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DEVELOPMENTAL MEDICINE & CHILD NEUROLOGY ORIGINAL ARTICLE

Walking ability and predictors of performance on the 6-minute walk


test in adults with spastic cerebral palsy
GRETHE MAANUM 1 | REIDUN JAHNSEN 1 , 2 | KATHRINE F FRØSLIE 1 , 2 , 3 | KERSTIN L LARSEN 1 | ANNE KELLER 4

1 Sunnaas Rehabilitation Hospital, Nesoddtangen, and Faculty of Medicine, University of Oslo, Oslo, Norway. 2 Oslo University Hospital, Rikshospitalet, Oslo, Norway.
3 Department of Biostatistics, Institute of Basic Medical Sciences, University of Oslo, Oslo, Norway. 4 Oslo University Hospital, Ullevaal Hospital, Oslo, Norway.
Correspondence to Dr Grethe Maanum, Sunnaas Rehabilitation Hospital, 1450 Nesoddtangen, and Faculty of Medicine, University of Oslo, Oslo, Norway. E-mail: grethe.manum@sunnaas.no

PUBLICATION DATA AIM To describe walking ability and identify factors predicting walking capacity in adults with
Accepted for publication 7th December 2009. spastic cerebral palsy (CP) assessed with the 6-minute walk test (6MWT).
Published online 12th February 2010. METHOD A cross-sectional clinical study as part of the recruitment process for a randomized con-
trolled trial on the effects of botulinum toxin A. Data analysed were the 6MWT, Timed Up and Go
LIST OF ABBREVIATIONS (TUG) test, Borg Scale, spasticity, muscle strength, popliteal angle, pain, fatigue, type of CP, foot
6MWT 6-minute walk test deformity, Gross Motor Function Classification System (GMFCS) levels, Functional Mobility Scale
6MWD 6-minute walk test distance (FMS) scores, a gait questionnaire, interview, and demographic data.
FMS Functional Mobility Scale RESULTS In total, 126 persons were included (53 males, 73 females; mean age 39y [SD 12y]; 59
SF-36 General Health Survey Short-Form 36 with unilateral and 67 with bilateral spastic CP; GMFCS level I, n=12; level II, n=94; level III, n=20).
TUG Timed Up and Go
Mean distance on the 6MWT was 485m (SD 95m) with FMS scores reflecting independent walking
performance in daily life. Multiple regression analysis identified sex, type of CP, popliteal angle,
pain, and TUG values as significant predictors, with TUG values as the strongest predictor
(standardized regression coefficient =)0.57, p<0.001).
INTERPRETATION Our results demonstrate that 39% of the participants had declined one GMFCS
level from adolescence to their present age, and that the TUG was the strongest predictor for the
6MWT. This implicates the importance of focusing specifically on the different elements of func-
tional mobility in further studies.

Cerebral palsy (CP) is a group of permanent disorders of complex ability to stay upright and be moving at the same
movement and posture, often accompanied by secondary mus- time.7 Walking in the community also requires sensory, per-
culoskeletal problems.1 Literature on children with CP sup- ceptual, and cognitive abilities to anticipate and react ade-
ports a link between the levels of impairment and walking quately to crowds and unexpected events.8
ability.1 However, the relation between self-reported declined In studies on gait and mobility ⁄ balance, the 6-minute walk
walking ability and data on clinical measurements in adults test (6MWT)9 and the Timed Up and Go (TUG) test10 are
needs more investigation. frequently used measures for the activity dimension of the
Surveys of adults with CP report that contractures, spastic- International Classification of Functioning, Disability and
ity, pain, fatigue, as well as reduced muscle strength and bal- Health (ICF).11 The 6MWT, which measures the maximum
ance, are associated with declined walking ability.2–4 distance a person can walk within 6 minutes, was originally
The Gross Motor Function Classification System developed as an endurance measure in chronic heart failure.12
(GMFCS) categorizes gross motor function into five levels, For elderly and stroke populations, the 6MWT is considered
with level I describing the highest level of function and level V a general measure of overall functional walking capacity,
the lowest.5 A study of individuals with CP aged up to involving the integrated response of multiple body sys-
21 years showed that those classified at GMFCS levels III to tems.9,13,14 The TUG was originally developed as a balance
V have their peak motor function in childhood, whereas indi- test for the elderly.15 By measuring, in time, the ability to
viduals at levels I or II have a stable gross motor function.6 stand up, walk, turn around, and sit down, the TUG captures
However, surveys of adults with CP indicate that even persons the complex interaction between balance and movement,
at GMFCS levels I or II experience increasing walking diffi- including planning, initiating, executing, and completing a ser-
culties in young adulthood.4 ies of linked movements that are common in daily activities.8
Adequate postural stability, selective motor control, muscle The purpose of this study was to describe walking ability in
strength, and endurance are necessary prerequisites for per- a population of adults with spastic CP experiencing declined
forming basic gross motor skills such as walking, including the walking function, and to identify clinical and demographic

