Professional Documents
Culture Documents
1 Department of Rehabilitation Medicine, EMGO Institute for Health and Care Research, VU University Medical Center, Amsterdam, the Netherlands. 2 Adelante, Valkenburg,
the Netherlands. 3 Rehabilitation Centre Heliomare, Wijk aan Zee, the Netherlands. 4 Mytyl and Tyltylschool and Rehabilitation Center Amsterdam, the Netherlands.
Correspondence to Dr Annet J Dallmeijer, at VU University Medical Center, Department of Rehabilitation Medicine, PO Box 7057, 1007 MB Amsterdam, the Netherlands. E-mail: a.dallmeijer@vumc.nl
PUBLICATION DATA AIM To evaluate the effectiveness of functional progressive resistance exercise (PRE) strength
Accepted for publication 17th November 2009. training on muscle strength and mobility in children with cerebral palsy (CP).
Published online 28th January 2010. METHOD Fifty-one children with spastic uni- and bilateral CP; (29 males, 22 females; mean age
10y 5mo, SD 1y 10mo, range 6y 0mo–13y 10mo; Gross Motor Function Classification System
LIST OF ABBREVIATIONS levels I–III) were randomized to the intervention group (n=26) or the control group (n=25, receiving
LSU Lateral Step-Up test usual care). The intervention group trained for 12 weeks, three times a week, on a five-exercise
PRE Progressive resistance exercise circuit, which included a leg-press and functional exercises. The training load progressively
STS Sit-To-Stand test increased based on the child’s maximum level of strength, determined by the eight-repetition
maximum. Muscle strength (measured with hand-held dynamometry and a six-repetition maxi-
mum leg-press test), mobility (measured with the Gross Motor Function Measure, two functional
tests, and a mobility questionnaire), and spasticity (measured by the appearance of a catch) were
evaluated before, during, directly after, and 6 weeks after the end of training by two blinded
research assistants.
RESULTS Directly after training, there was a statistically significant effect (p<0.05) on muscle
strength (knee extensors +12% [0.56N ⁄ kg; 95% confidence interval {CI} 0.13–0.99]; hip abductors
+11% [0.27N ⁄ kg; 95% CI 0.00–0.54]; total +8% [1.30N ⁄ kg; 95% CI 0.56–2.54]; six-repetition maxi-
mum +14% [14%; 95% CI 1.99–26.35]), but not on mobility or spasticity. A detraining effect was
seen after 6 weeks.
INTERPRETATION Twelve weeks of functional PRE strength training increases muscle strength up
to 14%. This strength gain did not lead to improved mobility.
Cerebral palsy (CP) describes a group of disorders in the uncontrolled studies which showed that strength training can
development of movement and posture, causing activity limi- improve lower-limb muscle strength in children with CP
tation, that are attributed to non-progressive disturbances without increasing spasticity.3,4 Although studies have shown
that occurred in the developing brain.1 It is the most com- there to be sufficient evidence for its effectiveness on muscle
mon cause of movement disability in childhood. Children strength, these effects are probably overestimated because of
with CP may experience a variety of impaired muscle func- the low methodological quality of the studies.5,6 The few
tions, such as spasticity, muscle weakness, and loss of selective uncontrolled studies that have evaluated the effectiveness of
motor control. Although all impaired muscle functions limit strength training on mobility outcomes in children with CP
the performance of daily life activities and participation in a have reported limited effectiveness.3,7 Three recently pub-
child with CP, a recent study has shown that muscle weak- lished randomized clinical trials8–10 evaluated both muscle
ness showed a stronger association with mobility limitations strength and mobility in children with CP, but conflicting
in children with CP than spasticity.2 Strength training for results were reported.11,12 One of the explanations for these
these children is, therefore, expected to improve or maintain conflicting results could be the high variability in training
their mobility. characteristics, such as type of training, training intensity, and
Until recently, strength training in children with CP was training period. To be successful, strength training must be
discouraged as it was assumed that it would increase spastic- individualized, and involve a progressive increase in intensity,
ity. However, this was not supported by the results of earlier thereby stimulating strength gains that are greater than those
ª The Authors. Journal compilation ª Mac Keith Press 2010 DOI: 10.1111/j.1469-8749.2009.03604.x e107
associated with normal growth and development (i.e. ‘over- What this paper adds
load’). This is known as progressive resistance exercise (PRE). • Insight into the true effectiveness of strength training in children with CP, using
According to the US National Strength and Conditioning a blinded randomized controlled study.
