Exercise Programs for Children with Cerebral Palsy, Pediatria(2), MPDZ
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Authors:
Olaf Verschuren, BSc, PT
Marjolijn Ketelaar, PhD
Tim Takken, MSc, PhD
Paul J.M. Helders, MSc, PhD, PCS
Jan Willem Gorter, MD, PhD
Cerebral Palsy
Affiliations:
From the Center of Excellence,
Rehabilitation Center “De
Hoogstraat,” Utrecht, the Netherlands
(OV, MK, JWG); Department of
Pediatric Physical Therapy & Exercise
Physiology, University Hospital for
Children and Youth “Het Wilhelmina
Kinderziekenhuis,” University Medical
Center Utrecht, Utrecht, the
Netherlands (OV, TT, PJMH);
Department of Rehabilitation and
Rudolf Magnus Institute of
Neuroscience, University Medical
Center Utrecht, Utrecht, the
Netherlands (MK, JWG); and Partner
of NetChild, Network for Childhood
Disability Research, the Netherlands
(OV, MK, TT, PJMH, JWG).
LITERATURE REVIEW
Exercise Programs for Children with
Cerebral Palsy
A Systematic Review of the Literature
ABSTRACT
Verschuren O, Ketelaar M, Takken T, Helders PJM, Gorter JW: Exercise pro-
grams for children with cerebral palsy: a systematic review of the literature. Am J
Phys Med Rehabil 2008;87:404–417.
Disclosures:
This study was funded by the Dr.
W. M. Phelps Foundation.
The purpose of this literature review, regarding all types of exercise programs
focusing on cardiovascular fitness (aerobic and anaerobic capacity) and/or lower-
extremity muscle strength in children with cerebral palsy (CP), was to address the
following questions: (1) what exercise programs focusing on muscle strength,
cardiovascular fitness, or a combination are studied, and what are the effects of
these exercise programs in children with CP? (2) What are the outcome mea-
sures that were used to assess the effects of the exercise programs? (3) What is
the methodological quality of the studies?
Correspondence:
All correspondence and requests for
reprints should be addressed to Olaf
Verschuren, Rehabilitation Center
“De Hoogstraat,” Rembrandtkade 10,
3583 TM Utrecht, The Netherlands.
We systematically searched the literature in electronic databases up to October
2006 and included a total of 20 studies that were evaluated. The methodological
quality of the included trials was low. However, it seems that children with CP
may benefit from improved exercise programs that focused on lower-extremity
muscle strength, cardiovascular fitness, or a combination. The outcome measures
used in most studies were not intervention specific and often only focused on the
International Classification of Function, Disability and Health body function and
activity level. There is a need to determine the efficacy of exercise programs to
improve the daily activity and participation level of children with CP and increase
their self-competence or quality of life.
0894-9115/08/8705-0404/0
American Journal of Physical
Medicine & Rehabilitation
Copyright © 2008 by Lippincott
Williams & Wilkins
DOI: 10.1097/PHM.0b013e31815b2675
Key Words:
Fitness, Exercise, Cerebral Palsy, Muscle Strength, Review
C
erebral palsy (CP) describes a group of disorders of the development of
movement and posture, causing activity limitation, that are attributed to non-
progressive disturbances that occurred in the developing fetal or infant brain.
1
The motor disorders of CP are often accompanied by disturbances of sensation,
cognition, communication, perception, and/or behavior, and/or by a seizure
disorder.
1
Because of the impairments, many children and adolescents
2
with CP
have at least difficulty with activities such as walking independently, negotiating
stairs, running, or navigating safely over uneven terrain.
3
(The term
childhood
generally refers to the period of 2–12 yrs of age, and adolescence refers to the
period of 13–21 yrs of age.
2
In this review, children and adolescents are referred
404
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Vol. 87, No. 5
to as
children
.) Improving one’s ability to walk or
to perform other functional activities are often the
primary therapeutic goals for children with CP.
4
Exercise refers to planned structured activities
involving repeated movement of skeletal muscles
that result in energy expenditure and seeks to
improve or maintain levels of physical fitness above
the intensity of activities of daily living.
5
Exercise
in children with CP has often been avoided because
of the concern about the negative effect of such effort
on muscle spasticity and children’s movement pat-
terns.
6
Several factors have contributed to a recent
shift in perspective about the use of exercise in chil-
dren with CP. Studies evaluating the effect of exercise
on children with CP reported no adverse effect on
patterns of movement,
7,8
flexibility,
8,9
or spasticity.
10
These findings have influenced current practice.
Most exercise programs for children with CP
are primarily designed for the lower extremity. The
most common functions of the lower extremity
tend to be gross motor activities that involve re-
petitive, reciprocal, coordinated motions of both
extremities to move through space and that often
require little conscious effort once under way.