e126 DOI: 10.1111/j.1469-8749.2010.03614.x ª The Authors. Journal compilation ª Mac Keith Press 2010
14698749, 2010, 6, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/j.1469-8749.2010.03614.x by Cochrane Chile, Wiley Online Library on [30/08/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
factors predicting walking capacity, as measured with the What this paper adds
6MWT. • Thirty-nine per cent of ambulant adults with spastic CP had declined
one GMFCS level from adolescence to their present age.
METHOD • 6MWD and TUG values varied significantly over the GMFCS levels.
This study is based on data from the baseline screening before • The TUG test was the strongest predictor of 6MWT.
• Age effects and TUG elements need specific investigation.
inclusion in a randomized controlled trial on the effects of bot-
ulinum toxin A in adults with spastic CP at the Sunnaas Reha-
bilitation Hospital, Oslo, Norway.
The participants were recruited through advertisements in rating mobility over three distances (5m, 50m, and 500m), was
newspapers and on the websites of the Norwegian Cerebral used as a measure of walking performance and rated based on
Palsy Association and Sunnaas Rehabilitation Hospital from information from the participants. The FMS has established
2007 to 2008. The first author was in contact with all the psychometric properties for children with CP (interrater reli-
responders. Potential eligible responders were questioned by ability ICC=0.95; construct validity r=0.75–0.89).18 The self-
semi-structured telephone interview about changes in walking administered questionnaire on walking function (Appendix SI)
function from adolescence to their present age. included information on causes of change in walking ability,
Inclusion criteria were (1) uni- or bilateral spastic CP, (2) walking performance in different contexts, and on limiting
age 18 to 65 years, (3) GMFCS levels I to III, (4) ability to symptom(s) related to walking. It was developed by studying
walk independently continuously for 6 minutes, (5) ability to other questionnaires used in adult CP research,2,3 and through
provide informed consent, and (6) a phone interview reported workshops with the present authors and the named person
declined walking ability compared with adolescence such as acknowledged. Applicability was assessed in the ordinary clinic
reduced walking distance, increasing stiffness, spasticity, pain, before the start of the study.
or balance difficulties. Exclusion criteria were (1) other condi- The 6MWT was used to measure functional walking capac-
tions that could affect the walking ability, (2) orthopaedic sur- ity. The participants were instructed to walk as fast and as long
gery within the past 18 months, (3) injections of botulinum as possible for 6 minutes. Each participant underwent the
toxin A within the preceding 3 months, (4) pregnancy, and (5) 6MWT in a quiet hospital corridor with a 30m marked track,
intellectual disability documented in medical records. and chairs as turning points instead of cones to support turn-
The Commissioner for the Protection of Privacy in ing if needed. The investigator repeated set phrases every min-
Research and the Regional Ethics Committee in south-eastern ute during the walk.9 The total distance walked was recorded
Norway approved the study. All participants gave written as the 6MWT distance (6MWD, to the nearest metre). The
informed consent. 6MWT has demonstrated test–retest reliability for adults with
CP (ICC=0.97).19 Perceived exertion was graded immediately
Measurements after 6 minutes using the Borg Scale (rating of perceived exer-
A test protocol provided detailed descriptions and instructions tion, 6–20), a measure of exercise intensity with reported cor-
for all data elements collected during one visit to Sunnaas relations of ratings and heart rates of r>0.80 and test–retest
Rehabilitation Hospital. Data from medical records, classifica- reliability of ICC=0.72.19,20
tion of type of CP according to the Surveillance of Cerebral The TUG was used to measure functional mobility. The
Palsy in Europe,16 functional level by GMFCS,5,17 and Func- participants sat on a standardized armchair. They were
tional Mobility Scale (FMS),18 6MWT, TUG test, and self- instructed to rise, walk as quickly and safely as possible to a
administered questionnaires were performed or collected by marker on the floor 3m away, turn around, walk back to the
the first author. The first author, assisted by one of three chair, and sit down on set cues. Timing started on the word
skilled physiotherapists, used a consistent technique with stan- ‘start’ and finished once the participant’s back touched the
dardized procedures for the outcomes of body structure ⁄ func- back of the chair.10 Participants performed the test three
tion. No formal reliability testing was performed; however, times; the fastest time measured in seconds with one decimal
the measurements were the same as those used in the motion place was used. The TUG has demonstrated test–retest reli-
laboratories at Sunnaas Rehabilitation Hospital and at Oslo ability of ICC=0.99 in children with CP.21 Despite some simi-
University Hospital, developed and adjusted through joint larity with 6MWT, the TUG has several tasks involved that
training sessions with consensus processes. are potentially destabilizing, thus testing functional mobility
and anticipatory aspects of postural control.8
Activity ⁄ participation Pain intensity and impact on daily life were measured using
Walking ability was determined using the GMFCS, the FMS, the ‘bodily pain’ domain of the General Health Survey Short-
and a questionnaire on walking function (Appendix SI, pub- Form 36 (SF-36), on a scale from 0 (worst pain) to 100 (no
lished online). The GMFCS level at adolescence was rated on pain).22 The SF-36 is validated in the Norwegian popula-
information from the participants. Present GMFCS level was tion,23 and the ‘bodily pain’ domain has demonstrated test–
rated based on observations. The GMFCS has demonstrated retest reliability of ICC=0.90.24
validity and reliability for classification of present and earlier Fatigue was measured with the Fatigue Severity Scale, a
gross motor function in adults with CP (intraclass correlation questionnaire using a scale from 1 to 7.25 The questionnaire is
coefficient [ICC]=0.93–0.95).17 The FMS, a 6-level scale validated in the Norwegian population, with average scores of

