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Intensive Group Training in a
Local Community Setting forChildren with Cerebral Palsy
Methodological Aspects and Change in Motor Functioning

Anne Brit Sørsdahl

Department of Public Health and Primary Health Care
Section for Physiotherapy Science
University of Bergen
2010

iii

Acknowledgments
Many persons and institutions have been involved in this doctoral work.
I wish to express my sincere gratitude and warm thanks to:-

All the children and their parents who kindly used of their time to attend theassessments in the project, and who have shared their experiences and opinions
regarding participation in the intensive group training.

-

Associate professor, dr.philos. Liv Inger Strand, Section for Physiotherapy
Science, University of Bergen, my main supervisor, for your excellent
guidance, support and knowledge. I have learned a lot from you.

-

Professor, dr.philos. Rolf Moe-Nilssen, Section for Physiotherapy Science,
University of Bergen, my co-supervisor, for your enthusiasm, advice andcritical questions, which have always inspired me.

-

Helga K Kaale, Jannike Rieber, Bjørg Ringheim, Elisabeth Skarstein Waalerand Eva Jellestad, for all the fun during the development and piloting of theintervention model, the phase when we prepared for the studies and the datacollection. Each of you is in your special way highly skilled professionals, andit has been a pleasure to work with you all these years. Without yourprofessional and personal support, the studies could not have been carriedthrough.

-

Margot Andenes, Regine Benz, Kristin Daling Felde, Reidun Haavik, Jone
Strand Helgesen, Synnøve Iversen, Anne Strand Kiperberg, Tone Grinde
Seeberg, Marit Anne Smørdal, Walter Søiland and Bente Hole Vik who
conducted the group training.
You did a great job! A particular
acknowledgement to Ambjørg Løyning for all her effort regarding organizationof groups and data collection in Haugesund.

-Anne Marie Mandujano, Helse Bergen and Karin Berg, Bergen University
College, who scored the video clips in the intervention study.

-

Eli Hereide, Drude Malmin, Grete Opsal, Janne Sponland, Trine Sande and Liv
Marie Torbergsen who participated in the data collection.

-

Arnlaug Steine and Line Irgens Solheim for the assistance with recruitment ofchildren to the methodological studies.

iv-

Astrid Hatlø, Helse Sunnmøre; Marie Mellingen, Helse Fonna; Olav Roti,
Helse Førde and Randi Schiøtz, Helse Stavanger, the leaders of theparticipating habilitation units, for their enthusiasm and support during theintervention study.

-

The staff at the Section for Physiotherapy Science, University of Bergen, who
has provided a very good scientific learning environment.

-

The staff of highly skilled professionals at the Department of Physiotherapy,
Bergen University College, who has always been very supportive.

-

Reidun Jahnsen for your encouragement and positive support throughout thework with this thesis.

-

Else Mari Larsen and Elaine Møller for all the valuable discussions we have
had over the years, and for all the fun and laughter we have shared.

-

Kjersti Wilhelmsen, Aud Marie Øyen and Tove Dragesund, my fellow
students, for interesting discussions, support and encouragement.

-

Ingunn Aalvik and Eva Brusgaard, The Ministry of Health and Care Services;
Bjørg Halvorsen and Rutti Østensjø, the Directorate of Health and Social
Affairs, for their effort of improving habilitation services in Norway.

-

The Norwegian CP-association for their encouragement and support.

-

Vivienne Knowles for revising the English text.

-

Torbjørn, Simen and Line, my family, and also my mother Inger Lise, whoalways have believed in me and supported me, and have reminded me of all theother important aspects of life.

This work was made possible with financial support from: The Norwegian Fund for
Post-Graduate Training in Physiotherapy, Bergen University College, Directorate of
Health and Social Affairs and Ragna Sofie and Christian Rieber. All are gratefully
acknowledged.

v

Summary
The purpose of this thesis was to examine measurement properties of three outcome
measures of motor function for children with cerebral palsy (CP), and to investigatechange in their motor functioning following three weeks of intensive, activity-focusedand goal-directed physiotherapy in a group setting.

A portable electronic walkway has been found feasible and reliable when measuring
gait parameters in adults with neurological disorders and in children with typicaldevelopment, but measurement properties of the assessment tool have not been
examined in children with CP. Test-retest reliability of gait parameters from theelectronic walkway was investigated in 17 children with CP. A defined procedure to
calculate speed dependent gait parameters at a normalised gait speed was used. In a
short time span, the electronic walkway was found to be highly reliable for assessing
gait parameters in children with CP.

A subsequent study examined the inter-observer and intra-observer reliability of two
quality of movement measures when scored from video clips. Quality of movement
measures can be challenging and time consuming to score in a clinical setting due tothe complexity of the construct. The impact of quality of movement on motor
development has been sparsely investigated, but good quality is presumed by many
professionals to increase efficiency and safety of activities and decrease efforts inchildren with CP. Hence an efficient way of assessing quality of movement seems
important. Twenty-six children with CP participated in a reliability testing of theGross Motor Performance Measure (GMPM) and the Quality of Upper Extremity
Skills Test (QUEST). Performance of the test items were videotaped, edited andindependently scored by two assessors on two occasions. The intra-observer and inter-vi
observer reliability of the total scores of the two measures were found satisfactory,
and the total scores can thus be recommended for research use. Reliability, however,
was not satisfactory for the sub-scales and single items, and cannot be recommended
for use as separate measures.

The above three measures quantify different aspects of motor functioning in children
with CP and were found to be reliable and feasible for use in clinical research,
including research carried out as multicentre studies.

There is little scientific knowledge about the optimal type, dosage and onset ofphysiotherapy for children with cerebral palsy. Recent work has indicated that more
intensive, goal-directed and functional training than commonly offered byphysiotherapists might be beneficial. Furthermore, there is little knowledge about the
impact of movement quality on motor development in children with CP. The third
study investigated change of motor functioning in children with CP who participatedin intensive, activity-focused and goal-directed physiotherapy in a group setting forthe first time.

Twenty-two children aged three to nine years in five training groups from different
places in Western Norway participated in the study. A repeated measures design wasapplied with three baseline measurements before and two follow up measurements
after the intervention. The intervention aimed to attain individual goals regarding
basic motor abilities and motor abilities in everyday activities, and consisted of threehours of physiotherapy, five days a week for a three-week period. After theintervention period, the children had gained significant improvements in basic motor
abilities assessed by the Gross Motor Function Measure (GMFM) and high attainmentof predetermined individual goals was found. The children’s parents reported
significant improvement in the children’s ability to perform self-care activities in theviihome environments and a decreased need for caregiver assistance in mobility andself-care activities assessed by the Pediatric Evaluation of Disability Inventory(PEDI).

A positive trend in improved quality of movement as measured by the Gross Motor
Performance Measure (GMPM) and Quality of Upper Extremity Skills Test (QUEST)
was revealed, but not statistically significant. A significant improvement in movement
quality was found in items of the GMFM which improved during the study period, but
not in items that reminded stable. Thus, acquisition of gross motor functions seemed
to be related to better quality of movement, however, more research is needed to
substantiate this observation.

The intensive physiotherapy training in a group setting was experienced as fun andmotivating, even though the children and their families also found the training period
strenuous. Defined periods of intensive, focused training to attain specific goals seemto be a feasible way of optimising motor functioning in children with CP.

viii

List of papers
The present thesis is based on the following original articles:

I

Sorsdahl AB, Moe-Nilssen R, Strand LI.
Test-retest of spatial and temporal gait parameters in children withcerebral palsy as measured by an electronic walkway.
Gait and Posture. 2008; 27: 43-50.

II

Sorsdahl AB, Moe-Nilssen R, Strand, LI.
Observer Reliability of the Gross Motor Performance Measure and the
Quality of Upper Extremity Skill Test, based on Video Recordings.
Developmental Medicine and Child Neurology. 2008; 50: 146-151.

III

Sorsdahl AB, Moe-Nilssen R, Kaale HK, Rieber J, Strand LI.
Change in basic motor abilities, quality of movement and everyday
activities following an intensive activity-focused goal-directed
physiotherapy program in a group setting for children with cerebral palsy.
BMC Pediatrics. 2009 (under review after revisions have been performed).

ix

Table of contents
Acknowledgements
Summary
List of papers
Table of contents
Abbreviations
Definitions

ii
iii
vi
vii
ix
x

Introduction

1

Background4

Cerebral palsy - diagnosis and functional classifications
Motor disorders and motor development in children with CP
Measuring change in motor functioning – theoretical frameworks
A shift in theoretical basis of physiotherapy for children with cerebral palsy
Intensive motor training approaches for children with cerebral palsy
Pilot study

Aims of the present project
General aims
Specific aims

Methods
Designs
Participating children
Participating professionals
Assessment tools
Goal setting process and goals
Videotaping and editing
Intervention
Statistical analysis
EthicsSummary of the papers
Methodological studies
Intervention study

4
7
9
11
13
14

16
16
17

18
18
18
22
22
31
31
32
33
34

35
35
37

x

Main findings
Methodological studies
Intervention study

Discussion
Methodological studies
Reliability of the electronic walkway GAITRite
Feasibility and recommendations for use of the GAITRite as a research tool
Reliability of the quality of movement measures GMPM and QUEST
Feasibility and recommendations for use of the GMPM and QUEST as research tools
Strengths and limitations of the methodological studies

Intervention Study

38
38
38

40
40
40
42
44
45
46

48

Change of functioning in a capacity and performance perspective
Change of functioning in an individual and a group perspective
Quality of movement and basic motor abilities
Content and description of the intervention
Assessment tools
Strengths and limitations of the intervention study

48
51
53
54
56
58

Ethical considerations
Intensive periods of physiotherapy in habilitation services

60
62

Conclusions and further research

63

References

65

Papers I-III

Appendices
1.
2.
3.
4.
5.
6.
7.

Gross Motor Function Classification System (GMFCS), Norwegian translation
Manual Ability Classification System (MACS), Norwegian translation
Gross Motor Development Curves
Content of the Pediatric Evaluation of Disability Inventory (PEDI)
Goal Attainment Scale (GAS)
Procedure for video uptakes
Information to participantsxiAbbreviations
AACPDM

American Academy for Cerebral Palsy and Developmental Medicine

ANOVA

Analysis of variance

CIMT

Constrained Induced Movement Therapy

CP

Cerebral Palsy

GAS

Goal Attainment Scaling

GMAE

Gross Motor Ability Estimator

GMFCS

Gross Motor Function Classification System

GMFM

Gross Motor Function Measure

GMFM-66

Gross Motor Function Measure 66-item version

GMFM-88

Gross Motor Function Measure 88-item version

GMPM

Gross Motor Performance Measure

ICC

Intraclass Correlation Coefficient

ICF

International Classification of Functioning, Disabilities and Health

ICF-CYInternational Classification of Functioning, Disabilities and Health Children and Youth Version

MACS

Manual Ability Classification System

MTSCI-1

Motor Teaching Strategies Coding Instrument

MIC

Minimal Important Change

NDT

Nevrodevelopmental therapy

OT

Occupational Therapist

PEDI

Pediatric Evaluation of Disability Inventory

PT

Physiotherapist

QUEST

Quality of Upper Extremity Skills Test

RCT

Randomised Controlled Trials

SCPE

Surveillance of Cerebral Palsy in Europe

SDD

Smallest Detectable DifferenceSW

Within subject standard deviation

WHO

World Health Organisation

xii

Definitions
Definitions of concepts used in the thesis and/or in the articles:
Anti-spastic medication: Medication given to reduce muscle tone, e.g. Botulinum
toxin type A given as injection in muscles, and Baclofen given throughan infusion pump [1].
Basic motor abilities: Basic abilities like rolling, crawling, sitting and walking [2].
Bonferroni adjustment: Adjustment of the chosen level of significance due to multiple
comparisons. The selected significance level is divided by the numberof tests to obtain a more stringent p-value [3].
Coefficient of determination (R2): An indication of the percentage of variance that isshared by two variables [3].
Condition-specific measure: Measurement tool designed to document the status ofindividuals having a specific diagnosis or condition [3].
Construct validity: The ability of a measure to converge with other indicators or
measures of the same construct and discriminate unrelated indicators or
measures [4].
Content validity: The extent to which the components (items) of the scale cover all
aspects of the attribute to be measured, in a balanced way [4].
Criterion validity: The extent to which a measure correlates with a pre-existing one of
the same concept, preferable a “gold standard” [4].
Criterion referenced measure: A measure where there is an external criterion against
which people are judged [5].
Effectiveness: The extent to which a specific intervention, procedure, regimen, orservice does what it is intended to do for a defined population [6].
Efficacy: The extent to which a specific intervention, procedure, regimen, or service
produces a beneficial result under ideal conditions [6].
Everyday activities: Activities like mobility, self-care, social function and play indaily environments [7].
Feasibility: The ability to use the measure within its application area e.g. in terms ofcost, training, equipment and respondent burden [6].
Functioning: An umbrella term encompassing all body functions, activities andparticipation [8].
Generic measures: Measures that have been developed to measure constructs that are
relevant to the general population [5].