11
There has been an increased interest in developing
and implementing exercise programs that improve
the cardiovascular fitness (aerobic and anaerobic
capacity) and/or lower-extremity muscle strength
of children with CP.
Two systematic reviews have been published
that examined the effects of strengthening in the
CP population.
12,13
To date, there is no systematic
review that examined all types of exercise programs
focusing on cardiovascular fitness (aerobic and an-
aerobic capacity) and/or lower-extremity muscle
strength in children with CP.
The purpose of the present paper was to sys-
tematically review the literature regarding exercise
programs in children with CP to address the fol-
lowing questions: (1) what exercise programs fo-
cusing on lower-extremity muscle strength, car-
diovascular fitness or a combination are studied
and what are the effects in children with CP? (2)
what are the outcome measures that were used to
assess the effects of the exercise programs? and (3)
what is the methodological quality of the studies?
In many systematic reviews, a meta-analysis is
performed, statistically combining the results of the
various studies into a single estimated effect size.
However, meta-analysis has been described specifi-
cally for randomized controlled trials (RCTs). We
expected most of the studies to be observational stud-
ies, a situation in which the use of meta-analysis is
generally not recommended.
14
Therefore, a qualita-
tive systematic review on the effects of all types of
exercise programs focusing on cardiovascular fitness
(aerobic and anaerobic capacity) and/or lower-ex-
tremity muscle strength in children with CP was
performed.
METHOD
Search Strategy
The following electronic databases were searched
from their respective inceptions to September 2006:
MEDLINE, PubMed, EMBASE, CINAHL, Sports Dis-
cus, Cochrane, and PEDro. Search terms included
subject headings and text words based on (I) cerebral
palsy; (II) exercise (in combination with
strength,
fitness, working capacity, aerobic power, anaerobic
power, endurance, cardiorespiratory physical train-
ing or program
); (III) lower extremity; and (IV) clin-
ical trials. Inclusion criteria were (1) children and
adolescents with CP, (2) intervention (exercise pro-
grams focusing on lower-extremity muscle strength,
cardiovascular fitness, or a combination), and (3)
outcome (measurement of change in body function
and structure, activity, or participation). Exclusion
criteria were (1) doctoral dissertations, (2) reports
published in books, (3) reports published in confer-
ence proceedings, and (4) studies that included chil-
dren with CP as well as children with other diagnoses.
Titles and available abstracts of all items iden-
tified by the electronic searches were scrutinized
by one author (O.V.).
Data Extraction
Included papers were read in full by three
(arbitrarily chosen out of a sample of five for each
paper) independent reviewers with their back-
ground in pediatric physical therapy, exercise phys-
iology or rehabilitation. They all recorded details of
the study design, practice setting, participants, inter-
ventions, outcome measures, results, and conclu-
sions on a data extraction form. Any disagreements or
discrepancies were resolved through discussion and
checking the original papers. Where key information
was not reported, efforts were made to contact the
authors to obtain further details.
Lower-extremity strength training
was de-
fined as prescribed exercises for the lower limbs,
with the aim of improving strength and muscular
endurance, that are typically carried out by making
repeated muscle contractions resisted by body
weight, elastic devices, masses, free weights, spe-
cialized machine weights, or isokinetic devices.
15
Aerobic (fitness) training
was defined as aiming to
improve the cardiorespiratory component of fit-
ness, typically performed for extended periods of
time.
15
Anaerobic (fitness) training
refers to exer-
cises that require large bursts of energy over short
(
May 2008
Exercise Programs for Cerebral Palsy
405
30 secs) periods of time.
15
Mixed (physical fit-
ness) training
was, on the basis of the United States
Department of Health and Human Services,
15
de-
fined as a planned, structured regimen of regular
physical exercise deliberately performed to improve
one or more components of physical fitness (i.e.,
muscle strength, aerobic and anaerobic capacity,
flexibility, and body composition).
Included trials were divided in four categories:
lower-extremity strength training, aerobic training,
anaerobic training, and mixed training. Because in
some studies it can be difficult to distinguish between
the different categories, any disagreements among
the three reviewers were resolved by a discussion
until a consensus was reached.
The outcome measures used in the studies were
categorized by using the International Classification
of Function, Disability and Health (ICF)
16
framework
for the description of health. In this framework, a
person’s disability can be considered in terms of im-
pairment on the body function or structure level,
activity limitations and participation restrictions. In
line with the ICF we consider a person’s functioning
as a dynamic interaction between the health condi-
tion (in this case, CP) and personal and contextual
factors such as the environment.
in internal validity. So, the ideal method for deter-
mining efficacy of a treatment is through RCTs, but
such trials are often difficult to pursue.
