2013

http://informahealthcare.com/dreISSN 0963-8288 print/ISSN 1464-5165 online

Disabil Rehabil, Early Online: 1–11! 2013 Informa UK Ltd. DOI: 10.3109/09638288.2013.805820

The efficacy of GMFM-88 and GMFM-66 to detect changes in grossmotor function in children with cerebral palsy (CP): a literature review

Madawi Alotaibi1, Toby Long2, Elizabeth Kennedy3, and Siddhi Bavishi4

1Department of Physical Therapy, Doctor of Health Science, University of Indianapolis, Indianapolis, IN, USA, 2Department of Pediatrics, Center for

Child and Human Development, Georgetown University, Washington DC, USA, 3Department of Physical Therapy, University of South Alabama,

Mobile, AL, USA, and 4Department of Physical Therapy, Master of Health Science, University of Indianapolis, Indianapolis, IN, USA

Abstract

Aim: The purpose of this study was to review published research on the use of the Gross MotorFunction Measure (GMFM-88) and (GMFM-66) as outcome measures to determine if these toolsdetect changes in gross motor function in children with cerebral palsy (CP) undergoinginterventions. Methods: A comprehensive literature search was conducted using Medline andPubMed to identify studies published from January 2000 through January 2011 that reportedthe accuracy of GMFM-88 and GMFM-66 to measure changes over time in children with CPundergoing interventions. The keywords used for the search were ‘‘GMFM’’ and ‘‘CP’’. Two ofthe authors (M.A. and S.B.) reviewed the titles and abstracts found in the databases. Themethodological quality of the studies was assessed by using the Critical Review Form-Quantitative Studies. Results: Of 62 papers initially identified, 21 studies fulfilled the inclusioncriteria. These articles consist of three longitudinal studies, six randomized controlled trials, fourrepeated measure design, six pre–post test design, a case series and one non-randomizedprospective study. The included studies were generally of moderate to high methodologicalquality. The studies included children from a wide age range of 10 months to 16 years.According to the National Health and Medical Research Council, the study designs were level II,III-2, III-3 and IV. Conclusion: The review suggests that the GMFM-88 and GMFM-66 are useful asoutcome measures to detect changes in gross motor function in children with CP undergoinginterventions.

� Implications for Rehabilitation

� Accurate measurement of change in gross motor skill acquisition is important to determineeffectiveness of intervention programs in children with cerebral palsy (CP).

� The Gross Motor Function Measure (GMFM-88 and GMFM-66) are common tools used byrehabilitation specialists to measure gross motor function in children with CP.

� The GMFM appears to be an effective outcome tool for measuring change in gross motorfunction according to a small number of randomized control studies utilizing participantpopulations of convenience.

Keywords

Cerebral palsy, gross motor function measure,intervention, outcomes

History

Received 17 April 2012Revised 20 January 2013Accepted 13 May 2013Published online 26 June 2013

Introduction

Cerebral palsy (CP) describes ‘‘A group of disorders of thedevelopment of movement and posture causing activity limita-tions that are attributed to non-progressive disturbances thatoccurred in the developing fetal or infant brain. The motordisorders of CP are often accompanied by disturbances ofsensation, cognition, communication, perception and/or behaviorand/or a seizure disorder’’ [1]. These neurological impairmentsresult in activity limitations affecting a person’s lifelong oppor-tunity to fully participate in daily activities. Until recently, it wasbelieved that the prevalence of CP had remained steady at 2–2.5per 1000 live births, however, there is indication that theprevalence is higher, more closely approximating 3.1 per 1000

births [2]. Incidence is slightly higher in premature neonates andin neonates who are small for their gestational age. CP is slightlymore common in males than in females. Clinical presentation ofCP is varied with spastic CP as the most common type of CP,affecting �50% of individuals with a diagnosis of CP. AthetoidCP affects �20% of people with CP, ataxic CP accounting foranother 10% and the remaining 20% of individuals diagnosed withCP considered mixed [3].

Contemporary management of people with CP is based on aframework that considers effective intervention programs as thosethat promote optimum function throughout the life span. A widerange of medical, therapeutic and rehabilitation interventions arecurrently utilized to improve motor performance, mobility, fitnessand independence in daily living. There is an on-going need forevidence to substantiate the values of these programs. A primarygoal of therapeutic intervention in children with CP is to promotegross motor skill performance as essential components offunctional mobility. Accurate measurement of change in gross

Address for correspondence: Madawi Alotaibi, Department of PhysicalTherapy, Doctor of Health Science, University of Indianapolis,Indianapolis, IN, USA. E-mail: pt_madawi@hotmail.com

Dis

abil

Reh

abil

Dow

nloa

ded

from

info

rmah

ealth

care

.com

by

Uni

vers

ity L

ibra

ry U

trec

ht o

n 08

/17/

13Fo

r pe

rson

al u

se o

nly.

motor skill acquisition is important to document effectiveness ofinterventions as well as to accurately describe an individualchild’s motor abilities [4]. Traditional measurement tools, suchas norm-referenced developmental tests, are not sensitive todocument subtle changes in functional motor skills over time forchildren with neuromuscular dysfunction such as CP [5]. Theliterature repeatedly supports the need for measurement instru-ments to objectively assess gross motor activities in thesechildren. Although many gross motor function evaluation toolshave been developed, few of these instruments fulfilled thecriteria of reliability and validity with respect to responsivenessto change in gross motor performance in children with CP [6].It is evident that reliable and valid instruments are needed todetect motor changes over time to increase the likelihood ofdetecting clinically important treatment effects, even if the effectis small [7].

A crucial element for assessing intervention effectiveness forchildren with CP is the capability of being able to reliablymeasure responsiveness to change in gross motor abilities [8].Over the past 25 years, the Gross Motor Function Measure(GMFM) and its subsequent revisions, has become the mostcommon functional outcome measure used by rehabilitationspecialists to measure gross motor functioning in children with CPand other neurologically based conditions, such as Downsyndrome and traumatic brain injury [9,10]. The earliest versionof the GMFM is known as the GMFM-88 and the most recentversion is referred to as the GMFM-66 [11,12]. Both tools arestandardized criterion referenced measurement tools designed tomeasure gross motor function over time for children withdisabilities, ages 5 months to 16 years of age [11,13,14]. Eachtool has been validated to measure change in children with CP[15] but only the original GMFM-88 has been validated with otherpopulations, such as Down syndrome [16,17], traumatic braininjury [18] and osteogenesis imperfecta [19]. The uniqueness ofthese tools lies in the fact that the tools provide outcome scoresthat reflect how much of an activity a child can accomplish(function) rather than how well the activity is performed [11,13].The scores provide an enhanced understanding of activityoutcomes; ultimately leading towards achievement of contextualparticipation goals specific to the individual child. Althoughspecialist training is not required to administer the tool, theauthors recommend that administrators need to be familiar withassessing motor skills in children and the GMFM administrationguidelines [11,13]. International acceptance of the tools is notedwith translation into Spanish, French, Dutch, German andJapanese [20].

Organization of test items reflects developmental gross motormilestones. Items on the GMFM-88 are grouped into fivedimensions: lying and rolling (17 items), sitting (20 items),crawling and kneeling (14 items), standing (13 items), andwalking, running and jumping (24 items). The items are scored onfour-point ordinal scales (0¼ cannot initiate; 1¼ initiates;2¼ partially completes item; 3¼ completes item independently)[21]. Any item that has been omitted or the child is unable, orunwilling to attempt is scored as 0. The child is allowed amaximum of three trials on each item. Percentage scores arecalculated within each dimension and averaged to obtain a totalscore that ranges from 0 to 100. Higher scores indicate bettercapacity [11,13]. The GMFM-88 reliability values range from0.87 to 0.99.9 [11,13,21]. The 66-item GMFM was developedusing Rasch analysis in an attempt to improve the interpretabilityand clinical usefulness of the earlier measure. Sixty-six of theoriginal 88 items were retained and represent the uni-dimensionalconstruct of gross motor ability [12,22]. The GMFM-66 providesdetailed information on the level of difficulty of each item therebyproviding much more information to assist with goal setting.

The items are administered in the same way as with the GMFM-88. The Gross Motor Ability Estimator (GMAE), a computerprogram, is used to convert individual scores into an interval levelscoring system [11]. Russell et al. [12] found the test–retestreliability for the GMFM-66 to be high with an intraclasscorrelation coefficient of 0.99.

The GMFM is used by a variety of rehabilitation specialists forclinical and research purposes to measure change over time, andthe effectiveness of interventions to affect change [23]. Thestandardized measures provide objective information in an easy tounderstand format. As stated earlier, the GMFM has become thestandard tool for measuring change in gross motor function overtime for children with CP. Given its unique purpose, its wide use,and the amount of research that has been conducted using thisoutcome measure, examining the usefulness as related to inter-vention is important to measure change over time for childrenwith CP. Therefore, we conducted a systematic review to examinethe efficacy of the GMFM-88 and GMFM-66 to detect change ingross motor function in children with CP undergoing interven-tions. Our focus on children under 17 years old was chosenbecause children with CP often show significant change in grossmotor skill abilities during this time, are most often receivingtherapeutic intervention during this period, and the tool includesitems that are often accomplished by children at this age[12,14,24–27].

