed and efforts were
made to standardize the AOT intervention: the AOT intervention was clearly described
in terms of setting, object sequences, therapist behaviour and video presentation. In
addition, the AOT intervention was tailored according to the baseline motor impairment
of the child, according to the house functional classification scale but similar (in range of
motion, hand orientation, hand opening, reach trajectory) for both groups. Another
strength which makes this study unique, is the valuable qualitative information that was
gained from the participating children. As an addition to the CP-QOL and LIFE-H
questionnaires completed by the parents, this study included the perspectives and
experiences of children with UCP. In-depth interviews revealed a good group dynamics
and unique shared experience of the camp to meet and play with other children with
hemiplegia. Several parents indicated that the camp positively influenced the self-image
of their child. Next to that, the camp may have contributed to a better understanding how
to stimulate the affected side in everyday activities. In the end the camp has also indirectly
contributed to valuable connections between parents and children with similar limitations.
35
Further research may focus on determining factors on the effects of mCIMT and AOT.
Factors such as type of CP and location of the brain lesion, age, motor severity and
cognitive factors could be investigated. More in depth investigation of children who
maximally benefit from these therapies may be important for the future. Our current study
sample did not allow subgroup analysis but this should be conducted in studies with an
accurate sample size. Knowledge of the demographic and clinical predictors of children
who benefit most from the different intervention models is fundamental in order to
implement effective therapies in practice.
36
Conclusion
The findings of this randomized trial suggest that intensive upper limb training in a camp
model can impact the QOL of children with UCP in the area feelings about functioning
and that these changes last for six months. Positive effects and perceived functional
improvement in the affected side were also reported by the children through in-depth
interviews. Nevertheless, measurements of participation and quality of life revealed no
meaningful significant results, as an outcome of the intervention, between the two groups.
Further research is needed to provide more insights in AOT as a rehabilitative approach
in children in UCP.
37
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44
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Disability and Health. Geneva: WHO.
45
Appendix I, Day structure
Day structure of a day at the therapy camp
Example the second day of the first camp in Leuven, 2014
Tuesday 19/08/2014
09h00 09h05
09h05 09h55
10h00 10h50
10h50 11h05
11h05 12h00
12h00 13h00
13h00 13h55
13h55 14h50
14h50 15h05
15h05 15h55
15h55 16h00
* Abbreviations: n = number
Welcome with 'Zora'
AOT/placebo AOT (n=4) or group activity (n=4)
Group activity (n=4) or AOT/placebo AOT(n=4)
BREAK
Individual therapy (n=8)
LUNCH
AOT/placebo AOT (n=4) or group activity (n=4)
Group activity (n=4) or AOT/placebo AOT (n=4)
BREAK
Group activity (n=8)
End with 'Zora'
46
Appendix II, List of Goal-directed actions of Action Observation Treatment (House Functional Classification 4-5) Task
number
Description of sub-activity 1 Description of sub-activity 2 Description of sub-activity 3
1 Lift up the box covering a candy and place it next to the candy Pick up a candy from a small wooden block and drop it in a cup Pour water from a small plastic bottle into a cup containing a candy and put the bottle back
2 Pick up a colored card from a small wooden block and place it on an
identical colored card on a raised rectangular box (there are three possible vertically placed cards on the rectangular box)
Pick up a colored card from a card holder and place it on a square with the
same color (three possible, vertically placed, squares)
Pick up a colored card, turn it over, and lay it down
3 Pick up an animal-shaped sponge stamp and place it in a circle on a paper Pick up an animal-shaped sponge stamp (oblique on the right or left upper
corner of the paper) and make a print in a circle on a paper, and put the stamp back
Pick up an animal-shaped sponge stamp (oblique from the
bottom side of the paper) and make a print in a circle on a paper, and put the stamp back
4 Pick up a coin from a holder and put it into a box through a slot on top of
the box