19
As a result,
many studies employ less-well-controlled research
designs. The variety of research designs in the liter-
ature mandates use of a method to help evaluate
diverse studies and give weight to their findings. To
determine the degree of confidence that can be placed
in the evidence available about an intervention, a grad-
ing system developed by the American Academy for
Cerebral Palsy and Developmental Medicine (AACPDM)
can be used.
20
For evidence levels, see Table 1.
RESULTS
Search Results
The initial search of the electronic databases
and the manual search of reference lists identified
581 citations. On the basis of title and abstract, we
excluded 559 studies that did not meet our inclu-
sion criteria. Of the remaining 22 articles that were
read full text, 4 articles were excluded because the
intervention did not meet the criteria. Screening of
references of these studies led to another 2 studies
being included. In total, 20 studies remained and
were included in the present systematic review
(Fig. 1): 11 studies on strength training interven-
tions, 5 studies on aerobic training interventions, and
4 studies on mixed training interventions. All infor-
mation was obtained directly form the articles.
No article focused on anaerobic training; there-
fore, the included trials were divided into three cate-
gories: lower-extremity strength training, aerobic
training, and mixed training.
Quality Assessment
Obtained reports were assessed by the same
three reviewers that performed the data extraction
for each specific paper. Empirical studies that met
inclusion criteria were rated for methodological
quality with the PEDro Scale, based on the Delphi
list described by Verhagen et al.
17
With the PEDro
Scale, the following indicators of methodological
rigor were scored independently as either absent
(zero points) or present (one point) by the review-
ers: (1) specification of eligibility criteria, (2) ran-
dom allocation, (3) concealed allocation, (4) prog-
nostic similarity at baseline, (5) subject blinding,
(6) therapist blinding, (7) assessor blinding, (8)
85% follow-up for at least one key outcome, (9)
intention-to-treat analysis, (10) between-group sta-
tistical analysis for at least one key outcome, and
(11) point estimates of variability provided for at
least one key outcome. Points are only awarded
when a criterion is clearly satisfied and reported in
the trial report.
According to the PEDro guidelines, criteria 2
through 11 are used for scoring purposes so that a
score from 0 to 10 can be obtained. The PEDro scale
has shown moderate levels of interrater reliability
(intraclass correlation coefficient
Intervention Characteristics and Effects
Lower-Extremity Strength Training
Table 2 shows the characteristics of the 11
included strength training interventions
9,21–30
in
children with CP, varying in age from 6 to 20 yrs.
Exercise interventions lasted for 6 wks in seven
trials,
21–24,26,27,30
8 wks in three trials,
9,25,29
and 9
mos in one trial.
28
All exercise frequencies were
three times a week. Nine programs were individu-
ally based,
9,21–25,27,28,30
and two programs were
group programs.
26,29
In six studies,
22–24,26,28,30
the
supervisor was a physical therapist or parent/part-
ner, in four studies
9,21,25,27
the supervisor was not
described, and in one study the supervisor was a
research assistant.
29
All studies reported outcome results on the ICF
body structure and function level, and eight stud-
ies
22,23,25–30
reported on the activity level. In two
RCTs,
23,24
small improvements in performance on
tests of muscle strength were found for the experi-
mental group. In one RCT,
29
only significant change
in the perception of body image and a more upright
posture were found. Another RCT
28
found no signifi-
0.54; 95% confi-
dence interval (CI), 0.39 – 0.71).
18
To improve the
reliability of this scale, any disagreement between the
reviewers were resolved by discussion with an inde-
pendent reviewer until consensus was reached.
Evidence Assessment
RCTs are the best method to ensure that any
differences in outcome were attributable to the treat-
ment and not other factors. They give one confidence
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Verschuren et al.
Am. J. Phys. Med. Rehabil.
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TABLE 1
American Academy for Cerebral Palsy and Developmental Medicine (AACPDM) levels
of evidence
Level
Nonempirical
Group Research
Outcomes
Research Single-Subject Research
I
Randomized controlled
trial
All-or-none case series
N-of-1 randomized
controlled trial
II
Nonrandomized controlled
trial
Prospective cohort study with
concurrent control group
Analytic survey ABABA design
Alternating treatments
Multiple baseline across
subjects
III
Case–control study
Cohort study with historical
control group
ABA design
IV
Before-and-after case series
without control group
AB design
V
Descriptive case series or
case reports
Anecdote
Expert opinion
Theory based on physiology,
bench, or animal research
Common sense/first principles
cant changes at all. Five trials reported significant im-
provements in tests of muscle strength after strength
training programs lasting 6 – 8 wks.
9,21,22,25,27
Dodd et
al.,
23,24
Mac Phail et al.,
25
Morton et al.,
27
Unger et al.,
29
and Patikas et al.
