Clinical Prognostic Messages From a Systematic Review on … · Clinical Prognostic Messages From a Systematic Review on Cerebral Palsy abstract OBJECTIVE: To summarize evidence on

Clinical Prognostic Messages From a SystematicReview on Cerebral Palsy

abstractOBJECTIVE: To summarize evidence on the rates of co-occurringimpairments, diseases, and functional limitations with cerebralpalsy into succinct clinical messages.

METHODS: A search was conducted of the databases PubMed, Medline,CINAHL, and PsycINFO, and the results were supplemented with handsearches. Two independent reviewers determined whether retrievedabstracts met the following inclusion criteria: human subjects;.90% were children or adults with cerebral palsy; published after1999; and population-based data. Articles were appraised, analyzingdesign, participants, level of evidence, rates of impairments, andfunctional implications. Methodologic quality was rated by usinga standardized checklist.

RESULTS: A total of 1366 papers were identified in the search; 82 wereappraised and 30 were included in the meta-analyses. High-levelevidence existed, as rated on the Oxford 2011 Levels of Evidence: 97%of prevalence studies were level 1. The data were of a moderate to highquality grade (with the exception of sleep disorders), allowing plainEnglish clinical messages to be developed.

CONCLUSIONS: Among children with cerebral palsy, 3 in 4 were in pain;1 in 2 had an intellectual disability; 1 in 3 could not walk; 1 in 3 had a hipdisplacement; 1 in 4 could not talk; 1 in 4 had epilepsy; 1 in 4 had a be-havior disorder; 1 in 4 had bladder control problems; 1 in 5 had a sleepdisorder; 1 in 5 dribbled; 1 in 10 were blind; 1 in 15 were tube-fed; and 1in 25 were deaf. Pediatrics 2012;130:1–28

AUTHORS: Iona Novak, PhD, MSc (Hons), BAppSc (OT),a,b

Monique Hines, PhD, BAppSc (SP),a Shona Goldsmith,BPhty (Hons),a,b and Richard Barclay, BSc (Hons)Psychologya

aCerebral Palsy Alliance Research Institute, Sydney, Australia;and bSchool of Medicine, University of Notre Dame, Sydney,Australia

KEY WORDScerebral palsy, meta-analytic methods, parent friendly, populationregister, prognosis, systematic review

ABBREVIATIONSCI—confidence intervalGMFCS—Gross Motor Function Classification SystemID—intellectual disabilitySCPE—Surveillance of Cerebral Palsy in EuropeUKCP—United Kingdom Cerebral Palsy register

www.pediatrics.org/cgi/doi/10.1542/peds.2012-0924

doi:10.1542/peds.2012-0924

Accepted for publication Jun 19, 2012

Address correspondence to Iona Novak, PhD, MSc (Hons), BAppSc(OT), Head of Research, Cerebral Palsy Alliance Research Institute,PO Box 184, Brookvale NSW 2100, Australia. E-mail:[email protected]

PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, 1098-4275).

Copyright © 2012 by the American Academy of Pediatrics

FINANCIAL DISCLOSURE: The authors have indicated they haveno financial relationships relevant to this article to disclose.

FUNDING: No external funding.

PEDIATRICS Volume 130, Number 5, November 2012 1

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mailto:[email protected]

Parents of children with cerebral palsyoften ask pediatricians and alliedhealth professionals, “will my childwalk?,” “will my child talk?” and “will mychild work and live independently?”1 Inessence, parents want to know “howbad is it?” and what will their child’sfuture look like.1,2 The answer to thequestion depends on the severity ofphysical disability, the type of motorimpairment, and the presence ofcomorbid conditions. Research existsto help clinicians identify impairmentsand predict future function, such aswalking, in children with cerebralpalsy. Yet parents report that they arerarely given prognostic information.3

Parents believe professionals withholdprognostic information in an attemptto protect them from bad news. Re-search, however, suggests that theabsence of prognostic informationmakes it more difficult, not easier, forparents to cope.4 Dissatisfaction withdelayed receipt of diagnostic in-formation has been linked to higherrates of parental depression. In quali-tative studies, parents advise pro-fessionals that they want and needprognostic information to assist themwith planning services.3 In addition,parents recommend that medical in-formation be presented in “parent-friendly” language to facilitate theirunderstanding and acceptance of in-formation.5

