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Authors:

Heidi Anttila, MSc (Health Sci), PTJutta Suoranta, MSc (Health Sci), PTAntti Malmivaara, PhD, MDMarjukka Makela, PhD, MD, MSc

(Clin Epi)Ilona Autti-Ramo, PhD, MD

Affiliations:

From the Finnish Office for HealthTechnology Assessment, NationalResearch and Development Centre forWelfare and Health, Helsinki, Finland(HA, JS, AM, MM, IAR); TampereSchool of Public Health, University ofTampere, Tampere, Finland (JS);Department of General Practice,University of Copenhagen, Denmark(MM); The Social Insurance Institute,Helsinki, Finland (IAR); andDepartment of Child Neurology,Hospital for Children andAdolescents, University of Helsinki,Finland (IAR).

Correspondence:

All correspondence and requests forreprints should be addressed to HeidiAnttila, Finnish Office for HealthTechnology Assessment, PO Box 220,FIN-00531 Helsinki, Finland.

Disclosures:

This study was funded by Finohta, anational government-fundedorganization for health technologyassessment, and by a grant from the

Academy of Finland. The authorshave no financial or personalconflicts of interest.

0894-9115/08/8706-0478/0

American Journal of Physical

Medicine & Rehabilitation

Copyright 2008 by Lippincott

Williams & Wilkins

DOI: 10.1097/PHM.0b013e318174ebed

Effectiveness of Physiotherapy andConductive Education Interventionsin Children with Cerebral PalsyA Focused Review

ABSTRACT

Anttila H, Suoranta J, Malmivaara A, Makela M, Autti-Ramo I: Effectiveness ofphysiotherapy and conductive education interventions in children with cerebralpalsy: a focused review. Am J Phys Med Rehabil 2008;87:478501.

We conducted a criteria-based appraisal of systematic reviews on the effective-ness of physiotherapy and conductive education interventions in children withcerebral palsy (CP). Computerized bibliographic databases were searched with-out language restriction up to August 2007. Reviews on trials and descriptivestudies were included. Two reviewers independently identified, selected, andassessed the quality of the reviews using the criteria from the Overview QualityAssessment Questionnaire complemented with decision rules. Twenty-one re-views were included, six of which were of high methodological quality. Altogether,the reviews included 23 randomized controlled trials and 104 observationalstudies on children with CP. The high-quality reviews found some evidencesupporting strength training, constraint-induced movement therapy, or hippo-therapy, and insufficient evidence on comprehensive physiotherapy and occupa-tional therapy interventions. Conclusions in the other reviews should be inter-preted cautiously, although, because of the poor quality of the primary studies,most reviews drew no conclusions on the effectiveness of the reviewed interven-tions. Reviews on complex interventions in heterogeneous populations should userigorous methods and report them adequately, closely following the Quality ofReporting of Meta-Analyses recommendations.

Key Words: Physiotherapy, Systematic Review, Cerebral Palsy, Quality Assessment,

Clinical Applicability

478 Am. J. Phys. Med. Rehabil. Vol. 87, No. 6

LITERATURE REVIEW

Cerebral Palsy

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The principles of evidence-based practice arewidely accepted among professionals.1,2 The stron-

gest support for evidence-based decision making

comes from updated, high-quality systematic re-

views (SR). Such reviews identify the relevant stud-

ies, appraise their quality, and summarize the re-

sults, using sound scientific methodology.3,4 They

can also help clinicians to find relevant answers to

clinical questions in a time-efficient and reliableway.3 Professionals treating children with CP often

have limited time, skills, and resources to search

for evidence and to interpret effectiveness studies.5

Cerebral palsy (CP) is an umbrella term for

nonprogressive but often-changing motor impair-

ment syndromes secondary to lesions or abnormal-

ities of the brain arising in the early stages of

development.6 Basic management of the motor

disability in CP includes physiotherapy (PT) and a

wide spectrum of other therapeutic interventions.7

Motor learning goals may also be incorporated into

educational programs such as conductive educa-tion (CE) instead of separate rehabilitation inter-

ventions provided by different professionals.8

An appreciation of the quality of an SR is

essential before deciding whether its conclusions

should be followed. Such quality may mean the

rigor of the review methods, or quality of report-

ing. Previous evaluations of SRs in many fields

imply that readers should not accept them uncriti-

cally, and there is a need for improvement of the

methodological quality and guidelines for report-

ing.9 Cochrane reviews are usually more rigorously

conducted and reported than non-Cochrane re-views.912

There are at least 24 instruments to assess the

quality of SRs.13 A rigorously developed and vali-

dated tool, the Overview Quality Assessment Ques-

tionnaire (OQAQ), has been constituted by Oxman

and Guyatt.14,15 Hoving et al.16 have slightly mod-

ified this tool and applied it in rehabilitation re-

search. The Quality of Reporting of Meta-Analyses

statement describes the preferred way to present

the abstract, introduction, methods, results, and

discussion sections of a report of meta-analysis,

including a flow diagram of the article identifica-tion and selection process.17 Balanced interpreta-

tion of the applicability and clinical relevance re-

quires accurate information of the reviewed

populations, interventions, comparison interven-

tions, and outcomes.1820

An essential feature of SRs is critical appraisal

of the methodological quality of the included pri-

mary studies.3,21 Lack of adherence to defined qual-

ity criteria may explain the different results of

studies on the same topic.22 Published SRs have

heterogeneous approaches to assess methodologi-

cal quality, and this has been infrequently reportedand incorporated into the analyses.10,23,24