Walking Ability in Adults with CP Grethe Maanum et al. e127


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at least 5 defined as severe fatigue,25 and has demonstrated dures are valuable tools when asymptotic properties of the
test–retest reliability of kappa=0.74 for the average score.26 estimators can be debated.36
The principal component analysis, ridge modelling, and the
Body structure ⁄ function bootstrap routines were performed in the R language, version
Foot deformity, defined as tibiocalcaneal angle being R.2.9.1 (http://www.r-project.org/). All other analyses were
greater than 5 varus or valgus, was determined by visual cate- performed with SPSS software (version 15.0; SSPS Inc., Chi-
gorization with the participants in standing position.27 This cago, IL, USA). Probability values less than 0.05 were consid-
assessment showed no disagreement with a secondary photo- ered statistically significant.
graph-based assessment for 66 of the participants.
Unilateral popliteal angle, expressing ‘functional’ hamstring RESULTS
length,7 was used as an indicator of range of motion in lower Of 201 respondents, 156 met the inclusion criteria and were
limbs (Appendix SII, published online). Intrarater reliability of enrolled for further testing. After clinical investigation another
ICC=0.77 is reported for children with CP.28 30 individuals were excluded, and 126 individuals were
The Modified Ashworth Scale, which measures the degree included in the study (Fig. S1, published online). Demo-
of resistance to fast passive movement, was used as an indica- graphic and clinical characteristics are presented in Table I.
tor of spasticity in lower limbs (Appendix SII).29,30 Interrater
reliability with a general kappa value of 0.74 to 0.82 is Walking ability
reported for comparable populations.30 Most participants (n=94) were classified at GMFCS level II.
Manual muscle testing as described by Hislop,31 which However, according to GMFCS level at adolescence, 32 had
scores muscle strength on a scale from 0 to 5, was used as an
indicator of muscle strength in lower limbs (Appendix SII). Table I: Demographic, social, and clinical data of participants (n=126)
This has demonstrated convergent construct validity with
dynamometry (r>0.76).32 Because manual muscle testing is n (%)a
prone to examiner bias, all the tests were performed by the
Age in years (y), mean (SD) 39 (12)
same assessor.31,32
Range, y 18–65
As impairment measures for the lower limbs, the popliteal Sex
angles, Modified Ashworth Scale, and manual muscle testing Female 73 (58)
Male 53 (42)
scores were individually transformed into summary measures
Type of cerebral palsyb
calculated as the mean of the measurements.33 Spastic unilateral 59 (47)
Spastic bilateral 67 (53)
GMFCS levelc
Statistical analysis
I 12 (10)
Descriptive statistics were used to characterize the sample. II 94 (74)
The TUG displayed a skewed distribution and was log trans- III 20 (16)
Education (duration)
formed. There were no missing data. Differences in TUG
First level (9y) 25 (20)
scores and 6MWD between the GMFCS levels were analysed Second level (12y) 49 (39)
with the Kruskal–Wallis test and the Mann–Whitney U-test. Third level (>12y) 52 (41)
Work ⁄ source of income
Bivariate relations between variables were explored in cross-
Paid work ‡20% 81 (64)
tables, box plots, and scatter plots, with corresponding Pear- Student 16 (13)
son’s and Spearman’s correlation coefficients. Disablement benefit ⁄ unemployedd 51 (40)
Comorbiditiese 61 (48)
Relevant literature was considered to identify possible pre-
Epilepsy 1 (1)
dictors for performance on the 6MWT, reflecting both Allergy 26 (21)
patient characteristics and the different ICF domains. The Anxiety ⁄ depression 16 (13)
Hypertension 11 (9)
regression model building included univariate regression anal-
Hypothyroidism 7 (6)
yses, analyses of several custom models considered to be of Otherf 4 (3)
importance, and stepwise procedures. Every step of the analy- Orthopaedic surgery lower limbg 62 (49)
Gastrocnemius ⁄ soleus lengthening 47 (37)
sis involved F-tests, Akaike’s Information Criterion and model
Hamstring lengthening 11 (9)
diagnostics, and discussions of the clinical importance of the Iliopsoas lengthening 11 (9)
findings. Interdependencies between predictors were explored Otherh 9 (7)
by principal component analysis34 and multicolinearity diag- a
Numbers presented unless otherwise stated. bBased on data from
nostics. The impact of this on the regression coefficients was medical records and clinical assessments. cClassified by first author.
explored by ridge regression.34 The final multiple model was d
For many with less than 100% paid work and some of the students
based on variables identified in these procedures, with redun- the income was combined with disablement benefit, so the
percentages of work ⁄ studies and disablement benefit add up to more
dant variables removed. Bootstrap methods with 10 000 repli- than 100. eObtained from medical records. fNamely diabetes type II,
cates were used to calculate the regression confidence intervals psoriasis, and hypercholesterolemia. gObtained from medical records.
(CIs), as the traditional formula-based CIs do not account for For several participants, this was more than one operation, so the
percentages of orthopaedic surgery of lower limbs add up to more
potential bias in the marginal distributions of the regression than 100. hRotational osteotomies and foot ⁄ toe corrections. GMFCS,
coefficients.35 Despite their shortcomings, bootstrap proce- Gross Motor Function Classification System.

e128 Developmental Medicine & Child Neurology 2010, 52: e126–e132


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Table II: Present walking ability described by GMFCS level, FMS score, and questionnaire (n=126)

GMFCS levela, n (%)

Adolescence Present Shift to lower GMFCS levels

I 44 (35) 12 (10)
II 80 (64) 94 (74) 32 (25) from level I to II
III 2 (2) 20 (16) 18 (14) from level II to III

