1

Lithuanian University of Health Sciences

Faculty of Medicine

Department of Pediatrics Rehabilitation

Title of Master’s Thesis:

The efficacy of interventions to increase participation of children with cerebral palsy

Author:

Sherine Bahrou

Supervisor:

Ph.D., Assoc. Prof. Audrone Prasauskiene

Kaunas

2017-2018

2

Table of Contents

SUMMARY ……………………………………………………….………………….……….…. 3

ACKNOWLEDGMENTS …………………………………………………….…………….…… 5

CONFLICTS OF INTEREST …………………………………………………………………… 6

PERMISSION ISSUED BY THE ETHICS COMMITTEE ……………………………..……… 6

LIST OF ABBREVIATIONS …………………………………………………………………… 7

CHAPTER 1: INTRODUCTION ………………………………………………………..……… 9

CHAPTER 2: AIMS AND OBJECTIVES ……………………………………………………… 10

CHAPTER 3: LITERATURE REVIEW ………………………………………………………... 11

3.1 Definition, prevalence, types, and etiology of cerebral palsy ………………….. 11

3.2 ICF framework …………………………………………………………………...…… 13

3.3 Participation: definition, construct, and related concepts ………….………..… 14

3.4 Participation of children with CP ……………………………………………… 15

3.5 Systemic review …………………………………………………………...…... 16

CHAPTER 4: RESEARCH METHODOLOGY AND METHODS …………………………... 19

CHAPTER 5: RESULTS ………………………………………………………………………. 20

CHAPTER 6: DISCUSSION OF RESULTS ……………………………………………….…. 25

CHAPTER 7: CONCLUSIONS …………………………………………………………...…... 27

CHAPTER 8: PRACTICAL RECOMMENDATIONS ……………………………………….. 28

CHAPTER 9: REFERENCES ………………………………………………………………... 29

CHAPTER 10: ANNEXES ……………………………………………………………………. 32

3

SUMMARY

Author name: Sherine Bahrou

Research title: The efficacy of interventions to increase participation of children with cerebral palsy.

Aim: To determine the efficacy of interventions aimed at increasing the level of participation in children

with cerebral palsy.

Objectives:

1. Identify the extent and range of the existing literature on the efficacy of interventions aimed to increase

participation of children with cerebral palsy.

2. Systematically review evidence on the impact of rehabilitation interventions on the participation of

children with cerebral palsy.

3. Identify gaps in the research and to delineate the future research needs.

Methodology: The systematic review was conducted in line with the Preferred Reporting Items for

Systematic Reviews and Meta-analyses statement (PRISMA). The data sources include Medline (PubMed),

along with reference lists of relevant reviews and included articles. Study eligibility criteria were studies

that assess the efficacy of interventions to increase participation of children with cerebral palsy.

Results: Eleven studies were included in the review. Hand and arm bimanual therapy and constrain-

induced movement therapy had positive impact on social participation, while robot-assisted upper-limb

therapy didn’t have effect on participation. Studies investigating effect of physiotherapy interventions

show equivocal results. Positive results on participations were seen in studies that combined

physiotherapy with context- based therapies and used goal-oriented approach for treatment planning.

Conclusion: Child-focused and context-focused interventions to promote participation of children with CP

are equally effective. The goal setting is critical component of successful intervention.

Keywords: Cerebral palsy, participation, children, early intervention.

4

Santrauka

Autorius: SherineBahrou

Darbo tema: Intervencijų, skirtų cerebrinį paralyžių turinčių vaikų dalyvumui gerinti, efektyvumo

apžvalga.

Tikslas: Nustatyti intervencijų, skirtų cerebrinį paralyžių turinčių vaikų dalyvumui gerinti, efektyvumą

Uždaviniai:

1. Atrinkti mokslinius straipsnius, kuriuose analizuojamas intervencijų, skirtų cerebrinį paralyžių

(CP) turinčių vaikų dalyvumui gerinti, efektyvumas

2. Sistemiškai apžvelgti reabilitacijos intervencijų, skirtų CP turinčių vaikų dalyvumui gerinti,

mokslinį pagrįstumą.

3. Įvertinti kokios intervencijos ištirtos nepakankamai, nustatyti kokie papildomi tyrimai reikalingi

šioje srityje.

Metodika: Sisteminė apžvalga atlikta vadovaujantis sisteminių apžvalgų ir meta-analizių rengimo

rekomendacijomis PRISMA. Atrinkti moksliniai straipsniai iš Medline (PubMed) duomenų bazės, ir į šią

apžvalgą įtrauktų straipsnių literatūros sąrašų. Įtraukimo į apžvalgą kriterijai – tyrimai, analizuojantys

intervencijų, skirtų CP turinčių vaikų dalyvumui gerinti, efektyvumą.

Rezultatai: Į apžvalgą įtraukti 11 tyrimų. Rankos suvaržymo terapija ir abiejų rankų lavinimas turėjo

teigiamą poveikį dalyvumui socialinėse veiklose. Rankos lavinimas robotizuoto treniruoklio pagalba

dalyvumui įtakos neturėjo. Studijų, tyrusių kineziterapijos (KT) efektyvumą, duomenys prieštaringi.

Teigiamas poveikis dalyvumui rastas studijose, kuriose KT buvo derinama su į aplinkybes nukreiptomis

intervencijomis ir kai buvo išsikelti konkretūs terapijos tikslai.