In this study, we wanted to evaluate the meth-odological validity of SRs and their clinical useful-

ness when targeting a heterogeneous populationand looking at variably applied interventions such

as PT and CE in children with CP. The primaryobjective was to appraise the methodological qual-

ity of the reviews on the effectiveness of PT or CE

interventions in children with CP, and to explorewhat needs to be done to enhance the quality ofreviews. The secondary aims were to make conclu-

sions about the effectiveness of the reviewed inter-ventions, and to consider the included study designs,

populations, interventions, outcome measures, andresults of various PT interventions on children with

CP to allow interpretation of possible evidence intoclinical practice. Finally, our aim was to use all this

information to make suggestions for future studies inthis field.

METHODS

Locating and Selecting the Reviews

Only published SR articles were considered.To be included, these publications were required

to have descriptions of the searched databases,search time period, and selection criteria for

population and interventions. This review in-cluded interventions usually provided by physio-

therapists and requiring therapeutic manage-ment7for instance, neurodevelopmental therapy

(NDT), strength training, saddle riding, physical

activity, swimming programs, functional therapy,and targeted training. In addition, interventionsthat in some countries or organizations may be

provided either by physiotherapists or occupationaltherapists (upper-limb interventions) or special

teachers (CE) were included. The main focus was to

include reviews on therapeutic management with-out specialized equipment; thus, interventions of

solely devices (electrical stimulation, biofeedback,orthotic, or other assistive devices) were excluded.

The patients were children or adolescents (aged 3mos to 20 yrs) with diagnosed CP.

If the review had included other interventionsor populations, it was included only if at least 80%

of the included populations or interventions weresimilar to our criteria, or if the results of only the

CP population and PT interventions were presentedseparately. Further, the review should report the

results of the included studies. Reviews in Danish,English, Finnish, German, Norwegian, or Swedish

were accepted.We searched Medline, CINAHL, the Cochrane

Database of Systematic Reviews, Database of Ab-stracts of Reviews of Effects, American College of

Physicians Journal Club, Health Technology As-

June 2008 Effectiveness of Interventions in CP 479

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sessment database, and the Physiotherapy Evi-dence Database (http://www.pedro.fhs.usyd.edu.au/

index.html) without language restrictions back tothe earliest time available and up until August

2007. An experienced information scientist plannedthe search strategies. High sensitivity search strat-

egies for Medline and CINAHL databases developedby the University of York25 were employed and

complemented with Medical Subject Headings or

text words for populations and interventions. Thesearch strategy for Medline is shown Table 1. FromJanuary 2003 to August 2007 the search results

from Medline and CINAHL were limited to sys-tematic reviews or review articles using the im-

proved filters provided by these databases. The ref-erences of the identified review articles were

checked by two reviewers (H.A., J.S.) to identifypossible reviews. We also searched our personal

files of studies and reviews on children with CP.Two reviewers (H.A. and R.K. or I.A.R.) inde-

pendently screened the titles or abstracts identified

in the initial search strategy for inclusion andexclusion criteria. When the title and abstract didnot clearly indicate whether an article should be

included, two reviewers (H.A., I.A.R.) evaluated thefull article for inclusion criteria. The reviewers

were not blinded to the names of authors andinstitutions, sources of funding or results of the

review.

Data Extraction

One of two reviewers (either H.A. or J.S.) ex-

tracted the data. The included articles were allo-

cated equally, and data from one review was ex-tracted by both reviewers to ensure similarity. Afterdata extraction the results were checked by the

other reviewer. We tabulated the review focus,search strategies and inclusion criteria, data of the

included populations, interventions, settings, out-

come measures; number of studies and the studydesigns in each review; methods used in quality

assessment and analyses; and the main results andconclusions, and reported adverse effects. For

quantitative data we extracted the effect sizes of alloutcome measures used.

Assessment of the MethodologicalQuality

The methodological quality of the included

SRs was analyzed using a modified version16 of themethod described and validated by Oxman et

al.14,15 This checklist evaluates nine items coveringsearch methods, selection of the articles, validity

assessment and methods for synthesis, The modi-fication, previously applied in the field of rehabili-

tation, consists of the addition of decision rules toincrease transparency of the assessment.16 Each

item is scored from 0 to 2, with a maximum total

score of 18 (Appendix A). Reviews fulfilling all

points, except the item of selection bias (as using

two or more assessors for independently judging

and selecting studies with predetermined criteria,

and/or blinding reviewers to identifying features of

study, or to treatment outcome), were regarded as

being of high quality. Two evaluators (H.A., J.S.)

separately assessed the quality of the included re-

views. The discrepancies in evaluations were solved

by discussion, and remaining disagreements weredecided by a third reviewer (A.M.).