FMS, n (%)

Score Description 5m 50m 500m

6 Independent on all surfaces 14 (11) 5 (4) 1 (1)


5 Independent on level surfaces 103 (82) 101 (80) 87 (69)
4 Use one crutch or two sticks 8 (6) 12 (10) 25 (20)
3 Use two crutches 1 (1) 7 (6) 6 (5)
2 Use walking frame 0 (0) 1 (1) 5 (4)
1 Use wheelchair 0 (0) 0 (0) 2 (2)

Questionnaireb, n (%)

Is able to use public transport 98 (78)


Walking for exercise 83 (66)
Reported changes compared with adolescencec
Increased stiffness 100 (79)
Decreased balance 68 (54)
Decreased walking distance 76 (60)
Increased pain 81 (64)
Reported limitations in walking functiond
Fatigue 69 (55)
Stiffness 81 (64)
Balance 65 (52)
Pain 73 (58)

a
Classification of Gross Motor Function Classification System (GMFCS) level in adolescence based on interview data compared with present
classification based on observation. bQuestionnaire developed for this study. c,dThe percentages add up to more than 100 owing to changes or
limitations caused by more than one of the items. FMS, Functional Mobility Scale.

declined from GMFCS level I to level II (Table II). Examples tively. Because most participants were at GMFCS level II and
of statements from the interviews and questionnaires were, FMS 5, and because of colinearity in models with the TUG
‘Now I need a rail while climbing stairs’ and ‘I don’t do moun- and FMS, GMFCS and FMS were not included in the multi-
taineering anymore’. ple regression analysis. Descriptive statistics of the variables in
TUG values and 6MWD varied significantly across the the final multiple model are presented in Tables I and III.
GMFCS levels (p>0.001). The median scores (interquartile Univariate analyses (Table IV) showed that neuromuscular
range) on the TUG were as follows: GMFCS level I, 5.2s and musculoskeletal impairments, such as increased spasticity,
(4.7–5.9); level II, 6.5s (5.9–8.4); and level III, 10.8s (9.9–13.8). reduced muscle strength, increased popliteal angle, equinova-
Median 6MWDs were as follows: GMFCS level I, 611m rus or -valgus foot, as well as increased pain, fatigue, and time
(542–652); level II, 491m (435–543); and level III, 376m (317– used in the TUG, were related to decreased 6MWD. Sex was
423). included in the multiple regression analyses as a known
FMS scores showed that the participants were more likely to predictor for 6MWD.
use assistive devices outdoors (Table II). Additional informa- In the final multiple regression analysis, sex, type of CP,
tion from the interviews showed a trend towards increasing fre- popliteal angle, pain, and TUG values were identified as sig-
quency in the use of assistive devices. Stiffness was the most nificant predictors of 6MWD. The adjusted estimates and CIs
frequently reported cause of declined walking ability (Table II). are shown in Table IV. Females had a shorter 6MWD than
males, as did bilateral CP compared with unilateral CP. An
Predictors of 6MWT increase of 1 in popliteal angle resulted in a 2.6m shorter
During the model-building process, we noted that type of CP, 6MWD; transferring this result to clinical practice, a 10
spasticity, muscle strength, popliteal angle, FMS, GMFCS, increase in popliteal angle would have resulted in 26m shorter
and the TUG test were interrelated, with bivariate correlation 6MWD. The regression coefficient for pain reflected a 1.3m
coefficients ranging from r=0.31 (0.14–0.46) to r=0.63 (0.51– longer 6MWD, with a higher score on the 0 to 100 scale indi-
0.73; GMFCS ⁄ FMS), all factors having a correlation with cating less pain. The regression coefficient for a log-trans-
6MWD of r>0.38 (0.22–0.52). Both the GMFCS and FMS formed TUG of )377 reflected a shorter 6MWD with
were significantly associated with 6MWD in the univariate increasing TUG values (in seconds). Participants with a TUG
analysis (r=0.58 [0.45–0.69] and r=0.56 [0.43–0.67]) respec- value at the lower quartile for the group (5.9s) walked on