Išvados: Į vaiką orientuotos ir į aplinkybes orientuotos terapijos buvo vienodai efektyvios vaikų su CP

dalyvumui. Tikslų išsikėlimas - būtina intervencijų sėkmės sąlyga.

Raktiniai žodžiai: cerebrinis paralyžius, dalyvumas, vaikai, ankstyvoji intervencija

5

ACKNOWLEDGMENTS

Many thanks to my supervisor Ph.D. Assoc. Prof. Audrone Prasauskiene for her guidance and

encouragement throughout my research. Also, I would like to thank my family for all their help and

support specially my father DR. Abdullah Bahrou and my mother DR. Chaza al Naeb. Finally, special

thanks to my beloved husband for being always with me.

6

CONFLICTS OF INTEREST

The author reports no conflicts of interest.

PERMISSION ISSUED BY THE ETHICS COMMITTEE

The author no needed permission from Ethics committee.

7

LIST OF ABBREVIATIONS

APCP - Assessment of Preschool Children's Participation

BTX – Botox

BMFM - Bimanual Fine Motor

CAPE - Children’s Assessment of Participation and Enjoyment

CP - Cerebral Palsy

CT - Computed Tomography

CIMT - Constraint-Induced Movement Therapy

GMFCS - Gross Motor Function Classification System

HAS - Habitual Activity Survey

HABIT-ILE - Hand and Arm Bimanual Intensive Therapy Including Lower Extremity

ICF-CY - International Classification of Functioning, Disability and Health-Children and Youth

ICF - International Classification of Functioning, Disability, and Health

ID - Intellectual Disability

LAQ - Lifestyle Assessment Questionnaire

MRI- Magnetic Resonance Imaging

MACS – Manual ability Classification System

PEM-CY - Participation and Environment Measure for Children and Youth

PRE - Progressive resistance exercise

PODCI - Pediatric Outcomes Data Collection Instrument

PRISMA - Preferred Reporting Items for Systematic reviews and Meta-Analyses

PEDI - Pediatric Evaluation of Disability Inventory

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RAT - Robot-Assisted Therapy

SCPE - Surveillance of Cerebral Palsy in Europe

WHO - World Health Organization

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CHAPTER 1: INTRODUCTION

Cerebral palsy (CP): “a group of permanent disorders that affect the development of movement and

posture, and cause activity limitation, that attributed to nonprogressive disturbances that occurred in the

developing fetal or infant brain. The disorders of motor system of cerebral palsy are often accompanied by

disturbances of sensation, perception, cognition, communication, and behavior, by epilepsy, and by

secondary musculoskeletal problem”. [1]

Participation is a key outcome of health for all children, and defined as involvement and engagement in

life situations, including domestic life, school, leisure, and recreation activities. Children’s participation

relates to their behavioral and emotional well-being, social relationships, development of social and

physical competence, and their sense of meaning and purpose in life. Children with a chronic disability

such as CP have reduced opportunities to participate in meaningful or purposeful activities. They need

support and individually tailored interventions to promote and support their participation. In Imms [2]

published a systematic review of 40 studies investigating different aspects of participation of children with

CP. Authors found that children with CP had a reduced level of participation, especially those who had

greater functional impairments. Although children participated in a wide range of activities, involvement

in these activities was reduced. Interestingly, authors of the review didn’t find studies analyzing the effect

of interventions aimed to promote participation. The aim of this review was analyzing research evidence

related to interventions aimed to promote participation of children with CP as well as to determine the

efficacy of these interventions.

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CHAPTER 2: AIMS AND OBJECTIVES

Aim: To determine the efficacy of interventions aimed at increasing the level of participation in children

with cerebral palsy.

Objectives:

1. Identify the extent and range of the existing literature on the efficacy of interventions aimed to increase

participation of children with cerebral palsy.

2. Systematically review evidence on the impact of rehabilitation interventions on the participation of

children with cerebral palsy.

3. Identify gaps in the research and to delineate the future research needs.

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CHAPTER 3: LITERATURE REVIEW

3.1 Definition, prevalence, types, and etiology of cerebral palsy

Cerebral palsy (CP): “describes a group of permanent disorders that affect the development of movement

and posture, and cause activity limitation, that attributed to nonprogressive disturbances that occurred in

the developing fetal or infant brain. The disorders of motor system of cerebral palsy are often

accompanied by disturbances of sensation, perception, cognition, communication, and behavior, by

epilepsy, and by secondary musculoskeletal problems”. [1] The prevalence of CP in children is 2-2.5 per

1000 live births in developed countries [3] .

Surveillance of Cerebral Palsy in Europe (SCPE) has classified CP into three groups depending on

neurological signs;

1. Spastic CP: patients have increased muscle tonus, and pathological reflexes (e.g., hyperreflexia,

Babinski response) and impairment of movement and posture. This type is caused by a lesion of the upper

motor neuron. This type is divided in two subtypes: spastic unilateral CP when limbs in one body side are

affected; and bilateral, when CP affects symmetrical parts of the body.

2. Dyskinetic CP: is characterized by presence of uncontrolled, recurring, and occasionally rapid

movements. This type is due to damage to basal ganglia, which is responsive for movement control. SCPE

[9] divides this type to dystonic and choreo-athetotic CP subtypes. Dystonic CP is diagnosed when main

signs of CP are abnormal postures and hypertonia. Choreo-athetotic CP is diagnosed when the main

clinical disturbances of movement and posture are hyperkinesia and hypotonia.