Pick up a coin from a holder and put it into a box through a vertical slot Pick up a coin from a holder and put it into a box through a
horizontal slot
5 Pick up a stamp and place it on a drawn circle on a paper Pick up a stamp, make a print in a drawn circle on a paper, and put it back Pick up a stamp (positioned oblique from the bottom side of the paper), make a print in a circle on a paper, and put it back
6 Pick up a tube containing a small ball and place it on a paper more to the
left or right
Pick up a tube containing a small ball, pour the ball on the toy shovel that
is connected with a block through an elastic and place the tube back
Push down the end of the toy shovel that is containing a small
ball and is connected with a block through an elastic
7 Pick up a tube with a cap containing shimmery powder and place it more to the left or right
Lift up the cap from a tube containing shimmery powder and put it on the table
Pick up the tube without a cap, and pour some shimmery powder on a drawing, and place it back
8 Pick up a magnet and place it on a piece of paper Pick up a magnetic bar, catch a flower magnet with one side, put the bar
with the other side in a holder on the side
Take the flower magnet from the magnetic bar in the holder
and put it into a bowl
9 Pick up a soft ball and put it into a bowl filled with water Pick up a bottle of detergent, pour some into the bowl and place it back Pick up the ball out of the water in the bowl, squeeze it out and put it in a bucket on the right
10 Move a clip from the edge of a box to another one more on the left or
right
Pick up a clip and put it vertically on the tale of a cardboard animal Pick up a clip and put it horizontally on the tale of a
cardboard animal
11 Place your hand on a ball of clay and flatten it Roll the hand on a roll of clay, up and down for several seconds Pick up the ball of clay, squeeze it for several seconds, and put the clay down on the table
12 Pick up a tube of paint, squeeze some of the paint in a plastic cup and put
the tube back
Pick up a brush and put it in the cup filled with some paint Pick up the brush in the cup, color the paper using a template,
and put the brush back in the cup
13 Pick up a seed from the table and drop it in a flower pot Pick up a small sand shovel from a bin filled with sand, pour it into the pot
and place the shovel back in the bin
Lift up a watering can, pour some water in the pot filled with
sand, and place it back
14 Pick up a spider from a web that is placed on the table, and drop it in a can on the right
Lift up a cut-out part of a box with a handgrip, and place it next to the box Lift up the can filled with the spiders, pour it out in the box, and place the can back on the table
15 Pick up a plastic bag filled with short pieces of colored wire, and put the
bag in a large bowl
Take the plastic bag out of the bowl, so that the pieces of wire remain in the
bowl, and place the bag next to the bowl
Pick up a small strainer in the bowl filled with some pieces of
wire, put the pieces on a drawing next to the bowl and place the strainer back in the bowl
47
Appendix III, List of Goal-directed actions of Action Observation Treatment (House Functional Classification 6-7) Task number
Description of sub-activity 1 Description of sub-activity 2 Description of sub-activity 3
1 Lift up the box covering a candy and place it next to the candy Pick up a candy from the table and drop it in a cup Pour water from a small plastic bottle into a cup containing a
candy and put the bottle back
2 Pick up a card from a small wooden block and place it on an identical card on a raised rectangular box (there are three possible vertically
placed cards on the rectangular box)
Pick up a colored card from a card holder and place it on a square with the same color
Pick up a colored card (located on a raised rectangular box), turn it over, and lay it down
3 Pick up an animal-shaped sponge stamp and place it in a circle on a paper Pick up an animal-shaped sponge stamp and make a print in a circle on a paper, and put the stamp back (stamp at the middle of the upper side of the
paper)
Pick up an animal-shaped sponge stamp and make a print in a circle on a vertical paper, and put the stamp back
4 Pick up a coin from a holder and put it into a box through a vertical slot Pick up a coin from a holder and put it into a box through a horizontal slot Pick up a coin from a holder and put it into a box through an
oblique slot
5 Pick up a small stamp and place it on a drawn circle on a paper Pick up a small stamp on the right, make a print in a drawn circle on a
paper, and put it back
