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The natural history of

gross motor
development in
children with cerebral
palsy aged 1 to 15 years
E Beckung* PT PhD, Department of Physiotherapy,
Institution of Neuroscience and Physiology, Sahlgrenska,
Gteborg University, Sweden.
G Carlsson PhD, Department of Paediatrics, University of
Schleswig-Holstein/Campus Kiel, Germany.
S Carlsdotter Research Assistant;
P Uvebrant MD PhD, The Queen Silvia Childrens
Hospital/Sahlgrenska, Gteborg University, Sweden.
*Correspondence to the first author at Department of
Physiotherapy, Sahlgrenska University Hospital, Hus A4,
SU/Mlndal, SE-431 80 Mlndal, Sweden.
E-mail eva.beckung@vgregion.se

The aim of this study was to explore motor development in


children with cerebral palsy (CP) using developmental curves
for CP, subtypes, and the five severity levels of the Gross Motor
Function Classification System (GMFCS). The Gross Motor
Function Measure (GMFM) and the GMFCS were applied to 317
children (145 females, 172 males) with CP, aged between 1 and
15 years. The CP type distribution was spastic diplegia in 157
(49%), spastic hemiplegia in 101 (33%), spastic tetraplegia in
11 (3%), dyskinesia in 38 (12%), and ataxia in 10 (3%).
Forty-five physiotherapists were trained in the GMFM and
intra- and interrater reliability was tested. The GMFM was
measured prospectively every 6 months up to the age of 4 years
and once a year thereafter. Developmental curves were
constructed for 258 children with spastic CP. About threequarters of the children at GMFCS Level I reached 90% of the
maximum GMFM score at 5 years of age. The performance
peaked at 7 years of age. Children at GMFCS Level II reached
90% at a median age of 5 years, which was also the upper limit,
reached by about three-quarters at 7 years of age. The majority
of children at GMFCS Level III reached 80% of the GMFM by 7
years of age and most of the children at GMFCS Level IV
reached 30% at 5 years and remained there. The median score
for children at GMFCS Level V was 20%. The intra- and
interrater reliability for the GMFM 88 among physiotherapists
were Spearmans rank correlation coefficient 0.91 and 0.99
respectively. There were 931 measurements with a median of 2
(111) per child. The gross motor development was
demonstrated for the five GMFCS levels in children with spastic
CP. These kind of curves may be useful for monitoring and
predicting motor development, for planning treatment, and for
evaluating outcome after interventions.

Gross motor development in children is commonly described


as the acquisition of motor milestones such as unsupported sitting, crawling, and walking. A child with cerebral palsy (CP) will
be recognised by delayed gross motor development and the
presence of abnormal movement and posture patterns.1 CP
occurs in about 2 in every 1000 live births, making it the most
common cause of physical disability in childhood.2,3 In a population-based study, Himmelmann et al. reported about half of
the children to have mainly motor function affected, whereas
the remainder also had accompanying major impairments
adding to the disability, which affected several areas of activity
and participation.46
Gross motor development in CP can be described and
predicted in terms of motor growth curves using the Gross
Motor Function Measure (GMFM)7,8 and the Gross Motor
Function Classification System (GMFCS)9 in combination.10
Both the measure GMFM, and the classification GMFCS, have
been found to be valid and reliable tools that are widely used
in clinical work and research involving children with CP. The
GMFM is frequently used to evaluate the effects of various
interventions in children with CP, such as intensive physiotherapy,1114 botulinum toxin treatment,15 selective dorsal
rhizotomy,16 and orthopaedic surgery.1719 The GMFM is a
test specially designed and validated for measuring gross
motor function and the change over time in children with CP.
The test contains 88 items of gross motor function distributed over five dimensions: lying and rolling; sitting; crawling
and kneeling; standing and walking; and running and jumping.7 A percentage score was calculated for each dimension
and an overall score was obtained as the quotient of the sum
of percentages divided by the sum of dimensions. The GMFM
is constructed to measure quantitative aspects, i.e. how
much children can do, not the quality of their performance.
All the items are considered to be achievable by 5-year-olds
with normal motor function.
The GMFCS was developed to provide a standardized
classification of the patterns of motor disability in children
with CP aged 1 to 12 years.9 The focal point is the childs selfinitiated movements. The GMFCS is based on a five-level
ordinal grading in which the distance between the levels
should not be considered equal and it should be remembered
that children with CP are not expected to be equally distributed between the levels (Table I). To differentiate between
the GMFCS levels, interest focuses on functional limitations
and the need for assistive technology, including mobility
devices and wheeled mobility rather than quality of movement.9 When it comes to the reliability of the GMFCS, interrater
reliability of 0.75, reliability of 0.93 from chart reviews, established content validity, and very good predictive validity have
been reported for children over the age of 2 years.9,20,21 The
validity of the GMFM and GMFCS and the construction of
motor growth curves have contributed valuable knowledge to
help us understand how children with CP develop gross motor
ability and can thus serve as a reference when evaluating interventions.10 Furthermore, Rosenbaum et al.10 were able to present prognostic information on walking ability to parents of
children with CP based on the Ontario Gross Motor Growth
Curves (OMG). In the Swedish population-based studies of the
prevalence of CP, we found that the GMFCS, when applied to
the various CP types, further refined the potential to delineate
impairment severity subgroups.4 By adding information about
CP subtype, qualitative aspects of movement could also be