Analysis of the Reviewers Conclusions

The included reviews were classified according

to the intervention types: (1) comprehensive PT

approaches (e.g., neurodevelopmental or neuro-

physiological PT, home programs or Vojta), (2)

strength training, (3) constraint-induced move-

ment therapy (CIMT), (3) postural control, (4) soft

tissue treatment, (6) hydrotherapy, (7) hippo-

therapy, 8) CE and (9) various (several of the aboveinterventions in one review). For each group of

interventions we considered and weighed up the

conclusions according to the methodological qual-

ity of the SR. We also observed the number and

type of included studies and their overlaps between

the reviews to obtain a comprehensive overview of

the research volume in this field.

RESULTS

Article Identification and Selection

Figure 1 shows a flow chart of the literaturesearches and article selection. We found 21 SRs:

four reviews on comprehensive PT,2629 two on

strength training,30,31 one on CIMT,32 one on pos-

tural control,33 one on soft tissue treatment,34 one

on hydrotherapy,35 two on hippotherapy,36,37 four

on CE,3841 and five reviews covering a wide range

of various interventions.4246

Methodological Quality

The methodological quality scores of the re-

views are presented in Table 2. The search meth-

ods and inclusion criteria were at least partiallydescribed in all reviews, as these were our man-

datory criteria for inclusion. Six reviews fulfilled

all criteria other than blinding reviewers from au-

thor and outcome information.26,30,32,33,36,42

Twelve reviews26,29,30,3234,3639,42,43 had defined

quality-assessment criteria, and all but one37 used

these. Many reviews had inadequacies in search

and synthesis methods. The median quality score

was 11 out of 18 points (range 317).

A summary of the reviews focus and methods

is given in Table 3. The methods of qualitative

480 Anttila et al. Am. J. Phys. Med. Rehabil. Vol. 87, No. 6

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TABLE 1 High-sensitivity search strategy for identifying review articles in Medline, developed by theUniversity of York25

1. Cerebral palsy/rh, th [rehabilitation, therapy]

2. Cerebral palsy.mp. or cerebral palsy/3. Exp physical therapy techniques/4. (Physical therapy or physical therapies).ab,ti.5. Physiotherap$.ab,ti.6. Exp exercise therapy/7. (Physical activity or physical activities).ab,ti.8. Exp physical therapy (specialty)/9. Exp physical education and training/

10. Rehabilitation.mp. or REHABILITATION/11. (Vojta or bobath or neurodevelop$ or NDT or Rood or Kabat or vibroacoust$).ab,ti.12. Early intervention (education)/13. Conductive education.ab,ti.14. Conservative therap$.ab,ti.15. (Muscle strength$ or muscle training or motion or therapeutic exercise or excercise training or physical

exercise or fitness or aerobic training or kinetic chain).ab,ti.16. Movement.mp. or EXERCISE MOVEMENT TECHNIQUES/or MOVEMENT/17. SWIMMING/or swimming.mp. or hydrotherapy.mp.18. (Functional therapy or functional therapies).ab,ti.19. (Self-care training or motor control or motor learning).ab,ti.20. Occupational therapy.mp. or Occupational Therapy/

21. (Constraint adj induced).mp. [mp

ti, ab, tx, kw, ct, ot, sh, hw]22. Restraint, physical/23. (Forced adj2 treatment).mp. [mpti, ab, tx, kw, ct, ot, sh, hw]24. (Psychomotor performance or sensation).mp. [mpti, ab, tx, kw, ct, ot, sh, hw]25. Sensory integration.ab,ti.26. (Sensory adj perceptual).mp. [mpti, ab, tx, kw, ct, ot, sh, hw]27. Parentchild relations/or parents/or parent education.mp.28. Physical stimulation.mp. or physical stimulation/29. (Posture or positioning).mp. [mpti, ab, tx, kw, ct, ot, sh, hw]30. Facilitat$.ti,ab.31. 3 or 4 or 5 or 6 or 7 or 8 or 9 or 10 or 11 or 12 or 13 or 14 or 15 or 16 or 17 or 18 or 19 or 20 or 21 or

22 or 23 or 24 or 25 or 26 or 27 or 28 or 29 or 3032. 2 and 3133. 1 or 3234. Controlled.ab.

35. Design.ab.36. Evidence.ab.37. Extraction.ab.38. Randomized controlled trials/39. Meta-analysis.pt.40. Review.pt.41. Sources.ab.42. Studies.ab.43. Or/344244. Letter.pt.45. Comment.pt.46. Editorial.pt.47. Or/444648. 43 not 47

49. 33 and 48Limitations (from January 2003 to August 2007):50. Limit 49 to systematic reviews51. Limit 49 to review articles52. 51 and 50


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categorization and synthesis varied considerably.Eight reviews classified the study results into out-

come-related categories by dimensions of disability,27

ICIDH-2 (International Classification of Impair-

ments, Disabilities and Handicaps),34,38,43 ICF (In-ternational Classification of Functioning, Disability

and Health),30,35,36 or own classification.41 One re-

view applied meta-analysis on randomized con-trolled trials (RCT).43 Effect sizes and confidenceintervals were available from three reviews.30,32,34

Nine reviews applied levels of evidence analysis.