3. Ataxic CP: is caused by a lesion of the cerebellum. Main symptoms of this type are: (1) loss of

muscular coordination, which result in performing the movements with abnormal force, precisity, and

rhythm, typical features are ataxic gait (disturbed balance) (2) Tremor (3) Low tone.

4. Mixed CP: is diagnosed when child has movement problems that fall into several types of CP (e.g. child

has spasticity together with ataxia or dyskinesia). While the SCPE describe a child’s presentation, they do

not define any criteria for recording the functional abilities of the child. [9-4]

Further functional classification systems for children with CP were developed: The Gross Motor Function

Classification System (GMFCS), the Manual Ability Classification System (MACS), Communication

Function Classification System, and Eating and Drinking Ability Classification System for Individuals

with Cerebral palsy. [10]

The GMFCS was developed to describe gross motor function in children with CP and has its focus on

12

self-initiated movements, in particular sitting and walking. It is an age-related five-level system in which

level I represents the least limitation and level V the most. [10]

Level 1: children can walk independently without support, can climb, run with decreased speed.

Level 2: children walk without restrictions, limitations walking outdoors and, in the community,

Level 3: Children walk with assistive mobility devices, limitations walking outdoors and in community.

Level 4: Self mobility with limitations, children are transported or use power mobility outdoors and in the

community.

Level 5: Self mobility is severely limited, even with use of assistive technology. (Figure 1)

Figure 1. Gross Motor Function Classification System. Source (Paulson et. al., 2017)[10]

The MACS describes how well children aged 4–18 years with CP use their hands when handling objects

in usual daily activities. It reflects the child's manual performance, not the maximal capacity. Like the

GMFCS, the MACS is a five-level system where level I represents the best manual ability and level V

indicates that the child does not have any active hand function. [10]

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Level 1- Handles objects easily and successfully

Level 2- Handles most objects but with somewhat reduced quality and/or speed of achievement, some

activities achieved with little difficulty.

Level 3- Handles objects with difficulty, needs help to prepare or modify activities.

Level 4- Handles a limited selection of easily managed objects in adapted situations; performs parts of

activities with effort and with limited success.

Level 5- Does not handle objects and has severely limited ability to perform even simple actions, need

total assistance.

3.2 ICF framework

Functional consequences of CP can be displayed using the International Classification of Functioning,

Disability, and Health (ICF). It was developed by World Health Organization (WHO) and approved for

use by the World Health Assembly in 2001. The ICF is a conceptual framework is based on a model of

variety dimensions is made apparent by bidirectional arrows reflecting the ongoing influence of

environmental factors on body functions, activities, and participation. Contextual factors in the conceptual

framework are the personal factors such as age, education, and income status ‘‘features of the individual

that are not part of health conditions or health states’’. [11] Although the influence of personal factors

recognized in the conceptual model. Body functions and body structures involve physiological functions,

including psychological and anatomical parts of the body. Impairments defined as problems in body

function or structure. The activity described as a person does a task or action. Participation is limited

defined as involvement in a life situation, and participation restrictions are difficulties that a person may

experience in a life situation. Environmental factors can be physical, social, and attitudinal that people

experience during their lives. [11]

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Figure 3: Conceptual framework for International Classification of Functioning, Disability, and

Health (ICF). (WHO, 2001) [8]

3.3 Participation, definition, construct and related concepts

The publication of the World Health Organization’s (WHO) International Classification of Functioning,

Disability, and Health (ICF) has resulted in an intensification of international interest in ‘participation’ as

the ultimate health outcome. [11] The ICF define participation as ‘involvement in a life situation’. [11] ICF

is a classification of health and health-related domains. Several studies define participation as a family of

construct. [9,11] Participation consists of two related concepts: attendance and involvement. Other authors

[12-13] has also suggested these two concepts. Attendance defined as ‘being there’ and measured as

frequency of attending, and the range or diversity of activities in which an individual takes part.

Involvement: the experience of participation while visiting, including elements of motivation, persistence,

social connection, and effect. [14] (Figure 2) shows participation family.

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Figure2: A family of participation and participation-related constructs. (Imms, 2016) [15]

3.4 Participation of children with CP

There were studies about the participation of children with CP which aims to explore what children who

have cerebral palsy do. Children with CP have greater participation restriction comparing to healthy

children, and they have weaker participation outcomes comparing with other disability groups. Law et al.

[16] discovered that children with neurological impairments they have decreased the intensity of

participation. Restriction of involvement reported in educational, social and recreational domains. These

studies focused on factors associated with the children. Diagnosis was only found to be significant by

Longmuir. [14] Other studies show that level of severity of impairment affects the degree of participation

restriction. Morris et al. found that social conjunction predicted by movement ability, intellectual delay, and

manual ability. [17] There was no substantial effect of educational level of parents or number of children in

a family on participation. Six studies were about the influence of the environment on the participation of

children with disabilities. They examined physical dependence, limitation of mobility, educational

exclusion, and restriction of social interaction. The location has shown an effect on participation, with a

16

possible 16-point difference in LAQ score depending on the district the child lived. Different factors have

effects on participation, such as Social supports and physical accessibility. [18-20] There was no study

describing the intervention to promote participation of children with CP, and this was the reason to conduct

the review of papers published after 2009. The aim of the study to determine the efficacy of interventions

aimed at increasing the level of participation in children with CP.