Cerebral palsy is the most commonphysical disability in childhood, occur-ring in2 to2.5/1000births.6 Pediatriciansand allied health professionals there-fore need to have up-to-date prognosticinformation readily available to com-municate with families at diagnosisand throughout the child’s life spanto develop interventions. The definitionof cerebral palsy has recently beenexpanded to include the impairmentscommonly associated with the condi-tion because of their substantial im-pact on the child. “Cerebral palsy

describes a group of permanent dis-orders of the development of move-ment and posture, causing activitylimitation, that are attributed to non-progressive disturbances that oc-curred in the developing fetal or infantbrain. The motor disorders of cerebralpalsy are often accompanied by dis-turbances of sensation, perception,cognition, communication, and behav-iour, by epilepsy, and by secondarymusculoskeletal problems.”7 The in-clusion of secondary impairments andfunctional limitations within the defi-nition heightens the importance ofunderstanding and communicating theimpact of these co-occurring impair-ments, diseases, and functional limi-tations to parents to help predictoutcomes. Furthermore, althoughthe brain injury in cerebral palsy isnonprogressive, the co-occurring im-pairments, diseases, and functionallimitations change over time, reducingfunction and quality of life.8 Definitiveinformation about the real extent of co-occurring impairments, diseases, andfunctional limitations with cerebralpalsy is essential for parents inchoosing services and for providers interms of pediatric resource alloca-tion.4 Moreover, the psychological andphysical health of parents of childrenwith cerebral palsy is strongly influ-enced by the child’s behavior and thecomplexity of caregiving demands.9

Thus, the opportunity to introducepreventative mental and physicalhealth measures for parents and chil-dren exists when professionals proac-tively identify and forecast the extent ofa child’s disability.

The diagnosis of cerebral palsy is typi-cally made in the toddler years, aftermetabolic and degenerative conditionshave been ruled out.10 In the period of“wait and see” leading up to diagnosis,parents experience great distress.4 Thedescription and ultimately the diagnosisof cerebral palsy can in some cases

be made earlier; for example, if a pre-term child has abnormal brain imagingcoupled with abnormal motor signs ontools with good predictive psychomet-rics (such as General Movements or theTest of InfantMotor Performance).11 MRIis useful for identifying the presenceand location of an injury in ∼89% ofchildren with cerebral palsy.10 The lo-cation and type of brain injury generallycorrelate with subtype of cerebral palsy,which can provide physicianswith someguidance in prognosticating futurefunction.12 Despite this, significant limi-tations exist in the accuracy of MRIpredicting the severity of cerebral palsyand long-term functional outcomes.13 Inaddition, cerebral palsy is an umbrellaterm for many different brain lesions(with the type and size of the lesionresponsible for the different motorimpairments and accompanyingimpairments), which explains whyprofessionals encounter so many dif-ficulties in giving an accurate progno-sis to parents. Despite the lack of anevidence-based tool to prognosticatethe severity of cerebral palsy early,many parents anecdotally report beingtold that their child will be profoundlydisabled and will not walk, talk, orwork. For this reason, prognostic evi-dence drawn from more sources thanan abnormal MRI is needed to helpaccurately answer parents’ questions.

Because no guideline or systematicreview about prognosis of cerebralpalsy existed, we conjectured that thismight explain why clinicians have notyet developed clear clinical messagesfor communicating to parents. The lackof synthesized information may com-pound professionals’ preference toavoid giving bad news. Recom-mendations for intervention mightbe based on specialists’opinions abouttheir own area of expertise basedon experience, rather than on anoverview of evidence from cerebralpalsy population data. Given that

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interdisciplinary care is believed to bebest practice, we considered the lack ofsynthesized clinical messages to bea major gap in cerebral palsy knowl-edge. Our answerable clinical ques-tions therefore were: What are therates of co-occurring impairments,diseases, and functional limitations incerebral palsy? In addition, what is theprognosis of individuals with cerebralpalsy? The objective of the currentstudy was to systematically reviewthe highest levels of evidence availablerelating to rates of co-occurring impair-ments, diseases, and functional limi-tations in cerebral palsy, presentedin parent-friendly clinical prognosticmessages. We hypothesized that it waspossible to develop from the literaturea series of succinct clinical messagesin plain English for clinicians to com-municate to parents of children with ce-rebral palsy, to promote understandingand to inform intervention planning.

METHODS

Search Strategy

We conducted this systematic reviewusing a protocol based on recom-mendations for conducting systematicreviews from the Cochrane Collabo-ration and PRISMA statements andin accordance with the quality ofreporting of meta-analysis of obser-vational studies statements.14,15,16 Weidentified relevant articles by search-ing PubMed (1999–2011), Medline(1999–2011), CINAHL (1999–2011), andPsycINFO (1999–2011). Searches weresupplemented by hand searchingbibliographies of included articlesand review articles. Relevant studiesknown to the investigators throughprevious research work were also in-cluded. The search of published stud-ies was originally performed inJanuary 2009, and an updated searchwas conducted in January 2011.