Four reviews26,31,35,43 used a method described bySackett47,48 and one review36 modified this method2

to include Physiotherapy Evidence Database rat-ings. Three reviews27,33,34 applied American Acad-

emy for Cerebral Palsy and Developmental Medi-cine (AACPDM) methodology,49 and one review33

used the methodology of AACPDM and Sackett.47

One review42 applied the evidence synthesis

method described by van Tulder.50

Characteristics of the Review Contents

The reviews were based altogether on 31 RCTs

and 199 observational studies. Ten reviews in-cluded non-CP children,26,28,29,34,35,37,40,41,45,46 and

four reviews also included interventions that wereoutside the scope of this review,33,42,43,45 which

were excluded from the analyses. Twenty-threeRCTs and 104 observational studies were on chil-

dren with CP; of these, 13 RCTs and 29 observa-tional studies were included in more than one

review (Table 4).

A full description of the characteristics of re-viewed populations, interventions, outcome mea-

sures, and results of studies on children with CP isin Appendix B. The population in terms of age, type

and severity of CP, the interventions, and the out-come measures are heterogeneous in all reviews

and intervention groups. The included studies were

conducted in various settings (clinic, home,school, or community). The settings are suffi-ciently reported in only four reviews.30,38,39,42 Ex-

cept for one review,32 the content of each interven-

tion is described only with a short title. Thenumber of different outcome measures reported

varies from 6 to 30 per review. Two reviews do notreport any outcomes.40,45

Conclusions on Effectiveness of theInterventions Included in the ReviewsComprehensive PT

One high-quality26 and three low-qualitySRs2729 on comprehensive PT approaches have

evaluated 15 RCTs and 28 observational studies, ofwhich 9 RCTs6169 and 19 observational studies

were on children with CP. Seven of the 9 RCTs(total number of children, n 309) and 5 of the 19

observational studies (n 493) are included inmore than one review. The high-quality review

concludes that the current research . . . does notclearly demonstrate the efficacy or inefficacy of

NDT as a treatment approach.26(p242) Conclusionsin the low-quality reviews are similar: The prepon-

derance of the results . . . did not confer any advan-

FIGURE 1 Flow chart of the article selection process.


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TABLE

2

Methodologicalassess

mentscoresoftheincluded21

systematicreviews

First

Au

thor

(Year)

Searc

hMe

tho

ds

Se

lectio

nMe

tho

ds

Va

lidity

Assessm

en

t

Syn

thes

is

To

tal

Po

ints

(Max

18)

Searc

hM

etho

ds

Searc

h

Compre

hensiveness

Inc

lusion

Cri

teria

Avo

idanceo

f

Se

lec

tion

Bias

De

fin

itiono

fthe

Va

lidity

Assessmen

t

Cri

teria

Useo

fthe

Qua

lity-Assessmen

t

Cri

teria

Syn

thesis

Me

tho

ds

Accep

tability

of

the

Syn

thes

is

Me

tho

ds

Conc

lusions

Supporte

dby

Da

taAna

lysis

Comprehensivephysiotherapy

Brown

26

(2001)

2

2

2

1

2

2

2

2

2

17

Butler2

7

(2001)

2

2

2

0

0

0

2

1

2

11

Parette2

8

(1991)

1

1

1

0

0

0

0

0

0

3

Tirosh

29

(1989)

1

1

1

0

2

2

0

1

2

10

Strengthtraining

Dodd30

(2002)

2

2

2

1

2

2

2

2

2

17

Darrah31

(1997)

2

1

1

1

0

0

2

2

2

11

Constraint-inducedmovement

therapy

Hoare

32

(2007)

2

2

2

1

2

2

2

2

2

17

Posturalcontrol

Harris33

(2005)

2

2

2

1

2

2

2

2

2

17

Soft-t

issuetreatment

Pin34

(2006)

2

1

2

0

2

2

2

0

0

11

Hydrotherapy

Getz3

5

(2006)

2

2

2

1

0

0

2

2

2

13

Hippotherapy

Snider3

6

(2007)

2

2

2

0

2

2

2

2

2

16

Sterba3

7

(2007)

2

1

2

0

2

0

0

0

0

7

Conductiveeducation

Darrah38

(2003)

2

1

2

0

2

2

2

1

2

14

Ludwig39

(2000)

2

2

2

1

2

2

0

1

0

12

Pedersen

40

(2000)

1

1

1

0

0

0

0

0

0

3

French

41

(1992)

1

2

2

0

0

0

1

1

2

9

Variousinterventions

Steultjens4

2

(2004)

2

2

2

1

2

2

2

2

2

17

Boyd43

(2001)

2

2

2

0

2

1

1

1

0

11

Woolfson

44

(1999)

1

2

2

0

0

0

0

0

1

6

Hur4

5

(1995)

1

1

1

0

0

0

0

1

2

6

Horn

46

(1991)

2

2

2

0

0

0

0

1

2

9

Yes

15

13

16

0

12

10

11

8

14

Partially

6

8

5

8

0

1

2

8

1

No

0

0

0

13

9

10

8

5

6

Scoring:2,

thecriterionisfulfilled;1

,partiallyfulfilledorcannottell;0,notfulfilledornotreported;thedecisionrulesare

inAppendixA.