xiii
Habilitation: A process aimed at enabling persons with disabilities to reach andmaintain their physical, sensory, intellectual, psychiatric and/or social
functional levels, thus providing them with the tools to change theirlives towards a higher level of independence. It includes a wide range ofmeasures and activities from more basic and general rehabilitation togoal-oriented activities, for instance vocational rehabilitation [9].
Heteroscedasticity: An assumption in regression analysis that the residuals at eachlevel of the predictor variable(s) have unequal variances [10].
Homoscedasticity: An assumption in regression analysis that the residuals at eachlevel of the predictor variable(s) have equal variances [10].
Intra-observer reliability: The extent to which repeated observations of a single
observer agree [11], often expressed as a correlation coefficient.
Inter-observer reliability: The extent to which observations of the same thing made bymore than one observer agree [11], often expressed as a correlation
coefficient.
Motor Function: In this thesis motor function is used as an umbrella term
encompassing the motor aspects of body functions, activities andparticipation, including quality of movements, gross motor function andhand motor function.
Minimal Important Change (MIC): Minimal change that is seen important [12], also
named Minimal Clinically Important Difference (MCID) [6]. Expressed
as cut off point in measurements, or via parents, children’s and/or
professionals judgement [13].
Prevalence: The proportion of a given population experiencing a condition at a given
time e.g. the current cases in a population [11].
Quality of movement: An aspect of a motor activity e.g. coordination or stability [14].
Rasch analysis: Using a set of items from a sample the analysis produces an interval
scale that estimates the difficulties of the items (item difficulty) and theabilities associated with total raw score (child ability) [15].
Reliability: The extent to which measurements are repeatable [3]. Different types ofreliability exist e.g. inter-observer, intra-observer and test-retest
reliability.
Responsiveness: The ability of a measure to detect clinically important change overtime, even if these changes are small [12].
Smallest detectable difference (SDD) [16]: The limit of change an individual has toexceed to say there is a change beyond measurement error [3], also
named Minimal Detectable Difference (MDD)[17], Minimal Detectable
Change (MDC) [6] or Smallest Detectable Change [12]. Based on Sw
the SDD between two measurements for the same subject can bexivcalculated using the equation
observations [18].

2 × 1.96 × Sw = 2.77 Sw for 95% of pairs of

Sphericity: The extent to which all differences between pairs of scores are equallyvariable [19].
Standardised measure: A published measurement tool, designed for a specific purpose
in a given population, with detailed instructions provided on
administration and scoring and the results of reliability and validity
testing published in peer-reviewed journals [6].
Test-retest reliability: The extent to which repeated applications of a test provide
consistent results [6].
T-score: Standardised score from the raw scores, e.g. with a mean of 50 and standard
deviation of 10 [5].
Validity of a measure: Concerns what a test measures and how well it does so [20].
Different types of validity exist e.g. construct, content and criterion
validity.
Validity of a study: The extent to which the conclusions are believable and useful [3].
Internal validity concerns whether other than the independent variable
could be related to changes in the dependent variable, whereas external
validity concerns the generalizability of the results.
Within subject standard deviation (Sw): Standard deviation of repeated measurementson the same subject [18]. Also named Standard Error of Measurement(SEM) [3].

Mathematical and statistical notation is used as suggested by Altman 1991 [21].

1Introduction
In developed countries, cerebral palsy (CP) is the most common disorder ofmovement and posture in children, with prevalence of 2.0-2.5 per 1000 live births[11]. The prevalence of CP in Norway is reported to be 2.1 per 1000 live births [22].
A large range of habilitation services might be offered to children with CP and theirfamilies including medical interventions such as anti-spastic medication andorthopaedic surgery, special education services and speech therapy. Physiotherapy isseen as an integral part of the habilitation services, and children with cerebral palsy
are one of the largest groups of children receiving services from pediatricphysiotherapists [23]. In a Norwegian survey, Jahnsen et al. [24] found that 92
percent of adults with CP reported to have received physiotherapy during theirchildhood until the age of 15. Over the years, different physiotherapy interventions
have been offered to children with CP (e.g. Mayston [25] and Damiano [26] foroverviews), however, the effects have been sparsely investigated, and the quality of
the research has earlier been hampered by small samples, lack of sensitive measuresand poor descriptions of the interventions [27,28]. Even if methodological quality has
improved in recent years, there is still little scientific documentation regarding theeffects of physiotherapy interventions on motor development in children with CP[29,30].

An evolving understanding in the habilitation field has emerged during recent years
that theories and knowledge from the social sciences should complement thebiomedical model in habilitation [31], and that children’s engagement in activities andparticipation in kindergarten, school, home and leisure settings are important goals forhabilitation efforts [26,32,33]. In the 1990s, new knowledge in the field of movement
science and pediatric neurological physical therapy implied that goal-directed,

2functional and family-centred interventions might be more effective than traditional
neurodevelopmental focused interventions [34-36].

It has, in addition, beenquestioned by parents and professionals whether more intensive physiotherapy would
influence motor development in children with disabilities to a larger extent. In the late
1990s an increasing number of families were seeking out more intensive habilitationand training modalities abroad, which were not offered by the Norwegian healthsystem [37,38]. Moreover, there has been an increased requirement from official
health authorities of evidence based practice in health services, including
physiotherapy and habilitation services.

As a consequence of these trends and insights, and as parents of children withcerebral palsy strongly signalled that the amount and intensity of physiotherapy
programs were not sufficient, a project was developed in 1998 which impliedcollaboration between a physiotherapy institute for children in Bergen “Barnas
Fysioterapisenter” (BFS), the Physiotherapy Service in Bergen municipality and the
University College of Bergen (HiB). The project had an initial phase where an
intensive physiotherapy program in a group setting was developed and pilot tested[39], and a second phase where aspects of functioning in children with CP and waysof implementing intensive periods in habilitation plans were explored [40]. Finally,
the project had a third and last phase in which the intervention model wascommunicated to professionals and parents in seminars, and physiotherapists whowanted to start similar intensive training groups were offered supervision. An article
describing aspects of the intervention model was published in “Fysioterapeuten” [41]which is the main physiotherapy journal in Norway. The last phase also included aqualitative research arm aimed to explore and describe parents’ experiences with theintensive group training, parents’ view of the children’s change, and participating
physiotherapists’ description of and motivations for the intervention model. The
results were reported in another Norwegian publication [42].

3During the period from 2002 to 2008, the Ministry of Health and Social Affairs
funded developmental projects regarding habilitation in Norway [43]. Fivehabilitation units in Western Norway located in the cities of Stavanger, Haugesund,Bergen, Førde, Ålesund and collaborating partners from BFS, HiB and the Universityof Bergen (UiB) applied for grants in an umbrella project regarding intensive training
and habilitation. Each of the six applicants contributed with own project plans in the
joint application for grants, and had their own project leaders. Four of the five
habilitation units implemented the model of intensive physiotherapy that wasdeveloped in Bergen.

The research project leading to the present thesis was conducted in collaboration with
BFS and the four habilitation units in Stavanger, Haugesund (in cooperation with
Gard private institute for physiotherapy), Førde (in cooperation with the municipalityof Gloppen), and Ålesund; the children participating in intensive group training forthe first time. The group training was carried out during the period from August 2004
to October 2005. The project was performed as a doctoral work at the Department of
Public Health and Primary Health Care, Section for Physiotherapy Science at the
University of Bergen. The objective of the project was to investigate whether children
with CP changed their motor functioning as a result of participation in intensive
physiotherapy in groups. The study was performed as a multicentre study, in a part of
Norway with long travel distances, and there was a need for evaluation measures thatwere portable. To ease the assessment burden of children and assessors, it was
intended that advanced observational measures should be scored by skilled assessors
from video uptakes. The methodological part of the thesis aimed to examine
reliability of three evaluation measures that seemed useful for multicentre studies
including children with CP.

4Background
Cerebral palsy - diagnosis and functional classifications
Cerebral palsy is a neurodevelopmental condition beginning in early childhood andpersisting through the lifespan [44]. A classic definition of cerebral palsy from 1964
is “a disorder of posture and movement due to a defect or lesion of the immature
brain” [45 p.9]. However, this clinical descriptive term includes a heterogeneous
population with a variety of movement disorders, commonly accompanied by other
impairments. A new definition of cerebral palsy that covers the heterogeneity and thecomplex nature of the diagnosis is proposed by an international expert group [44 p.
9]:“Cerebral palsy (CP) describes a group of permanent disorders of movement and posture, causing
activity limitations that are attributed to non-progressive disturbances that occurred in the developing
fetal or infants’ brain. The motor disorders of cerebral palsy are often accompanied by disturbances of
sensation, perception, cognition, communication, and behaviour, by epilepsy, and by secondary
musculoskeletal problems”

CP is traditionally divided into subgroups based on topology of affected limbs andtone disturbances, e.g. hemiplegia, diplegia, quadriplegia, ataxia or dyskinesia, alongwith the degree of involvement e.g. mild, moderate or severe [11]. However, this subgrouping has been questioned because of its inaccuracy, poor reliability and lack ofincluding functions and body parts often affected, like bulbar function and trunk
involvement [44,46]. In a newer classification from a network of CP registers andsurveys, “Surveillance of CP in Europe (SCPE)” [47], the subgroups bilateral andunilateral spastic cerebral palsy, ataxia and dyskinesia are used. Criteria were definedand a training manual was developed to improve reliability of sub-grouping children
with CP, but considerable variation in assignment of CP subtype was revealed acrossthe network, hence reliability is still a challenge [48]. Little additional information
about the children’s functional ability has been derived by using diagnostic subgroups

5of CP, so in recent years rather functional classification systems of gross motor andarm/hand function have been developed and are increasingly being used in research aswell as in clinical practice.

The Gross Motor Function Classification System (GMFCS) [49] has received
international acknowledgement and is now a commonly used classification system ofchildren’s locomotor and sitting ability [50]. The GMFCS is a five-level agecategorized system developed to describe severity of motor involvement in children
with CP based on functional abilities, need for assistive technology and wheeledmobility. Children classified to GMFCS-level I can walk at home, at school, outdoors
and in the community. They can climb stairs, run and jump, but speed, balance andcoordination are impaired. Children classified to level V are transported inwheelchairs in all settings, their ability to maintain head and trunk postures against
gravity is limited, and their self-mobility even with the use of assistive technology isseverely limited. The GMFCS has demonstrated good measurement properties when
professionals as well as caregivers have classified functional abilities of children with
CP [49,51-53] and high stability of the classification has been found [54,55] implyingthat children tend to remain in the same GMFCS-level over time. The classification
system is, however, less precise in infants [49] and the GMFCS-level might bepreliminary until the age of 2 years [56] An age expanded and slightly revised versionof the GMFCS was released in 2007 [57]. The revised version has not yet undergone
reliability testing, nor has it been systematically compared to the old version, andtherefore one cannot know whether the two versions can be used interchangeably[58]. In this study, the original version was used.

The Manual Ability Classification System (MACS) [59] is in the same manner a fivelevel category system developed to describe arm and hand motor function in children
with CP based on the children’s abilities to handle objects in daily activities, their

6need for assistance and adaptations. Children classified as MACS-level I handle
objects easily and successfully, while children classified as level V do not handle
objects and require total assistance. Good measurement properties are demonstrated
when professional and families classify hand function [59-61], but for children under2 years MACS has shown only moderate reliability. MACS has shown stable levels
over 12 months [62].

Use of the GMFCS and MACS requires familiarity with the classification systems,
the user instructions and the child, but requires no formal training [49,59]. The
classifications have been translated into Norwegian (Appendix 1 and 2). The
correlation of GMFCS and MACS levels has been shown to be moderate to high[59,62,63], indicating that the classifications are only partly built on the same
construct. Recommendations are made to use both classifications systems in research
since they are regarded as complementary [59, 62-64].