Method

Data source

A literature search was performed in Medline, and PubMedelectronic databases created by the United States National Libraryof Medicine. These two databases were chosen for the systematicreview because of the breadth and large number of citationscontained in these databases with a focus on biomedical citations.A follow up search, using same search strategies, was undertakenusing the electronic database Scopus to determine if initialresearch was complete (http://www.info.sciverse.com.libprox-y2.usouthal.edu/scopus/about). Searches were limited to articlespublished between 1 January 2000 and 31 January 2011 to obtainthe most current evidence. The keywords used for the search were‘‘GMFM’’ and ‘‘CP’’.

Study selection and inclusion/exclusion criteria

The following inclusion criteria were used: (1) sample includedchildren with diagnosis of CP between the ages 10 months and16 years, (2) studies of all research designs including case reports,(3) fully published studies in peer-reviewed journals (notabstracts), (4) intervention studies that used the GMFM 88 orGMFM-66 as an outcome measure and (5) studies published inEnglish as translation expertise was not available for otherlanguages. Studies were excluded if they included children overthe age of 16 or the study was a systematic review. The searchstrategy yielded 62 studies appearing to meet the inclusioncriteria.

Data extraction

Two of the authors (M.A., S.B.), both pediatric physicaltherapists, read together the title and abstracts of all paperssourced to determine each publication’s suitability for inclusioninto the final systematic review based on participants, outcomemeasure, study design and language criteria. If there was adisagreement (mostly during screening the papers), the full text ofthe publication was retrieved. The two reviewers then assessedand discussed the relevant aspects of a specific paper until amutual decision regarding publication appropriateness for this

2 M. Alotaibi et al. Disabil Rehabil, Early Online: 1–11

Dis

abil

Reh

abil

Dow

nloa

ded

from

info

rmah

ealth

care

.com

by

Uni

vers

ity L

ibra

ry U

trec

ht o

n 08

/17/

13Fo

r pe

rson

al u

se o

nly.

review was achieved. Research intervention was not defined asthis criterion may have limited the efficacy of the GMFM testusefulness. Twenty-one studies met all inclusionary criteria[10,28–47]. These 21 articles were reviewed in detail on theusefulness of the GMFM-88 and GMFM-66 to measure changeover time in children with CP.

Data analysis

Data was independently extracted by the same members of theresearch team using the data collection sheet (Table 1). Allauthors were involved to verify accuracy of data extraction andanalysis.

Quality of the evidence

All studies meeting inclusion criteria were assessed for methodo-logical quality using a modified version of the Critical ReviewForm – Quantitative Studies by Law et al. [48] (Table 2).

This generic critical appraisal tool was modified to contain 12criteria, each representing key elements of the methodologicalquality of a research study. Specifically, this template guided theanalysis of study purpose, literature review, study design, studysample, outcomes, intervention, results, conclusions and clinicalimplications for all types of quantitative studies. Each criterionwas awarded a score of one if the response was ‘‘yes’’ and zero ifthe response was ‘‘no’’ or ‘‘not addressed’’. Scores wereconverted to percentages for ease of interpretation and todetermine how much of the criteria were met [48]. For example;a score of 12/12 would indicate that the study addressed all 12elements, receiving a perfect score of 100%. Two investigatorstogether completed a critical review form for each study. Anydisagreements were resolved through discussion until consensuswas achieved.

Finally, each of the included publications was assigned anumerical quality score to correspond with a defined level ofevidence by the National Health and Medical Research Council(NHMRC) as defined in Table 3 [49]. In 1999, Australianresearchers (NHMRC) designed a hierarchy ranking the body ofevidence into four levels from systematic reviews of randomizedtrials at the top of the hierarchy to case series and case reports atthe bottom of the hierarchy. Table 4 includes the level of evidenceassigned to each study using this method.

Results

The initial search identified 62 potentially relevant articles.The search results are summarized in Figure 1. After applicationof the inclusion criteria to the titles and abstracts, 19 articles wereexcluded as they did not meet the inclusion criteria. Of theremaining 43 full-text papers screened, 22 were excluded as theyturned out not to meet the inclusion criteria. The remaining 21studies [10,28–47] that met the inclusion criteria underwent thedata extraction and critical appraisal process. As seen in Figure 1,six articles were excluded as they were only available as abstracts;nine articles did not meet the age criteria; five articles werewritten in languages other than English; 20 articles were excludedbecause they did not used the GMFM as an outcome measures ofeffectiveness of interventions and one other study was excludedbecause it was a systematic review.

Table 1 provides details of the 21 studies, including the studypurpose, eligibility criteria, sample size and study features. Allstudies included in the analysis found the GMFM to be a validmeasure to detect change in gross motor function in children withCP. The quality scores derived from the critical appraisal aresummarized in Table 4. Findings from this process revealeda wide variability in quality scores indicating variable

methodological quality. Scores ranged from a low 75%[28,32,40,41,46] to a high 100% [47]. A system to indicate thevalue of the scores was established based on previous studiescategorization schemes. Using the Methodological Approach toAssessing the Evidence, scores 485% are considered goodstudies, scores between 84% and 60% are fair and scores of 59and below are considered poor [50,51]. This scheme is similar tothat used to interpret reliability. A reliability quotient over 0.85 isgenerally considered high reliability. Based on this information,we decided that a score of 85% or higher indicated a highmethodological quality, scores between 75% and 84% indicatedmoderate quality and scores 575% indicated low or poormethodological quality. Accordingly, the included articles weregenerally of moderate to high methodological quality since thescores ranged from 75% to 100%.

The trial designs include three longitudinal studies, sixrandomized controlled trials, four repeated measure design, sixpre–post test design, a case series and one non-randomizedprospective study. The type of intervention (surgical, pharmaco-logical, therapeutic) and its characteristics (equipment used, timespent training within a session, total intervention period) showedwide variability. The studies also demonstrated large variability inparticipants’ ages (10 months to 16 years) and levels of impair-ment (Gross Motor Function Classification System (GMFCS)levels I–IV). According to the National Health and MedicalResearch Council [49], the study designs were level II, III-2, III-3and IV.

Discussion

Since initial publications, the two versions of GMFM have made aconsiderable impact in the rehabilitation field as it is used acrossrehabilitation settings and has been translated into many lan-guages [20]. The purpose of this study was not to compareeffectiveness of intervention programs but to explore if theGMFM-88 and GMFM-66 are useful as outcome tools to measureintervention effectiveness for children with CP. Knowledge gainedfrom this type of review study serves to provide additionalconfidence in using the GMFM as an outcome tool especiallysince these standardized assessments are practical and easy toadminister without the need for expensive equipment [11–13,15–19,21,52]. Each of the studies included in this review providescontinuing evidence of the usefulness of these tools and evidencethat the GMFM is a valid outcome tool with clinical relevance.A further discussion of results and outcomes of the includedstudies is necessary to obtain a deeper insight into the usefulnessof the GMFM across settings.

A review of these studies provides an understanding howresearchers used the GMFM to make conclusions about theeffectiveness of a wide variety of intervention programs acrossvaried research designs. Van den Broeck et al. [28] implemented aprospective, double blind research study; using the GMFM as anoutcome measure to evaluate functional outcomes in 16 childrenwith CP after a 6-week general training program followed by anindividually defined training program. The researchers were ableto quantify a significant improvement in functional outcomes tosuggest a specific training program is more effective as comparedto a general training program for children with CP. Conclusionsfrom this study are supported by Meyer-Heim et al. [30] in asingle-case experimental design of 22 children with CP during a3- to 5-week period of task-specific training. The researchers inthis latter study reported a significant improvement in the GMFMafter a specific training program. Similar results were found inother reports [32,34,43,45,46].

In contrast, other researchers used the GMFM to discoverspecific interventions were not effective in producing clinical

DOI: 10.3109/09638288.2013.805820 A literature review 3

Dis

abil

Reh

abil

Dow

nloa

ded

from

info

rmah

ealth

care

.com

by

Uni

vers

ity L

ibra

ry U

trec

ht o

n 08

/17/

13Fo

r pe

rson

al u

se o

nly.

Tab

le1

.C

har

acte

rist

ics

of

stu

die

sw

hic

hd

eter

min

edG

MF

M-8

8o

rG

MF

M-6

6as

val

ido

utc

om

em

easu

res

for

chil

dre

nw

ith

CP.