Pick up a small stamp on the top of the paper, make a print in a
circle on a paper, and put it back
6 Pick up a spray can and place it on a paper more to the left Pick up a spray can and lay it down in a shaped foam Spray with the spray can (fixed in a styrofoam shell) into a cup
(positioned on the shaped foam)
7 Pick up a tube with a cap containing shimmery powder and place it down
to the left or right
Lift up the cap from a tube containing shimmery powder and put it on the
table
Pick up the tube without a cap, and pour some shimmery
powder on a drawing, and place it back
8 Pick up a magnet and place it on a piece of paper Pick up a toy fishing rod, catch the animal magnet with it and put the rod
in a large cup
Take the animal magnet from the fishing rod in the cup and put
it in a small plate
9 Grab a cloth that is spread out, and put it into a bowl filled with water Pick up a bottle of detergent, pour some into the bowl and place it back Pick up the cloth from the water in the bowl, wring it out and
put it in a bucket on the right
10 Take a wooden clip from an edge of a box on a higher level and lay it down (wooden clip on his side)
Pick up a clip from a small wooden block and put it horizontally on the tale of a cardboard animal
Pick up a clip from a small wooden block and put it vertically on the tale of a cardboard animal
11 Place your hand on a ball of clay and flatten it Move a clay form from a paper to the middle of the flattened clay Make a pattern in the clay by pressing on the top of the form,
pick up the form and put it on a paper
12 Pick up a tube of paint, squeeze some of the paint in a plastic bowl and put the tube back
Pick up a brush and put it in the bowl filled with some paint Pick up the brush in the bowl, color the paper using a template, and put the brush back in the bowl
13 Pick up a seed from the table and drop it in a flower pot Pick up a small sand shovel from a bin filled with sand, pour it in the pot and place the shovel back in the bin
Lift up the watering can, pour some water in the pot filled with sand, and place it back
14 Pick up a spider from a web that is placed on the table, and drop it in a
can on the right
Lift up a cut-out part of a box with a handgrip, and place it next to the box Lift up the can filled with the spiders, pour it out in the box, and
place the can back on the table
15 Pick up a plastic bag filled with short pieces of colored wire, and put the bag in a large bowl
Take the plastic bag out of the bowl, so that the pieces of wire remain in the bowl, and place the bag next to the bowl
Pick up a clamp next to the bowl, grab some pieces of wire in the bowl, put them on a drawing next to the bowl and place the
clamp back on the table
48
Appendix IV, psychometric properties LIFE-H & CP-QOL on the basis of the criteria of Terwee et al. (2007)
Meet-
instrument
Inhouds-
validiteit
Interne
consistentie
Criterium-
validiteit
Construct-
validiteit
Reproduceer-
baarheid
(test-hertest)
Reproduceer-
baarheid
(absolute
meetfout)
Respons-
iviteit
Vloer- en
plafond
effecten
Interpreteer-
baarheid
Somscore
LIFE-H + (1) + (2) + (3) ? (3) +/0 (4) ? (5) ? 0 (6) ? 4
CP-QOL + (7) + (8) ? (9) 0 (10) + (11) ? ? + (12) ? 4
Rating: + = positive; 0 = intermediate; - = poor; ? =no information available
49
LIFE-H
(1)
inhouds-
validiteit
(2)
Interne
consistentie
Bronnen
(Noreau, Fougeyrollas,
& Tremblay, 2005)
(Sakzewski, Boyd, &
Ziviani, 2007)
Verklaring
Twaalf internationale experts waaronder onderzoekers, professionals (ergotherapeuten, verpleegkundigen, sociaal
werkers en psychologen) en vertegenwoordigers van patiëntengroepen werden betrokken in de evaluatie van de
inhoud. De experts werden gevraagd het volgende te beoordelen: de duidelijkheid van de verwoording en de
relevantie van elk item, definities van de termen die gebruikt worden in het assessment, volgorde niveaus van
vervulling. De opmerkingen van de experts werden allemaal individueel door de leden van het onderzoeksteam
geanalyseerd. De tweede versie van de LIFE-H werd op de resultaten gebaseerd van deze validatiestudie. In 1998
gebeurde dit proces opnieuw om de derde versie van de LIFE-H vorm te geven.
Noreau, de auteur van de test, voerde een laatste validatiestudie uit in 2007. In dit onderzoek gaven meer dan 90%
van de panelleden (n=29) aan dat de formulering van de items gemakkelijk te begrijpen waren. De panelleden
bestonden uit ouders van kinderen met een functionele beperking (n=11), onderzoekers (n=3) en pediatrische clinici
en experten ( n=15). De meest experten (64-90%, afhankelijk van de dimensie) oordeelde de representativiteit van
de items als adequaat.