Walking Ability in Adults with CP Grethe Maanum et al. e129


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DISCUSSION
Table III: Variables used in the regression analysis (n=126)
This study investigated walking ability and the influence of
Variablea Mean (SD)b Range
demographic, clinical, and self-reported factors on functional
walking capacity in adults with CP who reported a declined
Dependent variable walking function compared with their function during adoles-
6-minute walk test distance (m) 485 (95) 260–730
Potential predictor
cence. We identified CP-related neuromuscular deficits, pain,
Borg Scalec (6–20) 13.5 (2.1) 9–20 and sex as important factors predicting functional walking
Foot deformityd, n (%) 75 (60) Yes ⁄ no capacity. Self-reported walking function in adolescence com-
Popliteal anglee (degrees) 51 (13) 25–90
Spasticityf 1.2 (0.8) 0.1–3.3
pared with current GMFCS classification indicated a shift in
Muscle strengthg 4.4 (0.4) 3.4–5.0 GMFCS level for 39% of the participants.
Timed Up and Goh (s), median (Q1–Q3) 6.8 (5.9–9.6) 3.6–24 As expected, type of CP was a significant predictor for
Log-transformed Timed Up and Go 0.86 (0.14) 0.56–1.38
Bodily paini (0–100) 54.8 (25.2) 0–100
6MWD, which implies that individuals with spastic bilateral
Fatigue severity scalej (0–7) 4.8 (1.4) 1.1–7.0 CP have more neurological impairments.1 Because CP is a
a
multifactorial disorder, and there is conflicting evidence on
Age, sex, and type of CP are described in Table I. bNumbers
how motor impairments relate to each other and to function
presented unless otherwise stated. cPerception of physical exertion,
higher scores indicating more exertion. dOccurrence of equinovarus or for children with CP,29,33 it seems logical that our results indi-
-valgus deformity. eUnilateral popliteal angle for the right and left side cate a complex interrelation between the variables investi-
calculated as the mean of the measurements, higher score indicating
gated.
reduced ‘functional hamstring length’. fModified Ashworth Scale (0–4)
for eight different muscle groups calculated as the mean of the Increased mean popliteal angle was associated with
measurements, higher score indicating more spasticity in lower limb.
g
decreased 6MWD. By choosing unilateral popliteal angle
Manual muscle testing (0–5) for 14 different muscle groups calculated
instead of bilateral popliteal angle,7 we measured the ‘func-
as the mean of the measurements, lower score indicating less muscle
strength in lower limbs. hHigher score indicating more functional tional hamstring length’ with the potential effect of a contra-
mobility impairment. iGeneral Health Survey Short-Form 36 (SF-36) lateral hip flexor tightness, a factor considered relevant when
domain of bodily pain, higher score indicating less pain and less effect
choosing a lower-limb range of motion measure related to
of pain on daily activities. jHigher score indicating more fatigue.
Q1–Q3, interquartile range. walking function. The effect of mean spasticity in the adjusted
analyses may be confounded by other effects, such as the mean
average 100m further than those at the upper quartile for the popliteal angle. This is supported by the association between
group (9.6s). Log-transformed TUG became the strongest reduced range of motion and increased spasticity ⁄ more
predictor, with a standardized regression coefficient of )0.57 impairment in spastic CP,7,29 the ‘increased stiffness’ as the
(Table IV). The coefficient of multiple determination (R2) most frequently reported cause of declined walking function,
showed that this model explained 67% of the variance of the and the fact that the modified Ashworth Scale does not dis-
6MWD. These results were similar to the results from ridge criminate between dynamic spasticity and spastic contrac-
regression models. tures.29

Table IV: Results from univariate and multiple linear regression analyses with 6-minute walk test distance as the dependent variable (n=126)