3.5 Systemic review

Systematic review: Summarizes the results of available studies that are selected by specific criteria.

Differences between narrative and systemic analysis are; in the narrative, data chosen without specific

criteria for selection based on author selection, these are for descriptive study. The systemic review uses

Several steps to follow-on;

1- formulate a focused question that you need to get an answer for it, and title of the research is better to

keep it as shorter as possible.

2- Define inclusion and exclusion criteria, for example, age of people involved in the study, interventions,

outcomes.

3- Develop search strategy and locate reviews, It is essential to come up with a comprehensive list of key

terms (i.e., “MeSH” terms) while using the references to find the studies.

4- Select studies: This process of review is done by at least two reviewers to establish reliability. The

authors should keep a log of all reviewed studies with reasons for inclusion or exclusion.

5- Extract data: is helpful to use and create data extraction form for organizing the information extracted

from each reviewed study (e.g., authors, publication year, number of participants, age range, study design,

outcomes, included/excluded).

6- Assess study quality: There are comprehensive recommended guidelines and standards available such

as the Consolidated Standards of Reporting Trials (CONSORT Statement; http://www.consort-

statement.org/), as well as articles providing recommendations for improving quality in RCTs and meta-

analyses for psychological interventions.

7- Analyze and interpret results: There are various statistical programs available to calculate effects sizes

for meta-analyses.

8- Disseminate findings: Although reviews should publish online.

PRISMA Checklist used in literature (Table1).

Table 1. PRISMA Checklist

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Section/topic # Checklist item

TITLE

Title 1 Identify the report as a systematic review, meta-analysis, or both.

ABSTRACT

Structured

summary

2 Provide a structured summary including, as applicable: background;

objectives; data sources; study eligibility criteria, participants, and

interventions; study appraisal and synthesis methods; results; limitations;

conclusions and implications of key findings; systematic review

registration number.

INTRODUCTION

Rationale 3 Describe the rationale for the review in the context of what is already

known.

Objectives 4 Provide an explicit statement of questions being addressed with reference

to participants, interventions, comparisons, outcomes, and study design

(PICOS).

METHODS

Protocol and

registration

5 Indicate if a review protocol exists, if and where it can be accessed (e.g.,

Web address), and, if available, provide registration information

including registration number.

Eligibility

criteria

6 Specify study characteristics (e.g., PICOS, length of follow-up) and

report characteristics (e.g., years considered, language, publication

status) used as criteria for eligibility, giving rationale.

Information

sources

7 Describe all information sources (e.g., databases with dates of coverage,

contact with study authors to identify additional studies) in the search and

date last searched.

Search 8 Present full electronic search strategy for at least one database, including

any limits used, such that it could be repeated.

Study

selection

9 State the process for selecting studies (i.e., screening, eligibility, included

in systematic review, and, if applicable, included in the meta-analysis).

Data

collection

process

10 Describe method of data extraction from reports (e.g., piloted forms,

independently, in duplicate) and any processes for obtaining and

confirming data from investigators.

Data items 11 List and define all variables for which data were sought (e.g., PICOS,

funding sources) and any assumptions and simplifications made.

Risk of bias

in individual

studies

12 Describe methods used for assessing risk of bias of individual studies

(including specification of whether this was done at the study or outcome

level), and how this information is to be used in any data synthesis.

Summary

measures

13 State the principal summary measures (e.g., risk ratio, difference in

means).

Synthesis of

results

14 Describe the methods of handling data and combining results of studies,

if done, including measures of consistency (e.g., I2) for each meta-

analysis.

Risk of bias 15 Specify any assessment of risk of bias that may affect the cumulative

18

across studies evidence (e.g., publication bias, selective reporting within studies).

Additional

analyses

16 Describe methods of additional analyses (e.g., sensitivity or subgroup

analyses, meta-regression), if done, indicating which were pre-specified.

RESULTS

Study

selection

17 Give numbers of studies screened, assessed for eligibility, and included

in the review, with reasons for exclusions at each stage, ideally with a

flow diagram.

Study

characteristics

18 For each study, present characteristics for which data were extracted

(e.g., study size, PICOS, follow-up period) and provide the citations.

Risk of bias

within studies

19 Present data on risk of bias of each study and, if available, any outcome

level assessment (see item 12).

Results of

individual

studies

20 For all outcomes considered (benefits or harms), present, for each study:

(a) simple summary data for each intervention group (b) effect estimates

and confidence intervals, ideally with a forest plot.

Synthesis of

results

21 Present results of each meta-analysis done, including confidence intervals

and measures of consistency.

Risk of bias

across studies

22 Present results of any assessment of risk of bias across studies (see Item

15).

Additional

analysis

23 Give results of additional analyses, if done (e.g., sensitivity or subgroup

analyses, meta-regression [see Item 16]).

DISCUSSION

Summary of

evidence

24 Summarize the main findings including the strength of evidence for each

main outcome; consider their relevance to key groups (e.g., healthcare

providers, users, and policy makers).

Limitations 25 Discuss limitations at study and outcome level (e.g., risk of bias), and at

review-level (e.g., incomplete retrieval of identified research, reporting

bias).