Opmerkelijk was dat een hoger aantal ouders de representativiteit van sommige dimensies zoals mobiliteit en
communicatie hoger beoordeelden, vergeleken met andere panelleden. Het omgekeerde was waarneembaar voor
de dimensies educatie en recreatie. Onderzoeken naar de inhoudsvaliditeit bij andere populaties gaven ook een
adequate tot excellente content validiteit van het assessment aan.
(Cronbach’s alpha, N=48 kinderen met CP)
Daily activities: Alpha= 0.97 → EXCELLENT
Social roles: Alpha= 0.90 → EXCELLENT
All categories: Alpha = 0.73-0.90 → MODERATE TO EXCELLENT
Interpersonal relationships: Alpha= 0.40 --> POOR
= Criteria Terwee (2007) Goede interne consistentie: Chonbach’s alpha tussen de 0.70 en 0.95
50
(3)
Criterium
validiteit
(4)
Reproduceer-
baarheid
(test-hertest)
(5)
Reproduceer-
baarheid,
Absolute meetfout
(Sakzewski et al., 2011)
(Magasi & Post, 2010)
(Noreau et al., 2007)
(Sakzewski, Boyd, &
Ziviani, 2007)
(Sakzewski, Boyd, &
Ziviani, 2007)
De criteriumvaliditeit werd in de literatuur enkel, op een betrouwbare manier, nagegaan met de PEDI ( Pediatric
Evaluation Disability Inventory) en WeeFIM (Functional Independence Measure for Children) (Spearman’s rho,
N=91 ouders van kinderen met CP)
PEDI en LIFE-H: ‘Zelfzorg’ en ‘mobility dimensions’ waren sterk geassocieerd met ‘persoonlijke
verzorging’ en ‘ ‘housing dimensions’ ( 0.79<r<0.88). ‘social function’ van de PEDI werd sterk
geassocieerd met categorieën: ‘Communication’ en ‘resonsibility( r=0.80-0.81)
WeeFIM: Hoge correlaties tussen dimensie ‘housing’ LIFE-H en ‘personal care’ van de weeFIM (r = 0.90-
0.94) en hoge correlatie tussen dimensie‘communicatie’ LIFE-H en ‘communicatie’ van de WeeFIM (r=
0.89)
= Terwee (2007): Een goede beoordeling voor criterium validiteit wordt gegeven wanneer de correlatie met de
gouden standaard ten minste 0,70 is
( ICC’s, N= 48 kinderen met CP)
Short form ( LIFE-H for children): ICC= 0.67: POOR (?)
Long form ( LIFE-H for children): ICC= 0.73: ADEQUATE
= Terwee: 0.70 en een sample van 50 patiënten is een minimum standaard voor betrouwbaarheid
Standard error of measurement (SEM) werd enkel berekend voor geriatrische cliënten bij een andere versie van de
LIFE-H
51
(6)
vloer- & plafond-
effecten
(Noreau et al., 2007)
Bijzondere aandacht moet gegeven worden aan de categorie ‘interpersoonlijke vaardigheden’ van de LIFE-H. 75% van de
kinderen in het onderzoek van Noreau, et al (2007) haalde een score van 8 of hoger wat op een plafond-effect kan wijzen.
= Terwee: Vloer -& plafondeffecten zijn aanwezig wanneer meer dan 15% van de respondenten de hoogst of laagst
mogelijke score scoort.
CP-QOL
(7)
Inhouds-validiteit
(8)
Interne
consistentie
(9)
Criterium-
validiteit
(Waters & Mackinnon,
2007)
(Waters & Mackinnon,
2007)
(Carlon et al., 2010)
“The development of the CP QOL-Child involved qualitative interviews with primary caregivers and children, reviewing
and developing items and response scales used in other QOL questionnaires, and piloting the CP QOL-Child using
interviews with both the primary caregivers and children. Seven Items included in the CP QOL-Child were based on
transcripts of interviews with children with CP and their primary caregivers.”