The distribution of the classification levels of a population of Swedish children with
CP is shown in Figure 1. Approximately one third of the children were classified as
GMFCS and MACS level I. Similar information about distribution of classificationlevels is not available for Norwegian children with CP, however, Andersen et al. [22]have estimated GMFCS levels from register data and classified 55% of a populationof 374 children as GMFCS-level I or II, and 17%, 20 % and 8% as levels III-V,
respectively. In the Norwegian population approximately 75% of the children were
sub-diagnosed as having spastic type of CP, 6 % as dyskinesia, 5% ataxic type and
7% were not classified. The proportion of children in the different subgroups of CP isessentially similar to the proportions reported in a European population of children
with CP [22]. In the Norwegian population approximately 4 % of the children had
severe hearing impairments, 5 % severe vision impairments, 28% epilepsy and 31 %

7general learning disabilities [22]. More severe GMFCS-levels have been found to be
associated with larger proportions of accompanying impairments [65,66].

Figure 1.
Distribution of GMFCS and MACS levels in a population-based studyof 359 children with CP in southern Sweden.
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Motor disorders and motor development in children with CP
Abnormal gross and fine motor functioning and organisation are the core features ofCP [44]. This may include problems with force generation, abnormal muscle tone,
altered reflexes, poor selective control of muscle activity, and reduced ability tocontrol posture and to learn movements [23]. The impairments should be seen as
coexistent rather than isolated, and may affect each other [1]. The motor impairments
can lead to difficulties in everyday activities like walking, feeding and swallowing,
coordination of eye movements, articulation of speech, and secondary problems withbehaviour, musculoskeletal problems, and participation in society [44].

8
The motor development in children with CP is delayed, and over the years clinicians
and researchers e.g. Bobath [67] and Levitt [68] have described the expected motor
development. Effort has been made to predict motor function, like walking ability
from the age when independent sitting is achieved [69-71]. An influential
contribution in this field of prediction is the creation of gross motor developmental
curves for children with CP [72,73] (Appendix 3). The trajectory lines of expecteddevelopment have made prediction of gross motor development from GMFCS-levels
possible. According to these curves, children with CP will reach their potential inbasic gross motor function in pre-school or in the first years of primary school age;children classified in GMFCS-level V, the earliest, and children classified in
GMFCS-level I, the last. Similar curves for hand motor function development has
been published [74], but their use is not widespread. Development of hand function
according to MACS-levels in children with unilateral CP has recently been described[16].

In addition to the development of basic motor abilities, professionals and parents
often pay attention to the movement patterns or “quality of movement” in children
with CP. This might include aspects like stability, weight shift or the cosmetics of amovement pattern [75-77]. The construct of quality of movement is complicated[14,78], but is often referenced to an optimal or “normal” movement pattern. There is,however, little scientific knowledge about the role that movement quality plays in thedevelopment of basic motor abilities in children with CP.

In recent years the focus on motor development in CP in a lifetime perspective has
increased, realizing the increasing musculoskeletal consequences, fatigue, pain anddetoriation of function experienced in many adolescents and adults [24]. Whether
these emerging disabilities are caused by overuse of muscles and joints, disuse, or a
combination of both, is yet to be decided [79].

9

Measuring change in motor functioning – theoreticalframeworks
The concept of measurement and the World Health Organization (WHO)’s
International Classification of Functioning, Disability and Health, Children and Youthversion (ICF-CY) [8] have set the framework for the studies in this thesis.

Measurement is “a process that involves an assessment, calculation, or judgment of
the magnitude, quantity or quality of a characteristic or attributes” [80 p.14]. Validmeasurement tools are a prerequisite in research and denotes that a tool actually
measures the concept in question and that the concept is measured reliably [81].
Highly reliable measurement tools are a prerequisite for detecting true change inoutcomes and ensuring that the observed change is not merely a result of
measurement error [6]. Measurement tools developed for evaluative purposes are in
addition required to demonstrate responsiveness to change before they can be usedconfidently as outcome measures [82]. This means that the measure must be able todetect change when change has occurred and show stability when no change has
occurred [14]. When observational skills are required to assign scores to a measureand observers are a part of the measurement process, high observer-reliability isrequired to secure valid results [6]. Test-retest reliability of an assessment tool isinvestigated when the similarity in results in individuals who are supposed to have astable performance is of concern [6].

The measurement property is not an invariant characteristic of a measure, but must be
demonstrated in the population and setting of interest [12]. In this thesis, this means in
the ages and functional levels of children with CP who were participants in theintervention study. For a measure to be declared reliable, it must demonstrate ability
to differentiate among children and provide consistent values with small errors.

10
Relative reliability concerns the ability to differentiate between individuals and isexpressed in some form of a correlation coefficient [3]. Absolute reliability concerns
the absolute measurement error e.g. how close the scores on repeated measures are,expressed in the unit of the measurement scale at issue [12]. From absolute reliability
values, the smallest detectable difference (SDD) can be calculated [18], and if achange exceeds the SDD, one can be 95% confident that the change is not due tomeasurement error. However, the SDD may not necessarily indicate a clinicallyimportant change.

The ICF-CY belongs to the family of international classifications developed by the
WHO for application of various aspects of health. It provides a framework formeasuring change in children with CP’s functioning which incorporates biologicaland social perspectives [83]. The ICF-CY builds upon and contains the same
components as the ICF (Figure 2), but has included a developmental perspective [8].
The ICF is divided into two parts, and the first part “Functioning and Disability” is of
main interest in this thesis. This part includes three components of health: Body
functions and structures, defined as the physiological functions of body systems andanatomical parts of the body, respectively; activity defined as the execution of a task
or action by an individual, and participation defined as involvement in a life situation[8]. Part two “Contextual factors” contains environmental and personal factors, which
also to a large extent influence a child’s functioning. The group setting and the
involvement of the children’s parents and professionals represent this part of the ICF–
CY and were considered important aspects of the intervention model. Aspects ofenvironmental and personal factors have been addressed in a qualitative arm of thedevelopmental project [42].

In the ICF model a distinction in the activity and participation dimensions is madebetween “capacity” and “performance”. Capacity describes an individual’s ability to11execute a task or action in a standardized environment and indicates the highest
probable level of functioning. Performance describes what an individual does in his or
her current environment [8]. The bidirectional arrows of the ICF-model indicate all
possible interactions and influences (Figure 2) and the relations between thecomponents on children’s functioning and the relation between capacity andperformance are not fully understood [84-87].

Figure 2.
Components of International Classification of Functioning, Disability and Health(ICF) [8]. Reprinted with permission.

A shift in theoretical basis of physiotherapy for children withcerebral palsy
In the past, various treatment methods have been offered to children with CP and theirfamilies. In the 1950s a common treatment method was to strengthen separate muscle
groups and use orthoses, inspired by physiotherapy given to children with
poliomyelitis. The building up of motion from reptilian movement patterns to primate
erect walking, as recommended by Temple Fay and later followed by the DomanDelacato system, and the training of muscle synergies in spiral and diagonal patterns,
as developed by Krabat, Knott and Voss, are other examples of treatment methods[68]. In Conductive Education, originated by Andreas Petö, intensively performed

12
movement programs in a group setting and use of speech as a reinforcement of active
movement, was put into a system [88]. Neurodevelopmental treatment (NDT),
developed by Karl and Berta Bobath, was originally based on reflex inhibition andfacilitation. Newer theories of motor control have been incorporated in the treatment
concept [89], and the treatment is widely used internationally.

In recent years, the focus on single muscle groups, movement patterns andimpairments such as increased tone and reflex activity has been followed by a systemapproach with emphasis on the child as a member of a family and of society.
Physiotherapy as a part of habilitation services should support family and child toattain goals of importance in their everyday life [90], and a focus on task-oriented
physiotherapy has evolved [35,90]. Based on information from the clinical field, aneclectic approach seemed widely used in Norway, with physiotherapy 1-2 times aweek and emphasis on supervision of caregivers and professionals and also on
modification of the environments e.g. by the affordance of technical equipment. In
addition, physiotherapy has for a long time commonly been intensified after surgery
or anti-spastic treatment. The majority of the projects regarding intensive
physiotherapy or intensive multidisciplinary programs funded by the Ministry of
Health and Social Affairs in Norway in the period 2002 to 2008 [43] were continued
after the end of the project phases. There is, however, no published information oncontent and frequency of today’s physiotherapy services for children with CP in
Norway.

13

Intensive motor training approaches for children with cerebral
palsy
The concept of “intensive training” or “intensive physiotherapy” has been used todescribe various types of training. If not otherwise defined, the concept commonly
includes an increased frequency of physiotherapy in a defined period of weeks ormonths, whereas the duration of each session remains the same as ordinary therapy
sessions lasting mainly 45 to 60 minutes [91-95]. The Norwegian Knowledge Centre
for the Health Services has reviewed international research concerning intensive
training/rehabilitation of children with brain damage [30]. Their definition of
intensive intervention was (p. 11): “Systematic and focused training and habilitation
efforts with a minimum range of 3 times a week up to several times a day for one andmore periods of time”. Seven systematic reviews and 20 separate studies met theinclusion criteria, and revealed that only studies of Constrained Induced Movement
Therapy (CIMT) and early intervention with a focus on parent education had
sufficient methodological strength. The two approaches revealed low to moderate
evidence of effectiveness [30]. For other intensive interventions, including intensive
training related to body functions such as strength training and functional activityfocused training, the review was inconclusive due to inconsistent results, few
participants and methodological weaknesses in the included studies. However, acloser look at single studies regarding activity-focused interventions reveals atendency of positive outcomes in favor of increased intensity in most studies [34,91,92,94-96], implying that this is a subject of further investigation.

There is no consensus regarding the optimal dosage of training. Intensive
physiotherapy program for children with CP have differed in frequency and duration;e.g. five sessions a week over six months [94], four sessions a week over four weeks[92], or several daily sessions over five months [97].

In two studies it was

14commented that most of the motor gains seemed to appear during the first weeks ormonths of the intervention [94,97], and in one study with increased frequency ofphysiotherapy lasting six months, children and parents seemed tired after the long
period of therapy [94]. Hence, shorter, focused periods of training with longer daily
duration might be beneficial. There are only a few studies examining the outcome ofintensive physiotherapy training in a group setting [98-100], and as group training isfound useful regarding motivation and effort [98,100], this might be an advantageous
way of organizing the training.

Pilot study
A pilot study [39] was conducted prior to the studies included in this thesis. Theintervention model included three hours of intensive physiotherapy five days a week
in a three-week period. Six children with CP aged 2 ½ to 5 years participated in two
periods of intensive physiotherapy, in the spring term and in the following autumn
term. The program consisted of activities in a gym and in a pool. A repeated measuresdesign with two measurements prior to the intervention period and two measurements
after was applied for four children, and one pre- and one post measurement for twochildren. The pilot study indicated improvement in basic motor abilities as measured
by the GMFM-88, particularly in connection with the periods of intensive
physiotherapy (Figure 3).

15

Total score (%)

7060Child 1
50Child 2Child 340

Child 430

Child 5
Child 620

18.11.1999

18.10.1999

18.09.1999

18.08.1999

18.07.1999

18.06.1999

18.05.1999

18.04.1999

018.03.1999

10

Figure 3.
Total scores of the GMFM-88 from six children with CP who participated in
two periods of intensive physiotherapy [39].
Gray bars indicate intervention periods.

16

Aims of the present project
Results from the pilot project indicated improvement in basic motor abilities whenchildren participated in a three-week period of intensive physiotherapy. In the present
project we wanted to broaden the scope of assessments, in line with the dimensions of
the ICF, and investigate change of motor functioning in a larger group of children
who participated in intensive training groups for the first time.

General aims
The first overall aim of this thesis was to adapt and evaluate measurement tools.
Using a multicentre approach in the intervention study where several habilitation units
were invited to participate, there was a need for outcome measures that could be usedreliably in different sites, and at the same time that impose as little strain on children
and assessors as possible. Electronic equipment like an electronic walkway connected
to a computer and video recording of parts of the assessment with subsequent editingby use of PC software and scoring from video clips, appeared to be feasible and ease
the assessments. The methodological part of this thesis aimed to examine reliability of
three evaluation measures that might be used in the outcome study of children withCP.

The second aim of this thesis was to investigate aspects of functional change inchildren with CP who participated in a course of intensive physiotherapy in their local
environments.