Conclusions 26 Provide a general interpretation of the results in the context of other

evidence, and implications for future research.

FUNDING

Funding 27 Describe sources of funding for the systematic review and other support

(e.g., supply of data); role of funders for the systematic review.

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CHAPTER 4: RESEARCH METHODOLOGY AND METHODS

Systematic literature review where searches were conducted using Medline (PubMed), all articles

published from 2012 included in the searches with no more than five years search criteria used. The search

terms used were: "cerebral palsy" AND "participation, patient" one search; another search "cerebral

palsy" AND "community participation"; and another one "cerebral palsy" AND "participation, social".

Inclusion and exclusion criteria listed in the (Table2) below.

Table.2

Inclusion Exclusion

Children study Adult studies

Human studies Animal studies

Full-text articles Reviews

Cerebral palsy Other neurological diseases

Participation studies Not participation studies

Within five years More than five years

Intervention studies Studies without intervention

Studies in English Other languages

For all selected studies, the full text retrieved and examined. Data on the interventions aimed to enhance

the participation of children with CP were extracted using a standardized data extraction form. Data

obtained included: (1) general information about the study (author(s), year of publication, study location);

(2) specific information related to the study population, aims of the study, study design, intervention (type,

intensity, duration), outcome measures, main results. The information was analyzed using two approaches:

(1) table in which a descriptive summary (author, year, and country, the aim of the study, study design,

participants, intervention, results) provided. (2) A narrative synthesis to summaries the data on effects of

interventions aimed to improve participation within functional domains of leisure, school, and community.

The ICF framework and terminology employed as a guide for this review.”

The information was analyzed using a table in which a descriptive summary (author, year, and country,

the aim of the study, study design, participants, intervention, results) provided.

20

CHAPTER 5: RESULTS

After the search in PubMed 340 articles were found, and 11 met the inclusion criteria (figure 4).

Articles were from the USA (2 studies), Belgium (1 study), Taiwan (1 study), Canada (2 studies),

Australia (2 studies), and Netherland (3 studies). Most studies were randomised-controlled (8 studies),

while others explored single-subject exploratory (1 study) or prospective, one group repeated-measures

design (2 studies).

In the studies selected for the review, five different outcome measures for participation were used:

Assessment of Life Habits (Life-HABITs), [21,22,26,27,29], (PEM-CY), [28], (PODCI), [31], (APCP),

[24], (CAPE) [27,29,30] and (ICF checklist) [23,25].

Three studies focused on interventions targeted to improve upper limb function: hand and arm bimanual

intensive therapy (HABIT) [21], robot-assisted therapy [22], and constraint-induced movement therapy

[27]. Results of these studies indicate that arm and hand training addresses through either CIMT or

HABIT achieved gains in hand function and social participation. Robot-assisted upper-limb therapy

improved upper limb functions, but not functional activities and social participation.

Six investigated the effect of physiotherapy interventions: physical fitness training [26,29], progressive

resistance exercise (PRE) [30], running interventions, hippotherapy [23,28], and classical ballet program

[25]. Findings of these studies show equivocal results. No effects on participation found in the studies that

analysed outcomes of PRE and lifestyle intervention (e.g., physical fitness training, counselling sessions

focused on physical behaviour and sports participation). Running interventions had an effect on school

participation but didn't affect participation at home or community. Six-month physical activity stimulation

program consisting of motivational interviewing, home-based physiotherapy, and 4 months of fitness

training as well as hippotherapy improved social participation of children with CP. The children who

participated in a classical ballet program reported high enjoyment level and desire for more classes. The

parents of children reported perceived therapeutic benefit, and the therapists viewed the class as a positive

adjunct to therapy as well.

An interesting study from Canada [24] evaluated the efficacy of a child-focused versus context-focused

intervention on voluntary, day-to-day activities outside a preschool. In the child-focused approach group,

therapists identified the impairments underlying a functional limitation (e.g., tone, posture, the range of

motion), and provided therapy to remediate the impairments and practice specific movements and tasks

(e.g. stretching, casting, strength training, a practice of different functional activities). In the context-

focused approach group, children were encouraged to use compensatory strategies to achieve functional

21

tasks. In the context-focused group, therapists were modifying physical characteristics of the environment,

task, materials or tools, practice of functional mobility activities, changing a task instruction, adding

adaptive equipment, and providing education/instruction to the family. Both approaches resulted in

equivalent and significant improvements in self-care, mobility, and participation outcomes of the children

with CP. Wright et al. [31] analysed effect of botulinum toxin (BTX) therapy on participation and had

found it effective as measured by PODCI scales.

Figure 4. Flowchart of the study design

Records identified through

database searching

(n =345)

Scre

enin

g In

clu

ded

El

igib

ility

Id

enti

fica

tio

n

Additional records identified

through other sources

(n =0)

Records after duplicates removed

(n =55)

Records screened

(n =290)

Records excluded

(n =242)

Full-text articles assessed

for eligibility

(n = 48)

Full-text articles excluded,

with reasons (n =32)

• Not children studies

n=9

• Not cerebral palsy n =

3

• Not intervention study

n=9

• Validity and reliability

n = 2

• Systematic review n=6

• Not participation

studies n= 3

Studies included in

qualitative synthesis

(n =11)

22

Articles identified with the search, of studies that investigated interventions aimed to improve

participation of CP children (Table3).