Stu

dy

auth

ors

Pu

rpo

seS

amp

lesi

zean

dfe

atu

res

Res

ult

sR

esea

rch

des

ign

Van

den

Bro

eck

etal

.[2

8]

Eval

uat

eef

fect

iven

ess

of

ind

ivid

ual

lyd

efin

edP

To

nfu

nct

ion

and

gai

t.N¼

16

,A

ge

ran

ge:

3–

12

yea

rsF

eatu

res:

spas

tic

dip

leg

ia,

wal

kin

gw

ith

ou

tas

sist

ive

dev

ices

,ap

pro

pri

ate

use

of

ort

ho

ses,

co-o

per

atio

nfo

ras

sess

men

t,re

gu

lar

rou

tin

eo

fP

Tb

efo

rein

clu

sio

nin

stu

dy.

Imp

rovem

ents

seen

inw

alk

ing

,ru

nn

ing

,ju

mpin

gof

GM

FM

-88

signif

ican

tly

mo

rep

ron

ou

nce

daf

ter

ind

ivid

ual

ized

PT

(p5

0.0

5)

com

par

edto

gen

eral

trai

nin

gp

rog

ram

.

Pro

spec

tive

do

ub

leb

lin

dre

pea

ted

mea

s-u

res

des

ign

.

Ru

ssel

lan

dG

ort

er[2

9]

Det

erm

ine

sen

siti

vit

yo

fG

MF

M-8

8to

wit

hin

-ch

ild

chan

ges

du

eto

use

of

amb

ula

tory

aid

or

ort

ho

ses.

N¼

25

7,

Ag

era

nge:

2–

15

yea

rsF

eatu

res:

all

typ

eso

fC

P,

GM

FC

Sle

vel

sI–

V.

Pai

red

t-te

sts

ind

icat

edim

pro

vem

ents

inG

MF

M-8

8w

ith

use

of

ort

ho

sis

and

/or

amb

ula

tory

aid

e.S

ign

ific

ant

chan

ges

var

ied

by

GM

FC

Sle

vel

for

ort

ho

sis

gro

up

.

RC

T.

Mey

er-H

eim

etal

.[3

0]

Mea

sure

fun

ctio

nal

gai

tim

pro

vem

ents

of

rob

oti

c-as

sist

edlo

com

oti

on

trai

nin

gin

chil

dre

nw

ith

CP.

N¼

22

,A

ge

ran

ge:

4–

12

yea

rsF

eatu

res:

chil

dre

nw

ith

CP,

GM

FC

Sle

vel

sII

–IV

,w

ith

ou

tad

dit

ion

altr

eat-

men

tw

ith

Bo

tox

or

surg

ery

wit

hin

the

last

3m

on

ths

bef

ore

the

stu

dy.

Excl

ud

edch

ild

ren

wit

hse

ver

eL

Eco

ntr

actu

res,

frac

ture

s,o

sseo

us

inst

abil

itie

s,o

steo

po

rosi

s,d

isp

rop

or-

tio

nal

bo

ne

gro

wth

,u

nh

eale

dsk

inle

sio

ns

of

LE

,th

rom

bo

emb

oli

cd

is-

ease

,ca

rdio

vas

cula

rin

stab

ilit

yan

dag

gre

ssiv

eo

rse

lf-h

arm

ing

beh

avio

rs.

Th

em

ean

sco

reo

fd

imen

sio

nD

of

the

GM

FM

-66

imp

roved

sig

nif

ican

tly

(p5

0.0

5)

afte

rin

terv

enti

on

per

iod

.T

he

mea

nsc

ore

of

dim

ensi

on

Eo

fth

eG

MF

M-6

6sh

ow

edin

crea

seb

ut

did

no

tre

ach

stat

isti

csi

gn

ific

ance

.

Are

pea

ted

mea

sure

des

ign

.

Dav

iset

al.

[31

]E

xam

ine

imp

act

of

ther

apeu

tic

ho

rseb

ack

rid

ing

on

fun

ctio

n,

hea

lth

and

qu

alit

yo

fli

feo

fch

ild

ren

wit

hC

P.

N¼

99

,A

ge

ran

ge:

4–

12

yea

rsF

eatu

res:

chil

dre

nat

GM

FC

Sle

vel

sI–

III

wit

ho

ut

pri

or

ho

rse

rid

ing

exp

erie

nce

.

No

stat

isti

call

ysi

gn

ific

ant

dif

fere

nce

inG

MF

M-6

6af

ter

the

inte

rven

tio

np

erio

d(p¼

0.4

5).

Pre

–p

ost

test

des

ign

.

Ch

enet

al.

[32

]D

eter

min

eth

efe

asib

ilit

yo

fu

sin

go

lfac

-to

ryen

shea

thin

gce

lls

totr

eat

chil

dre

nan

dad

ole

scen

tsw

ith

CP.

N¼

33

,A

ge

ran

ge:

1–

12

yea

rsF

eatu

res:

chil

dre

nw

ith

no

his

tory

of

exp

erim

enta

lm

edic

ine

or

trea

tmen

tsw

ith

inth

ela

st1

.5yea

rs,

no

med

ical

con

dit

ion

that

inte

rfer

edw

ith

the

mea

sure

men

tsto

ols

or

pro

hib

ited

par

-ti

cip

atio

n,

no

his

tory

of

DM

nee

din

gin

suli

n,

ble

edin

gab

no

rmal

itie

so

rim

mu

no

def

icie

ncy

.E

xcl

ud

edch

ild

ren

wh

oh

adan

yp

reex

isti

ng

con

dit

ion

likel

yto

resu

ltin

dea

thw

ith

inth

en

ext

12

mo

nth

s,h

adan

yin

fect

ion

sat

the

site

of

surg

ery,

chil

dre

nw

ith

lack

of

fam

ily

sup

po

rt.

Fo

llow

ing

trea

tmen

tG

MF

M-6

6sc

ore

of

the

trea

tmen

tg

rou

psi

gn

ific

antl

yim

pro

ved

(p¼

0.0

37

).F

or

con

tro

lg

rou

pG

MF

M-6

6sc

ore

dec

reas

edb

ut

no

tsi

gn

ific

antl

y(p¼

0.3

07

).

Sm

all

case

seri

esw

ith

in-s

ub

ject

com

-p

aris

on

des

ign

(pil

ot

stu

dy

).

So

rsd

ahl

etal

.[3

3]

Inves

tigat

eim

pac

to

fin

ten

sive,

sho

rtte

rmg

rou

p-b

ased

,g

oal

-dir

ecte

dP

To

nm

oto

rfu

nct

ion

,qu

alit

yo

fm

ovem

ents

and

ever

yd

ayac

tiv

itie

s.

N¼

22

,A

ge

ran

ge

3–

9yea

rs.

Fea

ture

s:ch

ild

ren

wit

hal

lty

pes

of

CP

atal

lG

MF

CS

and

MA

CS

level

s.

Mai

nef

fect

of

tim

ew

assh

ow

nin

GM

FM

-6

6m

ean

chan

ge

(p5

0.0

1).

Sig

nif

ican

tim

pro

vem

ent

inG

MP

Msc

ore

sfo

un

din

item

so

fG

MF

Mw

hic

hsh

ow

edim

pro

vem

ent.

Pre

–p

ost

test

stu

dy

des

ign

.

4 M. Alotaibi et al. Disabil Rehabil, Early Online: 1–11

Dis

abil

Reh

abil

Dow

nloa

ded

from

info

rmah

ealth

care

.com

by

Uni

vers

ity L

ibra

ry U

trec

ht o

n 08

/17/

13Fo

r pe

rson

al u

se o

nly.

Bar

-Hai

met

al.

[34

]D

eter

min

eco

ntr

ibu

tio

no

fra

nd

om

per

-tu

rbat

ion

sto

gro

ssm

oto

rfu

nct

ion

and

mec

han

ical

effi

cien

cyd

uri

ng

inte

nsi

ve

PT

.

N¼

20

,A

ge

ran

ge:

5.9

–1

2.9

yea

rsF

eatu

res:

chil

dre

nat

GM

FC

Sle

vel

sI,

III,

and

IV,

no

ort

ho

ped

icsu

rger

yo

rsp

asti

city

-red

uct

ion

inte

rven

tio

nd

uri

ng

the

pre

vio

us

6m

on

ths,

no

maj

or

con

trac

ture

so

flo

wer

lim

bs

mu

scle

s.

GM

FM

-66

sco

res

incr

ease

db

y�

1in

bo

thg

rou

ps.

RC

Tu

sin

ga

mu

ltip

leb

asel

ine

des

ign

.

To

ms

etal

.[3

5]

Co

mp

are

two

typ

eso

fca

nes

.N¼

18

4,

Age

ran

ge:

4–

11

yea

rsF

eatu

res:

chil

dre

nat

GM

FC

Sle

vel

sII

Iin

go

od

hea

lth

.