17

Specific aims•

Examine test-retest reliability of gait parameters using the electronic walkway
GAITRite£ in a sample of children with CP (Study I).

•

Examine inter- and intra-observer reliability of the Gross Motor Performance
Measure (GMPM) and Quality of Upper Extremity Skills Test (QUEST)
when scored from video clips (Study II).

•

Investigate the impact on functioning of a 3-week period of intensive goaldirected and activity-focused physiotherapy in a group setting for children
with CP (Study III).

•

Examine the relationship between achievement of basic motor abilities andquality of movements (Study III).

18

Methods
Designs
Study I

Same-day test-retest design [6] where children with CP walked eight times over anelectronic walkway at three different speeds. Retest was performed after an averageof 25 minutes.

Study II

Intra-observer and inter-observer reliability design [6] where two observers
independently scored video clips of the GMPM and QUEST over a one-week period.
The video clips were rescored after six weeks.

Study III

Repeated measures design [3] with three baseline assessments before the intervention
period and two follow up assessments after the intervention period. The baseline,
intervention and follow-up phases all lasted three weeks.

Participating children
Study I

The participating children were a convenience sample of children with CP withindependent gait function recruited from municipality 1 (Bergen) and 2 (Stavanger) in
Western Norway.

Four paediatric physiotherapists recruited the children, twoworking in private practice, one in a school for disabled children and one in a
habilitation unit. Inclusion criteria were children with CP from 2 to 15 years of age
who were able to walk without assistive walking devices (GMFCS-levels I and II).

19
Eighteen children with CP were referred to the study and were all included. In one
child (girl, seven years, GMFCS-level II), not sufficient data from the retest wereregistered, and the child was excluded (Figure 4). Characteristics of the seventeen
participating children are presented in Table 2. The study group represented aheterogeneous group of children with CP with different walking abilities.

Study II

The participating children were a convenience sample of children with CP recruitedfrom municipality 1 (Bergen) and 3 (Voss) in Western Norway. Six paediatricphysiotherapists working in private practice or primary schools recruited the children.
Inclusion criteria were children with CP from 2 to 15 years of age. Twenty-six
children were referred to the study and were all included (Figure 4 and Table 2). All
levels of the GMFCS and MACS were represented in the sample. Eight of thechildren were also included in the sample of study I.

Municipality 1

Municipality 2

Municipality 3

25 children

6 children

5 children

1 child

Excluded:
21 children
12 children

Sample:5 children
5 children

Study I

Study II

17 children

26 children

Figure 4.
Children from three municipalities were included in Study I and II.

20
Study IIIChildren with CP from four habilitation units in the cities of Stavanger, Haugesund,Førde and Ålesund in Western Norway, who were invited to participate in intensive
physiotherapy training for the first time, were included in the third sample. The
habilitation units recruited the children and composed the groups. The inclusion
criteria were children with CP in preschool or first years of primary school living
within one hour travelling time from the training location. Exclusion criteria werechildren with other diagnosis than CP and children who, according to the view ofprofessionals at the habilitation units, had suffered extensive strain due to for example
repeated hospitalisations or serious health problems in the past year. Twenty-fivechildren were referred to the study and were all initially included (Figure 5). One
child (girl, six years, GMFCS-level II) dropped out during the intervention period due
to long travel distance. Two children were excluded: One participated in less than halfof the intervention period due to illness (girl, four years, GMFCS-level III), and one
was found to have another neurological condition than CP (boy, four years).

The study sample included twenty-two children with CP, who accomplished theintervention, from five training groups in four different sites of Western Norway(Figure 5). All levels of the GMFCS and MACS were represented in the sample(Table 2). Five of the children received anti-spastic medication in the study period.

21

Site 1Site 2Site 3

Group 1

Group 2

Group 3

Group 4

Group 5

3 children*

6 children

5 children

6 children

5 children

Site 4Drop out:
1 child

Excluded:
1 child

1 child

Study IIISample:22 children

Figure 5.
Children from four sites and included in five training groups participated in Study III.
* An additional three children participated in this training group, but did not meet the inclusion criteria.

Table 2.
Demographics of participating children.
Study

Sex
boys/girls

Age
years

GMFCS

MACS

no

n

n

mean; range

level

n

I

17

8/9

7.0; 3-13

I
II

11
6

II

26

14/12

7.5; 2-13

I
II
III
IV
V

5
6
5
4
6

I
II
III
IV
V

III

22

15/7

5.5; 2-9

I
II
III
IV
V

8
2
6
5
1

I
II
III
IV
V

GMFCS: Gross Motor Function Classification System
MACS: Manual Ability Classification System

level

Diagnosis
n

type

n

Hemiplegia
Diplegia

11
6

4
9
5
5
3

Hemiplegia
Diplegia
Quadriplegia
Dyskinesia

6
12
6
2

8
7
5
1
1

Hemiplegia
Diplegia
Quadriplegia
Dyskinesia

7
11
2
2

22

Participating professionals
In Study I-III, two PTs administered the assessment tools and videotaped theassessment. In addition, several professionals participated in study II and III.
Study II

Four pediatric PTs participated in development of the procedure for videotaping. Twopediatric PTs with respectively 10 and 17 years of clinical experience scored thevideo clips.

Study III

Twelve pediatric PTs most with long clinical experience, mean 13 years, range 1 to 27
years, conducted the group training. Seven PTs and OTs from the participating
habilitation units participated in the data collection, mainly performing parent
interviews with the PEDI. The professionals had participated in training courses andwere experienced in using the assessment tool. Four pediatric PTs from Barnas
Fysioterapisenter supervised the group leaders before and during the intervention andparticipated in the data collection. Two pediatric PTs scored video clips of theassessments. The professionals, who collected data, did not participate in the grouptraining, and the PT who supervised the group leaders did not participate in datacollection in the groups they supervised.

Assessment tools
As functioning in children can be described using several components according to
the ICF and the relationship between the dimensions of the ICF are not fully
understood, a broad specter of assessment tools related to motor and everyday
functioning was chosen, reflecting a range of dimensions of the ICF-CY (Tables 3
and 4).

23
The electronic walkway GAITRite£ has demonstrated high validity and reliability inmeasuring temporo-spatial parameters of gait in adults with and without neurologicaldisabilities as well as in children aged 1 to 12 years without disabilities [101-108], but
the measurement properties of gait parameters have not been examined in children
with CP. The walkway is portable and seemed feasible to use in the population ofinterest. However, concern remained as to whether children with cerebral palsy at
different ages and at different functional levels could manage to follow the
instructions for walking on the walkway, and whether their walking pattern could berecorded reliably in a short-time perspective when no change was expected to occur.

Measurement properties of quality of movement measures developed for children
with CP have been tested by their developers or persons affiliated to the group thatdeveloped the measures and found satisfactory regarding reliability, validity andresponsiveness (Table 5). However, as the measures were to be scored from video
clips in the present project, the reliability of scores under such conditions had to be
examined.

In the intervention study, standardized condition-specific and generic measures that
had been found reliable, valid and responsive to change in children with CP werechosen (Table 5). The assessment tools were selected to cover several dimensions of
the ICF-CY related to motor and everyday functioning, and the length and ease ofadministration were taken into consideration, as well as their feasibility for use in a
multicenter study. The measures also had to be available in Norwegian or English. In
addition, an individualized goal attainment measure regarded as being particularly
responsive to change in children with disabilities [109] was used (Table 5), while
individual goals remained a main component of the intervention model. Pilot testingof the assessment protocol was conducted with children who participated in themethodological studies. The electronic walkway GAITRite£ was also included in the24
protocol of the intervention study, but due to unexpected practical circumstances, theportable walkway was not applied.

Table 3.
Assessment tools used in the different studies of the thesis.
Assessment tool*

Study IGAITRite®
GMPM
QUEST
GMFM-66
PEDI
GAS

x

Study II

Study III

x
x

x
x
x
x
x

* For abbreviations see page xi

Table 4.
Assessment tools related to components of the ICF-CY.
Component

Functioning/disability assessed

Assessment tool*

Body function and structure

Gait pattern
Movement functions

GAITRite®,
GMPM, QUEST, GAS

Activity and participation

Basic gross motor abilities
Hand activities
Mobility
Self-care
Social function

GMFM-66, GMPM, GASQUEST, GAS
PEDI, GAS
PEDI, GAS
PEDI

* For abbreviations see page xi

25
Table 5.
Reported measurement properties of the selected assessment tools in samples of children in pre- and primaryschool age with CP (condition specific assessment tools) or disabilities (generic assessment tools).
Assessment
tool*

Measurement properties

GMPM

Intra-tester reliability: ICC = 0.92-0.96 for total score[110,111], 0.90-0.97 for attributes [110].
Inter-tester reliability: ICC= 0.92-0.93 for total score[110,111], 0.84-0.94 for attributes[110,112].
Test-retest reliability: ICC = 0.96 for total score [109,110], 0.89-0.96 for attributes [110].
Content validity: Evaluated by 13 international experts when the assessment tool wasdeveloped [113].
Criterion validity: Not established; low correlation with parents’ and therapists’ ratings [114].
Construct validity: A priori hypotheses of differences between groups of children confirmed[114].
Responsiveness: Change in stable and responsive groups similar to therapists’ judgments [114].

QUESTInter-tester reliability: ICC = 0.90-0.96 [115,116].
Test-retest reliability: ICC = 0.95 for total score [115], 0.51-0.96 for domains [115].
Content validity: based on literature review and consultations with experts [115,117].
Criterion validity: Correlation with Peabody Fine Motor Scale r=0.84 [115,117].
Construct validity: Correlation with therapists’ ratings r =0.72 and 0.58 for left and right hand,
respectively. Correlation with age r= 0.33 [115,117].
Responsiveness: Responsiveness to change demonstrated [118].

GMFM

Intra-tester reliability: ICC = 0.99 [119,120].
Inter-tester reliability: ICC = 0.80-1.0 [119-121].
Test-retest reliability: ICC = 0.76 – 1.0 [119,121,122].
Content validity: Pilot tested by therapists [15]. Item hierarchy shown [120].
Construct validity: A priory hypotheses regarding change in scores in different ages andfunctional levels confirmed [119,120,123].
Responsiveness: Change correlated with change seen by parents and clinicians [119,120] anddemonstrated in effect size and standardized response means [82,118].

PEDIInter-tester reliability: ICC = 0.72-1.0 [7,77,124].
Test-retest reliability: ICC = 0.8–0.98 [124].
Content validity: Evaluated by 31 experts when the assessment tool was developed [7].
Criterion validity: Concurrent validity with related assessment tools of motor function and selfcare; r= 0.59-0.97 [7,77,125].
Construct validity: Support for hypotheses regarding increased scores with age [7].
Responsiveness: Responsiveness to change demonstrated in effect size and standardized
response means [82,118]. Change correlated with changes seen by parents [77,82] andtherapists [88].

GAS

Intra-tester reliability: ICC = 0.96 [126].
Inter-tester reliability: ICC = 0.51-0.96 [126,127].
Content validity: Supported when goals are appropriate, reasonable, relevant and complete, butreliant of clinical skills of goal setters [126,127]. Goals for children with CP supported byexpert panel of experienced PTs [128].
Criterion validity: Low correlation with Peabody gross motor scale [128] r=0.25.
Responsiveness: Good responsiveness for detecting meaningful clinical change [127-129].

* For abbreviations see page xi

26
GAITRITE (Study I)

The electronic walkway GAITRite®, (CIR Systems Inc., NJ, USA) is connected to a
portable computer and constructed to record gait parameters (Figure 6). As subjects
walk across the electronic walkway, sensors are activated under the pressure of thefeet and deactivated when the pressure is released. The geometry and the relative
arrangement of each footfall as a function of time is recorded [130]. The computer’ssoftware processes the raw data into footfall patterns and computes a large range ofspatial and temporal gait parameters [130]. From clinical experience five gaitparameters were chosen as clinically relevant in this study. Definitions of the spatial
parameters stride length, step length and step width selected in Study I are illustrated
in Figure 7. The temporal parameters cadence defined as steps per minute and stancetime on one leg were also considered relevant, and two asymmetry measures of step
length and stance time were in addition calculated from the gait parameters.

Figure 6.
Portable walkway [130] .
Reprinted with permission.

Stride length
Step width
Step length

Figure 7.
Definitions of spatial gait parameters illustrated.