Unadjusted estimates Adjusted estimates

Bootstrap Bootstrap

Potential predictors Ba p Betab Ba 95% CI Ba p Betab Ba 95% CI

Sex )29 0.089 )0.15 )29 )63; )4 )29 0.008 )0.15 )29 )50; )9
Age )2.3 0.001 )0.31 )2.3 )3.6; )1.0
Type of cerebral palsy )98 <0.001 )0.52 )98 )126; )70 )31 0.007 )0.16 )31 )55; )9
Foot deformityc )43 0.011 0.22 )43 )9; )78
Popliteal angled (degrees) )2.6 <0.001 )0.39 )2.6 )3.6; )1.4 )1.5 0.001 )0.20 )1.5 )2.3; )0.6
Spasticitye )40 <0.001 )0.45 )40 )53; )28
Muscle strengthf 133 <0.001 0.51 133 96; 173
Borg Scaleg )12.8 0.001 )0.29 )12.8 )20; )6
Log-transformed TUGh )508 <0.001 )0.77 )508 )606; )424 )377 <0.001 )0.57 )377 )478; )291
Bodily paini 1.3 <0.001 0.34 1.3 0.7; 1.9 0.5 0.009 0.14 0.5 0.1; 1.0
Fatigue Severity Scalej )16.2 0.008 )0.24 )16.2 )28; )4.9

Eleven clinically relevant potential predictors were included. The adjusted estimates are those from the final model, using multiple linear
regression analysis (R2=0.67). aRegression coefficient. bStandardized regression coefficient. Coding of categorical variables: male=0, female=1;
spastic unilateral=0, spastic bilateral=1. cOccurrence of equinovarus or -valgus deformity classified as no=0, yes=1. dUnilateral popliteal angle for
the right and left side calculated as the mean of the measurements, higher score indicating reduced ‘functional hamstring length’. eModified
Ashworth Scale (0–4) for eight different muscle groups calculated as the mean of the measurements, higher score indicating more spasticity in
lower limb. fManual muscle testing (0–5) for 14 different muscle groups calculated as the mean of the measurements, lower score indicating less
muscle strength in lower limbs. gPerception of physical exertion, higher score indicating more exertion. hHigher score indicating more functional
mobility impairment. iGeneral Health Survey Short-Form 36 (SF-36) domain of bodily pain (0–100), higher score indicating less pain. jHigher score
indicating more fatigue. TUG, Timed Up and Go.