No

n-s

tati

stic

ally

sig

nif

ican

tch

ang

ein

sco

res

inG

MF

M-8

8.

RC

T,

pro

spec

tive.

Par

ket

al.

[36

]D

eter

min

eb

enef

ito

fse

rial

cast

ing

and

bo

tuli

nu

mto

xin

typ

eA

inje

ctio

no

nam

bu

lati

on

of

chil

dre

nw

ith

aneq

uin

us

foo

tp

rese

nta

tio

n.

N¼

38

,A

ge

ran

ge:

2.3

–8

.3yea

rsF

eatu

res:

chil

dre

nw

alk

ind

epen

den

tly

wit

ho

ut

aid

s,h

adan

equ

inu

sfo

ot

wit

hm

ild

sho

rten

ing

of

the

calf

mu

scle

that

pre

ven

tsan

kle

do

rsif

lex

ion

pas

t1

0�

wit

hk

nee

exte

nd

edh

adn

ot

yet

dev

el-

op

edjo

int

or

bo

ne

def

orm

itie

s,h

adn

ot

had

ort

ho

ped

icsu

rger

y.

Dim

ensi

on

Do

fG

MF

M-6

6si

gnif

ican

tly

imp

roved

ing

rou

pA

bu

tn

ot

ing

rou

pB

(co

ntr

ol

gro

up

).T

her

ew

ere

no

sig

nif

ican

tch

ang

esin

dim

ensi

on

Ein

GM

FM

-66

inei

ther

gro

up

.

Pro

spec

tive

lon

git

ud

inal

inte

rven

tio

nst

ud

y.

Sch

olt

eset

al.

[37

]D

eter

min

eth

eco

mb

ined

mu

ltil

evel

bo

tu-

lin

um

tox

inty

pe

Aan

dco

mp

reh

ensi

ve

reh

abil

itat

ion

on

mo

bil

ity.

N¼

46

,A

ge

ran

ge:

4–

12

yea

rsF

eatu

res:

chil

dre

nd

iag

no

sed

wit

hsp

asti

ch

emip

leg

iao

rd

iple

gia

,G

MF

CS

level

sI–

IV,

spas

tici

tyin

2o

rm

ore

LE

mu

scle

gro

up

sin

terf

erin

gw

ith

mo

bil

ity,

gai

tch

arac

teri

zed

by

per

sist

ent

flex

ion

of

the

kn

ee(�

10

)in

mid

-sta

nce

,2

or

mo

rem

usc

leg

rou

ps

in1

lim

bn

eed

ing

BT

X-A

inje

ctio

n,

un

der

stan

din

stru

ctio

ns,

adeq

uat

ek

now

led

ge

of

the

Du

tch

lan

gu

age.

Co

mb

ined

trea

tmen

tp

rov

ided

asi

gn

ifi-

can

tly

gre

ater

imp

rovem

ent

at1

2(p¼

0.0

2)

and

24

wee

ks

(p5

0.0

1)

on

the

GM

FM

-66

.

Pre

–p

ost

test

stu

dy.

Ch

rist

ian

sen

and

Lan

ge

[38

]C

om

par

eth

eef

fect

of

inte

rmit

ten

tv.

con

tin

uo

us

PT

.N¼

25

,A

ge

ran

ge:

1–

8.1

yea

rsF

eatu

res:

chil

dre

nw

ith

GM

FC

Sle

vel

sI–

V,

excl

ud

edch

ild

ren

wh

ose

par

ents

nee

ded

anin

terp

rete

r,an

dw

ho

wer

eca

nd

idat

esfo

rsu

rger

yo

rm

edic

atio

nth

atm

igh

tin

flu

ence

the

ou

tco

me

mea

sure

s.

GM

FM

-66

sco

res

incr

ease

dd

uri

ng

inte

r-ven

tio

nfo

rin

term

itte

nt

gro

up

(p¼

0.0

28

)an

dco

nti

nu

ou

sg

rou

p(p¼

0.0

38

).N

osi

gn

ific

ant

dif

fere

nce

com

par

ing

del

tasc

ore

sb

etw

een

gro

up

s(p¼

0.8

1).

Co

mp

lian

cew

assi

gn

ifi-

can

tly

hig

her

inin

term

itte

nt

gro

up

(p¼

0.0

05

),n

oas

soci

atio

nb

etw

een

GM

FM

sco

rean

dco

mp

lian

ce.

Pro

spec

tive,

ran

do

miz

edco

ntr

oll

edd

esig

n.

Low

ing

etal

.[3

9]

Inves

tigat

eg

ross

mo

tor

fun

ctio

nan

dg

oal

atta

inm

ent

inch

ild

ren

wit

hC

Pb

efo

re,

du

rin

g,

afte

rg

oal

-dir

ecte

dfu

nct

ion

alth

erap

y,to

eval

uat

eb

od

yfu

nct

ion

s,an

dex

plo

rere

lati

on

ship

s.

N¼

22

,A

ge

ran

ge:

1–

6yea

rsF

eatu

res:

un

ilat

eral

or

bil

ater

alC

P,

GM

FC

San

dM

AC

Sle

vel

sI–

V,

un

der

stan

din

stru

ctio

ns,

excl

ud

edan

yo

ne

wh

oh

ado

rth

op

edic

surg

ery

or

exte

nsi

ve

trea

tmen

tw

ith

oth

erin

ter-

ven

tio

ns

du

rin

gth

eti

me

of

the

stu

dy.

Imp

rovem

ents

dem

on

stra

ted

inG

MF

M-

66

du

rin

gin

terv

enti

on

(mea

nd

iffe

r-en

ce:

5.0

7,

CI:

3.8

–6

.4,

p5

0.0

01

).

5-y

ear

lon

git

ud

inal

stu

dy.

(co

nti

nu

ed)

DOI: 10.3109/09638288.2013.805820 A literature review 5

Dis

abil

Reh

abil

Dow

nloa

ded

from

info

rmah

ealth

care

.com

by

Uni

vers

ity L

ibra

ry U

trec

ht o

n 08

/17/

13Fo

r pe

rson

al u

se o

nly.

Tab

le1

.C

on

tin

ued

Stu

dy

auth

ors

Pu

rpo

seS

amp

lesi

zean

dfe

atu

res

Res

ult

sR

esea

rch

des

ign

No

rdm

ark

etal

.[4

0]

Eval

uat

elo

ng

-ter

mfu

nct

ion

alo

utc

om

esan

dsi

de-

effe

cts

5-y

ears

po

stS

DR

.N¼

35

,A

ge

ran

ge:

2.5

–6

.6yea

rsF

eatu

res:

dip

leg

icC

Pu

nd

erg

oin

gS

DR

,G

MF

CS

level

sI–

V.

Fo

rG

MF

CS

sub

gro

up

sI,

II,I

IIan

dIV

–V

sig

nif

ican

tim

pro

vem

ents

du

rin

gth

efi

ve

yea

rsw

ere

seen

inca

pac

ity

of

gro

ssm

oto

rfu

nct

ion

(p¼

0.0

01

).

Pre

–p

ost

test

des

ign

.

Dep

edib

iet

al.

[41

]D

eter

min

eth

eef

fect

of

bo

tuli

nu

mto

xin

typ

eA

inco

nju

nct

ion

wit

hP

To

nsp

asti

city

and

fun

ctio

nal

dev

elo

pm

ent.

N¼

18

,A

ge

ran

ge:

3–

10

yea

rsF

eatu

res:

excl

ud

edan

ych

ild

wit

hst

atic

def

orm

itie

sac

com

pan

ied

by

sever

eat

het

osi

san

dth

ose

wit

hID

wh

oco

uld

no

tco

mm

un

icat

e.

GM

FM

(p5

0.0

01

)im

pro

ved

sig

nif

i-ca

ntl

y.T

he

GM

FM

ver

sio

nis

no

tre

po

rted

inth

est

ud

y.

Pro

spec

tive,

pre

–p

ost

test

des

ign

.

Fo

ote

r[4

2]

Det

erm

ine

effe

cts

of

ther

apeu

tic

tap

ing

on

sitt

ing

con

tro

lin

chil

dre

nw

ith

CP.

N¼

18

,A

ge

ran

ge:

3.4

–1

1.7

yea

rsF

eatu

res:

chil

dre

nw

ith

spas

tic

qu

adri

-p

leg

iaan

dat

het

osi

s,G

MF

CS

level

sIV

and

V,

rece

ivin

gP

Tfo

ra

min

imu

mo

f1

ho

ur/

mo

nth

,in

go

od

hea

lth

,u

nab

leto

sit

inco

nven

tio

nal

clas

sro

om

chai

rw

ith

ou

tas

sist

ance

,an

dn

ot

hav

ing

par

tici

pat

edin

any

pre

vio

us

tria

lsw

ith

adh

esiv

eta

pe

totr

un

km

usc

ula

ture

.