Developmental Medicine & Child Neurology 2007, 49: 751756 751

taken into account, in addition to the level of motor function. We wanted to study our data in comparison with the
OMG work to see if the results were similar and if adding the
CP subtype would differentiate the curves.
The aim of this study was to analyze the way gross motor
development, according to the GMFM, progresses over time in
children with CP by constructing gross motor developmental
curves for the five GMFCS levels for all children with CP and
for the spastic subtypes of hemiplegia and diplegia.
Method
PARTICIPANTS

All children with the diagnosis of CP are included on the west


Sweden CP register. Of the 409 children with CP born between
1981 and 2002, 317 participated in this study, representing
78% of the children included in the regional CP register during
the period. All children with CP attending a habilitation centre
were invited by their physiotherapist to join the study. CP was
defined as a group of disorders, i.e. it is an umbrella term; it is
permanent but not unchanging; it involves a disorder of movement and/or posture and of motor function; it is due to a nonprogressive interference/lesion/abnormality; this interference/
lesion/abnormality is in the developing immature brain.2 The
Hagberg classification of CP subtypes was applied.22
PROCEDURE

The GMFM assessment and the GMFCS classification was


applied.
Educational and training sessions were run for the paediatric physiotherapists in order to learn to perform the GMFM
assessment. Subsequently, an intra- and interrater reliability
study comprising 45 paediatric physiotherapists working in
the area was conducted. The GMFCS classification was made

Table I: The Gross Motor Function Classification System9


Level I: Walks without restrictions, limitations in more advanced
gross motor skills
Level II: Walks without restrictions, limitations walking outdoors
and in the community
Level III: Walks with assistive mobility devices, limitations walking
outdoors and in the community
Level IV: Self-mobility with limitations, children are transported or
use power mobility outdoors and in the community
Level V: Self-mobility is severely limited, even with the use of
assistive technology

and the GMFM was measured prospectively every 6 months


in children with CP up to the age of 4 years and then once a
year after their fourth birthday. A standardized data collection form was developed.
Physiotherapists from 14 habilitation centres participated
and reported their GMFM data to a common database at the
regional habilitation centre at The Queen Silvia Childrens
Hospital/Sahlgrenska University Hospital in Gteborg, Sweden.
The content of the database and definitions of variables
were discussed until consensus was reached. Children with a
diagnosis of CP were included and no exclusion due to treatment was made. The treatments used were physiotherapy,
orthopaedic surgery, the use of splints and orthoses, botulinum toxin injections, selective dorsal rhizotomy, and intrathecal baclofen treatment.
The study was approved by the Ethics Committee at the
Medical Faculty at the University of Gteborg. Informed consent was obtained from the parents.
STATISTICS