Table3. studies that investigated interventions aimed to improve participation of CP children

Author/year

Country

, sample

N (age)

Study design,

interventions

Participatio

n measures

Results

Bleyenheuft

et al. (2015)

[21]

USA,

N=24

(6 -13)

Randomized controlled

I group (n=12) -

immediate HABIT-ILE;

II group (n=12) -

delayed HABIT-ILE;

duration - 10 days = 90

hours

Life-

HABITs

Social

participation

performance, as

well as

satisfaction

of the parents,

improved

Gilliaux et

al. (2015)

[22]

Belgiu

m,

N=16

(7-18)

Randomized controlled

I group (n=8) -

conventional

therapy 5 sessions per

week over 8 weeks

II group (n=8) - 3

sessions of conventional

and 2 sessions of robot-

assisted therapy per

week over 8 weeks

Life-

HABITs

Robot-assisted

therapy improved

upper limb

functions, but not

functional

activities and

social

participation

Hsieh et al.

(2017)

[23]

Taiwan,

N=14

(3–8)

Single case (ABA)

Hippotherapy 30 min,

once weekly for 12

weeks

Withdrawal phase – 12

weeks

ICF-CY

checklist

Participation of

children at

GMFCS levels I–

III showed

significant

improvement, but

those at GMFCS

levels IV and V

remained

unchanged

Law et al.

(2011)

[24]

Canada,

N=128

(1-5)

Randomized controlled I

group (n=71) - child-

focused and II group

APCP

Significant

changes for both

treatment groups

23

(n=57) - context-focused

intervention over 6

months

found for play

intensity

(p<0.04), physical

activity intensity

and diversity

(p<0.001), and

total score

intensity

(p<0.01).

Lopez-Ortiz

et al. (2012)

[25]

USA,

N=16

Pilot exploratory

Classical ballet training

once weekly over 5-8

weeks

ICF-CY

checklist

The children

expressed the

desire for more

classes

(p=0.0001), a

high enjoyment

level (p=0.0001),

new interest in

participation in a

school group

(p=0. .04), new

interest in

watching a dance

show (p=0.0001)

and new interest

in attending an art

show ( p=0.004).

Slaman et

al. (2015)

[26]

Netherl

ands

N=456

(16-24)

Randomized controlled

6-month lifestyle

intervention: physical

fitness training,

counseling sessions

focused on physical

behavior and sports

participation

Life-

HABITs

No intervention

effects were noted

for social

participation

Sakzewski

et al. (2011)

[27]

Australi

a, N=64

(5-14)

Randomized controlled

CIMT delivered in day

camps (total 60 h over

10 d) using a circus

theme with goal directed

training

Life-

HABITs,

CAPE

Changes in

participation in

specific life habits

achieved, which

corresponded

with goals

identified by

children and their

caregivers.

Gibson et

al. (2017)

[28]

Australi

a, N=42

(9–18)

Randomized controlled

12-week running

intervention

PEM-CY There was

improvement in

participation in

24

the school

environment

(mean difference

1.18:

95%, CI 1.00–

1.39, p=0.045).

Wely et al.

(2014)

[29]

Netherl

and,

N=49

(7–13)

Randomized controlled

6-month physical

activity stimulation

program: motivational

interviewing, home-

based physiotherapy and

4 months of fitness

training.

Life-

HABITs,

CAPE

Intervention

resulted in a

positive effect on

social

participation in

domestic life

(mean between-

group difference

= 0.9, 95%

confidence

interval (CI) = 0.1

to 1.7 [1–10

scale], P = 0.03),

but not in

recreation and

leisure.

Scholtes et.

al. (2012)

[30]

Netherl

and,

N=51

(6-13)

Randomized controlled

I group (n=26) – 12

weeks functional PRE-

circuit training, for 3

times a week

II group (n=25) –

conventional therapy

CAPE The intervention

had no significant

effect on CAPE.

Wright et

al. (2015)

[31]

Canada,

N=85

(3-12)

Prospective, one group

repeated-measures

BTX for leg muscles

PODCI There were

changes of at

least 3.0 points

(maximum

p<0.001) by 6

months for all

PODCI subscales

except the upper

extremity.

25

CHAPTER 6: DISCUSSION OF RESULTS

Participation reported as a key outcome of health and main human right for all children. Participation of

children with CP investigated. The more recent review of Imms (2008) provided analysis of 40 studies

describing the participation of children with CP. The results of these 40 studies indicate that children with

CP have a reduced level of participation, and those with greater functional impairment are the most

restricted. Children with CP were reported to be less involved in activities as well. However, interventions

aimed to promote and support the participation of children with CP are less investigated. Our search has

identified only eleven studies of interventions focused mainly on body structures and functions but less on

contextual factors.

Adair et al. (2015) has published a systematic review of 29 studies aimed to promote participation of

children with all kind of disabilities. Authors of the review didn’t find that interventions targeted to

improve body structures or functions could be effective. In contrast, individually tailored education and

mentoring were reported to be highly effective. Differently, from these authors, we have found that some

interventions aimed to improve body structures and functions might be effective and to promote

participation of children with CP. It seems that main key to success is a goal-oriented approach to

intervention planning. For example, goal oriented upper limb interventions resulted in the better social

participation of children with hemiplegic CP in all studies. In contrast, robotic-assisted upper-limb

training following standard (not individual) protocols improved upper limb kinematics but not functional

activities or participation of children with hemiplegic CP.