Association, children should be trained at an 8- to 15-repeti- • It evaluates the effectiveness of a strength training program in which appropri-
tion maximum, which is the number of repetitions that can be ate guidelines for strength improvements are applied, based on the key ele-
ments of PRE.
completed before fatigue.13 To stimulate strength progres- • It shows that PRE based strength training is effective on muscle strength
sion, the amount of resistance should be increased as strength improvement, but not on mobility improvement.
increases. Most previous strength training studies did not
present full details of their training programme, or used insuf- during training (T1; a subset of outcomes), directly after train-
ficient intensities. Adequate interpretation of study results is ing (T2), and at the 6-week follow-up (T3). The measurement
not possible when key principles of the PRE are not followed. points of main interest were T2 and T3.
Further, earlier reported training programmes lasted 5 to
8 weeks but some have argued that a 6-week training pro- Intervention
gramme may not be long enough to induce functional adapta- The control group continued their conventional physical
tions.14 Therefore, the purpose of this study was to evaluate therapy programme. Children in the control group received
the effectiveness of a sufficiently long and intensive 12-week one to three sessions a week. The training group followed a
functional PRE strength training programme, by applying 12-week progressive functional strength-training programme
current guidelines.13 We hypothesize that this training would for the lower extremities. This replaced their conventional
improve muscle strength and, subsequently, lead to improved physical-therapy programme. Training was given three times
mobility in children with CP, without increasing muscle a week for 45 to 60 minutes at school in small groups (four
spasticity. or five children). Each training session consisted of one
exercise on a child-adapted leg-press (Enraf Nonius, Delft,
METHOD the Netherlands) and three functional exercises (sit-to-stand,
Participants lateral step-up, half knee-rise), loaded with a weighted vest.
Ambulatory children with spastic unilateral or bilateral CP1 During the training, intensity progressively increased, based
were pre-selected by a paediatric physiatrist. The inclusion on repeated estimation of the eight-repetition maximum. A
criteria were (1) age between 6 and 13 years, (2) able to detailed description and characteristics of the PRE training
accept and follow verbal instructions, (3) able to walk inde- programme can be found elsewhere.15
pendently indoors, with or without walking aids (Gross
Motor Function Classification System [GMFCS] levels I–III), Outcome measures
and (4) able to participate in a group training programme. The primary outcome, ‘mobility’, was measured with the 66-
The exclusion criteria were unstable seizures, any treatment item version of the Gross Motor Function Measure (GMFM-
for spasticity or surgical procedures from 3 months (for botu- 66), the Sit-To-Stand (STS) test, and the Lateral Step-Up
linum toxin type A injections) to 6 months (for surgery) (LSU) test. The GMFM-66 is a standardized observational
before the study (or planned during the study period), any instrument, based on interval scaling.16 It reports on the
change in medication expected during the study period, and child’s level of capacity, and has been internationally validated
suffering from any other diseases that interfered with physical for evaluating change in the gross motor activities of children
activity. with CP. The STS test assesses the number of repetitions (full
movement from standing up to sitting down) that the child
Study design can perform in 30 seconds on a child-sized chair with a
This study, performed between April 2007 and May 2008, had height-adaptable seat (no backrest, no armrest). The LSU test
a single-blind randomized controlled design. It was conducted assesses the number of repetitions (full movement from step-
at three special schools for children with physical disabilities ping up to stepping down) that the child can perform in
in the Netherlands (Amsterdam, Wijk aan Zee, Valkenburg). 30 seconds on a 21cm (GMFM I–II) or 11cm (GMFCS III)
The study protocol was approved by the medical ethics com- step. Subjectively reported mobility was assessed with the 28-
mittee of the VU University Medical Center in Amsterdam. item version of the Dutch mobility questionnaire (MobQues-
Full written informed consent was obtained from all parents 28), which measures caregiver-reported mobility limitations.