e130 Developmental Medicine & Child Neurology 2010, 52: e126–e132


14698749, 2010, 6, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/j.1469-8749.2010.03614.x by Cochrane Chile, Wiley Online Library on [30/08/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
When adjusted, mean lower-limb muscle strength was not a with the general population.40 The results show that even per-
significant predictor. This may reflect the narrow range of sons at GMFCS level I experience declined walking ability. As
muscle strength and the clinical impression that the partici- others have demonstrated stability until young adulthood,6
pants had satisfactory muscle strength for walking.37 However, our results demonstrate the need for studies on adults. In addi-
we recognize that manual muscle testing has limited ability to tion, there are few studies exploring different mobility aspects
discriminate muscle strength scores.31,32 Thus, we hypothesize that, potentially, protect from or predispose to declined walk-
that muscle strength is important for maintaining functional ing in individuals at GMFCS levels I and II. Thus, our results
mobility in more demanding mobility tasks than walking in a support the need for a measure differentiating the children at
corridor.8,38 Foot deformity was excluded in the adjusted anal- GMFCS level I.41
ysis as a consequence of other variables being more important. We recognize that a limitation of this study is the selected
As the TUG is a measure of functional mobility, the group of high-level independent walkers. The recruitment
interrelations between the TUG, GMFCS, FMS, and through advertisements also led to more female participants
6MWD were expected. We considered it important to use than males. Though widespread in clinical use, the lower limb
a measure with a potential ability to detect anticipatory assessments have limitations to their reliability.28,29,32 To
aspects of postural control, as well as sensory and percep- compensate for this we used a strict protocol, and one experi-
tual abilities required in functional mobility,8 thus challeng- enced clinician conducted all the assessments. The use of
ing the complex impairments mentioned in the definition mean scores may have limited the measurements for popliteal
of CP.1 The literature is conflicting about the ability of the angle, spasticity, and muscle strength. However, the purpose
TUG to differentiate between the different GMFCS lev- was to have clinically relevant scores in spastic lower limbs,
els.21,39 We found a significant difference in TUG values and therefore sum scores were judged to be sufficiently pre-
across these levels. Thus, we suggest that the TUG can cise.33
give useful information about functional mobility in daily Although we have attempted to explore a wide range of
activities in comparable populations. Recognizing the different subsets of predictor variables to support our final
limitations of the TUG as a specific measure of balance, it model, subset selection procedures tend to be slightly over-
may be sufficient for assessing changes of functional bal- fit, and the effect size estimates may, therefore, be biased.42
ance in follow-up programmes. Although the 6MWT and Our bootstrap estimation ensured that possible biases in
TUG are different measures, there is some overlap in tasks, the distribution of the estimators were accounted for in the
which may overestimate the effect of the TUG on calculations of the CIs. However, a potential slight overes-
6MWD. Further, it is recognized that the performance of timation of effect sizes remains, with other subsets of pre-
the TUG is related to age, possibly because of its depen- dictor variables potentially being equally good or superior