We searched electronic databases by us-ing EBSCO host software including the

following search terms: (i) cerebral palsyOR hemipleg* OR dipleg* OR quadripleg*;AND (ii) incidence OR prevalence ORprognosis OR rate OR proportion;AND (iii) behavior*/behavior*/emo-tion*/psycholog*/psychiatr*/autis*OR continen*/incontinen* OR drool*/saliva/sialorreah OR eat*/dysphagia/mealtime*/feed*/aspirat*/nutrition*/videofluoroscopy/video fluoroscopy/swallow*/deglutition OR epilepsyOR hearing OR hip/disloc*/displac*/scoliosis/lordosis/kyphosis/windsw*OR intellectual/intelligen*/mental*retard*/learning disab*/cognit* ORpain OR sleep OR communication/nonverbal communication/speech/intelligibility/articulation/dysarthria/anarthria/apraxia/dyspraxia/language/augmentative communication/alternativecommunication/AAC/speech generatingdevice/SGD/communication methods/communication aid* OR walk*/motor/ambulat* OR vision/visual/nystagmus/strabismus/hemianopia/blind*; AND (iii)population OR registry OR register. Thelist of associated impairments searchedwere generated from the definitionof cerebral palsy and seminal texts,6

and assigned plain English terms forparents.

Inclusion Criteria

Studies published in English regardingthe prevalence of co-occurring impair-ments, diseases, and functional limi-tations fulfilling the following criteriawere included:

1. Type of study: Observational studieswere specifically sought, because itis neither logical nor ethical to pro-spectively randomize individuals torisk association studies. To improvethe quality of the review and mini-mize bias, studies using a popula-tion sample (ie, a cerebral palsyregister) were preferentially sought.There was a scarcity of researchabout some functional limitations

(eg, behavior, bladder control, drib-bling, pain, sleep), and more gener-ous inclusion criteria were requiredto achieve the study aim for thesetopics (eg, nonpopulation studies).The search strategy only includedstudies with greater levels of biaswhen no higher-quality observa-tional studies were available.

2. Type of outcome: studies that in-volved ascertaining the ratesof co-occurring impairments, dis-eases, and functional limitationsfor children or adults with cerebralpalsy.

3. Types of subjects: studies that ex-plicitly involved human subjects inwhich .90% of the participantswere children or adults with cere-bral palsy.

Studies were excluded from the system-atic review if: (1) theywerenonpopulation\-based surveys (unless populationevidencewas unavailable); (2) a secondpublication of the same study waspublished, which found the sameresults; (3) they were not available inEnglish; and (4) they were publishedbefore1999,given themarkedadvancesinobstetricandneonatal care in the last10 years.

Data Abstraction

A data abstraction sheetwas developedbased on the Cochrane recommen-dations. Two independent assessorsdetermined eligibility of the abstractsidentified from searches for furtherreview. Abstracts were retained forfull review if they met the inclusioncriteria or if more information wasrequired from the full text to ascertaineligibility. The same 2 reviewersthen reviewed full-text versions of allretained articles and all additionalarticles identified by hand searching.Full-text articles were retained if theymet inclusion criteria; 100% agreementwas reached on inclusion and exclusionassignment for the full-text articles.

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Data extracted from included studiesincluded: authors and date of study;number of participants; participants’diagnosis; study design; and de-scription of findings. The data extrac-ted from each included study aresummarized in Table 1. All the datarequired to answer the study questionswere published within the papers, sono contact with authorswas necessary.

Quality Assessment

Evidence experts acknowledge thatconforming to gold standard system-atic reviewconventions is difficultwhencritiquing etiologic, risk factor,and prognostic studies.17,18 This diffe-rence is because evidence hierarchiesheavily favor treatment studies.18 Ourreview of the cerebral palsy literatureconfirmed this problem: a lack ofagreement existed in literature re-garding a preferred quality ratingscale for observational studies, andstudies had for the most part examinedonly single groups, which thereforeprecluded standard meta-analyses.17

In the absence of an endorsed sys-tematic review guideline for epidemi-ologic studies, the following course ofaction was chosen: First, we rated thelevel of evidence on the Oxford 2011Levels of Evidence as per conven-tion.19 Second, we assessed qualityby using the standardized checklistdeveloped by Boyle.20 Third, theoverarching body of prevalence evi-dence was rated by using the GRADEsystem.21

Ethics

The Human Research Ethics Committeewaived the need for ethical approvalbecause the study did not involve anycontact with humans.