Analogous conclusions done from the analysis of studies investigating physiotherapy interventions.

Previously, Adair et al. (2015) in their review emphasized that exercise programs, where participation was

a secondary outcome, generally demonstrated a little effect for children with disabilities. The results of

our review show that physiotherapy could be effective if physiotherapy program involves lifestyle

interventions such as counseling to motivate and coach the child to adopt a more active lifestyle. Both

parents and children with CP should be involved in such counseling interventions. In addition, skill-based

movement programs that combine therapeutic principles of movement rehabilitation with community

involvement (e.g., dance programs, hippotherapy) seems to be evidence-based rehabilitation method to

promote participation, self-confidence, and well-being of children with CP.

26

Results of Law et al. (2011) who investigated 128 children with CP confirm that child- or context-focused

therapy approaches can be equally effective. The goal setting may be a critical component of the

successful intervention.

Main gaps in research of interventions aimed to improve participation of children with CP is that almost

all studies that we have managed to find were investigating social participation. Home and school

participation weren't addressed in papers we have reviewed. Another gap is high assessment bias of

studies using ICF-CY checklist. Nowadays, more advanced participation measures such as PEM-CY are

available that can provide a lot of information on participation at home, school, community as well as

broad range of environmental characteristics.

27

CHAPTER 7: CONCLUSIONS

1. Interventions aimed to promote and support the participation of children with CP are insufficiently

investigates.

2. Research that shows that both child-focused and context-focused interventions could be effective. The

goal setting is a critical component of the successful intervention.

3. More research is needed to address gaps in current research, such as outcome measurement bias, lack of

research data regarding interventions aimed to promote home and school participation.

28

CHAPTER 8: PRACTICAL RECOMMENDATIONS

Recommendations for the research: most of studies focused on body function of children with CP, future

studies should focus on other factors like environmental, and personal factors that also have effect on

children with CP.

Recommendations for the clinicians: practitioners should combine child- and context- focused

interventions to achieve best outcomes. For example, it would be helpful to combine conventional

physiotherapy with parent training.

Recommendations for the medical education institutions: goal setting is a critical component of any

rehabilitation intervention and should be discussed with both child and parents.

29

CHAPTER 9: REFERENCES

1- Rosenbaum, P, Paneth, N, Leviton, A, Goldstein, M, Bax, M, Damiano, D, Dan, B, Jacobsson, B. "A

report: The definition and classification of cerebral palsy. April 2006". Developmental Medicine &

Child Neurology Supplement. 109: 8–14

2- Adair B, Ullenhag A, Keen D, Granlund M, Imms C. The effect of interventions aimed at improving

participation outcomes for children with disabilities: a systematic review. 2015. Developmental

medicine Child Neurol. 57(12):1093-104.

3- Blair E, Love S. Definition and classification of cerebral palsy. 2005. Dev Med Child Neuro. l47:510.

4- Cans C, McManus V, Crowley M, Guillem P, Platt M J, Johnson A, Arnaud C. Cerebral palsy of post-

neonatal origin: characteristics and risk factors. 2004. Pediatric Perinat Epidemiol. 18:214-220

5- Colver A, Sethumadhavan T. The term diplegia should be abandoned. 2003. Arch Dis Child. 88: 286-

290

6- Sanger TD, Delgado MR, Gaebler-Spira D, Hallett M, Mink JW. Classification and definition of

disorders causing. 2003

7- Surveillance of Cerebral Palsy in Europe (SCPE). Prevalence and characteristics of children with

cerebral palsy in Europe. 2002. Developmental medicine Child Neuro. l44 633-640.

8- World Health Organization. International Classification of Functioning, Disability, and Health:

Children and Youth Version (ICF-CY). 2007. Geneva: World Health Organization.

9- Granlund M. Participation – challenges in conceptualization, measurement, and intervention. 2013.

Child Care Health Developmental. 39: 470–3.

10- Andrea Paulson, Jilda Vargus Adams. Overview of Four Functional Classification Systems

Commonly Used in Cerebral Palsy. 2017. USA

11- King G. Perspectives on measuring participation: going forward. 2013. Child Care Health

Developmental. 39: 466–9.

12- Maxwell G, Alves I, Granlund M. Participation and environmental aspects in education and the ICF

and the ICF-CY: findings from a systematic literature review. 2012. Developmental Neurorepair. 15:

63–78

13- Law M, Finkelman S, Hurley P, Rosenbaum P, King S, King G, Hanna S. Participation of children

with physical disabilities: Relationships with diagnosis, physical function and demographic variables.

2004. Scand J Occupy Therapy. 11:156 – 162.

14- Longmuir PE, Bar-Or O. Factors influencing the physical activity levels of youths with physical and

sensory disabilities. 2000. Adaption Physical Active Q. 17:40 – 53

30

15- Christine Imms, Brooke Adair, Deb Keen, Anna Ullenhag, Peter Rosenbarum, Mats Granlund.

Participation’: a systematic review of language, definitions, and constructs used in intervention

research with children with disabilities. 2016. Australia

16- Law M, Finkelman S, Hurley P, Rosenbaum P, King S, King G, Hanna S. Participation of children

with physical disabilities: Relationships with diagnosis, physical function and demographic variables.