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TABLE

3

Continued

Rev

iew

(Year)

Obj

ec

tiveso

fthe

Rev

iew

Designs

Inclu

de

d*

Me

tho

dso

fAna

lyses

(Searc

hP

erio

d,

Me

tho

do

logica

lQua

lityAssess

men

t

(QA),Ca

tegoriza

tiono

fthe

Re

sults,

Syn

thesis

Me

tho

d)

Qua

lityScore

Conc

lusionso

fRev

iew

*

Posturalcontrol

HarrisandRoxborough33

(2005)

Efficacy

andeffectivenessof

postu

ralcontrolintervention

strate

giesforchildrenwithCP

OD(12)

Search(1990

2004)

QA:AACPDMQuality

AssessmentScalebyButler

49

Categorizationbystudydesigns

(group/singlesubject)

Levels-o

f-evidenceanalysesby

Sackett47

forgroupdesigns;

Butler4

9

forsingle-subject

designs

17

Externallygeneratedmovements:

Postural

perturbations:reactive

balance

GroupNDT

orpractice:

Soft-t

issuetreatment

Pinetal.3

4

(2006)

Effectivenessofpassive

stretc

hingbyusingICIDH-2

inchildrenwithCP

RCT(4)

OD(3)

SearchuntilApril2006

QA:PEDroscale5

4

Categorizationbyoutcomes

(ICIDH-2

)

Levels-o

f-evidenceanalysesby

Butler4

9

Meaneffectsizesandconfidence

intervalscalculated

11

Passivestre

tching:ROM

,spasticity

Sustaine

dstretchingvs.manual

stretchin

g:ROM,spasticity

Hydrotherapy

Getzetal.3

5

(2006)

Effectivenessofaquatic

interventionswithregardto

theICFdimensionsin

childrenwithneuromotor

impairments

RCT(1)

OD(10)

Search(1966January2005)

QA:No

Categorizationby

outcomes(ICF)

Levels-o

f-evidencesynthesisby

Sackett48

13

Hydrothera

py:respiratoryfunction,

activity?

,participation?

Hippotherapy

Snideretal.36

(2007)

Effectivenessofhippotherapy

andtherapeutichorseback

ridingonimpairments,

activi

ties,andparticipation

inchildrenwithCP

RCT(3)

OD(6)

Search(1806

2005)

QA:RCTsbyPEDroscale

54;otherdesignsby

NewcastleOttawascale

55

Categorizationbyinterventionsand

outcomes(ICF)Levels-o

f-evidence

synthesisbySackett,2

modifiedt

oinclude

PEDroscale54

17

Hippotherapy:musclesymmetry

Therapeutic

horsebackridingor

hippotherapy:activities,participation?

Sterba

37

(2007)

Effectivenessofhorseback

ridingusedastherapyto

improvegrossmotor

functioninchildrenwithCP

RCT(0)

CCT(3)

OD(7)

Search(1981December2005)

QA:16criteria,CriticalReviewFor

m

for

QuantitativeStudiesbyLawetal.56

Categorizationbyinterventions

Descriptiveanalyses

7

Allinterven

tioncategories:grossmotor

function

Conductiveeducation

Darrahetal.38

(2003)

Current

stateofevidenceof

CEprogramsinchildren

withCP

RCT(1)

OD(14)

Search(1966

Fall2001)

QA:7criteria(inclusion/exclusioncriteria,

intervention,measuresused,

blinding,

statisticalevaluation,

dropouts,c

ontrolling

thevariablesandlimitingbias)

Categorizationbyoutcomes(dimen

sion

ofdisability,

ICIDH-2

)

Descriptiveanalyses

14

CE:?


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TABLE

4

IncludedRCTsonchildrenwithCP

andtheiroverlapsin

the21

systematicreviews

Ran

dom

ize

dCon

tro

lle

dTria

ls

Systema

tic

Rev

iews

(First

Auth

or)

No.

ofRev

iews

Inc

luding

the

Study

First

Au

thor

(Year)

Sample

Size

Brown26

Butler27

Parette28

Tirosh29

Dodd30

Darrah31

Hoare32

Harris33

Pin34

Getz35

Snider36

Sterba37

Darrah38

Ludwig39

Pedersen40

French41

Steultjens42

Boyd43

Woolfson44

Hur45

Horn46

Sung71

(2005)

31

1

1

Cherng78

(2004)

14

1

1b

2

Benda7

9

(2003)

15

1

1

Deluca7

2

(2002),

Taub73

(2004)

18

1

1

McConachie82

(2000)

58

1

1

Reddihough81

(1998)

34a

1

1

1

1b

4

Law

61

(1997)

50

1b

1

1

1

4

Steinbok62

(1997)

28

1b

1

Dorval77

(1996)

20

1

1

MacKinnon

80

(1995)

19

1

1b

2

ODwyer7

4

(1994)

15

1

1

Law

63

(1991)

73

1b

1

1

1

4

Richards7

5

(1991)

19

1

1

Palmer6

4

(1990),

Palmer6

5

(1988)

48

1b,c

1

1b,c

1b

1b,c

1

1b

7

Tremblay7

6

(1990)

21

1

1

Hanzlik83

(1989)

20

1b

1b

1

3

McGubbin70

(1985)

30

1

1

1

3

Sommerfeld66

(1981)

29

1

1b

1

1

1

5

Talbot84

(1981)

59

1

1

Sellick85

(1980)

20

1

1

2

Scherzer6

7

(1976)

22

1b

1

1b

1

1

1

6

Carlsen

68

(1975)

12

1b

1

1b

1

1

1

6

Wrigth69

(1973)

47

1

1b

1

1

1

5

Sum

702

7

8

5

4

1

1

2

0

3

1

3

2

1

1

1

0

6

5

1

5

6

a

Thetrialincludedadditional32non

randomizedchildren;

b

Notclassifiedasan

RCTbytheauthors;conlythePalmeretal.65

publicationwasincludedinthereview.