For reliability tests Spearmans rank coefficient (rho) was


used. The GMFM and GMFCS data were presented in a
descriptive form with median values.
Results
The intra- and interrater reliability for the GMFM 88 among
physiotherapists were rho 0.91 and 0.99 respectively.
The 317 children were 1 to 15 years of age. The dominant
CP type was the spastic subtype comprising 157 (49%) children with diplegia, 101 (33%) with hemiplegia, and 11 (3%)
with tetraplegia. Dyskinesia was present in 38 (12%) children and ataxia in 10 (3%).
The distribution of CP subtypes with corresponding GMFCS
levels in the 317 participants is shown in Table II. The study
population represented 78% (317 of 409) of the children with
CP included on the regional CP register during the period.
All children with CP attending a habilitation centre were invited
to join the study but 87 families did not want to participate.
The children who did not participate in the study functioned
at GMFCS Levels I (9%), II (8%), and V (5%).
A total of 931 GMFM measurements were reported in 317
children. Of these, 12% of the measurements were performed
in the under-2 year band, 29% in the 2 to 4-year band, 23% in
the 4 to 6-year band, and 35% in the 6 to 12-year band of the
GMFCS (Table III). The median number of measurements
was 2 (range 111). In 62 children 5 measurements were
recorded.

Table II: Distribution of cerebral palsy (CP) subtypes by Gross Motor Function Classification System
(GMFCS) levels in 317 patients with CP
CP subtype

Spastic hemiplegia
Spastic diplegia
Spastic tetraplegia
Dyskinesia
Ataxia
Total

752

I
n (%)

II
n (%)

III
n (%)

GMFCS level
IV
n (%)

V
n (%)

Total
n (%)

82 (81)
44 (28)
0
3 (8)
4 (40)
133(42)

14 (14)
44 (28)
0
1 (3)
6 (60)
65 (21)

2 (2)
25 (16)
0
7 (18)
0
34 (11)

3 (3)
38 (24)
0
8 (21)
0
49 (15)

0
6 (4)
11 (100)
19 (50)
0
36 (11)

101 (100)
157 (100)
11 (100)
38 (100)
10 (100)
317 (100)

Developmental Medicine & Child Neurology 2007, 49: 751756

100
90
80
70
60
50
40
30
20
10
0

7 8 9 10 11 12 13 14 15
Age (y)

100
90
80
70
60
50
40
30
20
10
1

7 8 9 10 11 12 13 14 15
Age (y)

Figure 1c: Gross Motor Function Measure scores in patients


with spastic diplegia at 1 to 15 years of age. Gross Motor
Function Classification System Level III.

100
90
80
70
60
50
40
30
20
10
0

7 8 9 10 11 12 13 14 15
Age (y)

Figure 1b: Gross Motor Function Measure scores in patients


with spastic diplegia at 1 to 15 years of age. Gross Motor
Function Classification System Level II.

Gross Motor Function Measure score (%)

Gross Motor Function Measure score (%)

Figure 1a: Gross Motor Function Measure scores in patients


with spastic diplegia at 1 to 15 years of age. Gross Motor
Function Classification System Level I.

Children at GMFCS Level II (n=65) reached 90% of the


GMFM at a median age of 5 years, which was also the upper
limit, reached by 75% of the children by the age of 7. The
group mainly comprised children with diplegia (n=44;
Fig. 1b).
The majority of children at GMFCS Level III (n=34)
reached 80% of the GMFM by the age of 7 years. This group
also mainly comprised children with diplegia (Fig. 1c).
The maximum performance for children at GMFCS Level IV
(n=49) was 70%, but the majority only reached the 30% level
at the age of 4 to 5 years and then remained there. This was
also true for the children with diplegia (Fig. 1d).
The median score for children at GMFCS Level V (n=36)
was 20%, which was reached at the age of 7 years. The group

Gross Motor Function Measure score (%)

Gross Motor Function Measure score (%)

Half the children at GMFCS Level I (n=133) had obtained


90% of the total 100% GMFM scores at 4 years of age and more
than 75% had done so at the age of 5. Their performance
seemed to level off at the age of 7 years. Between the ages of 5
and 9 years, the scores could range from 50 to 100%.
When the children with diplegia at GMFCS Level I (n=44)
were described separately, the GMFM scores were more
homogeneous, especially after the age of 6 years (Fig.1a),
when the GMFM scores were above 90% and remained so up
to the age of 15 years.
Children with hemiplegia at GMFCS Level I (n=82) almost
reached the 100% score, but the range for children aged 6 to
10 years was fairly wide and varied between 50% and 100%
(Fig. 2a).