No

sig

nif

ican

td

iffe

ren

ces

fou

nd

for

the

GM

FM

-88

sco

res

bet

wee

ng

rou

ps

over

tim

e(p5

0.0

5).

Pil

ot

stu

dy

(are

pea

ted

mea

sure

sd

esig

n).

Kn

ox

and

Evan

s[4

3]

Eval

uat

eth

eef

fect

so

fB

ob

ath

ther

apy

inch

ild

ren

wit

hC

P.

N¼

15

,A

ge

ran

ge:

2–

12

yea

rsF

eatu

res:

chil

dre

nw

ith

all

typ

eso

fC

Pat

GM

FC

Sle

vel

sI–

IVE

xcl

usi

on

crit

eria

:re

ceip

to

fm

edic

alp

roce

du

res

likel

yto

affe

ctm

oto

rfu

nct

ion

such

asB

oto

xo

ro

rth

op

edic

surg

ery.

Sig

nif

ican

tim

pro

vem

ent

inG

MF

Mto

tal

sco

re(p¼

0.0

09

)an

dg

oal

sco

re(p¼

0.0

01

)fo

llow

ing

Bo

bat

hth

erap

y.T

he

GM

FM

ver

sio

nis

no

tre

po

rted

inth

est

ud

y.

Lo

ng

itu

din

alan

dst

rati

fied

anal

ysi

s.

Ko

nd

oet

al.

[44

]D

eter

min

eef

fect

iven

ess

of

mu

scle

rele

ase

surg

ery

for

chil

dre

nw

ith

CP

usi

ng

lon

git

ud

inal

and

stra

tifi

edan

alysi

s.

N¼

25

,A

ge

ran

ge:

4–

16

yea

rsF

eatu

res:

chil

dre

nsp

asti

cd

iple

gia

,qu

adri

ple

gia

,an

dat

het

osp

asti

cqu

adri

ple

gia

,G

MF

CS

level

sI–

IV.

Sig

nif

ican

td

iffe

ren

cefo

un

daf

ter

surg

ery

inG

MF

CS

level

sII

I,IV

(p5

0.0

5).

Imp

rovem

ent

inG

MF

Msc

ore

sb

etw

een

1w

eek

bef

ore

surg

ery

and

12

mo

nth

saf

ter

surg

ery

fou

nd

inal

lG

MF

CS

level

s.T

he

GM

FM

ver

sio

nis

no

tre

po

rted

inth

est

ud

y.

Pre

–p

ost

test

stu

dy.

Lee

[45

]A

sses

sth

eef

fect

iven

ess

of

LE

stre

ng

then

ing

exer

cise

sto

imp

rove

mu

scle

stre

ng

than

dgai

t.

N¼

16

,A

ge

ran

ge:

4–

12

yea

rsF

eatu

res:

spas

tic

dip

leg

ico

rh

emip

le-

gic

,G

MF

CS

level

sII

–II

I,E

xcl

ud

edif

they

wer

en

ot

able

tofo

llow

com

man

ds

fro

mth

erap

ists

,h

adfi

xed

con

trac

ture

atth

ek

nee

or

hip

join

t42

5� ,

had

con

dit

ion

sp

reven

tin

gex

erci

seo

rh

ado

rth

op

edic

surg

ery

of

LE

or

inje

ctio

no

fan

anti

-sp

asti

cd

rug

.

GM

FM

score

Dan

dE

signif

ican

tly

imp

roved

inth

eex

per

imen

tal

gro

up

atp

ost

-tra

inin

g.

Th

eG

MF

Mver

sio

nis

no

tre

po

rted

inth

est

ud

y.

RC

T.

6 M. Alotaibi et al. Disabil Rehabil, Early Online: 1–11

Dis

abil

Reh

abil

Dow

nloa

ded

from

info

rmah

ealth

care

.com

by

Uni

vers

ity L

ibra

ry U

trec

ht o

n 08

/17/

13Fo

r pe

rson

al u

se o

nly.

Boy

det

al.

[10

]D

eter

min

eef

fect

iven

ess

of

bo

tuli

nu

mto

xin

typ

eA

and

SW

AS

Hfo

rch

ild

ren

wit

hm

od

erat

eto

sever

eC

P.

Det

erm

ine

iftr

eatm

ent

affe

cts

lon

g-t

erm

hip

dis

-p

lace

men

tan

dn

eed

for

surg

ery.

N¼

39

,A

ge

ran

ge:

1.7

–4

.10

yea

rs.

Fea

ture

s:b

ilat

eral

spas

tic

CP

wit

hh

ipd

isp

lace

men

t,G

MF

CS

level

sII

–V

Excl

usi

on

crit

eria

:fi

xed

con

trac

ture

of

the

hip

add

uct

ors

,w

ith

anab

du

ctio

nra

nge5

20�

inei

ther

hip

or

aco

mb

ined

abd

uct

ion

ran

ge

of5

40� ,

pre

vio

us

hip

surg

ery,

init

ial

MP4

40

%,

hip

flex

ion

con

trac

ture

so

f4

30� ,

sco

lio

sis

wit

hC

ob

ban

gle4

20� .

Bo

thg

rou

ps

show

edn

on

-sig

nif

ican

tch

anges

into

tal

GM

FM

sco

re.

Sim

ilar

imp

rovem

ents

on

GM

FM

go

alsc

ore

san

dG

MF

M-6

6sc

ore

s,w

ith

no

add

-it

ion

altr

eatm

ent

effe

cts

Mu

ltip

lere

gre

ssio

no

fch

ang

ein

tota

lG

MF

Mby

GM

FC

Sfo

rea

chg

rou

psh

ow

edg

reat

erim

pro

vem

ent

inth

eto

tal

sco

res

fro

mb

asel

ine

inB

TX

-A/S

WA

SH

trea

ted

gro

up

than

con

tro

lg

rou

p.

Th

eG

MF

Mver

sio

nis

no

tre

po

rted

inth

est

ud

y.

No

n-r

and

om

ized

pro

spec

tive

des

crip

tive

stu

dy.

En

gsb

erg

etal

.[4

6]

Co

mp

are

ou

tco

mes

of

3p

roce

du

res

(hee

lco

rdad

van

cem

ent,

hee

lco

rdle

ng

then

ing

acco

rdin

gto

Vu

lpiu

s,an

dh

eel

cord

len

gth

enin

gac

cord

ing

toW

hit

e)in

chil

dre

nw

ith

CP.

N¼

32

chil

dre

nM

ean

age

for

the

3g

rou

ps

4.3

,6

.8,

8.6

Fea

ture

s:sp

asti

cd

iple

gia

,G

MF

CS

level

sI.

All

surg

erie

sin

dic

ated

sig

nif

ican

tim

pro

vem

ents

inG

MF

M-8

8.

RC

T.

Tra

han

and

Mal

ou

in[4

7]

Det

erm

ine

effe

ctiv

enes

so

fsh

ort

-ter

min

ten

sive

ther

apy

for

chil

dre

nw

ith

sever

eC

P.

N¼

5,

Age

ran

ge:

10

–3

7m

on

ths

Fea

ture

s:G

MF

CS

level

sIV

and

V.

Excl

ud

edch

ild

ren

wh

ow

ere

can

di-

dat

esfo

rsu

rger

yo

rw

ith

any

oth

erco

nd

itio

ns

that

cou

ldp

ote

nti

ally

mo

dif

yth

ere

hab

ilit

atio

np

rog

ram

.

Sig

nif

ican

tin

crea

ses

inG

MF

M-8

8sc

ore

sin

thre

ech

ild

ren

(p5

0.0

5)

and

all

par

tici

pan

tsm

ain

tain

edth

eir

mo

tor

per

form

ance

du

rin

gth

etw

o8

-wee

kre

stp

erio

ds.

Th

ein

term

itte

nt

pro

gra

mle

dto

imp

rovem

ents

inm

oto

rfu

nct

ion

that

wer

em

ain

tain

edover

the

rest

per

iod

s.

Mu

ltip

le-b

asel

ine

des

ign

(pil

ot

stu

dy

).

GM

FM¼

Gro

ssM

oto

rF

un

ctio

nM

easu

re,

PT¼

Physi

cal

Th

erap

y,C

P¼

Cer

ebra

lP

alsy

,G

MF

CS¼

Gro

ssM

oto

rF

un

ctio

nC

lass

ific

atio

nS

yst

em,

RC

T¼

Ran

do

miz

edC

lin

ical

Tra

il,

LE¼

Low

erE

xtr

emit

y,D

M¼

Dia

bet

esM

elli

tus,

MA

CS¼

Man

ual

Ab

ilit

yC

lass

ific

atio

nS

yst

em,

GM

PM¼

Gro

ssM

oto

rP

erfo

rman

ceM

easu

re,

BT

X-A¼

Bo

tuli

nu

mT

ox

inT

yp

eA

,C

I¼

Co

nfi

den

ceIn

terv

al,

SD

R¼

Sel

ecti

ve

Do

rsal

Rh

izo

tom

y,ID¼

Inte

llec

tual

Dis

abil

ity,

SW

AS

H¼

Sit

tin

gW

alk

ing

and

Sta

nd

ing

Hip

,M

P¼

Mu

scle

Pow

er.