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tage to NDT over the alternatives . . .27(p22);. . . only four studies used a rigorous design, and

three of these concluded that no evidence exists forthe efficacy of the intervention . . .29(p555); or the

available literature offers some support for the ef-ficacy of therapeutic interventions for infants and

young children with cerebral palsy.28(p5)

Strength Training

We found one high-quality30 and one lower-quality31 SR on strength training in children with

CP, evaluating altogether 1 RCT and 11 observa-tional studies (n 102). Four studies are included

in both reviews, including the RCT.70 The conclu-sions are similar: strength training programs im-

prove muscle strength in children and youngadults with CP, with no adverse effects on spastic-

ity.30,31

CIMT

One high-quality Cochrane review32 analyzed

two RCTs7173 and one controlled clinical trial (CCT)(n 94). This SR found a significant treatmenteffect [on bimanual performance] using modified

CIMT in a single trial. A positive trend favoring CIMTand forced use was also demonstrated.32(p10)

Postural Control

From one high-quality review33 on interven-

tions aiming to improve postural control, we in-cluded four observational studies on NDT, rocker

platform, and massed practice (n 22). The reviewconcludes with suggestive evidence for the effec-

tiveness of interventions comprising externallygenerated movement on the development of pos-

tural control, promising evidence for postural per-turbations improving reactive balance when a high

number of repetitions is provided, and moderatelystrong evidence for the lack of group-level effects of

1 wk of NDT or practice.

Soft-Tissue Treatment

One low-quality review34 evaluated threeRCTs7476 and two observational studies on passive

stretching in children with CP (n 89). The con-

clusion is that the effectiveness of passive stretch-ing remains weak, although some evidence indi-

cates that sustained stretching is preferable tomanual stretching in improving range of motion

and reducing spasticity.

Hydrotherapy

In one low-quality review on aquatic interven-tions,35 one RCT77 and four observational studies

address children with CP (n 68). Getz et al.35

conclude that hydrotherapy might improve respi-

ratory function in children with CP.

Hippotherapy

One high-quality36 and one lower-quality37 re-

view compare therapist-directed hippotherapy vs.recreational horseback riding therapy. These re-

views include three RCTs7880 and seven observa-tional studies (n 100). Of these, two RCTs78,80

and six observational studies are included in bothreviews. The results of Snider et al.36 indicate that

hippotherapy has short-term positive effects on

muscle symmetry in the trunk and hip and thattherapeutic horseback riding is no more effectivethan other therapies for improving muscle tone.

Observational studies have shown positive effects ofboth hippotherapy and therapeutic horseback

riding on activities. The low-quality review37 statesthat clinicians and therapists can recommend hip-

potherapy as an efficacious, medically indicatedtherapy for gross motor rehabilitation of children

with CP.

CE

The effectiveness of CE has been evaluated in

four reviews.3841 These include 1 RCT81 and 21observational studies (n 1264), with 7 of the

observational studies being included in more thanone review. The overall conclusions of these re-

views are concordant: the number of studies wastoo small, and the quality was too low, to make

conclusions about the effectiveness or ineffective-ness of CE.

Various Interventions

One high-quality42

and four low-quality re-views4346 include different types of interventions

from 13 RCTs61,63-70,81-85 and 47 observationalstudies. The reviewers conclusions unanimously

pinpoint the paucity of evidence. According toSteultjens et al.,42 evidence for the efficacy of oc-

cupational therapy is insufficient in all interven-tion categories. Horn et al.46 have found no evi-

dence of the effectiveness or ineffectiveness ofNDT, sensory integration or naturalistic program-

ming. No conclusions are made on treatment ap-proaches for upper-limb dysfunction,43 on training

and behavior-modification techniques in conjunc-tion with PT,45 or on multidomain developmentaland CE programs,44 because of the paucity of evi-

dence and methodological limitations.

DISCUSSION

We identified and critically analyzed 21 SRs on

PT and CE interventions in children and adoles-cents with CP. Our analysis of the quality of evi-

dence summaries and of the volume, characteris-tics, and effectiveness of primary studies in this

field provides insights into the current scientific


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basis for clinical decision making and future re-

search agendas.