and 12-year-old children before enrolment. Randomization It was found to be valid and reliable in a recent study in a
was performed for each school separately by one independent Dutch population.17
physiatrist. The children were pre-stratified according to three The secondary outcome, ‘muscle strength’, was measured
stratification variables: sex, GMFCS level (I, II–III), age with (1) a six-repetition maximum test on a leg-press and (2)
(youngest: 6–9y; oldest 10–13y), and subsequently randomized isometric strength tests of the hip flexor ⁄ abductor, knee
to one of two groups using sealed envelopes. Parents, thera- flexor ⁄ extensor, and ankle plantar flexor muscles of the most
pists, and schoolteachers were notified about the randomiza- affected leg, using a hand-held dynamometer (MicroFet; Bio-
tion group by letter. Two independent research assistants metrics, Almere, the Netherlands). At T1 only a subset of
performed all assessments and data entry. They were blinded these outcomes were tested: the STS, LSU, and six-repetition
for treatment allocation. Assessments were at baseline (T0), maximum.
Patients ineligible
n=4 (4× planned for surgery)
Randomized patients
n =51
Intervention Control
Personal characteristics group (n=24) group (n=25) p value 95% CI
Data are number of patients, unless otherwise indicated. aDerived from the Tanner stages of sexual maturation. bDerived from the Strength and
Difficulties Questionnaire. CI, confidence interval.
improved mobility, without any adverse effect on muscle spas- were found in the knee extensors (12%) and hip abductors
ticity. (11%), but not in the knee, hip, and ankle plantar flexors. This
This 12-week functional PRE strength training resulted in seems to underline the importance of specificity in strength
statistically significant larger improvements (8%) in total training; i.e. the exercise must be specific to the working of the
hand-held isometric muscle strength in the training group muscle. Because the circuit training specifically targeted
than in the control group. Isolated significant improvements strengthening knee and hip extensors (with the leg-press, STS,
Intervention Control
Regression coefficient
T n Mean (SD) n Mean (SD) (95% CI) p value
Primary outcomes
GMFM-66 (0–100) T0 24 76.1 (12.8) 23 71.8 (12.5)
T2 24 76.1 (11.8) 24 73.1 (12.4) )0.56 ()2.11 to 0.99) 0.48
T3 24 76.6 (13.0) 24 72.7 (12.8) 0.26 ()1.23 to 1.76) 0.73
STS test (reps) T0 24 12.9 (2.8) 23 10.8 (3.0)a
T1 24 13.3 (3.2) 24 12.1 (4.2) )0.47 ()2.28 to 1.33) 0.61
T2 24 13.6 (3.0) 24 12.7 (4.3) )0.75 ()2.21 to 0.72) 0.32
T3 24 14.3 (2.9) 23 12.7 (4.6) 0.03 ()1.43 to 1.49) 0.97
LSU test (reps) T0 23 15.6 (4.0) 23 13.3 (5.4) 0.95