Analyses

Our intention was to conduct a meta-analysis if the retrieved studies wereof sufficient clinical and statistical ho-

mogeneity.Weencountered2problems.First, the prevalence studies pre-dominantly only studied 1 group (ie,the cerebral palsy population). Thus,conducting 2-group prevalence meta-analyses by using standard Mantel-Haenszel techniques was not possible.14

For the prevalence meta-analysis, wetherefore needed to use simple de-scriptive statistics, calculating pop-ulation prevalence mean impairmentrates with 95% confidence intervals(CIs). To increase the rigor of thesedescriptive analyses, studies had tomeet these additional stringent crite-ria for inclusion within the meta-analyses: (1) study sample was .80%of population (as per public healthconventions) or the total sample sizeneeded to be .500 cases of cerebralpalsy; (2) study sample included allsubtypes of cerebral palsy so as not toskew the estimates; and (3) case datawere not duplicated in other pub-lications. Studies meeting these extracriteria are denoted in Table 1. Whenstudies included near-duplicate data,older and smaller duplicates werepreferentially excluded to improverigor, unless the newest study hadcollapsed data, in which case the nextbest available study was included.Review articles were not included inthe meta-analysis, as per convention.The second problem was that althoughwe originally intended to conductthe meta-analysis by using GrossMotor Function Classification System(GMFCS) levels, there were insufficientnumbers of studies reporting GMFCSdata for this to be possible.

RESULTS

A total of 1366 citations were identifiedin our search; 82 articles met the in-clusion criteria for full appraisal, butonly 30 studies met the additionalstringent inclusion criteria for meta-analyses (Fig 1).

Levels of Evidence

Evidence from these studies was gen-erally very highwhen rated by using theOxford 2011 Levels of Evidence; 97% ofthe prevalence articles included in themeta-analysis were rated as level 1evidence (“most relevant local andcurrent random sample survey orcensus”) because they were cerebralpalsy register studies capturing datafrom .80% of the population. Only 1lower-level evidence (level 5) prevalencearticle, on the topic of sleep difficulties,was included in the meta-analyses be-cause no higher-level evidence could befound in the literature.

Rating of Study Quality

Table 2 presents quality analysis forthe prevalence studies by using thestandardized checklist developed byBoyle.20

Prevalence of Co-occurringImpairments, Diseases, andFunctional Limitations

Average rates of impairments, dis-eases, and functional limitations as-sociated with cerebral palsy werecalculated by aggregating the findingsof the included prevalence studies toidentify a mean rate and a 95% CI. Thefindings were: (1) behavior (818 casesfor analysis): 26% (95% CI: 24–28) hadabnormal behavior (the rate and CIwere from Parkes et al22 because newCIs were unable to be calculated be-cause only 1 study met inclusion cri-teria); (2) bladder and bowel control(601 cases for analysis): 24% hadbladder control problems (95% CI un-able to be calculated because only 1study met inclusion criteria); (3) drib-bling (1119 cases for analysis): 22%had excessive drooling (95% CI unableto be calculated because only 1 studymet inclusion criteria); (4) eating (1299cases for analysis): 6% (95% CI: 3–9)were tube-fed; (5) epilepsy (12140 cases for analysis): 35% (95% CI:


TABLE1

Included

StudiesAccordingto

Alterations

ofBody

Function,Activity

Limitations,and

Co-occurring

Diseases

Study

Participants

Analyzed

nResearch

Design

Levelof

Evidence

Results

Included/Excluded

Meta-analysis

Alterations

ofabody

function

Behavior

Parkes

etal(2008)

22CP:Allsubtypes

818

Prospectivecross-sectionalsurveyfrom

8populationCP

registers(70%

participationrate)

126%hadabnorm

albehavior

(95%

CI:24–28)(as

measuredon

theStrengthsandDifficulties

Questionnaire)

Included:n

.500

32%hadpeer

problems(95%

CI:30–35)

31%hadhyperactivity

(95%

CI:29–33)

29%hadem

otionaldifficulties

(95%

CI:26–31)

8–12

y17%hadconductproblems(95%

CI:15–19)

Behavioralproblemsrateswerehigher

with

amoderateto

severe

intellectualimpairment

comparedwith

thosewith

mild

intellectual

impairmentor

norm

alintelligence(OR:3.2[95%

CI:2.1–4.8])

Ratesofbehavioralproblemswerehigher

inchildrenwith

severepaincomparedwith

children

withoutp

ain(OR:2.7[95%

CI:1.5–4.6])

Ratesofbehavioralproblemswerehigher

inchildrenwith

better

grossmotor

ability(OR:1.0

GMFCSI)comparedwith

childrenwithoutm

ore

severephysicaldisability(OR:0.2[95%

CI:0.1–0.3]

GMFCSV)

Russoetal(2008)

30CP:hem

iplegiaonly

107

PopulationCP

register:Prospective

assessment(75%participationrate,

100%

completionrate)

148%hadpain

Excluded:Notallsubtypesbut

included

inTable3

3–16

yRatesofbehavioralcompetencewerehigher

for

childrenwithoutpain(m

ean:3.3)than

forchildren

with

pain(m

ean:2.9)

(P,

.01)

Sigurdardottiretal

(2010)

31CP:GMFCSI–IV

CP:36

PopulationCP

register,Iceland

(69%

of1999–2004

birthcohort)

3[Prognostic]