Recommendations for SRs

SRs are based on critically appraised, high-

quality effectiveness research, usually RCTs. Six of

the identified SRs were of high quality.26,30,32,33,36,42All

reviews included observational studies, possibly be-

cause of the limited number of RCTs available. Theterminology in various observational study designs

was very mixed. Some reviews did not even recog-

nize RCTs among their included studies.26,28,37 As-

sessment of the included studies revealed that re-

views on the same topics included somewhat

different studies. No review had excluded studies

on the basis of quality. The differences may be

attributable to different search periods, search

terms and databases, or somewhat different foci on

inclusion criteria. We recommend that future re-

views clearly define what study designs are to be

included.Twelve reviews had defined quality-assess-

ment criteria, and all but one used these. How-

ever, most quality criteria only suit RCTs, not

observational studies. In three reviews,27,33,34

quality was assessed by a tool49 that raises single-

case studies to the level of RCTs in the evidence

hierarchy. Today, the AACPDM methodology has

been updated86 to meet the criteria of evidence-

based evaluation.87 The variety of quality-assess-

ment tools reflects the lack of consensus as to

which components and what tools would best

assess trial quality.10,88,89 Previous research onthe role of nonrandomized studies and case se-

ries in SRs in other fields has been hampered by

both the paucity and the poor quality of these

studies.90,91 More research is needed on how the

methodological features of observational studies

affect outcomes in this field.

The qualitative synthesis methods were built

on different combinations of different aspects

across the reviews. The categorization of the re-

sults was made either by outcomes, interventions,

study designs, study quality, or populations. These

categories are then summarized, either descrip-tively by levels of evidence analyses, or by counting

for the numbers of studies in different categories.

Most of these hide important factors, such as the

number of patients included and the real effect

sizes. Only three reviews provide effect sizes to-

gether with the confidence intervals. A common

understanding on how to summarize findings on

individual studies in a qualitative synthesis is ob-

viously needed, as found earlier on Cochrane re-

views in PT and occupational therapy interven-

tions.92

Clinical Heterogeneity of the ReviewedInterventions

CP is a heterogeneous condition where the

developmental potential and goals for rehabilita-tion vary with age.93 Many reviews include non-CP

children, which may bias conclusions when resultsare not analyzed separately. Thus, it is difficult to

determine which patient groups may benefit fromthe studied interventions. An SR in this field can be

improved by focusing on clearly defined targetgroups.

Many older reviews include a variety of incom-parable interventions. Complex interventions with

several, often vaguely defined, interacting compo-nents can complicate analyses, decreasing clinical

applicability. Interventions may be insufficientlydescribed in the original studies,94 and interven-

tions in different countries may actually not becomparable at all, despite similar names.39 Nar-

rower inclusion criteria for interventions may al-low better comparison across studies, as seen in the

recent reviews.3037 First steps toward interna-tional intervention categories in PT with adults

have been taken,95 which may help future evidencesyntheses.

It is important to know whether all clinicallyrelevant outcomes have been reported. Numerous

noncomparable outcome measures were used inthe studies, and the clinical relevance of many of

them remains unclear. Without a consensus onmeasures to apply in CP94 or in rehabilitation in

general,96 the combination of results across studiesis problematic.

All these factors may complicate reviews ofcomplex interventions in this heterogeneouspopulation, as recognized earlier.92 Clinicians

and researchers would benefit from a more pre-cise description of the studies in terms of popu-

lation, interventions, comparison interventions,and outcomes to increase the clinical applicabil-

ity of reviews. Many problems were caused byinsufficient reporting of the details of the re-

viewed studies, possibly because of poor report-ing in original studies.97 We recommend using

guidelines such as the Quality of Reporting of

Meta-Analyses statement to increase the qualityof the review report.17

Effectiveness and Clinical Applicabilityof the Reviewed Interventions

The six high-quality reviews allow conclusions

on some of the interventions reviewed. Evidence ofcomprehensive PT approaches26 and occupational

therapy interventions42 is insufficient. The fourhigh-quality reviews on more focused interven-

tions provided positive evidence on some out-comes: strength training on muscle strength,30

intensive upper-extremity training on bimanual


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performance,32 hippotherapy on muscle symmetry

and activities,36 and effectiveness of externally gen-

erated movements and postural perturbations to

reactive balance.33

The four reviews posing targeted questions

may be clinically easier to apply because they in-

clude a limited number and type of interventions

and outcomes. For example, in the strength train-

ing review, the interventions, outcome measures,

and patient inclusion criteria are fairly unambigu-ous.30 The positive evidence on effectiveness is

based on only one RCT70 and several concordant

observational studies. The evidence-grading system

by the GRADE Working Group98 suggests upgrad-

ing for cohort studies, when two or more observa-

tional studies show a consistent association, with

no plausible confounders. This was the case for the

studies on strength training.

Some low-quality reviews have made conclu-

sions on indicative evidence of passive and sus-

tained stretching on range of motion and spastic-

ity,34 hydrotherapy on respiratory function,35 and

hippotherapy and horseback riding therapy for

gross motor performance.37 This evidence should

be interpreted cautiously because of the method-

ological limitations. In the other reviews, the au-

thors concordantly state that they can make no

conclusion on the effectiveness or ineffectiveness

of the reviewed interventions. These reviews thus

provide no support on applying the studied inter-

ventions in clinical practice.