27
GMPM (Study II and III)The Gross Motor Performance Measure (GMPM) [111] was developed to assess
quality of movements in gross motor activities like walking, sitting and crawling inchildren with CP. The test is observational and criterion referenced, and the children
are compared to themselves over repeated measurements. Twenty items derived froma sibling measure, the Gross Motor Function Measure (GMFM), are assessed on five
attributes: Alignment, Coordination, Stability, Dissociated movements, and Weightshift. For items that can be performed on both left and right side, only the child’s mostaffected side is tested and only items for which the child receives a score of 1 or moreon the GMFM, is scored on the GMPM. In each item, three single attributes arescored on a 1-5 point ordinal scale (Figure 8). A score of 1 is given when nopathology is observed in three repeated trials. A score of 5 is given when severe
pathology is observed. Percent scores for the attributes (scale 20-100 %) and a total
score (scale 0-100%) as the average of the five attribute scores, are calculated.

Figure 8.
Illustration of the scoring system of the Gross Motor Performance Measure (GMPM)

28
QUEST (Study II and III)The Quality of Upper Extremities Skill Test (QUEST) [115] was developed todescribe aspects of upper extremity quality of movement in activities like grasp,
weight bearing and protective extension, to plan intervention programs and evaluate
effectiveness of therapy in children who exhibit neuromotor dysfunction withspasticity. Quality of movement is assessed in four domains: Dissociated movements,
Grasp, Weight bearing, and Protective extension (Figure 9). The measure includes 33
items. Each item comprises, however, several sub-items and both left and right upper
extremity is assessed, so a total of 174 sub-items are scored on a dichotomous scale;“able to complete” or “not able to complete”. If the movement is not administered,this is reported as “not tested”. Percent scores for the domains (scale 0-100 %) and a
total score (scale 0-100 %) are calculated as the mean of domains actually tested.

Figure 9.
Illustration of the scoring system of the Quality of Upper Extremity Skills Test (QUEST)29
GMFM-66 (Study II and III)The Gross Motor Function Measure (GMFM) [119] was developed to evaluatechange in gross motor function in children with CP. The GMFM is a criterionreferenced, clinical observation tool designed to be used by all types of CP andwithout age restrictions. The items are grouped into five dimensions of basic motor
functions: Lying & rolling, Sitting, Crawling & kneeling, Standing, and Walking,running & jumping, and scored on a 0-3 ordinal scale. A score of 0 is given when achild is not able to perform the task. A score of 3 is given when the child fully
execute the motor task of the item. Two versions of the GMFM exist; the original
GMFM-88 and a more recent version, GMFM-66 [15]. In the GMFM-88, percentage
scores (scale 0-100 %) are calculated for each dimension and the total score (scale 0100 %) is the average of the five dimension scores. In order to improve theinterpretability and clinical usefulness, a Rasch analysis was applied to the GMFM88, resulting in a unidimensional hierarchical scale consisting of 66 items from theoriginal measure [120]. Using the Gross Motor Ability Estimator software [15] a total
score (scale 0-100) with interval-level properties is calculated for the GMFM-66.

PEDI (Study III)The Pediatric Evaluation of Disability Inventory (PEDI) [7] was developed todetermine chronically ill children’s functioning in three domains: Self-care, Mobility,
and Social function. The instrument contains three theoretical dimensions: Functional
skills, Caregiver assistance and Modification (environmental and technicaladaptations) (Appendix 4). In this thesis the Functional skills and Caregiver assistancedimensions were applied. In the Functional skills dimension, 197 items are scored ona dichotomous scale; “able to perform the item in most situations” or “unable toperform the item in most situations”. Caregiver assistance is determined for 20 items

30
on a 0-5 ordinal scale, where a score of 0 is given if the child needs total assistanceand a score of 5 is given if the child is independent in the activity. Normative standard
scores for the domains based on a sample of American children without disabilities
can be derived from the measure as well as scaled scores (scale 0-100) which
provides an indication of a child’s ability to perform the total number of tasks of the
PEDI domain. The PEDI is translated into Norwegian [131,132] and validated in
Norwegian children without disabilities [133]. The Norwegian norms deviatedsomewhat from the original norms, implying a cultural difference, however, thescaled scores are not age related, but provide a criterion referenced measure, and isapplicable in a Norwegian setting [133]. The measure is usually administered in a
structured parent interview.

GAS (Study III)

Goal Attainment Scaling (GAS) [109] was developed to capture individualized goal
attainment within a predetermined timeframe. For goal identified by family/child andprofessionals in collaboration, a scale containing five levels of outcome descriptions
is constructed: Expected outcome (0), two levels of less (-2 and -1) and two levels of
more (+ 1 and + 2) than expected outcome. The scales are scored in a follow-up
evaluation. Example of a follow-up scale is given in Appendix 5. When used inclinical practice, usually the caregiver and professionals construct the follow-up
scales. In research, however, it has been recommended that other than professionals
who conduct the intervention construct the scales [134].

31

Goal setting process and goals
Goals for the intervention period in Study III were decided through a collaborative
process including parents who made decisions about the content of the goals, andprofessionals who contributed to operationalize and refine the goals. A total of 98
goals were set for the training period, an average of 4 for each child. 57 goals wereclassified as activity goals, e.g. “Can put trousers on without help”, while 31 wereclassified as movement goals, e.g. “Can get the heels down when he stands
supported”, and 10 were combined. GAS follow-up scales were constructed for all
children, for one to three goals per child, a total of 53 scales. The follow-up scales
were scored by parents and professionals in collaboration at the final follow-up
assessment. The goal attainment of the remaining 45 goals was reported by parents
and professionals at the last follow up assessment. Type and number of goals scoredwith GAS and by oral report, respectively, are listed in Table 6.
Table 6.
Attainment scoring of goals.
Attainment scoring

GAS follow-up scale
5 levels
Oral report

Number ofactivity goals

Number of
movement goals

Number ofcombined goals

34

14

5

23

17

5

Videotaping and editingStandardized procedures for videotaping performances of the GMPM and QUEST
were developed, piloted and revised twice, in collaboration with the PT who later
videotaped the assessments in Study II and III. The final written procedures areshown in Appendix 6. In GMPM, each item was videotaped separately, while in the

32
QUEST, several test items were videotaped in one sequence due to the structure of
the measure.

The videos were edited using the Pinnacle Studio 8 software program (Pinnacle
Systems Inc) and stored on DVDs. A fixed procedure for editing video clips wasused. The duration of a video clip was for example standardized in items containing
static positions where no indication of time was given in the manuals. In the finalDVDs, each GMPM item was announced by the item number, while in the QUEST,
since the items are performed as consecutive movement sequences and several items
are scored during one sequence, the video clips were announced by the dimension
name.

Intervention
The intervention model in Study III aimed to provide an intensive, but limited periodof physiotherapy within the frame of the children’s local environment to support theirachievement of individual goals of motor and everyday activities. To ensure that theintervention could fit within the children’s everyday life, a model of three hours of
training, five days a week in a three-week period was chosen. The principles of theintervention model have been described in the pilot project [39] and are further
outlined in a report from the developmental project [42]. The following principles
have formed the base for the intervention model: 1) Functional goal directed training,
implying a focus on practicing specific activities of importance to the child [35], 2)
Family centered practice [36], implying that parents were involved in the goal setting
process and were active participants in the training, 3) Cooperation between groupleaders and parents as well as local professionals and other persons important to thechild, to secure carryover of knowledge and skills to the child’s everyday life, 4)

33
Applying recent knowledge of motor learning, using e.g. motivating activities,
stimulating environments and variation [135,136]. For a more specific description of
the intervention, one group training session at each site was video recorded and theamount and type of training and also the applied teaching and learning strategies,
were outlined (Appendix, Paper III).

The intervention model had been applied in clinical practice for several years at aphysiotherapy institute before the start of this study. To make sure that theintervention was similarly conducted, the group leaders participated in a workshop
with practical and theoretical lessons, lasting two days. The workshop included
observation of ongoing groups, theoretical lesions and discussions regarding
planning, organization and guidance of a group, contents of the intervention andinformation about the assessment protocol. In addition, the group leaders weresupervised three times during the course of the intensive training.

Statistical analysis
The children’s background data and raw data scores were described by values ofmean, standard deviation (SD), range, confidence interval (CI) and percentage.
Parametric statistics were applied when i) the variables were at interval or ratio levels
and in fine graded sum scores of variables at ordinal level, ii) the assumption of
normality was met by inspecting Q-Q plots and by examining for normality using theShapiro-Wilk test for small samples [21]. If the assumption of normality was not met,
non-parametric statistics were applied. An overview of statistical methods ispresented in Table 7.

34
Table 7.
Main statistical methods applied in the studies.
Statistical method

Study I

Study II

Intra Class Correlation
Cohen’s Kappa
One-way analysis of variance
Mixed factorial analysis of variance
Paired T-test
Spearman rank correlation
Friedman’s Test
Wilcoxon signed rank testxxxStudy III

x
x
x
x

x
x
x

P-values  0.05 were considered to be statistically significant, however, on repeatedmeasurements of the GMFM, and in PEDI where the dimensions have been shown to
be inter correlated [7], a stricter p-value (p< 0.01) was chosen. Criteria for acceptable
ICC, Kappa and percent agreement values were set in advance. Sample size
calculation based on results from the pilot study was performed in connection with theplanning of Study III. Raw GAS scores were transformed to standardized GAS Tscores using the formula given by the developers [109].

Ethics
The studies were performed according to the Helsinki Declaration. The protocol wasapproved in advance by the Regional Ethical Committee in Western Norway and the
National Data Inspectorate of Norway. Written informed consent was obtained fromthe children’s parents before participation in the studies (Appendix 7), and thechildren were given age appropriate information about the objectives of the studies.

35

Summary of the papers
Methodological studies
Study IThis study examined measurement properties of an electronic walkway in a sample ofchildren with CP. Gait parameters can be assessed in clinical observation, by using
equipments such as a stop watch or inkpads, and by advanced equipment in gaitlaboratories. Electronic walkways are relatively new technological equipment, used
to record gait parameters. They are said to be feasible in a clinical setting, with
automatic exportation of gait parameters to a computer for subsequent analysis.
Measurement properties of gait parameters have previously been examined in adults
with and without disabilities and in children with typical development, and haveshown high test-retest reliability. The objective of this study was to examine testretest reliability of seven selected temporal and spatial gait parameters and asymmetry
measures in children with cerebral palsy by an electronic walkway. Seventeen
children with CP between 3 and 13 years of age participated in the study. They
performed a standardized procedure of walks with different speeds along theelectronic walkway. The tests were repeated after approximately 25 minutes. The
scores were normalized to a walking speed of 1.1 m/sec to avoid the confounding
effect of gait speed on speed dependent gait parameters. Intraclass correlation
coefficients (ICC1,1 and 3,1), within subject standard deviation (Sw) and smallest
detectable difference (SDD) were calculated. The relative reliability of cadence, step
length, stride length and single stance time was high to excellent (ICC1,1 being
between 0.73 and 0.95 ), while it was poor for step width (ICC1,1 = 0.27 and 0.35).
The relative reliability for the two asymmetry measures were high for the step length
index (ICC1,1 = 0.82) and moderate for the single stance time index (ICC1,1 = 0.49).

36
The absolute reliability values for the gait parameters and asymmetry measures werealso calculated and reported. All children, except one, could easily walk over the matat different speeds, making it possible to calculate gait parameters at a normalized
walking speed. In one child, the walkway failed to register sufficient footsteps,
possibly due to the child’s shuffling gait pattern.

Study IIIn this study measurement properties of measures assessing quality of movement were
examined in a sample of children with CP. Quality of movement measures forassessing motor performances of children with CP are complex and time consuming
to score. Structured recordings and scoring from video clips may ease the assessment
burden on children and assessors. The objective of the study was to examine observer
reliability of the Gross Motor Performance Measure (GMPM) and the Quality ofUpper Extremity Skills Test (QUEST) based on scores from observation of video
clips. The tests were administered to 26 children with CP aged 2 to 13 years. The
children’s performances were recorded and edited according to fixed procedures. Twoexperienced paediatric physical therapists assessed the children from watching thevideo clips. Intra-observer and inter-observer reliability values (ICC1,1) of the total
scores varied from 0.69 to 0.97 with only one coefficient below 0.89. The ICCs of
sub-scores varied from 0.36 to 0.95, finding “Alignment” and “Weight shift” in
GMPM and “Protective extension” in QUEST highly reliable.