40%to50%hadabnorm

albehavior

asratedby

parentsand60%to65%hadabnorm

albehavioras

ratedby

teachers


included

inTable3

4–6y

Controls:110

Ratesofbehavioralproblemswere4tim

eshigher

inchildrenwith

CPcomparedwith

controls

Bladderandbowelcontrol

Roijenetal(2001)

32CP:Allsubtypes

601

Prospectivecohortstudy,6sites,the

Netherlands(76%

response

rate)

124%hadenuresis

Included:n

.500

4–18

yBladdercontrolw

asdelayedinCP.The

likelihoodof

developing

bladdercontroldecreased

over

time

97%who

haddaytimeenuresiswentontoachieve

nocturnalcontinence

Nocturnalenuresiswas

significantlydelayedifinitial

daytimebladdercontrolw

asachieved

after8y(P

,.001)

Childrenwith

spastic

quadriplegiaandIDwereless

likelytohavebladdercontrol(OR

:2.6[95%

CI:1.7–

3.5])

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TABLE1

Continued

Study

Participants

Analyzed

nResearch

Design

Levelof

Evidence

Results

Included/Excluded

Meta-analysis

Singhetal(2006)

33CP:Bilateralonly

55Cohortstudy(55%

participationrateof

SpecialN

eeds

Database)

5aDaytimeandnocturnalenuresisrateswerehigherin

nonambulant

childrencomparedwith

thosewho

couldwalk(85%

vs15%,and

77%vs

23%,

respectively)

Excluded:,

80%ofpopulation

andnotallsubtypes

but

included

inTable3

4-18

yDaytimeandnocturnalenuresisrateswerehigher

with

severe

IDcomparedwith

thosewithoutIDor

mild

tomoderateID(80%

vs20%and62%vs38%,

respectively)

33%hadstoolincontinence.Rates

ofstool

incontinence

werehigher

innonambulant

children(83%

GMFCSIV–V)

comparedwith

those

who

couldwalk(17%

GMFCSI–III).Ratesofstool

incontinence

werehigher

inchildrenwith

severe

ID(83%

)comparedwith

thosewithoutIDor

mild

tomoderateID(17%

)Sullivanetal(2000)

34CP:Allsubtypes

CP=

93%samplewith

neurologic

impairments

271

CPregister,Oxford,UK

(72%

response

rate)

126%hadconstipation

Excluded:,

80%ofpopulation

butincludedinTable3

Dribbling

Parkes

etal(2010)

35CP:Allsubtypes

1119

PopulationCP

register,NorthernIreland

(86%

of1980–2001

birthcohort)

122%hadexcessivedrooling

Included

$5y

Ratesofdroolingwerehighestamongchildrenwith

moresevere

physicaldisability(GMFCSIVOR

:4.8;

GMFCSVOR

:12.9)

Hearing

Arnaud

etal(2008)

28CP:Allsubtypes

818


8populationCP

registers(63%

participationrate)

12%

hadasevere

hearingimpairment

Excluded:partd

uplicatecases

with

Surm

anetal(2006)36

8–12

y

AustralianCP

Register

Group(2009)

37CP:Allsubtypes

1897

PopulationCP

register,VictoriaandWA,

Australia

(1993–2003

birthcohort)

13%

hadbilateraldeafness

Included

9%hadsomehearingimpairment

Himmelmannetal

(2006)

38CP:Allsubtypes

367

PopulationCP

register,W

estern

Sweden

(89%

of1911–1998

birthcohorts)

12%

hadahearingimpairment

Included

4–8y

Monganetal(2006)

39CP:Allsubtypes

75PopulationCP

register,WestIreland

(88%

of1990–1999

birthcohort)

110%hadahearingimpairment

Included

$5y

3%hadasevere

hearingimpairment

Parkes

etal(2001)

40CP:Allsubtypes

745

PopulationCP


(95%

of1981–1993

birthcohorts)

12%

hadasevere

hearingimpairment

Excluded:partd

uplicatecases

with

Surm

anetal(2006)36

5y

Shevelletal(2009)41

CP:Allsubtypes

243

CPregister,Quebec,Canada

(80%

of1999–2002

birthcohort,80%

response

rate)

111%hadasevere

hearingimpairment

Included

2–5y

Ratesofhearingimpairmentw

erehigher

inthose

with

moreseverephysicaldisability(18%GM

FCSIV

andV)

comparedwith

thosewith

less

physical

disability(8%GM

FCSI-III)(P

,.003)

Sigurdardottiretal

(2008)

42CP:Allsubtypes

148

PopulationCP

register,Iceland

(85%

had

dataavailablefrom

1985–2000

birth

cohort)

12%

hadsomehearingimpairment

Excluded:partd

uplicatecases

with

SigurdardottirandVik

(2011)

434–6y


TABLE1

Continued

Study

Participants

Analyzed

nResearch

Design

Levelof

Evidence

Results

Included/Excluded

Meta-analysis

Sigurdardottiretal

(2009)