Recommendations for ClinicalStudies in CP

Given the considerable variation in the CP

population, interventions, and outcome measures,

future studies would gain from agreement on a CP

definition.99 Careful compiling of the interven-

tions components and detailed reporting would

allow interpretation of the applicability to clinical

practice. Inconclusive evidence on the more com-

prehensive treatment approaches calls for better

understanding of the intervention components,94

and a phase-oriented approach may be useful.100

Evidence from studies on focused interventions,such as strength training and CIMT, could be used

when developing complex interventions. Indeed,

the many single components of PT interventions

could be studied separately.

Many treatment approaches have not been rig-

orously evaluated, and well-conducted studies are

still needed. Given the number of outcome mea-

sures used, a consensus on outcomes is needed.

Future studies should apply validated measures

covering all ICF components and health-related

quality of life.

CONCLUSIONS

SRs of PT or CE interventions in children with

CP require cautious interpretation of the findings.On the basis of six high-quality reviews, conclu-

sions on the effectiveness of some interventions on

specific outcomes could be made. Otherwise, theeffects remain unclear or unsupported by data. The

low number of RCTs resulted in the inclusion of alarge variety of observational studies in reviews.

Well-conducted studies on current treatment op-tions as well as new treatment approaches using

valid outcomes are obviously needed. Because re-views on rehabilitation within such a heteroge-

neous population as CP are demanding to conduct,compliance with methodological guidelines on re-

porting, such as the Quality of Reporting of Meta-Analyses statement, is recommended.

ACKNOWLEDGMENTS

The authors thank professor Regina Kunz, PhD,MD, MSc (Epi), for reviewing the abstracts until June

2003; information scientists Riitta Grahn, MSc, andJaana Isojarvi, MSocS, for their support in the liter-

ature search; and Mark Phillips, BA, for his help inreviewing the language of the article.

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APPENDIX A

Quality-assessment criteria for review articles.A modified version by Hoving at al.16 of an index

constituted by Oxman and Guatt14 and Oxman etal.15 Maximum total score is 18.

Search Methods

1. Were the search methods used to find evi-

dence (primary studies) on the primaryquestion(s) stated?

2 points: Yes; includes description of data-bases searched, search strategy, and

years reviewed. Described well enough toduplicate.

1 point: Partially; partial description ofmethods, but not sufficient to duplicate

search0 points: No; no description of search

methods2. Was the search for evidence reasonably

comprehensive?2 points: Yes; must include at least one

computerized database search as well as

a search of unpublished or nonindexedliterature (e.g., manual searches or let-

ters to primary authors)1 point: Cannot tell; search strategy par-

tially comprehensive (e.g., at least one ofthe strategies in the foregoing section

were performed)0 points: No; search not comprehensive or

not described well enough to make a

judgment

Selection Methods

3. Were the criteria used for deciding whichstudies to include in the review reported?

2 points: Yes; inclusion and exclusion cri-teria clearly defined

1 point: Partially; reference to inclusionand exclusion criteria can be found in the

paper but are not defined clearly enoughto duplicate

0 points: No; no criteria defined

4. Was bias in the selection of articles avoided?2 points: Yes; key issues influencing selec-

tion bias were covered. Two of three of

the following bias avoidance strategieswere used: two or more assessors inde-

pendently judged study relevance and se-

lection using predetermined criteria, re-viewers were blinded to identifying

features of study (i.e., journal title, au-thor(s), funding source), and assessors

were blinded to treatment outcome.1 point: Cannot tell; if only one of the three

strategies above were used0 points: No; selection bias was not avoided

or was not discussed

Validity Assessment

5. Were the criteria used for assessing thevalidity for the studies that were reviewed

reported?2 points: Yes; criteria defined explicitly

1 point: Partially; some discussion or ref-erence to criteria but not sufficiently de-

scribed to duplicate0 points: No; validity or methodological

quality criteria not used or not described6. Was the validity for each study cited as-

sessed using appropriate criteria (either inselecting studies for inclusion or in analyz-

ing the studies that are cited)?2 points: Yes; the criteria used address the

major factors influencing bias (for exam-ple: population, intervention, outcomes,

follow-up)1 point: Partially; some discussion of

methodological review strategy, but not


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clearly described with predetermined cri-teria

0 points: No; criteria not used or not de-scribed

Synthesis

7. Were the methods used to combine the

findings for the relevant studies (to reach aconclusion) reported?

2 points: Yes; qualitative or quantitativemethods are acceptable

1 point: Partially; partial description ofmethods to combine and tabulate; not

sufficient to duplicate0 points: Methods of combining studies

not stated or described8. Were findings of the relevant studies com-

bined appropriately relative to the primaryquestion the review addresses?

2 points: Yes; combining of studies seemsacceptable

1 point: Cannot tell; should be marked if indoubt

0 points: No; no attempt was made to

combine findings, and no statement

was made regarding the inappropriate-

ness of combining findings; should be

marked if a summary (general) esti-

mate was given anywhere in the ab-

stract, the discussion, or the summary

section of the paper, and the method of

deriving the estimate was not de-

scribed, even if there is a statementregarding the limitations of combining

the findings of the studies reviewed

9. Were the conclusions made by author(s)

supported by the data or analysis reported

in the review?

2 points: Yes; data, not merely citations,

were reported that support the main con-

clusions regarding the primary ques-

tion(s) that the overview addresses

1 point: Partially

0 points: No; conclusions not supported or

unclear


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