DOI: 10.3109/09638288.2013.805820 A literature review 7

Dis

abil

Reh

abil

Dow

nloa

ded

from

info

rmah

ealth

care

.com

by

Uni

vers

ity L

ibra

ry U

trec

ht o

n 08

/17/

13Fo

r pe

rson

al u

se o

nly.

change. Davis et al. [31] stated in their study on 99 children withCP and various levels of impairment that a 10-week specifictraining program of therapeutic horse riding did not increase theGMFM score compared to a control group. The scores of theGMFM were used in combination with additional quality of lifescores. A lack of measurable improvement in the outcome scoresprovided evidence for these researchers to conclude this horseriding intervention program was not effective in demonstratingclinical change in gross motor function for participants in thestudy. Toms et al. [35] used the GMFM to test benefits of aprototype walking aid as compared to conventional walking aidsin a pilot study of eight children with CP at a GMFCS Level III.The results showed improvement in GMFM scores for some butnot all children with overall outcomes not statistically significant.Conclusions of this study did provide insight regarding the marketability of the walking aid and data to assist in improving thedesign of assistive walking equipment for children with CP. Thesefindings were similar to the study that assessed the effects ofusing therapeutic taping to impact sitting performance in 18children with CP [42]. The GMFM failed to detect any significantfunctional changes that could be attributed to a taping protocol.The lack of clinical change may have been related to the severityof the motor involvement of the children studied as most of thechildren were classified at lower levels of function according tothe GMFCS. Both these studies, however addressed gross motorskills that are highlighted within the GMFM protocol, walkingand sitting thus the tool was probably the most appropriate.

The GMFM has also been used to describe the status ofchildren’s gross motor performance across a variety of conditions.Russell and Gorter [29] gathered data using GMFM-88 scores ina within child research program to identify functional differencesin children using a walking aid and orthoses as compared to thesame children walking barefoot. The results provide evidence thatGMFM scores are able to document differences based on withinchild variables related to mobility. A number of studies supportthese findings [10,36,37,41]. For example, Sorsdahl et al. [33]found an interaction between GMFM scores and the child specificGMFCS, indicating that children classified as level I or II hadhigher GMFM scores than children classified as levels III or IV.The results also suggested a significant interaction between timepassage and GMFCS level implying that children classified atGMFCS level I–II exhibited a greater improvement in GMFMscores over time as compared to children at levels III–V.

Christiansen et al. [38] found that GMFM scores increasedsignificantly in children who received intermittent or continuousphysical therapy. Trahan and Malcouin [47] also studied theeffects of intermittent intensive physical therapy in children withCP. These researchers used the total GMFM score as a primaryoutcome measure to document that increasing the frequency oftreatment, from twice a week to four times a week, improved thelevel of motor performance. The greatest improvement in theGMFM scores was seen in the children with less severe physicalimpairment and who were cooperative. These results also supportSorsdahl et al. [33] in which there was a strong correlationbetween GMFM scores and the GMFCS. Nordmark et al. [40]study also support these findings.

Contrary to these findings, Lowing et al. [39] in their study of22 children with CP who underwent goal-directed functionaltherapy found that children made obvious improvement inGMFM, irrespective of GMFCS level. Kondo et al. [44] reportedsimilar findings. These researchers reported significant changeafter selective muscle release surgery in children at GMFCS levelIII and IV with no significant improvement in the GMFCS level Iand II. This lack of improvement might occur because the aim ofthe surgery was weighted more for the correction of deformitythan for the improvement of motor function. In other words, theyT

able

2.

Mo

dif

ied

crit

ical

rev

iew

form

for

qu

anti

tati

ve

stu

die

s(L

awet

al.)

[48

].

Cat

ego

ryQ

ues

tio

nO

per

atio

nal

def

init

ion

Stu

dy

pu

rpo

se1

.W

asth

ep

urp

ose

stat

edcl

earl

y?

Ap

pli

cati

on

of

stu

dy

toth

erap

euti

cin

terv

enti

on

.

Lit

erat

ure

2.

Was

rele

van

tb

ack

gro

un

dli

tera

ture

rev

iew

ed?

Just

ific

atio

no

fst

ud

yw

asd

escr

ibed

.

Sam

ple

3.

Was

the

sam

ple

des

crib

edin

det

ail?

Was

pow

erd

iscu

ssed

?4

.W

asth

esa

mp

lesi

zeju

stif

ied

?T

he

char

acte

rist

ics

of

sam

ple

pro

vid

ed,

the

nu

mb

ero

fch

ild

ren

inth

esa

mp

lecl

earl

yst

ated

,g

rou

ps

wer

ecl

earl

yd

iffe

ren

tiat

ed.

Des

crib

eet

hic

sp

roce

du

re.

Was

info

rmed

con

sen

to

bta

ined

?

Ou

tco

mes

5.

Wer

eth

eo

utc

om

esm

easu

res

reli

able

&val

id?

Sp

ecif

yth

efr

equ

ency

of

ou

tco

me

mea

sure

san

do

utc

om

esu

chas

self

-car

e,p

rod

uct

ivit

y,le

isu

re.

All

mea

sure

su

sed

wer

ed

escr

ibed

.

Inte

rven

tio

n6

.In

terv

enti

on

was

des

crib

edin

det

ail?

7.

Co

nta

min

atio

n&

co-i

nte

rven

tio

nw

asav

oid

ed?

Des

crip

tio

no

fin

terv

enti

on

pro

vid

ed:

focu

s,w

ho

del

iver

edit

,h

ow

oft

en,

sett

ing

.C

ou

ldth

ein

terv

enti

on

be

rep

lica

ted

inp

ract

ice?

Res

ult

s8

.R

esu

lts

wer

ere

po

rted

inte

rms

of

stat

isti

cal

sig

nif

ican

ce?

9.

Wer

eth

ean

alysi

sm

eth

od

(s)

app

rop

riat

e?1

0.

Cli

nic

alim

po

rtan

cew

asre

po

rted

?1

1.

Dro

po

uts

wer

ere

po

rted

?

Res

ult

sd

escr

ibed

and

stat

isti

cal

sig

nif

ican

cep

rov

ided

,li

mit

atio

ns

of

stu

dy

pro

vid

ed,

dis

cuss

edef

fect

so

fu

sin

gm

ult

iple

ou

tco

mes

.D

iscu

ssed

clin

ical

imp

ort

ance

of

resu

lts

and

dif

fere

nce

sb

etw

een

gro

up

s.D

iscu

ssed

nu

mb

eran

dre

aso

ns

for

dro

p-o

uts

.

Co

ncl

usi

on

san

dcl

inic

alim

pli

cati

on

s1

2.

Co

ncl

usi

on

sw

ere

app

rop

riat

eg

iven

stu

dy

met

ho

ds

and

resu

lts

Co

ncl

usi

on

san

dim

pli

cati

on

scl

earl

yst

ated

and

dis

cuss

edin

wh

atw

ere

the

mai

nli

mit

atio

ns

or

bia

ses

inth

est

ud

y?

8 M. Alotaibi et al. Disabil Rehabil, Early Online: 1–11

Dis

abil

Reh

abil

Dow

nloa

ded

from

info

rmah

ealth

care

.com

by

Uni

vers

ity L

ibra

ry U

trec

ht o

n 08

/17/

13Fo

r pe

rson

al u

se o

nly.

Table 4. Calculated quality score of studies determined by modified Critical Review Form for Quantitative Studies and level of evidence determined bystudy design.