44CP:Allsubtypes

139

PopulationCP

register,Iceland

(100%of

1990–1996

and2997–2003

birth

cohort)

11%

to2%

hadsomehearingimpairment

Excluded:partd

uplicatecases

with


(2011)

43


(2011)

43CP:Allsubtypes

152

PopulationCP

register,Iceland

(100%of

1989–2004

birthcohort)

13%

hadsomehearingimpairmentordeafness

Excluded:Datacouldnotbe

separated

4–6y

Surm

anetal(2006)

36CP:Allsubtypes

6910

CPregister,UK.UKCP

=5CP

registers,2

arepopulationbased(Northern

IrelandandScotland)(1960–1997

CPbirthcohort)(87%

haddata

available)

17%

to8%


Included:n

.500

2%hadasevere

hearingimpairment

Surm

anetal(2009)

45CP:Allsubtypes

5019

CPregister,UK.UKCP

=5CP

registers,2



birthcohort)(73%

dataavailable)

18%


Excluded:partd

uplicatecases

with

Surm

anetal(2006)36

Intellectualfunction

Arnaud

etal(2008)

28CP:Allsubtypes

8–12

y818


8populationCP

registers(63%

consented)

153%hadan

IDExcluded:partd

uplicatecases

with

SCPE

(2000)

46

AustralianCP

Register

Group(2009)

37CP:Allsubtypes

1195

PopulationCP

register,Victoria,Australia

(1993–2003

birthcohort)

147%hadan

ID(IQ

,70)

Included

31%hadasevere

ID(IQ

,50)

Bairdetal(2000)

4CP:Allsubtypes

107

PopulationCP

cohort,South

East

Tham

es,UK(100%of1989–1992

birth

cohort)

153%hadan

IDExcluded:partd

uplicatecases

with

Surm

anetal(2009)45

31%hadasevere

ID

Dolketal(2006)

47CP:Allsubtypes

909

PopulationCP


(93%

1981–1987

birthcohorts)

146%hadan

IDExcluded:partd

uplicatecases

with

SCPE

(2000)

46

Dolketal(2010)

48CP:Allsubtypes

3758

CPregister:UKCP5

CPregisters(1984–

1997

birthcohorts)

120%hadasevere

IDExcluded:Rates

notprovidedfor

IQ50–70

El-Tallawyetal(2011)

49CP:Allsubtypes

52Populationsurvey,w

ithdoor

knocking,

Egypt(100%

of1990–2007

birth

cohort)

170%hadan

IDIncluded

Himmelmannetal

(2006)

38CP:Allsubtypes

4–8y

367

PopulationCP

register,W

estern

Sweden

(89%

of1991–1998

birthcohort)

140%hadan

IDIncluded

21%hadasevere

IDHimmelmannetal

(2009)

50CP:Dyskinesiaonly$5y

572

CPregister:SCPE(1976–1996

birth

cohorts)

152%hadasevereID(IQ

,50),comparedwith

33%of

childrenwith

thebilateralspasticsubtype


included

inTable3

Jarvisetal(2005)

51CP:Allsubtypes

3454

CPregister:SCPE(77%

of1976–1990

birthcohorts)

128%hadasevere

ID(IQ

,50)

Excluded:partd

uplicatecases

with

SCPE

(2000)

46

McM

anus

etal

(2006)

52CP:Allsubtypes

9128

SCPE

register

(1980-1996

birthcohorts)

130%hadasevere

ID(IQ

,50)

Excluded:partd

uplicatecases

with

SCPE

(2000)

46

Monganetal(2006)

39CP:Allsubtypes

75PopulationCP

Register,W

estIreland

(88%

of1990–1999

birthcohort)

156%hadan

ID(IQ

,70)

Included

$5y

35%hadasevere

ID(IQ

,50)

Nordmarketal

(2001)

53CP:Allsubtypes

167

PopulationCP

Register,South

Sweden

(100%of1990–1993

birthcohort)

152%hadan

ID(IQ

,70)

Included

6–9y

26%hadasevere

ID(IQ

,50)

Parkes

etal(2001)

40CP:Allsubtypes

5y

745

PopulationCP


(95%

of1981–1993

birthcohorts)

145%hadan

ID(IQ

,70)

Excluded:partd

uplicatecases

with

SCPE

(2000)

46

REVIEW ARTICLE


TABLE1

Continued

Study

Participants

Analyzed

nResearch

Design

Levelof

Evidence

Results

Included/Excluded

Meta-analysis

Parkes

etal(2010)

35CP:Allsubtypes

1357

PopulationCP


(89%

of1980–2001

birthcohort)

148%hadan

ID(IQ

,70)

Excluded:partd

uplicatecases

with

SCPE

(2000)

46$5y

31%hadasevere

ID(IQ

,50)

Rankinetal(2010)

54CP:Allsubtypes

acquired

excluded

1104

CPRegister:Surveillance

ofCerebral

PalsyinEurope

(SCPE),recordlinkage

forFrance,Denmark,andNorthern

England(87%

availabledata)

126%hadasevere

ID(IQ

,50)

Excluded:partd

uplicatecases

with

SCPE

(2000)

46

Ravn

etal(2010)

55CP:Allsubtypes

1185

PopulationCP

register,Denmark(.