T1 24 15.2 (3.9) 24 13.7 (4.3) )0.05 ()1.87 to 1.77) 0.63
T2 24 17.0 (5.1) 23 15.4 (4.3) 0.48 ()1.45 to 2.40) 0.9
T3 24 17.5 (4.8) 24 15.8 (6.6) 0.13 ()1.84 to 2.10)
Secondary outcomes
Isometric muscle strength (N ⁄ kg)
Knee extensors T0 24 4.78 (1.12) 23 4.36 (1.05)
T2 24 5.39 (1.10) 24 4.48 (1.21) 0.56 (0.13 to 0.99) 0.01
T3 23 5.20 (1.04) 24 4.46 (1.20) 0.35 ()0.16 to 0.85) 0.16
Knee flexors T0 24 2.73 (0.79) 23 2.25 (0.96)
T2 24 2.76 (0.75) 24 2.27 (1.02) 0.05 ()0.25 to 0.36) 0.71
T3 24 2.67 (0.86) 24 2.33 (0.90) )0.10 ()0.43 to 0.24) 0.58
Hip flexors T0 24 3.96 (0.75) 23 3.76 (0.99)
T2 24 4.43 (0.99) 24 4.12 (0.99) 0.16 ()0.22 to 0.55) 0.41
T3 24 4.46 (0.90) 24 4.43 (0.86) )0.12 ()0.50 to 0.27) 0.55
Hip abductors T0 24 2.66 (0.76) 22 2.41 (0.74)
T2 24 2.78 (0.85) 24 2.28 (0.70) 0.27 (0.00 to 0.54) 0.05
T3 24 2.90 (0.99) 24 2.45 (0.94) 0.23 ()0.10 to 0.56) 0.17
Ankle plantar flexors T0 24 3.90 (1.43) 23 3.11 (0.87)a
T2 24 4.53 (2.16) 23 3.54 (0.94) 0.23 ()0.47 to 0.93) 0.51
T3 24 5.01 (2.26) 24 4.38 (1.49) )0.16 ()1.01 to 0.69) 0.72
Total T0 24 18.04 (3.52) 22 15.94 (3.57)a
T2 24 19.88 (4.13) 23 16.65 (4.11) 1.30 (0.56 to 2.54) 0.04
T3 23 20.39 (4.49) 24 17.80 (4.01) 0.40 ()1.02 to 1.83) 0.58
Six-repetition maximum on leg- T0 23 112.78 (21.28) 23 93.76 (20.18)a
press (% body weight) T1 24 119.38 (26.61) 23 100.80 (23.72) 1.97 ()8.45 to 12.41) 0.71
T2 23 135.63 (31.87) 22 102.88 (26.76) 14.17 (1.99 to 26.35) 0.02
T3 24 129.90 (32.15) 23 111.99 (26.17) 3.42 ()8.62 to 15.46) 0.58
MobQues-28 (0–100) T0 22 68.42 (20.93) 22 64.77 (26.26)
T2 23 67.51 (24.58) 23 66.43 (25.93) )0.46 ()6.00 to 5.07) 0.87
T3 21 70.03 (23.49) 17 67.49 (20.33) )2.22 ()7.88 to 3.43) 0.44
Control outcome Median (SD) Median (SD)
a
Statistically significant baseline difference between the two groups. T, measurement point; T0, baseline; T1, 6 weeks; T2, 12 weeks (end of
training); T3, 18 weeks (follow-up); n, number of individuals; GMFM, Gross Motor Function Measure; STS, Sit-to-Stand test; LSU, Lateral Step-Up
test; reps, repetitions; MobQues-28, mobility questionnaire; CI, confidence interval.
forward step-up, and half-knee-rise exercises) and hip abduc- is impossible to compare these results with others. However,
tors (with the lateral step-up exercise), improvements in these one randomized study10 did use the one-repetition maximum
target muscles were, therefore, to be expected. Unfortunately, STS as an outcome. When adjusted for body weight, the
isometric hip extension strength was not assessed, owing to strength increase in favour of the intervention group was com-
this assessment’s low reliability, as found in a pre-pilot study. parable (approximately 15%).