CriteriaScores Scores %

Study 1 2 3 4 5 6 7 8 9 10 11 12 12/12 100% Level of evidence

Van den Broeck et al. [28] Y Y Y N Y Y Y Y Y N N Y 9/12 75% III-2Russell et al. [29] Y Y Y N Y Y Y Y Y Y N Y 10/12 83.33% IIMeyer-Heim et al. [30] Y Y Y N Y Y Y Y Y Y N Y 10/12 83.33% III-3Davis et al. [31] Y Y Y Y Y Y N Y Y Y Y Y 11/12 91.66% IVChen et al. [32] Y Y Y N N Y Y Y Y Y N Y 9/12 75% IVSorsdah et al. [33] Y Y Y N Y Y Y Y Y Y Y Y 11/12 91.66% IVBar-Haim et al. [34] Y Y Y N Y Y Y Y Y Y N Y 10/12 83.33% IIToms et al. [35] Y Y Y Y Y N Y Y Y Y Y Y 11/12 91.66% IIPark et al. [36] Y Y Y N Y Y Y Y Y Y N Y 10/12 83.33% III-2Scholtes et al. [37] Y Y Y N Y Y Y Y Y Y Y Y 11/12 91.66% IVChristiansen et al. [38] Y Y Y N Y Y Y Y Y Y Y Y 11/12 91.66% IILowing et al. [39] Y Y Y Y Y Y Y Y Y N N Y 10/12 83.33% III-2Nordmark et al. [40] Y Y Y N Y Y N Y Y Y N Y 9/12 75% IVDepedibi et al. [41] Y Y Y N Y Y N Y Y Y N Y 9/12 75% IVFooter et al. [42] Y Y Y Y Y Y N Y Y Y N Y 10/12 83.33% III-2Knox et al. [43] Y Y Y Y Y Y N Y Y Y Y Y 11/12 91.66% III-2Kondo et al. [44] Y Y Y Y N Y Y Y Y Y N Y 10/12 83.33% IVLee et al. [45] Y Y Y Y Y Y N Y Y Y N Y 10/12 83.33% IIBoyd et al. [10] Y Y Y N Y Y Y Y Y Y N Y 10/12 83.33% III-2Engsberg et al. [46] Y Y Y N Y Y N Y Y Y N Y 9/12 75% IITrahan et al. [47] Y Y Y Y Y Y Y Y Y Y Y Y 12/12 100% III-3

Y¼ yes; N¼ no; na¼ not addressed; NA¼ not applicable; Criteria: 1. study purpose clearly stated; 2. background literature reviewed; 3. sampledescribed in detail; 4. sample size justified; 5. outcome measure reliability and validity reported; 6. intervention described; 7. contamination and co-intervention avoided; 8. results reported in terms of statistical significance; 9. analysis methods appropriate; 10. clinical significance reported;11. drop-outs reported; 12. conclusions appropriate.

Level of evidence based on the designation used by the National Health and Medical Research Council (NHMRC) [49].

Figure 1. Flow diagram of search strategy.

Table 3. Levels of evidence (National Health and Medical Research Council) [49].

Level Study design

I Evidence obtained from a systematic review of all relevant randomized controlled trials.

II Evidence obtained from at least one properly-designed randomized controlled trial.

III-1 Evidence obtained from well-designed pseudo-randomized controlled trials (alternative allocation or some other method).

III-2 Evidence obtained from comparative studies (including systematic reviews of such studies) with concurrent controls and allocation notrandomized, cohort studies, case–control studies or interrupted time series with a control group.

III-3 Evidence obtained from comparative studies with historical control, two or more single arm studies or interrupted time series without aparallel control group.

IV Evidence obtained from case series, either post-test or pre-test/post-test.

DOI: 10.3109/09638288.2013.805820 A literature review 9

Dis

abil

Reh

abil

Dow

nloa

ded

from

info

rmah

ealth

care

.com

by

Uni

vers

ity L

ibra

ry U

trec

ht o

n 08

/17/

13Fo

r pe

rson

al u

se o

nly.

found very little change in GMFM score, whereas there wasremarkable improvement in gait parameters after surgery whichcould not be assessed with the GMFM.

There are many aspects affecting the studies’ quality includedsuch as not reporting clinical significance, drop-outs, contamin-ation and co-intervention, reliability and validity of outcomemeasures, unjustified sample size and intervention was notdescribed in detail. The advantages and disadvantages of theGMFM-88 and GMFM-66 have been widely reported. Consistentwith contemporary practice incorporating the InternationalClassification of Function, the GMFM has been criticized forexamining gross motor skill function only without measuring thechild’s activity and participation levels [12,53]. Furthermore,GMFM-88 and GMFM-66 measure child performance accordingto a controlled test protocol instead of within his/her naturalenvironment. The GMFM-88 is lengthy requiring children tocomplete all 88 test items to arrive at a valid score, which is adisadvantage for busy practitioners who have limited amount oftime to complete a thorough assessment of skill performance. TheGMFM-88 also has moderate ceiling and floor effects, indicatingthat only children with intermediate motor ability will havegreatest possible change with the GMFM-88 [12,54].

In comparison, the GMFM-66 has 22 fewer items than theGMFM-88 allowing for faster completion of the test and has amethod for obtaining a score for the child even in the event thatnot all items have been assessed [12,22]. This latter test also has afloor effect in children with low motor ability and ceiling effectsin children older than 5 years [12,25]. This finding means theGMFM-66 is much less useful when scoring children of low-motor ability and those older than 5 years. Although disadvan-tages have been noted, the overall preponderance of positivefindings from research supports the premise that the GMFM is auseful and accurate outcome tool for clinicians and researchers.

Conclusion

The results of this review suggest that both GMFM-88 andGMFM-66 version are useful and valuable in assessing functionalmotor abilities of children under 17 years of age with CP. Thesetools have been translated and validated in a variety of languagesand used in different cultures; adding to the usefulness of thetools. The GMFM-88 and GMFM-66 version are able to detectclinically significant change in gross motor function in childrenunder 17 years of age with CP based on children age, severity ofimpairment, intervention and frequency. Studies with largersample size showed more improvement in GMFM scorescompared with studies with smaller sample size. Moreover,combining therapy had the greatest impact on measured improve-ment in gross motor function when measured by GMFM. Somestudies needed longer time to show any improvement in theGMFM scores. The GMFM may not be sensitive for detectingfunctional change in children with severe motor disability who areat GMFCS level IV and V compared to less impaired children inGMFCS levels II and III. Also, some studies showed littleimprovement in the GMFM scores because the participants werealready functioning at a higher level in the GMFM dimensions,therefore leaving little room for improvement. Additionally,children receiving intensive program (without being tiring) andintermittent showed more improvement in GMFM scorescompared to non-intensive and continues program. Finally, werecommend clinicians consider the use of other outcome measuresin conjunction with the GMFM to provide a comprehensivepicture of a child’s functioning/activity and participation levels.

Declaration of interest

The authors report no conflicts of interest.

References

1. Bax M, Goldstein M, Rosenbaum P, et al. Proposed definition andclassification of cerebral palsy. Dev Med Child Neurol 2005;47:571–76.

2. Yeargin-Allsopp M, Van Naarden Braun K, Doernberg NS, et al.Prevalence of cerebral palsy in 8-year-old children in three areas ofthe United States in 2002: a multisite collaboration. Pediatrics 2008;121:547–54.

3. Springhouse. Professional guide to diseases. 8th ed. Ambler(PA):Lippincott Williams & Wilkins; 2005.

4. Oeffinger D, Bagley A, Rogers S, et al. Outcome tools used forambulatory children with cerebral palsy: responsiveness and min-imum clinically important differences. Dev Med Child Neurol 2008;50:918–25.

5. Parrette HP, Hourcade JJ. A review of therapeutic interventionresearch on gross and fine motor progress in young children withcerebral palsy. Am J Occup Ther 1984;38:462–8.

6. Ketelaar M, Vermeer A, Helders PJ. Functional motor abilities ofchildren with cerebral palsy: a systematic literature review ofassessment measures. Clin Rehabil 1998;12:369–80.

7. Guyatt GH, Walter SD, Norman G. Measuring change over time:assessing the usefulness of evaluative instruments. J Chronic Dis1987;40:171–80.

8. Rosenbaum PL, Russell DJ, Cadman DT, et al. Issues in measuringchange in motor function in children with cerebral palsy: a specialcommunication. Phys Ther 1990;70:125–31.

9. Krach LE, Kriel RL, Gilmartin RC, et al. GMFM 1 year aftercontinuous intrathecal baclofen infusion. Pediatr Rehabil 2005;8:207–13.

10. Boyd RN, Dobson F, Parrott J, et al. The effect of botulinum toxintype A and a variable hip abduction orthosis on gross motorfunction: a randomized controlled trial. Eur J Neurol 2001;8:109–19.

11. Russell DJ, Rosenbaum PL, Avery L, Lane M. Gross motorfunction measure (GMFM-66 and GMFM-88) user’s manual: clinicsin developmental medicine. London, England: Mac Keith Press;2002.

12. Russell DJ, Avery LM, Rosenbaum PL, et al. Improved scaling ofthe Gross Motor Function Measure for children with cerebral palsy:evidence of reliability and validity. Phys Ther 2000;80:873–85.

13. Russell D, Rosenbaum P, Gowland C, et al. Gross Motor FunctionMeasure Manual. 2nd ed. Hamilton, Canada: McMaster University;1993.

14. Russell D, Rosenbaum P, Cadman D, et al. The gross motor functionmeasure: a means to evaluate the effects of physical therapy. DevMed Child Neurol 1989;31:341–52.

15. Wei S, Su-Juan W, Yuan-Gui L, et al. Reliability and validity of theGMFM-66 in 0-3 year-old children with Cerebral Palsy. Am J PhysMed Rehabil 2006;85:141–7.