100
90
80
70
60
50
40
30
20
10
0

7 8 9 10 11 12 13 14 15
Age (y)

Figure 1d: Gross Motor Function Measure scores in patients


with spastic diplegia at 1 to 15 years of age. Gross Motor
Function Classification System Level IV.

Gross Motor Development in Subtypes of Cerebral Palsy E Beckung et al. 753

mainly represented children with dyskinesia (n=19) and


spastic tetraplegia (n=11).
Discussion
GMFM scores from 317 children with CP aged between 1 and
15 years were reported, and presented as gross motor developmental curves for spastic CP subtypes for the GMFCS levels. The study is not longitudinal but rather cross-sectional as
there were only about two measurements per child.
However, in 20% of cases there were more than five measurements per child. The corresponding mean number of measurements was four per child in the OMG study.10
More than 60% of the children were able to walk without
aids or in younger children were at GMFCS Levels I and II.
This was in accordance with previous reports from the
Swedish CP register.4,23 In this study, as in the Canadian and
an Israeli study, gross motor development reached a plateau
at the age of 6 to 7 years.10,24
To our knowledge, this study seems to be the first attempt
to create motor developmental curves for the CP subtypes.
The CP subtypes in this study differed to a small extent
from our CP register. Children at GMFCS Levels I, II, and V

Table III: Number of measurements per age band and Gross


Motor Function Classification System (GMFCS) level in 317
patients with cerebral palsy
Age band
II
n=65

36
90
96
148
370

31
67
51
71
220

GMFCS level
III
IV
V
n=34 n=49 n=36
9
37
24
41
111

15
31
25
51
122

20
47
22
19
108

Total
n=317
111
272
218
330
931

Gross Motor Function Measure score (%)

Gross Motor Function Measure score (%)

<2y
24y
46y
615y
Total

I
n=133

100
90
80
70
60
50
40
30
20
10
0

7 8 9 10 11 12 13 14 15
Age (y)

Figure 2a: Gross Motor Function Measure scores in patients


with spastic hemiplegia at 1 to 15 years of age. Gross Motor
Function Classification System Level I.

754

were fewer than expected but the sample was considered to


be representative for gross motor function distribution, as
this was very similar to the distribution of GMFCS levels in
the population-based study of 367 children reported by
Himmelmann et al.,4 with 32% at Level I, 29% at Level II, and
8%, 15%, and 16% at Levels III, IV, and V respectively. The distribution was also similar to that in another populationbased Swedish study of 175 children by Nordmark et al.,25
with Level I in 41%, Level II in 21%, Level III in 13%, Level IV
in 11%, and Level V in 14%. A sample of 657 children in a
Canadian community-based study10 was selected to be evenly distributed across the levels: Level I 28%; Level II 12%;
Level III 19%; Level IV 21%; and Level V 20%. In the two
Swedish samples, 69% and 75% of the children respectively
were able to walk with or without walking aids compared
with 59% in the Canadian study, which means that the distribution of children with CP per GMFCS levels in the Canadian
study is not representative for the CP population as in the
Swedish studies.4, 25
Our results showed great variability in GMFM scores between
the different GMFCS levels, which was also the case in the
Canadian study.10
A large number of paediatric physiotherapists contributed
measurements in this study. In spite of this, the reliability of the
GMFM was very good and similar to previous reports,7 possibly
due to the educational and training sessions before the study.
Considering the classification of CP types, no reliability
study has been performed but the Hagberg classification has
been consistently used during many years in western
Sweden. Gorter et al.26 report discrepancies in classification
and a poor correlation to GMFCS levels using limb distribution in addition to CP type classification in a Canadian sample, but a classification including limb distribution has not
been used in Sweden.
Some children at GMFCS Level I appeared to have gross
motor development that did not differ very much from
population norms when it came to the achievement of

Developmental Medicine & Child Neurology 2007, 49: 751756

100
90
80
70
60
50
40
30
20
10
0

7 8 9 10 11 12 13 14 15
Age (y)

Figure 2b: Gross Motor Function Measure scores in patients


with spastic hemiplegia at 1 to 15 years of age. Gross Motor
Function Classification System Level II.