28
were the only studies that used
long-term follow-up measurements, which varied from
4 wks up to 1 yr. Only three studies concluded that the
gained benefits on muscle strength,
23,25,27
gross motor
function,
23,25,27
scholastic competence and social accep-
tance,
24
and muscle tone
27
of training were maintained.
capacity, and nonsignificant improvements on an-
aerobic capacity, muscle strength, and fat mass,
was found. One study
35
investigated the activity
level, measured with the Gross Motor Function
Measure (GMFM; dimension D: standing; and E:
walking, running, jumping) of the subjects. Three
trials
31,32,34
reported statistically significant im-
provements of aerobic capacity.
Physical activity ratio,
31
fat mass,
31
anaerobic
capacity,
31
and the energy expenditure index
35
were
studied as well. No statistically significant changes
were found in the included studies.
In two studies, follow-up measurement took
place.
31,33
Both studies, including one RCT,
31
con-
cluded that inactivity during summer vacation (ap-
proximately 3 mos) significantly reduced the aero-
bic capacity.
Aerobic Training
Table 3 shows the results of the five stud-
ies
31–35
that focused the intervention on aerobic
exercise in children with CP. They varied in age from
7 to 20 yrs (except one subject in the study performed
by Berg et al.
33
who was 25 yrs old.) Exercise inter-
ventions varied from 6 wks to 16 mos, with exercise
frequencies varying from two to four times a week for
20 – 45 mins. The intensity of the training programs
varied from exercise at the anaerobic threshold
point,
32
training at an intensity of
70% of the heart
rate reserve
31
to various loads based on the maximal
cycling capacity.
33
One study did not describe the
intensity of the training.
34
Two programs
31,34
were
group programs, and three
32,33,35
were individually
based programs. In two studies,
33,34
the supervisor
was a physical therapist, and in three studies
31,32,35
the supervisor was not described.
All included studies, of which one was an
RCT,
31
reported results on the ICF level of body
function. In the RCT performed by Van den Berg-
Emons et al.,
31
Mixed Training
In Table 4, the results of four studies that
examined the effects of mixed training interven-
tions
36 –39
in children with CP, varying in age from
4 to 20 yrs, are shown. Exercise interventions varied
from 4 wks to 6 mos. Exercise frequencies varied
from two to three times a week and from 30 to 60
mins. All programs were group programs. However,
one study
38
combined the group program with an
individual swimming program. In three stud-
ies,
36,37,39
the supervisor was a physical therapist; in
one study,
38
the supervisor was not described.
All included studies reported results on the level
of body function. Two studies
36,39
found a significant
increase in muscle strength. One study
38,39
a significant increase in aerobic
reported
May 2008
Exercise Programs for Cerebral Palsy
407
FIGURE 1
Flow chart of included studies.
a significant increase in vital capacity, and another
study
36
reported no significant change in heart rate
and energy expenditure. The study performed by
Darrah et al.
36
showed a significant increase for self-
perception of physical appearance. Two studies inves-
tigated the effects on the level of activity.
38,39
Blundell
et al.
39
reported a significant increase in stride length,
and mixed results for walking speed. Darrah et al.
36
found a significant change in walking speed. There
were two studies that used a follow-up measure-
ment.
36,39
Blundell et al. concluded that all training
improvements were maintained after 8-wk follow-
up.
39
The results found by Darrah et al. show that the
significant changes in muscle strength were main-
tained 10 wks after completion of the program.
36
mometer,
25
the Cybex,
31
the spring scale,
9
the Lat-
eral Step-up Test,
39
the Motor Assessment Scale
(Sit-to-Stand),
39
a 10-repetition maximum,
24
and
the minimum chair height test
39
were used.
Spasticity and Muscle Tone
To measure spasticity and muscle tone the
modified Ashworth scale of Spasticity
25,28
and the
resistance to passive stretch
27
were used in all
included studies.
Fat Mass
Fat mass was measured using skinfold mea-
surement in one study.
31
Outcome Measures
The outcomes that were used in all included
studies were categorized by using the ICF
16
frame-
work for the description of health and can be ap-
preciated in Table 5.
Fitness Measures
The energy expenditure index,
22,25,26,28,35,36
which is defined as walking heart rate minus rest-
ing heart rate, divided by walking speed, expressed
in beats per meter,
40
was used to quantify the
energy consumed during walking. To measure the
aerobic capacity, the cycle ergometer (arm and leg)
was used in five studies.
31–34,36
One study
31
inves-
tigated the effects of an aerobic-focused interven-
tion on anaerobic performance, using the Wingate
Body Function and Structure
Muscle Strength
To measure muscle strength, the handheld
dynamometer,
21–23,27,35,36,39
the isokinetic dyna-
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