16. Russell D, Palisano R, Walter S, et al. Evaluating motor function inchildren with Down syndrome: validity of the GMFM. Dev MedChild Neurol 1998;40:693–701.

17. Gemus M, Palisano R, Russell D, et al. Using the gross motorfunction measure to evaluate motor development in children withDown syndrome. Phys Occup Ther Pediatr 2001;21:69–79.

18. Linder-Lucht M, Othmer V, Walther M, et al. Validation of theGross Motor Function Measure for use in children and adolescentswith traumatic brain injuries. Pediatrics 2007;120:880–6.

19. Ruck-Gibis J, Plotkin H, Hanley J, Wood-Dauphinee S. Reliabilityof the gross motor function measure for children with osteogenesisimperfecta. Pediatr Phys Ther 2001;13:10–7.

20. Ketelaar M, Vermeer A, Hart H, et al. Effects of a functional therapyprogram on motor abilities of children with cerebral palsy. Phys Ther2001;9:1534–45.

21. Finch E, Brooks D, Stratford P, Mayo N. Physical rehabilitationoutcome measures: a guide to enhanced clinical decision making.2nd ed. Hamilton, Ontario: BC Decker Inc; 2002.

22. Avery L, Russell D, Raina P, et al. Rasch Analysis of the GrossMotor Function Measure: validating the assumptions of the Raschmodel to create an intervallevel measure. Arch Phys Med Rehabil2003;84:697–705.

23. Damiano DL, Abel MF. Functional outcomes of strengthtraining in spastic cerebral palsy. Arch Phys Med Rehab 1998;79:119–25.

24. Vos-Vromans D, Ketelaar M, Gorter J. Responsiveness of evaluativemeasures for children with cerebral palsy: the Gross Motor Function

10 M. Alotaibi et al. Disabil Rehabil, Early Online: 1–11

Dis

abil

Reh

abil

Dow

nloa

ded

from

info

rmah

ealth

care

.com

by

Uni

vers

ity L

ibra

ry U

trec

ht o

n 08

/17/

13Fo

r pe

rson

al u

se o

nly.

Measure and the pediatric evaluation of disability inventory. DisabilRehabil 2005;27:1245–52.

25. Rosenbaum P, Walter S, Hanna S, et al. Prognosis for gross motorfunction in cerebral palsy. JAMA 2002;228:1357–63.

26. Wang H, Yang Y. Evaluating the responsiveness of 2 versions of theGross Motor Function Measure for children with cerebral palsy.Arch Phys Med Rehabil 2006;87:51–6.

27. Palfrey J, Singer J, Walker D, Butler J. Early identification ofchildren’s special needs: a study in five metropolitan communities.J Pediatr 1987;111:651–9.

28. Van den Broeck C, De Cat J, Molenaers G, et al. The effect ofindividually defined physiotherapy in children with cerebral palsy(CP). Eur J Paediatr Neurol 2010;14:519–25.

29. Russell DJ, Gorter JW. Assessing functional differences in grossmotor skills in children with cerebral palsy who use an ambulatoryaid or orthoses: can the GMFM-88 help? Dev Med Child Neurol2005;47:462–7.

30. Meyer-Heim A, Ammann-Reiffer C, Schmartz A, et al.Improvement of walking abilities after robotic-assisted locomotiontraining in children with cerebral palsy. Arch Dis Child 2009;94:615–20.

31. Davis E, Davies B, Wolfe R, et al. A randomized controlled trial ofthe impact of therapeutic horse riding on the quality of life, health,and function of children with cerebral palsy. Dev Med Child Neurol2009;51:111–19; discussion 88.

32. Chen L, Huang H, Xi H, et al. Intracranial transplant of olfactoryensheathing cells in children and adolescents with cerebral palsy: arandomized controlled clinical trial. Cell Transplantation 2010;19:185–91.

33. Sorsdahl AB, Moe-Nilssen R, Kaale HK, et al. Change in basicmotor abilities, quality of movement and everyday activitiesfollowing intensive, goal-directed, activity-focused physiotherapyin a group setting for children with cerebral palsy. BMC Pediatr2010;10:26 (1–11). doi: 10.1186/1471-2431-10-26.

34. Bar-Haim S, Harries N, Belokopytov M, et al. Random perturbation:a potential aid in treatment of children with cerebral palsy. DisabilRehabil 2008;30:1420–8.

35. Toms B, Harrison B, Bower E. A pilot study to compare the use ofprototypes of multipositionalpaediatric walking sticks and tripodswith conventional sticks and tripods by children with cerebral palsy.Child Care Health Dev 2007;33:96–106.

36. Park ES, Rha DW, Yoo JK, et al. Short-term effects of combinedserial casting and botulinum toxin injection for spastic equinus inambulatory children with cerebral palsy. Yonsei Med J 2010;51:579–84.

37. Scholtes VA, Dallmeijer AJ, Knol DL, et al. The combined effect oflower-limb multilevel botulinum toxin type A and comprehensiverehabilitation on mobility in children with cerebral palsy: arandomized clinical trial. Arch Phys Med Rehabil 2006;87:1551–8.

38. Christiansen AS, Lange C. Intermittent versus continuous physio-therapy in children with cerebral palsy. Dev Med Child Neurol 2008;50:290–3.

39. Lowing K, Bexelius A, Carlberg EB. Goal-directed functionaltherapy: a longitudinal study on gross motor function in childrenwith cerebral palsy. Disabil Rehabil 2010;32:908–16.

40. Nordmark E, Josenby AL, Lagergren J, et al. Long-term outcomesfive years after selective dorsal rhizotomy. BMC Pediatr 2008;8:54(1–15). doi: 10.1186/1471-2431-8-54.

41. Depedibi R, Unlu E, Cevikol A, et al. Ultrasound-guided botulinumtoxin type A injection to the iliopsoas muscle in the management ofchildren with cerebral palsy. NeuroRehabilitation 2008;23:199–205.

42. Footer CB. The effects of therapeutic taping on gross motor functionin children with cerebral palsy. Pediatr Phys Ther 2006;18:245–52.

43. Knox V, Evans AL. Evaluation of the functional effects of a courseof Bobath therapy in children with cerebral palsy: a preliminarystudy. Dev Med Child Neurol 2002;44:447–60.

44. Kondo I, Hosokawa K, Iwata M, et al. Effectiveness of selectivemuscle-release surgery for children with cerebral palsy: longitudinaland stratified analysis. Dev Med Child Neurol 2004;46:540–7.

45. Lee JH, Sung IY, Yoo JY. Therapeutic effects of strengtheningexercise on gait function of cerebral palsy. Disabil Rehabil 2008;30:1439–44.

46. Engsberg JR, Oeffinger DJ, Ross SA, et al. Comparison of three heelcord surgeries in children with cerebral palsy. J Appl Biomech 2005;21:322–33.

47. Trahan J, Malouin F. Intermittent intensive physiotherapy inchildren with cerebral palsy: a pilot study. Dev Med Child Neurol2002;44:233–9.

48. Law M, Stewart D, Pollock N, et al. Critical review form –quantitative studies. McMaster University: Occupational TherapyEvidence-Based Practice Research Group; 1998.

49. NHMRC: A guide to the development, implementation and evalu-ation of clinical practice guidelines. Canberra, ACT: NationalHealth and Medical Research Council, Commonwealth of Australia;1999.

50. Jarrett MS, Orlando JF, Grimmer-Somers K. The effectiveness ofland based exercise compared to decompressive surgery in themanagement of lumbar spinal-canal stenosis: a systematic review.BMC Musculoskelet Disord 2012;13:30 (1–9). doi: 10.1186/1471-2474-13-30.

51. Medlicott MS, Harris SR. A systematic review of the effectivenessof exercise, manual therapy, electrotherapy, relaxation training, andbiofeedback in the management of temporomandibular disorder.Phys Ther 2006;86:955–73.

52. Brunton LK, Bartlett DJ. Validity and reliability of two abbreviatedversions of the gross motor function measure. Phys Ther 2011;91:577–88.

53. Lundkvist Josenby A, Jarnlo GB, Gummesson C, Nordmark E.Longitudinal construct validity of the GMFM-88 total score andgoal total score and the GMFM-66 score in a 5-year follow-up study.Phys Ther 2009;89:342–50.

54. McCarthy ML, Silberstein CE, Atkins EA, et al. Comparingreliability and validity of pediatric instruments for measuringhealth and wellbeing of children with spastic cerebral palsy. DevMed Child Neurol 2002;44:265–9.

DOI: 10.3109/09638288.2013.805820 A literature review 11

Dis

abil

Reh

abil

Dow

nloa

ded

from

info

rmah

ealth

care

.com

by

Uni

vers

ity L

ibra

ry U

trec

ht o

n 08

/17/

13Fo

r pe

rson

al u

se o

nly.