motor milestones. To be able to measure the effects of interventions in these children and to predict gross motor development, complementary measures are needed such as the
Gross Motor Performance Measure that looks at qualitative
aspects of gross motor function or the BruininksOseretsky
Test of Motor Proficiency.27,28
At the other end of the spectrum, the children with severe
CP scored very low and did not change much, which was
obvious in this study where children at GMFCS Level V
reached only 20%. The GMFM does not discriminate at this
level. To evaluate interventions in these children, the outcome measure needs to target the goal of the intervention
more specifically than is possible using the GMFM described
in the study by Bower et al.11
For many children, there appeared to be a maximum
achievement at the age of 9 to 10 years in this study. After this
age, there was great variability in the data, due partly to the
small number of measurements. We would need longitudinal data for a large group of adolescents to study whether
and when gross motor deterioration will appear. A deterioration in walking ability in adulthood would be expected in CP
due to pain, fatigue, and a lack of adapted physical activity.
However, Andersson et al.29 report unchanged walking ability from childhood to the age of 20 to 58 years in half of participants with hemiplegia and in one-third of participants with
diplegia. In a Norwegian study of adults with CP, 70% of the participants with hemiplegia and 43% of participants with diplegia, self-reported an improved or unchanged walking ability.30
The majority of the children in this study performed at
GMFCS Levels I and II and mainly represented children with
spastic hemiplegia or diplegia. The gross motor development did not differ very much between these two CP subtypes at these levels. However, the GMFM score range was
wider for children with hemiplegia at GMFCS Level I compared with children with diplegia at the same level.
No children with hemiplegia were at GMFCS Levels IV or
V, whereas all the children with spastic tetraplegia and most
with dyskinesia performed at GMFCS Level V. There were,
however, some children with dyskinesia who performed at
higher levels. In this study, there were very few children with
ataxia and their results did not differ very much from those of
other CP types at corresponding GMFCS levels.
The CP subtype alone was not sufficient to predict the
gross motor development in a specific child as Gorter et al.26
report, especially in the case of children with spastic diplegia,
who were represented at all five GMFCS levels. This would
support the recommendation proposed by Rosenbaum et al.
to use the Surveillance of Cerebral Palsy in Europe classification of bilateral and unilateral spastic CP.6
In this study, as in a preliminary report on 211 children,31
we have described specific gross motor development curves
for the spastic hemiplegic and diplegic CP subtypes. These
CP type-specific curves offer more precise information about
the expected gross motor development than the CP type or
GMFCS level separately, and they may perhaps serve as reference norms in follow-up investigations after interventions.
Conclusion
If further developed, these general and CP type-specific
gross motor development curves may be used to estimate the
natural history of motor development in children with different types of CP at the five different GMFCS levels. The

GMFM curves can also help doctors, physiotherapists, and


parents to plan treatment and evaluate outcome after interventions by comparing the childs development with the
expected change over time.10
Accepted for publication 21st June 2007.
Acknowledgements
The Regional Research and Development Council, Vstra Gtaland,
the Folke Bernadotte Research Foundation, and the Sven Jerring
Research Foundation. We would like to thank all the
physiotherapists who participated in this study.
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European Academy of
Childhood Disability
20th Annual Meeting
Early Diagnosis Implies
Early Intervention
Zagreb June 57 2008

The programme of the meeting will address the theme of Early diagnosis implies early intervention
which we believe might ensure better functional recovery.
It will be a joint meeting of the EACD and European Paediatric Neurology Society (EPNS). Being able to
benefit from the expertise of both societies certainly will result in high level scientific contributions.
All topics of the programme will be presented by keynote lectures, parallel sessions, lectures, dedicated
papers, and posters, as well as workshops and instructional courses.
Satellite symposia arranged by EACD/EPNS will precede the official programme of the meeting. Also,
Parent Associations will contribute to the pre-conference events enriching the programme by their satellite
symposia.
Members are invited to apply to present an Instructional Course or an Oral Presentation on a specific
topic in the scientific programme. New to the programme is the session Meet the expert.
We look forward to welcome you to Zagreb and Croatia in 2008!
On behalf of the Organizing Committee,
Vlatka Mejaski-Bosnjak
For more information see the website: www.eacd2008.hr

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