The sub-scores“Dissociated movements” in GMPM and QUEST and “Grasp” in QUEST were theleast reliable. Kappa coefficients of single items ranged between 0 and 1 for bothmeasures. The video scoring was time-consuming and demanding, but offered many
advantages; the possibility to review performances, to use especially trained observers
for scoring (not available during the tests), and less demanding assessments for thechildren.

37Intervention study
Study IIIThe objective of the intervention study was to investigate the impact on basic motor
abilities and everyday activities of children with CP (n= 22) who participated inintensive activity-focused, goal-directed physiotherapy training for the first time. It
was further an objective to investigate the coherence between acquisition of basic
motor abilities and quality of movement during the study period. A rather stablebaseline phase in the repeated measures design was demonstrated by the Gross Motor
Function Measure (GMFM-66), being the main outcome measure. A main effect oftime was shown after the intervention period, mean change being 3.8 (p<0.01) at first
follow up increasing to mean 4.5 at the last follow up assessment. An interaction
between time and Gross Motor Function Classification System (GMFCS) levels wasfound, implying that children classified to GMFCS-levels I-II improved more thanchildren classified to GMFCS-levels III-V. There were no main or interaction effects
of age or anti-spastic medication. Change scores in the Pediatric Evaluation of
Disability Inventory (PEDI) ranged from 2.0 to 6.7, and the change was foundsignificant (p<0.01) in the Self-care domain of the Functional Skills dimension and
the Self-care and Mobility domains of the Caregiver Assistance dimension. The
children’s individual goals were on average attained, Mean Goal Attainment Scaling(GAS) T-score being 51.3. Video scoring by two blinded assessors gave nonsignificant improved scores on the GMPM and the QUEST. A significant
improvement in GMPM scores was found in the same items that improved in the
GMFM, but not in items that maintained the same score.

38

Main findings
Methodological studies•

Test-retest reliability in a short time span of gait parameters using an electronic
walkway, was high in five of seven gait parameters and asymmetry indexes inchildren with CP who were able to walk without assistive walking devices.

•

The walkway was feasible for recording gait parameters in children with CP, butreliability seemed limited in children who had difficulties in following theadministration procedure or understand the task.

•

Inter-observer and intra-observer reliability of GMPM and QUEST total scores
were mostly satisfactory high.

•Reliability of sub-scores and single items of the two quality of movement
measures differed, some showing high others low reliability.

•

Video scoring was time-consuming, but was found to offer many advantages, likepossibility to review performances, a less demanding assessment procedure forthe children and use of especially trained observers who were not available forscoring at the time of the tests.

Intervention study•

Basic motor abilities and self-care improved in young children with CP after
three weeks of intensive, activity-focused and goal-directed physiotherapy.

•

Individual goal attainment was high.

•

The children’s need for caregiver assistance in self-care and mobility decreased.

•

No significant change was found in the children’s social function or in their
quality of fine and gross motor movement patterns, however, in basic motor

39abilities that improved during the study, significant improvement in quality of
movement was found, whereas in basic motor abilities that remained stable, nosignificant improvement was found.
•

Intensive, goal directed physiotherapy over a limited period of time, withinvolvement of the children’s parents and local professionals seemed to be welltolerated, motivating and instructive for the participants.

•

To accomplish individualized training within a group context and at the sametime take care of the group process and supervise the children’s escorts, requires
competence and experience.

40

Discussion
Methodological studies
In the two methodological studies of this thesis, different types of reliability were
investigated depending on the nature and expected use of the measures.
Reliability of the electronic walkway GAITRite
Optimizing gait function e.g. the speed, effort and safety of gait is often anintervention goal in children with CP who are able to walk, and a portable walkway
might be a cost-effective and user-friendly way of quantifying temporal and spatial
gait parameters. Four gait parameters considered clinically relevant for children withCP (cadence, step length, stride length, single stance time) and an asymmetry index
based on step length, revealed high repeatability (Table 3, Paper I). The satisfactory
relative and absolute reliability of these gait parameters make them applicable inresearch. As variability in scores from test to retest was low, a change in theparameters could be considered mainly a result of a true change and not of
measurement error. Low reliability was demonstrated for the step width, as has also
been found in studies of other populations [102,103,108]. A test-retest design does
not separate different sources of error, e.g. errors resulting from the instrument, theadministrators or the participating children [6]. Hence, possible sources of error in thestep width parameter in this study might be due to the size of the sensors as suggested
by others [102], or that a hallmark of step width might be variability rather thanstability in children with CP. The asymmetry index stance time also showed low
reliability and this index as well as the step width parameter seem, accordingly, lessapplicable than the others in research.

41
Gait parameters in children with CP collected by use of other types of device, likedata-based equipment in a laboratory setting or observational measures, havedemonstrated moderate to high reliability [137-141]. However, neither the reliability
values nor the gait parameter values can automatically be compared acrossmeasurement devices since they might measure different aspects of gait, and measure
the parameters in slightly different ways. Footsteps computed from pressure sensitive
pads under the feet may for instance give different output compared to parametersfrom markers over the ankle joint recorded from a sagittal plane. Hence the reliability
is equipment, environmental and population specific and the gait parameter values
from an electronic walkway cannot directly be compared to values from otherequipment.

Concurrent with the pre-publishing on the net of Paper I, a study examining test-retest
reliability of the GAITRite£ in a sample of American children with disabilities was
published [142]. Reliability of six temporal-spatial gait parameters in children withCP (n=16), Angelman syndrome (n=2) and arthrogryphosis (n=1), was examined in
two conditions; barefoot and with shoes and orthoses. The pre determined minimum
reliability coefficient criteria of ICC = 0.80 was met and supports that the electronic
walkway is a reliable way of recording gait parameters in children with CP. This
study expanded the scope for examination of reliability as gait with shoes andorthoses were also investigated, in contrast to our study where only the barefootcondition was examined.

A large range of spatial and temporal gait parameters are computed by the software of
the GAITRite£ walkway [130]. In the present study, as well as in the study by
Wondra et al. [142], reliability was examined in gait parameters considered relevant
as outcome measures in children with CP. In the present study and in the study ofWondra, reliability of cadence and stride length was examined. The other gait42parameters examined in the two studies can hardly be compared since different
expressions of the parameter were examined, e.g. single stance time as percentage ofthe gait cycle in one study versus single stance time in seconds in the other. In
cadence and stride length, however, our study revealed less variability by smaller
absolute reliability values. The method of calculating gait parameter values at acommon reference speed might be a more reliable way of expressing gait parametervalues than using a single or the average of three trials, as in the study by Wondra etal. [142], where walking speed might be a confounding factor.

Reliability of gait parameters as measured by computerized gait analysis, has beenfound dependent upon GMFCS-level [143], children in GMFCS-level I exhibiting
higher reliability than children classified in GMFCS-levels II and III. In our study, we
did not find differences in reliability with respect to either GMFCS-level or age(Paper I). However, children classified in GMFCS-level III were not included. The
recording of gait parameters from children who usually use crutches, canes or otherwalking devices are, however, possible when using an electronic walkway, but might
require additional work to identify foot steps.

Feasibility and recommendations for use of the GAITRite as a research tool
The electronic walkway was tried out on two different sites during the reliability
study and it was easy to transport the walkway and use it in a location familiar to thechildren. Furthermore, it was easy to motivate the children to walk over the walkway,
the procedure of walking at three different speeds was easily understood by thechildren as young as three years, and the procedure required little test time;
approximately five minutes for each child (Paper I). There was no requirement ofplacement of any devices on the child and the raw data could be stored to be analyzed
by skilled professionals later.

43
Careful attendance of the administration procedure with observation of the child’s
ability and need for additional visual or verbal instructions might increase thereliability of the walkway (Paper I). Picking up toys at the end of the walk can serve
as motivation for performing repeated walks, and most children found it exciting tolook at their own footfalls on the computer screen when all the walks in the procedure
were carried out. However, for most children, additional motivation beyond
instructions seemed not needed.

The walkway seems to have limitations when children use a shuffling gait pattern, as
the registration of single footsteps by the sensors inherent in the walkway is thus
complicated. In a previous pilot study [144], the walkway was found to have
limitations in recording footfalls in young children possibly due to their low weight.
However, this was not found in the present study. The procedure of walking over the
walkway at three different speeds requires vision and cognitive abilities, and will
hence restrict the use of the walkway for groups of children with CP.

Compared to examination of gait parameters in a gait laboratory, the cost of the
walkway equipment, the training of administrators and the time used for preparation
and analysis of the gait data, are considerably less. The walkway has limitations
compared to gait analysis performed in a gait laboratory, since it records neither
kinetics nor kinematics of the gait pattern. However, the walkway might haveadvantages in investigating aspects of gait.

The impact on gait parameters ofdifferent interventions like strength training [145], NDT [146], treadmill training[147] and hippotherapy [148] have been investigated in children with CP, somestudies showing a large positive impact of the intervention on gait parameters [145147], whereas in other studies such impact was not found [148]. The walkway was
included in the protocol of the intervention study to investigate change in gaitparameters as a result of the activity-focused, intensive physiotherapy. However, this44
part of the project was not carried out due to illness of the PT who was to administer
the assessment with the walkway, and practical difficulties to recruit and train anotherin time for the assessments of the first groups. Hence an important outcome measureis lacking in the intervention study, but the research question is of interest and will befurther investigated.
Reliability of the quality of movement measures GMPM and QUESTGMPM and QUEST are observational measures constructed to describe and evaluate
quality of movement in children with CP. The scoring of the measures is complex
since several aspects of movement quality are to be scored concurrently in each item;
hence the observer-reliability is of concern.

Intra-observer and inter-observer reliability in items scored from video clips were
examined, and the total scores on both measures were found to be highly reliable(Paper II). Scoring of the GMPM and QUEST entirely from video clips have to ourknowledge not previously been performed, but the reliability of the total scores
correspond to results from studies performed by the developers of the GMPM
[110,111] and the QUEST [115,116] in a clinical setting, and in addition to a recent
study regarding inter-observer reliability of the QUEST [149].

The sub-scores Alignment and Weight shift in GMPM and Protective extension in
QUEST were likewise found highly reliable, whereas the reliability of the otherdomains of the measures and single items revealed a large range of reliability
coefficients (Table 2, Paper II). In the sub-scores with low reliability, the ICC values
were lower than ICC values found in other studies [110,112,115,116]. As the conceptof movement quality and scoring of the items were seen as challenging, as also
indicated by others [112,117] much effort was used to discuss the definitions and the45scoring of items, particularly in the GMPM. A total of 22 hours over a four-month
period were used for training. However, the training and the practice of the scoring
systems might not be sufficient, and the decision to focus on the GMFM at the cost of
the QUEST (Paper II) might have been a reason for the low reliability of some thesub-scores of the QUEST. The sub-scores with the lowest reliability, Dissociatedmovements in both measures, require observation of several body parts and joints
concurrently and seem to require good observation skills and knowledge of thescoring criteria. The sub-scores with high reliability; Alignment and Weight shift in
the GMPM and Protective extensions in the QUEST, could be seen as the quality
attributes most alike those clinician observe and describe in clinical practice, andhence be most familiar to the assessors.

It has been claimed that if inter-observer reliability is high, intra-observer willconsequently also be high and there is therefore no need for testing this [5]. In thisstudy, however, the inter-observer reliability in some scores was higher than intraobserver reliability, also implying that within assessors’ interpretation, the scores
might vary. Hence, in observer dependent measures, there might be a need for theexamination of intra-observer reliability.

Feasibility and recommendations for use of the GMPM and QUEST as
research tools
Quantifying quality of movement patterns is demanding and assessing quality of
movement from video clips was found to have many advantages for the assessors andthe children, such as the possibility to watch the video clips several times, a more
efficient scoring while the assessments were edited and a less demanding test
situation for the children while the assessment was not interrupted by scoring (Paper
II). However, the use of video scoring is also costly in terms of training, editing and46
scoring. An expanded manual and training-DVDs with examples of the scoring would
have clarified the scoring options and secured the understanding of the dimensions of
the measures.

The total scores of both measures demonstrated high reliability and are recommended
used for research. The total scores consist, however, of several sub-scores
representing different aspects of movement quality. As goal setting, intervention
planning and evaluation may relate to only some aspects of movement quality and achange in a particular attribute/domain may be masked in the total score; reliability ofseparate domain scores should be of concern. Even if the sub-scores of the GMPM
and QUEST were designed to capture clinically useful information, we recommendthat only the total scores are used for research purposes to avoid measurement error
obscuring the results.