“Internal consistency ranged from 0.74 to 0.92 (n= 205)”
= Terwee: Goede interne consistentie: Chonbach’s alpha tussen de 0.70 en 0.95
“Domains moderately correlated to those of KIDSCREEN and CHQ”
= exacte waarden niet terug te vinden in de literatuur
52
(10)
construct-
validiteit
(11)
Reproduceer-
baarheid
(test-hertest)
(12)
vloer- en plafond-
effecten
(Waters & Mackinnon,
2007)
(Waters & Mackinnon,
2007)
(Carlon et al., 2010)
“All domains of the CP QOL-Child were moderately correlated with global QOL (primary caregivers r=0.18-0.58; child
self-report r=0.41-0.64) and global health (primary caregivers r=0.21-0.56, child self-report 0.51-0.60). All correlations
were in the expected direction. Domains of the CP QOL-Child were moderately correlated with KID-SCREEN (r=0.30 -
0.51) except pain and impact of disability (r=-0.14). For child self-report, all domains on the CP QOL-Child were
moderately correlated with KIDSCREEN (r=0.61 -0.70).
Domains of the CP QOL-Child were also moderately correlated with functioning (r=0.18-0.62) except access to services
(r=0.11). Domains of the CP QOL-Child were also correlated with domains of the CHQ. Table IV contains the correlation
coefficients between CP QOL-Child and CHQ. The large majority of the correlations were significant and similar domains
of the CP QOL-Child and CHQ tended to be moderately correlated, e.g. functioning was correlated with physical
functioning (r=0.42) and emotional well-being was correlated with self-esteem (r=0.49).”
= Terwee: correlatie met de gouden standaard ten minste 0,70 is
For primary caregivers, 2-week test-retest reliability ranged from 0.76 to 0.89 (n= 205)
= Terwee: 0.70 en een sample van 50 patiënten is een minimum standaard voor betrouwbaarheid
Review: ‘geen vloer- en plafondeffecten’
53
Appendix V, ‘LIFE-H’
54
55
56
57
58
Appendix VI, ‘CP-QOL’
59
60
61
62
63
Appendix VII, Additional tables
64
Appendix VIII, ‘We are in the newspaper! ’
Published on http://nieuws.kuleuven.be
Kinderen met halfzijdige hersenverlamming verleggen grenzen tijdens
therapiekamp
28 Aug 2014
Met een robot dansen op Gangnam Style, een spelletje vier-op-een-rij spelen of fruit dippen
in een chocoladefontein: voor kinderen met een hemiplegie of halfzijdige hersenverlamming
is het meer dan alleen plezier maken. Tijdens een therapiekamp aan de Faculteit Bewegings-
en Revalidatiewetenschappen werkten acht kinderen tussen 10 en 12 jaar op deze manier aan
de verbetering van hun arm- en handfunctie.
Hemiplegie is het gevolg van een hersenletsel dat ontstaat voor, tijdens of kort na de
geboorte, en wordt ook hersenverlamming of cerebrale parese genoemd. Tijdens het kamp
kregen de kinderen zes uur per dag intensieve therapie om hun arm- en handfunctie te
verbeteren.
65
Een team van kinesitherapeuten onder leiding van Katrijn Klingels en doctoraatsstudente
Lisa Mailleux stimuleerde hen om op een speelse manier de aangedane arm meer en beter te
gebruiken. Dat gebeurt met Constraint-Induced Movement Therapy (CIMT): de kinderen
dragen een spalkje aan de goede hand, zodat ze alleen met de minder goede hand taken
kunnen uitvoeren. Zo wordt die hand intensief gestimuleerd.
Naast individuele therapie waren er ook groepsactiviteiten, zoals knutselen, koken, sport en
spel. Een revalidatierobot met de naam Zora speelde daarbij een belangrijke rol. Deze
interactieve robot kan spreken, zingen, dansen en stappen. Het ruimteschip van Zora was
neergestort en elke ochtend kregen de kinderen een opdracht om het schip in gereedheid te
brengen om terug te keren naar de ruimte. 's Avonds toonden de kinderen haar hun
knutselwerken en voerden ze gesprekken met haar over wat ze allemaal hadden meegemaakt.