Two other randomized studies have evaluated isometric mus- The results of this study show that training with a sufficient
cle strength, and found similar results, either in total muscle intensity, e.g. with a training load at which no more than 8 to
strength9 or isolated (targeted) muscle strength.8 The training 12 repetitions can be completed before muscular fatigue,
also resulted in a statistically significant improvement (14%) results in increases in muscle strength. With this result we
in six-repetition maximum leg-press strength, compared with hope to fill the gap that was recently addressed in a literature
the control group. To our knowledge, no other study has discussion19 on the effectiveness of strength training on
assessed this outcome measure in a similar patient group, so it muscle strength, and stressing the need for new randomized
90 22 160
d e f
6RM (% bodyweight)
*
20
#
70 120
18
60 100
16 80
50 Training
60 Control
40 14
0 6 12 18 0 6 12 18 0 6 12 18
Time (wks)
Figure 2: Estimated marginal means and standard errors of (a) Gross Motor Function Measure, 66 items (GMFM-66), (b) Sit-to-Stand, (c) Lateral Step-Up,
(d) Dutch mobility questionnaire (MobQues-28), (e) hand-held dynamometer (HHD)– total, and (f) six-repetition maximum (6RM) scores at measurement points
0 (baseline), 6, 12, and 18 weeks for the intervention and the control groups. aStatistically significant baseline difference. bStatistically significant difference in
change between intervention and control groups, corrected for baseline differences (p<0.05).
controlled trials that apply appropriate guidelines. We feel the longitudinal relation between muscle strength and mobil-
that our study adds to this discussion and the current evi- ity. Daily activities require only a certain amount (e.g. lowest
dence20 that PRE strength training is effective in increasing threshold) of muscle strength. Increases above these lowest
leg muscle strength in this group of patients. threshold values may be accompanied by increases in mobil-
There was a detraining effect, resulting in loss of muscle ity,23 but there might also be a point (e.g. highest threshold) at
strength at 6 weeks after the training ended, which was which further strength increases provide no additional advan-
expected, and has also been observed in healthy children.21 tage in mobility improvements.24 In that case, strength train-
Surprisingly, this effect was not observed in the few compara- ing would not be the appropriate choice of treatment when
ble (mostly uncontrolled) studies in children with CP3,9,12 with the aim is to improve mobility. Here, other components, such
a follow-up assessment. Based on the findings of our random- as balance and coordination, might influence mobility
ized controlled trial, we recommend that strength training improvement to a greater degree than muscle strength alone.
should be included in a regular exercise routine to maintain This should also be a subject for future research. In addition,
increased strength levels.22 the lack of effectiveness on mobility improvement found in
Unexpectedly, the 8 to 14% strength improvements were this study might also be explained by the lack of variation in
not accompanied by mobility improvements, according to the task contexts and individually tailored exercises. It is said that
GMFM-66, the functional strength tests, and the mobility functional improvement is unlikely to occur unless the task to
questionnaire. Two out of three comparable randomized con- be learned is practised under various different contexts.25 Even
trolled trials showed that their strength training resulted in though our training contained functional exercises that resem-
small, but significant, functional improvements on the bled daily mobility activities (e.g. rising from a chair), the
GMFM-66 in favour of the training group.8–10 So, despite the actual context of these exercises (i.e. chair height) remained
current popularity of strength training, the evidence from unchanged throughout the training period. The lack of effec-
current randomized controlled trials is inconclusive on the tiveness on mobility improvement might also be explained by
effectiveness of strength training to improve mobility in the non-individual specificity of the exercises. Selection of
children with CP. individually tailored mobility exercises with a more goal-ori-
Possible explanations for the lack of mobility improvement ented approach might improve the effectiveness of strength
in our study might be that the 8 to 14% improvements were training on mobility outcomes.26
too small to improve mobility, or that the number of individ- There was no change in spasticity (in either group) during,
ual muscles gaining strength was too limited. Therefore, directly after, or 6 weeks after the training, confirming the
future studies should focus on optimizing the training pro- results of similar studies.3 This supports the current belief that
gramme to reach maximum strength gains in children with strength training for patients with spasticity is not contra-indi-
CP. Besides this, we need to gain a better understanding of cated. Children were informed that they could experience a
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