Strengths and limitations of the methodological studies
Heterogeneous samples of children with CP from three municipalities in Western
Norway were included in the two studies. In Study I, this secured the examination ofreliability in a sample with different gait abilities. In Study II, all GMFCS- andMACS-levels were represented in the sample, which secured that all items were
included in the reliability study, which is essential when examining the reliability ofan assessment tool. The samples of the reliability studies were seen as representativewith regards to age, GMFCS- and MACS-levels of the population of interest in theintervention study.

The number of children in the reliability studies are in line with earlier
methodological studies in the field [110,112,115,116,149], but the samples are47
smaller than suggested (n=50) in a recent paper on quality criteria for measurement
properties [12]. The sample sizes were based on practical considerations; however, alarger sample might have given a more precise estimate of the reliability of the
GAITRite®, the GMPM and the QUEST. Larger samples might also have provided
better estimates for reliability of subgroups of children, and revealed differences
between functional levels, which were not uncovered in the present studies.

The results of the reliability studies are limited to children with CP aged 3 to 13 years.
The inclusion criteria of the studies were youths up to 15 years of age. It seemed,
however, difficult for children in the final years of primary school and secondary
school to participate during day-time, as is understandable, and if older children wereto participate, one might consider other times of the day and other ways of recruiting
the children.

In Study I, only children classified in GMFCS-levels I and II were included, but theelectronic walkway may also be a possible outcome measure for children classified in
GMFCS-level III. This subgroup could have been included to examine reliability inchildren who are able to walk with assistive devices and hence broadened the scope of
reliability study. In Study I, only one PT administered the assessment with the
walkway. Examining reliability with several administrators might also have revealed
whether the administrator influenced on reliability of the gait parameters.

In Study II, the assessors were trained to score the measures simply on the basis of thewritten test manuals, without training with more experienced users of the quality of
movement measures. In addition the reliability was examined on the basis of scorings
from video clips, and both these conditions might have affected the reliability. A
design that separated these two conditions might have revealed whether the trainingor the scoring from video-clips were the main source of variability in the scores. It is

48
evident that scoring of the quality of movement measures requires competence andexperience in movement analysis in children with CP and preferably also competence
in testing. Hence the results of this study can not be generalized to novice
physiotherapists. The administration of the measures with video uptakes for
subsequent scorings also requires good knowledge of the measures and experience intesting.

Intervention Study
When the intervention study was initiated, the body of knowledge regarding effects ofintensified physiotherapy programs for children with CP was limited. Few studies had
been performed and the results possibly biased by methodological weaknesses[27,28]. There were indications, however, that increased frequency and duration oftraining might provide more gains in basic motor abilities than common training at thetime, often implying one hour of weekly physiotherapy [34,91,92,94,96,98]. The
funding of developmental projects in the habilitation field in the time period of 2002
to 2008, made it possible to accomplish intensive physiotherapy training in groups forchildren with CP at four habilitation units in Western Norway, and to investigate theoutcome in a systematic way.

Change of functioning in a capacity and performance perspective
The main outcome measure in the study, the GMFM, is considered a measure of achild’s capacity, meaning what a child can do in a standardized environment [77]. The
study revealed significant improvement in basic motor abilities after the intervention
period as measured by the GMFM. The children with the highest functional levels(GMFCS-level I and II) obtained more gains in motor function than children

49classified to Levels III to V (Paper III), which is in line with development curves forchildren with different GMPCS levels, constructed by Rosenbaum et al. [72].
Different abilities to change, depending on GMPCS levels, have also been shown inother intervention studies [91,93]. However, also children classified at levels III to V
improved in gross motor function in the present study, implying a potential for change
when the children participated in three weeks of intensive physiotherapy training.

What the child is capable of doing in a standardized setting might, however, notcorrespond to what the child performs in everyday life with constraints like distance,
speed and safety [150]. The emphasis on assessing functioning in actual life situations
has increased over the last ten years [33,86,127,151].

Tieman [86] found thatchildren and youths with CP, well capable of walking without assistance, chosedifferent locomotor strategies in different environments; walking without walking
aids at home, with crutches or canes at school, and use of a wheelchair in thecommunity. Children with anticipated similar functioning according to GMFCS-level,
were found to use quite different locomotor methods in everyday life [152]. While
children with severe functional limitations gained improved GMFM scores after anintervention, these improvements were not found to be transferred to improved
abilities in home environments [153]. The intensive training in the present study was
accomplished in the child’s local community, but not in the everyday setting of thechild, like kindergarten, school or home environments. The transference of potential
achieved capacity to performance in home environments was hence of concern whenplanning the study, including PEDI as a supplementary measurement tool.

The PEDI is based on the parents’ perception of the child’s performance in everyday
environments, and our results did indicate a positive change after the intervention
period. The parents reported that their children had obtained increased participation in
everyday activities, and that they needed less assistance in mobility (Table 4, Paper

50III). This is in line with results of a 3-year longitudinal study by van Eck et al. [151]
who found increased motor capacity to be significantly related to improved motor
performance in children with CP. There might be several components in theintervention model, in addition to the motor practice, that contributed to thesechanges. The group training included extensive education and supervision of parents
and local professionals to secure knowledge transition and carryover to the child’s
daily environments. The developing child is strongly dependent on the caregivers andthe social environments, hence the influence of the environmental factors on
functioning may in this phase be even greater than later in the life span [8]. Increased
knowledge, skills and changed attitudes achieved through participation in theintervention period, might have influenced the children’s social environments andhence their performance in daily life.

Since the intensive training was performed in a group setting, the social function
domain of PEDI was also included in the assessment. In this domain, as well as in thechildren’s functional mobility domain, some improvement was indicated, but thechange was not statistically significant. In the interviews with the parents, allunderscored their child’s functional improvements, and exemplified change observedin the home environments within all dimensions of the ICF [42] such as; improved
ability to take turns, improved self-esteem and more engagement in play inkindergarten and at home. In paper III we have discussed our experiences with PEDI
which in some cases served as an educational tool for the parents at the preintervention

assessment

regarding

performances

of

their

children.

Otheradministrations of the PEDI, like informing the parents about the contents of the items
before the interview, or administration over two sessions might have improved thevalidity of the parent’s information, particularly at the beginning of the study. An
expanded assessment of the children’s performances in everyday settings with moreemphasis on participation e.g. by the “Children’s Assessment of Participation and51
Enjoyment” [154] or the “Test of playfulness” [155] might have revealed otheraspects of transference between capacity and performance. In other studies ofintensive therapy where PEDI has been used as an outcome measure, significant gains
in approximately the same domains as we have found, have been shown [96,97]. The
social domain, however, is seldom used as an outcome measure, but in one study
where the children intensively performed goal directed activities in their daily
environment, and in addition participated in one group session per week, significant
improvements in the social dimension were demonstrated [97]. This may raise aquestion of whether an ecological approach is more beneficial than intensive grouptraining, not taking improved social functioning in particular into consideration. Thisis a subject of further investigation, where the aim of the intervention should befurther discussed.

Change of functioning in an individual and a group perspective

A principle in the intervention model applied in the present study was to emphasize
the child’s own goals for the training period. In the written invitation as well as in theverbal information given prior to the study, the caregivers received information about
the goal setting process (Appendix 7), and were encouraged to reflect upon goals forthe intervention period and discuss possible goals with the child’s local professionals.
During the baseline period, the children were repeatedly tested with motor assessment
tools according to the protocol, and parents were interviewed regarding the children’s
performance of everyday activities. After the two first assessments in the baseline
phase, preliminary goals were written and finally formulated at the third baseline
assessment, allowing the decision of goals to be a process.

Due to the time consuming process of constructing GAS-follow up scales, such scales
were only constructed for half of the goals. For the remaining goals, parents and52
professionals reported orally after the intervention to what extent the goals wereattained. On average the children’s expected goals in the GAS scales were attained, asreported in Paper III. The individual goals were in the field of basic motor abilities,
self-care and movement patterns, and probably reflected the parents’ decision toparticipate in a physiotherapy training group. The contents and scores of the non-GAS
goals were not reported, but the type of goals and the goal attainments were quite
similar to the GAS follow-up scales. The participants in the groups seemed toappreciate the goal directed approach, as has also been found in other studies[34,35,97,156]. The individual goal setting approach also seemed feasible from aclinical point of view, and has been continued as a basis for training in later groups[41,42].

In the intervention study statistical significance of change scores for the total sample
was of concern. However, other frameworks for examining change might have beenused. Due to the heterogeneity of the population and the challenges of performing
randomized controlled trials (RCT-studies) or case-control studies in the CP
population, single subject approaches have been proposed [27], where children act astheir own controls. A complementary evidence judgement scale for single subject
research has been developed [157] and implemented in the American Academy for
Cerebral Palsy and Development Medicine (AACPDM) outcome reviews. In thepresent study a characteristic of the single subject paradigm was used by
implementing the repeated measures design. Each child acted as its own control, andvariability due to heterogeneity in the sample was removed [3]. A further extension of
the individual paradigm, would have been to calculate the percentage of individual
children who exceeded a minimal important change (MIC) in the outcome measures,or as suggested by Terwee et al.[12], use the equation MIC/ n to calculate a minimal
important change in group studies. However, as we have discussed in Paper III
regarding the GMFM, but also relevant for the other outcome tools, the distribution of

53
GMFCS-levels and ages in the sample would probably influence the expected MIC
value. Researchers have previously proposed different change scores of the PEDI
[99,118] and the GMFM [99,158] to be clinically relevant. Presently, there is noaccepted value of MIC for the developing children with CP, and for some children
merely the maintenance of motor and everyday function might be relevant goals,
according to the motor growth curves of CP [72,73,159]. The concept of MIC will,
however, be of value when more knowledge of change in different age groups andfunctional levels is gained.

Quality of movement and basic motor abilities
Quality of movement is a controversial term. The aim of attaining “normal”
movement patterns has been a guideline for clinical physiotherapy practice, forinstance in the NDT approach [89], but has been given little or no focus in task
oriented therapies and measurements [7,14,77]. As quality of movement is definedand used differently, it is a demanding construct to operationalize. Different aspectsof movement quality have been addressed by the two measures applied in this thesis,but the measures have been criticized for not having an underlying theoreticalconstruct [160,161], and not including all relevant aspects of quality of movement
like effort, force, timing and velocity [161]. However, to our knowledge at the start ofthis study, these were the only quality of movement measures developed and validated
for the population of children with CP.

The results of this study indicate that quality of movement as measured by the GMPM
improved when basic motor abilities as measured by the GMFM improved (Paper III);hence there seems to be a connection between quality and ability. Further studies areneeded to investigate this connection, whether development of abilities is aprerequisite for development of quality, or vice versa. The impact of quality of

54
movement in CP on e.g. effort and prevention of secondary impairments, is also an
important subject for research.

Manual guidance in therapy is often used as a tool to improve the quality of amovement. Recent theories of motor learning recommend, however, avoiding or
minimizing manual guidance to support the child’s own solution to a movement
problem [135]. When watching video uptakes from the groups, it was evident thatmanual guidance served different purposes; as postural support, to increase variationin movements and joints, and to make the child able to contribute in the group
activities (Appendix, Paper III). The challenge was to provide sufficient, but not too
much guidance. There were examples of children who seemed irritated and disturbed
in their execution of a task by the manual guidance. Hence if manual guidance is tobe given, it should be necessary, adapted, and gradually reduced. The child’s
caregivers who provide the manual guidance need, accordingly, substantial
supervision. However, in the intervention model of this study, there was no request of
normal quality of movement, but variation was a principle. According to the model of
Valvano [136] this could preferably be achieved through active interventions, but also
through passive intervention if necessary.

Content and description of the intervention
When the goals of each child were determined, the two group leaders designed thegroup program based on each child’s goals and also with elements aiming to prevent
secondary impairments, as suggested by Valvano [136]. The “intensity” in the groups
was defined as increased duration and frequency of physiotherapy in a three-weekperiod, as well as a strong “drive” and continuous activity in the groups, with few
breaks [41,42]. The number of repetitions of the activities was challenging to record
due to variations between and within the children. From video uptakes of the groups,

55
it was evident, as discussed in Paper III, that motivation and individually tailoreddemands on the child was a prerequisite for high intensity of training, meaning thatthe child performed many focused and repeated efforts to complete a task. As aconsequence, the training implied continuous adaptation of the tasks and the demandson the child, and in addition exploration of the appropriate level and type of support.
The ability to perform such individualized physiotherapy in a group setting requires
knowledge and experience in the group leaders.