85%

ascertainm

ent,1983–1998

birth

cohortofEasternDenm

ark)

162%hadan

IDIncluded

5–6y

SCPE

(2000)

46CP:Allsubtypes

3714


birth

cohorts)

123%to44%hadan

ID(IQ

,70)

Included:n.500

30%to41%hadasevere

ID(IQ

,50)

SCPE

(2002)

56CP:Allsubtypes

6502

CPregister:SCPE(73%

of1980–1990

birthcohorts)

131%hadasevere

ID(IQ

,50)

Excluded:partd

uplicatecases

with

SCPE

(2000)

46

Sigurdardottiretal

(2008)

42CP:Allsubtypes

148

PopulationCP

register,Iceland

(85%

had

dataavailablefrom

1985–2000

birth

cohort)

140%hadan

ID(IQ

,70)

Excluded:partd

uplicatecases

with


(2011)

434–6y

21%hadasevere

ID(IQ

,50)

Sigurdardottiretal

(2009)

44CP:Allsubtypes

139

PopulationCP

register,Iceland

(100%of

1990–1996

and1997–2003

birth

cohort)

140%to51%hadan

ID(IQ

,70)

Excluded:partd

uplicatecases

with


(2011)

43


(2011)

43CP:Allsubtypes

152

PopulationCP

register,Iceland

(100%of

1989–2004

birthcohort)

139%hadan

ID(IQ

,70)

Included

4–6y

21%hadasevere

ID(IQ

,50)

Surm

anetal(2003)

57CP:Allsubtypes

898

Oxford

Register

ofEarlyChildhood

Impairments,England

(1984–1995

CPbirthcohort)

150%hadan

IDExcluded:partd

uplicatecases

with

Surm

anetal(2009)45

Surm

anetal(2006)

36CP:Allsubtypes

6910

CPregister,UK.UKCP

=5CP

registers,2



CPbirthcohort)

139%to51%hadan

IDExcluded:partd

uplicatecases

with

Surm

anetal(2009)45

24%to31%hadasevere

ID

Surm

anetal(2009)

45CP:Allsubtypes

5019

CPregister,UK.UKCP

=5CP

registers,2



birthcohort)(73%

dataavailable)

148%hadan

IDIncluded:n

.500

Wichers

etal(2005)

58CP:Allsubtypes

6–19

y127

PopulationCP

study,Netherlands(100%

of1977–1988

birthcohort

population)

134%to43%hadan

ID(IQ

,70)

Included

Pain Arnaud

etal(2008)

28CP:Allsubtypes

818


8populationCP

registers(63%

participationrate)

172%hadpain

Included:n

.500

8–12

y54%hadmoderatepainand18%hadsevere

pain

Painwas

associated

with

poor

quality

oflifeinthe

physicalwell-being

(OR:5.2[95%

CI:2.7–9.7])(P,

.001)andpsychologicalw

ell-being

(OR:2.9[95%

CI:1.7–4.9])(P,.001)and

self-perception(OR:2.7

[95%

CI:1.6–4.4])(P

,.001)domains


TABLE1

Continued

Study

Participants

Analyzed

nResearch

Design

Levelof

Evidence

Results

Included/Excluded

Meta-analysis

Jahnsenetal(2004)

59CP:Allsubtypes

without

IDCP:406

Prospectivepopulationcohortstudy,

Norw

ay(53%

response

rate)

3[Prognostic]

78%hadpain;45%

hadmoderatetosevere

pain

Included:,

80%ofpopulation

butb

estp

opulationdata

availablebecauseofcase

controls

18–72

yControls:2287

28%hadchronicpaincomparedwith

15%inthe

population

82%hadpaininatleast1

body

part.Paininnumber

ofbody

parts:1only,18%

;2to3parts,43%;3

to5

parts,42%;and

$5parts,13%

Numberofpainfulbodypartswas

significantly

associated

with

numberofjointswith

contracture

(P,

0.001)

Painincreasedwith

overactivity

(73%

),inactivity

(26%

),andcoldweather

(14%

)Paindecreasedwith

rest(51%

),physiotherapy

(49%

),andmedication(35%

).andwarmweather

(28%

)Kennes

etal(2002)

29CP:Allsubtypes

408

Prospectivecohortstudy,Canada

(18%

ofpopulationcohortrandom

lysampled,96%

return

rate)