dence on muscle strength as well as anticipatory postural to those we have presented.34,42,43 Therefore, our model
control, both of which decrease with age.8 Thus the TUG requires further validation in studies on similar samples.
may confound the effects of age and muscle strength in The CIs in the final model may reflect both measurement
our final model. errors and the known heterogeneity in spastic CP.7 The
Our finding that pain was a significant predictor of func- results support the need for more studies with adequate
tional walking capacity, confirms the earlier reported impact sample sizes addressing subgroups, as well as studies with a
of pain on declined ambulation.2,4 In contrast, fatigue was longitudinal design exploring the effects of ageing on walk-
adjusted out as consequence of other variables being more ing ability.
important. However, it is reasonable to hypothesize a greater
impact from pain and fatigue if walking performance had been CONCLUSION
investigated.4 The present study demonstrates that in this population of
Despite the normally distributed age range from 18 to independent adult walkers reporting declined walking ability
65 years, we did not find any effect of age. This was surpris- compared with adolescence, 39% had changed GMFCS level.
ing,3 but may be due to the impact of the TUG or to other The multiple regression analyses identified sex, type of CP,
CP-related impairments being more important predictors popliteal angle, pain, and performance on the TUG as signifi-
than age. Moreover, we recognize the fundamental challenge cant predictors, with the TUG as the strongest predictor. This
of age effects in a cross-sectional study. implicates the importance of focusing specifically on different
As with samples of healthy populations, sex was a significant aspects of functional mobility in further studies with an ade-
predictor in our final model; height and body mass index are quate sample size and a longitudinal design.
likely to contribute to this finding.40 The Borg Scale was not a
significant predictor, which probably reflects that most of the ACKNOWLEDGEMENTS
participants were more limited by neuromuscular impairment This study was performed as part of a randomized controlled trial,
than by cardiovascular factors. This has also been demon- and the project was supported by the East Regional Health Adminis-
strated for stroke populations.14 tration and Sunnaas Rehabilitation Hospital (grant number 206
The walking capacity in these high-level ambulant partici- 24 503). We are grateful to all the adults with CP who participated.
pants was better than in a population of adults with CP We acknowledge Professor Katharina S Sunnerhagen, and the staff at
recruited from a rehabilitation centre,19 but reduced compared Sunnaas Gait laboratory.

Walking Ability in Adults with CP Grethe Maanum et al. e131


14698749, 2010, 6, Downloaded from https://onlinelibrary.wiley.com/doi/10.1111/j.1469-8749.2010.03614.x by Cochrane Chile, Wiley Online Library on [30/08/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
SUPPORTING INFORMATION Appendix SII: Clinical measures.
Additional supporting information may be found in the online version Please note: Wiley-Blackwell are not responsible for the content or
of this article: functionality of any supporting materials supplied by the authors. Any
Figure S1: Summary of recruitment of adults with CP responding to queries (other than missing material) should be directed to the corre-
the advertisement. sponding author of the article.
Appendix SI: Questionnaire concerning present walking ability.

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