The intervention model employed in this thesis was multifaceted and developed inaccordance with recent theories of motor development and learning [135,136]. It was
inspired by goal-directed and family centered practice [36,162] and by experiencesfrom clinical practice. In the developmental phase of the intervention model, several
meetings with participating parents and professionals were arranged before, during
and after the first pilot groups and after subsequent groups. This strengthened theuser’s perspective of the intervention and resulted in a program that might beconsidered “best practice” as seen by professionals and parents.

The interventions of earlier research have been criticized for being poorly described[27,28,30], and not sorting out the various elements [26]. One objective of Paper III
was to make a careful description of the intervention, beyond general terms describedin previous articles [39,41,42]. An inductive approach was used to describe theintervention from video recordings of a group session. Several ways of describing
interventions were identified [153,163,164], most describing individual interventions.
The “Motor Teaching Strategies Coding Instrument (MTSCI-1)” by Larin [165] wasfound the most appropriate; however, substantial adaptations of the coding scheme
were made in order to capture meaningful elements of a group session. One session ofeach of the groups had been videotaped by professionals from the habilitation unit.
Each video recording lasted between two and three hours, depending on the number

56
of breaks, and whether dressing and undressing were recorded or not. A more united
instruction for the videotaping would have secured that all activities in the groups had
been captured. All video recordings were viewed and broadly described by theinvestigator. One group setting was selected for further description as it was seen as
representative. The selected video uptake was viewed several times and describedaccording to the adapted MTSCI-1 (Appendix, Paper III). The careful description
gives an opportunity to get insight into the content of the intervention and to compare
this type of intervention with other types of interventions.

Assessment tools

The use of different assessment tools in previous research has made comparison of
results difficult across studies [30]. A common toolkit of measures would make itpossible, despite small samples and different designs, to compare results in children
with CP across different functional levels and ages [27,29]. No common toolkit has
been suggested, but the GMFM and PEDI are often used.

The ICF has increasingly been used as a framework for assessment in studies ofchildren with CP [77,118,166]. The model has been considered useful, but not able tocapture all aspects of functioning addressed in habilitation services [167]. The
assessment tools used in the present studies were chosen to reflect dimensions of the
ICF-CY. The GMFM was used to measure activity components of the ICF [2], anditems of the PEDI were mainly used to assess activity and participation components,
but the environmental factor is also inherited in the measure [168]. Depending on thegoal, GAS may reflect all components of the ICF-CY, and gait parameters are part ofthe body function component according to the coding system of ICF-CY. The twoquality of movement measures, GMPM and QUEST, include aspects of movement
patterns as is seen as a part of the body function component. In addition, both

57
measures seem to include aspects of the activity dimension, such as “stability of body
positions” assessed by the GMPM and “hand function” assessed by QUEST. Hence,the quality of movement measures are more difficult to link to a particular componentof the ICF-CY.

When administrating the GMFM and the QUEST, it is possible to adapt the sequenceof the items to what is suitable to the child and accomplish the items in a frame ofplay. The GMPM, however, is less adaptable, as the items have to be administered in
the order of the scoring form and each item is to be repeated three times. This requires
good cognitive skills, cooperativeness and endurance from the child A new versionof the GMPM, called Quality FM [169], is designed for use with children who are in
GMFCS-levels I to III and focuses on quality of movement related to ambulation.
This might be a simpler and more user-friendly assessment tool of movement quality.

There were few missing test data, despite the repeated and comprehensive
assessments. In the last follow-up assessments, however, four of the children weretired and extremely little motivated for the assessment, and the administrator decided
it was too detrimental to the children to complete the GMPM. This might haveinfluenced the results of the last follow-up assessment negatively as the scores of thefirst follow-up assessment were carried forward.

There is no recommendation about the time intervals of assessments in the manuals of
the assessment tools. One may question whether a change in everyday activities could
be apparent after 6 weeks. A longer follow up period would have captured a change
in a longer time-frame; however, the effect of maturation could then have blurred theresults.

58

Strengths and limitations of the intervention study

In intervention studies, the internal validity, meaning the extents to which otherfactors than the intervention cause the change in outcome, and the external validity,
the generalization of the results, is of concern [170,171]. This intervention study
investigated the effectiveness of a clinical intervention offered to children in theirlocal community. As compared to an efficacy approach, where the research
environment ideally is highly controlled, a homogeneous sample is included and thetreatment is highly standardized, an effectiveness study entails more threats to internal
validity [172]. Maturation and learning in the participating children, the small sample
size, anti-spastic medication, lack of standardization of the intervention and the lackof blinding of the assessors are sources of threat to the internal validity in the present
study.

The repeated measures design that was chosen and the stable baseline phase prior to
the intervention phase, control to a large extent for maturation and learning effects inthe children. A power analysis was performed prior to the intervention (Paper III), andthe sample size of the present study was suggested as sufficient. The effect of antispastic medication was investigated, but demonstrated no significant effect (Paper
III). A larger sample, however, might have provided better estimates of results in
subgroups of children, according to functional levels, age groups and effects ofadditional anti-spastic medication.

The group leaders participated in a two-day workshop and were in addition
supervised three times during the three weeks intervention period to secure that thetraining was accomplished according to the principles of the intervention model.
Assessors not involved in the intervention with experience in the use of the59assessment tools, administered and scored the outcome measures. Moreover, all theassessments were videotaped to give the possibility for validation of the clinicalmeasures. Blinded assessors scored video clips of two assessment tools, but thechange in quality of movements were less than in the other assessment tools. The
blinding of the other assessors was difficult and was not performed. This might havebiased the results, and the lack of blinding may be the largest limitation of this study.
Hence, compared to an efficacy study, where Randomized Controlled Trials (RCT)give the highest level of evidence in group research [173], and where internal validity
is strong, the results of this effectiveness study give a weaker indication of causerelationship between the intervention and the outcome.

Despite the methodological strengths, efficacy studies might, however, have
limitations in their ability to estimate treatment effects that can be expected in clinicalsettings [172]. Effectiveness studies evaluate treatment responses in settings that are
more representative of ordinary clinical practice and hence might provide higher
external validity [174]. In this study inclusion criteria were children with CP in pre- or
primary school, where the parents wanted a more intensive training program. The
sample was heterogeneous and seemed representative of children and families whowant periods of more intensive physiotherapy. The intervention was carried out atfour different sites, by different group leaders, within different organizations. In twosites, the intensive training was carried out in the habilitation units, in one site, at a
private physiotherapy institute and in the last site, in a municipality physiotherapy
service. The intervention model is hence applicable in different settings, and theoutcomes may be generalized to other similar intensive training groups for children
with CP aged three to nine years with a diagnosis of spastic or dyskinetic CP. The
study can be considered the first step in providing more evidence based practice inthis field.

60

Ethical considerations
The World Medical Association has developed the Declaration of Helsinki as astatement of ethical principles for medical research involving human subjects [175].
In the declaration it is stated that the well-being of the individual research subject
must take precedence over all other interests, and special precaution is necessary inresearch populations including humans that cannot give consent for themselves, e.g.
children under the age of eighteen.

The research protocol for this study contained descriptions of the ethicalconsiderations involved and how the principles in the Helsinki declaration wereaddressed, and it was approved in advance by the Regional Ethical Committee.
Written informed consent was obtained from the children’s parents before
participation in the study, and effort was made to give the children age appropriate
information about the aims of the studies. The parents might feel an obligation toparticipate in the research study since the intervention was carried out in a groupsetting with close contact between the parents and professionals from the habilitation
units. However, the children could participate in the group training without
participating in the research study as illustrated in one of the training groups. In
addition, the parents seemed to value the thorough examination of the children before
and after the intervention and appreciated a scientific evaluation of the physiotherapy
intervention.

In the intervention study, each assessment lasted for 60 to 90 minutes and the childwas escorted by its parent(s). Effort was made to put as little strain on the children as
possible, e.g. by accomplishing test items in a frame of play, and taking breaks whenneeded. In the methodological studies, the assessments were shorter and each child61
participated in only one assessment. We discussed in advance whether and in whatways the extensive testing before and after the intervention period could put strain on
the children and their parents. Four of the GMPM follow-up assessments in our study
are missing due to the PTs’ judgement that it would be too detrimental for thechildren to carry out the last GMPM assessment. These decisions ensured that thechildren’s well-being was taken care of, and that they were prioritized before
accomplishment of the assessments. Except for this, it is our experience that theassessments and also the parent’s interviews were well within acceptable limits forthe participating children and their parents. On the contrary, the parents appreciatedthe effort made to investigate and document changes in motor function.

To participate in a daily physiotherapy program over weeks may impose strain on
children and parents. The two PTs who conducted each group were experienced andthe training was individually tailored to the child’s current state. It was a goal thatevery child should experience mastery of the demands and get an individually tailored
program within a group frame. The group training was based on play with use ofsongs, stories and rhymes as motivating factors (Appendix, Paper III). However, if theparents or professionals found the intensive training too strenuous, the training would
be terminated. One child with family did leave the intensive training and continued
the ordinary physiotherapy program.

62

Intensive periods of physiotherapy in habilitation services
Children who need prolonged, combined services are entitled to an individual plan.
The purpose of an individual plan is to provide a complete, coordinated andindividually tailored set of services [176]. This study as well as several other studies[34,91,92,95-97] indicate that an increased amount of physiotherapy in a limited timeframe improves basic motor abilities and have positive implications for everyday lifein children with CP and their families. Most models of intensive physiotherapy andmultidisciplinary intensive habilitation developed with funding from the Directorate
of Health and Social Affairs have been implemented in clinical practice. Short,focused, intensive periods of training may be one possible and effective way of
attaining goals important to the child and the family. To secure priority, support andcoordination such periods must be incorporated in the child’s habilitation plan.

In older children and adolescents, the model of intensive physiotherapy presented here
is not sufficient and youths might enjoy and need other types of physical intervention
and leisure activities like strength training and sport. Challenges of becoming a part ofworking life and participating in leisure activities are reported to be more pronounced
in adulthood [177]. A lifetime perspective must hence be incorporated in habilitationand physiotherapy, encouraging the possibility of performing physical activity in a
lifetime perspective [24]. Possible overuse as well as disuse of the muscular andskeleton systems in different phases of the life span should also be of concern[79,178].

63

Conclusions and further research
The objective of this thesis was to gain increased knowledge of measurement
properties of three assessment tools which were considered to be useful as outcome
measures in multicentre studies including children with CP, and further investigate
whether intensive physiotherapy organized as group training affected functioning ofchildren with CP.

The measures revealed good measurement properties and are a supplement to otherresearch methods for the population. In the intervention study, positive, significant
changes in basic motor abilities and self-care, and reduced caregiver’s assistance inmobility and self-care were found after a three-week period of intensive activityfocused, goal-directed group training. The intervention seemed to be motivating andwell tolerated, and a feasible way of improving motor functioning in children withCP.

Further studies are already ongoing, where the outcome measures examined in thisthesis are included. One study uses the electronic walkway as an assessment tool
when children with CP in preschool age participate in intensive, activity-focused andgoal-directed group training. The aim of the study is to examine the relationshipbetween change in gait parameters and change in basic motor abilities. In anotherpilot study including six children with severe CP, change in quality of movements is
examined by scoring the assessment tools from video clips. Studies examining testretest reliability of gait parameters within a longer time frame and studies of
responsiveness to important change will elucidate the value of the walkway as anevaluative measure.

64
Several families want to participate in repeated periods of group training. In the pilot
project, further gain in basic motor abilities was found when children participated in asecond intensive training period. A model containing two periods of intensive
physiotherapy a year for children in pre and first years of primary school, with lessfocus on motor training in the periods in between, is presently being investigated.

A multicentre study in Norway where children diagnosed with CP are followed withsystematic assessments for three years from the time of diagnosis, is presently being
initiated. A prospective study over an extended period of time with careful
registration of the children’s age, functional level and habilitation services, including
periods of intensive training, would give accumulated knowledge of the outcomes ofdifferent approaches regarding the child and the family in a long time perspective. To
conclude, there are many challenges for further research in the field of intensive
training for children with CP.

65

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