2004. Scand J Occupy Therapy. 11:156 – 162

17- Morris C, Kurinczuk JJ, Fitzpatrick R, Rosenbaum P. Do the abilities of children with cerebral palsy

explain their activities and participation. 2006. Developmental medicine Child Neural. 48:954 – 961.

18- Forsyth R, Colver A, Alvanides S, Woolley M, Lowe M. Participation of young severely disabled

children is influenced by their intrinsic impairments and environment. 2007. Developmental medicine

Child Neurol. 49:345 – 349.

19- Heah T, Case T, McGuire B, Law M. Successful participation: The lived experience among children

with disabilities. 2007. Can J Occupy Therapy. 74:38 – 47.

20- Lawlor K, Mihaylov SI, Welsh B, Jarvis S, Colver A. A qualitative study of the physical, social and

attitudinal environments influencing the participation of children with cerebral palsy in northeast

England. 2006. Pediatric rehabilitation. 9:219 – 228.

21- Bleyenheuft, Yannick, Arnould, Carlyne, Brandao, Marina B, Bleyenheuft, Corrine, Gordon, Andrew

M. Hand and Arm Bimanual Intensive Therapy Including Lower Extremity (HABIT-ILE) in Children

with Unilateral Spastic Cerebral Palsy: A Randomized Trial. 2014. Neurorehabilitation and Neural

Repair, 645-657. doi:10.1177/1545968314562109

22- Gilliaux, Maxime, Renders, Anne, Dispa, Delphine, Holvoet, Dominique, Sapin, Julien, Dehez,

Bruno, Detrembleur, Christine, Lejeune, Thierry M, Stoquart, Gaƫtan. Upper Limb Robot-Assisted

Therapy in Cerebral Palsy: A Single-Blind Randomized Controlled Trial. 2014. Neurorehabilitation

and Neural Repair. 183-192. doi:10.1177/1545968314541172

23- Yueh-Ling Hsieh, Chen-Chia Yang, Shih-Heng Sun, Shu-Ya Chan, Tze-Hsuan Wang & Hong-Ji

Luo. Effects of hippotherapy on body functions, activities and participation in children with cerebral

palsy based on ICF-CY assessments. 2016. Disability and Rehabilitation. 39:17,1703-

1713, DOI: 10.1080/09638288.2016.1207108

24- Law MC, Darrah J, Pollock N, et al. Focus on function: a cluster, randomized controlled trial

comparing child- versus context-focused intervention for young children with cerebral

palsy. Developmental medicine and child neurology. 2011. 53(7):621-629. doi:10.1111/j.1469-

8749.2011.03962. x.

31

25- López-Ortiz C, Gladden K, Deon L, Schmidt J, Girolami G, Gaebler-Spira D. Dance program for

physical rehabilitation and participation in children with cerebral palsy. 2012. Arts & Health. 4(1):39-

54. doi:10.1080/17533015.2011.564193.

26- Slaman, J, van den Berg-Emons, HJG, van Meeteren, J, Twisk, J, van Markus, F, Stam, HJ, van der

Slot, WM, Roebroeck, ME. A lifestyle intervention improves fatigue, mental health and social support

among adolescents and young adults with cerebral palsy: focus on mediating effects. 2015. Clinical

Rehabilitation. 717-727. doi:10.1177/0269215514555136

27- Leanne Sakzewski PhD, B.OccThy, Jenny Ziviani PhD, MEd, BAppSc, O, David F. Abbott PhD,

Richard A. MacdonellMD, FRACP, FAFRM, Graeme D. Jackson MD, FRACP, Roslyn N. Boyd PhD.

Participation Outcomes in a Randomized Trial of 2 Models of Upper-Limb Rehabilitation for Children

with Congenital Hemiplegia. 2011. Australia. Physiotherapy. 531-539.

28- Noula Gibson, Annie Chappell, Amanda Marie Blackmore, Susan Morris, Gavin Williams, Natasha

Bear & Garry Allison. The effect of a running intervention on running ability and participation in

children with cerebral palsy: a randomized controlled trial. 2017. Disability and Rehabilitation.

DOI: 10.1080/09638288.2017.1367426

29- Van Wely, Leontien, Balemans, Astrid CJ, Becher, Jules G, Dallmeijer, Annet J. The effectiveness of

a physical activity stimulation programme for children with cerebral palsy on social participation, self-

perception and quality of life: a randomized controlled trial. 2013. Clinical Rehabilitation. 972-982.

doi: 10.1177/0269215513500971

30- Vanessa A.Scholtes, Jules G.Becher, Yvonne J.Janssen-Potten, HurnetDekkers, LindaSmallenbroek,

Annet J.Dallmeijer. Effectiveness of functional progressive resistance exercise training on walking

ability in children with cerebral palsy: A randomized controlled trial. 2011. Rehabilitation Centre

Heliomare. Doi.org/10.1016/j.ridd.2011.08.026-31.

31- F Virginia Wright, Peter L Rosenbaum, Charles H Goldsmith, Mary Law, Darcy L Fehlings. How do

changes in body functions and structures, activity, and participation relate to children with cerebral

palsy. 2015. DOI: 10.1111/j.1469-8749.2008.02037. x.

32

CHAPTER 10: ANNEXES

33