Samen met Zora sloten ze de dag af met een dansje. De interactie met de robot stimuleerde
en motiveerde de kinderen om telkens weer hun best te doen.
"Dit eerste kamp was een groot succes", zegt Katrijn Klingels. "Het is fantastisch om te zien
hoe flink de kinderen oefenen met hun moeilijke hand en hoe fier ze zijn over de vooruitgang
die ze boeken, hoe ze samen plezier maken en hun grenzen verleggen."
Nieuwe therapievorm
Het therapiekamp kadert in een onderzoeksproject aan het Departement
Revalidatiewetenschappen, onder leiding van professor Hilde Feys en Katrijn Klingels.
Omdat kinderen met een hemiplegie soms ook moeilijkheden hebben met het plannen van
bewegingen, gaan de onderzoekers na of een nieuwe therapievorm, actie-observatie-
therapie, in combinatie met CIMT beter is dan CIMT alleen. Tijdens actie-observatie-
therapie kijken de kinderen eerst naar een videofragment van een bepaalde taak, om die
daarna zelf uit te voeren, zegt Katrijn Klingels. Door eerst te kijken naar de taak, zal het kind
die taak beter leren plannen en uitvoeren. Deze nieuwe therapievorm is gebaseerd op het
spiegelneuronensysteem.
66
We willen ook onderzoeken of bepaalde kinderen meer baat hebben bij deze intensieve
therapievorm. We gaan na of de plaats en de grootte van het hersenletsel, de integriteit van
de motorische en sensorische hersenbanen, en de functionele reorganisatie van de hersenen
een invloed hebben op de arm- en handfunctie en op de vooruitgang die de kinderen kunnen
boeken met deze therapie. Hiervoor werken we samen met het Departement Ontwikkeling
en Regeneratie, het Departement Beeldvorming en Pathologie, en met ETH Zurich in
Zwitserland.
Het kamp werd georganiseerd in samenwerking met de KU Leuven, UZ Leuven
(CPreferentiecentrum o.l.v. professor Guy Molenaers en klinisch bewegingslaboratorium
o.l.v. professor Kaat Desloovere), QBMT (het bedrijf dat robot Zora ontwikkelde) en de vzw
Move To Improve.
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Robot laat kindjes met hersenverlamming dansen
26/08/15 - 06u00 Bron: Het Laatste Nieuws
Video: http://www.hln.be/hln/nl/33/Fit-Gezond/article/detail/2435132/2015/08/26/Robot-
laat-kindjes-met-hersenverlamming-dansen.dhtml
In het Diepenbeekse dienstencentrum Sint-Gerardus vindt een therapiekamp voor kinderen
met een hersenletsel plaats. De deelnemers worden daarbij ondersteund door zorgrobotje
Zora.
Een dansje doen, een spelletje spelen of de kinderen vragen wat ze die dag allemaal gedaan
hebben: Zora, het bekende robotje van het Belgische bedrijf QBMT, kan het allemaal. De
kinderen genieten er alvast van met volle teugen: ze begroeten Zora als was het een
deelneemster aan het kamp. "Deze week ontvangen we hier acht kinderen met hemiplegie.
Dat is het gevolg van een hersenletsel dat ontstaat voor, tijdens of kort na de geboorte en
wordt ook hersenverlamming genoemd. In dit therapiekamp wordt gewerkt op de verbetering
van de arm- en handfunctie", zegt Lisa Mailleux van de KU Leuven. "Dat doen we door een
spalkje te bevestigen aan hun 'goede hand' zodat enkel de minder goede hand gebruikt kan
worden om taken uit te voeren en op deze manier dus zeer intensief gestimuleerd wordt. Zora
stimuleert en motiveert hen daarbij elke dag bij het start- en slotmoment."
Kaat Valkeneers (10) uit Hasselt is alvast helemaal weg van het dansende robotje.
"Paardrijden en een taart maken was heel leuk, maar spelen met Zora is toch nog net iets
leuker", lacht ze.
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Appendix IX, toestemming tot consultatie