114%hadpain

Excluded:,

80%ofpopulation

butincludedinTable3

5–13

yPainwas

notrelated

tolevelofp

hysicaldisability

(GMFCS)

(P=.37)

Opheim

etal(2011)

60CP:Hem

iplegiaand

diplegiasubtypes

withoutID

288

Prospectivepopulationcohortstudy,

Norw

ay(51%

longitudinalfollowup

tostudyby

Jahnsenetal(2004)

59

5a83%hadpain


included

inTable3

18–72

yMorepeopleexperiencedpainastheyaged;22%

were

pain-free

initially,but

only17%werepain-free

7yearslater

Back/neck,foot,and

anklepainweremostcom

mon

Parkes

etal(2008)

22CP:Allsubtypes

818

Cross-sectionalsurveyfrom

8CP

registers:SCPE

(70%

participation

rate)

1Ratesofbehavioralproblemswerehigherinchildren

with

severe

paincomparedwith

children

withoutp

ain(OR:2.7[95%

CI:1.5–4.6])

Excluded:Duplicatecaseswith

Arnaud

etal(2008)

28but

included

inTable3

8–12

y

Russoetal(2008)

30CP:Hem

iplegiaonly

107

PopulationCP

register,prospective

assessment(75%participationrate,

100%

follow-up)

148%hadpain


included

inTable3

3–16

yRatesofbehavioralcompetencewerehigher

for

childrenwithoutpain(m

ean:3.3)than

forchildren

with

pain(m

ean:2.9)

(P,

.01)

Sleeping

REVIEW ARTICLE


TABLE1

Continued

Study

Participants

Analyzed

nResearch

Design

Levelof

Evidence

Results

Included/Excluded

Meta-analysis

Newman

etal(2006)

61CP:Allsubtypes

173

Prospectivecohortstudy,Ireland(70%

response

rate)

5a23%hadpathologicsleepdisorder

Included:Onlystudyavailable

6–11

y24%haddifficulty

initiatingandmaintaining

sleep

18%hadsleep–waketransitiondisorders

15%hadsleep-relatedbreathingdisorders

11%hadexcessivesomnolence

8%hadarousaldisorders

6%hadsleephyperhidrosis

Pathologicsleepwas

highlyassociated

with

uncontrolledepilepsy,single-parentfam

ilies,and

cosleeping

Disordersofinitiatingandmaintaining

sleepwere

associated

with

4-lim

binvolvem

ent(spastic

quadriplegiaanddyskinesia)andsevere

visual

impairment

Totalpathologicsleeprateinstudysamplewas

23%

comparedwith

5%innorm

alchild

population

Vision Arnaud

etal(2008)

28CP:Allsubtypes

818


8populationCP

registers(63%

consented,93%follow-up)

17%

hadfunctionalblindness

Excluded:partd

uplicatecases

with

McM

anus

etal(2006)

528–12

y

AustralianCP

Register

Group(2009)

37CP:Allsubtypes

1897

PopulationCP


Australia

(1993–2003

birthcohort)

15%

hadfunctionalblindness

Included

29%hadsomevision

impairment

13%hadstrabism

usonly

Himmelmannetal

(2006)

38CP:Allsubtypes

367

PopulationCP

register,W

estern

Sweden

(89%

of1991–1998

birthcohort)

119%hadsevere

visualimpairments

Included

4–8y

Ratesofvisualimpairmentw

erehigherinthosewith

severe

physicaldisability

McM

anus

etal

(2006)

52CP:Allsubtypes

9128

SCPE

register(1980–1996

birthcohorts)

112%hadasevere

visualimpairment

Included:n

.500

Monganetal(2006)

39CP:Allsubtypes

85PopulationCP


(88%

of1990–1999

birthcohort)

112%hadavisualimpairment

Included

$5y

6%hadasevere

visualimpairment

Nordmarketal

(2001)

53CP:Allsubtypes

167

PopulationCP

Register,South

Sweden

(100%of1990–1993

birthcohort)

122%hadavisualimpairment

Included

6–9y

Parkes

etal(2001)

40CP:Allsubtypes

5y

745

PopulationCP


(95%

of1981–1993

birthcohorts)

111%hadasevere

vision

impairment

Excluded:partd

uplicatecases

with

Surm

anetal(2006)36


TABLE1

Continued

Study

Participants

Analyzed

nResearch

Design

Levelof

Evidence

Results

Included/Excluded

Meta-analysis

Saunders

etal

(2010)

62CP:Allsubtypes

CP:118

PopulationCP


(100%ofdataavailable)

1High

refractiveerrorsweremorecommon

inCP

than

matched

controls

Excluded:partd

uplicatecases

with

Surm

anetal(2006)36

4–23

yControls:128

Ratesofhigh

myopiaweremorecommon

inCP

(9%)

comparedwith

controls(0%)(P

,.05)

Ratesoflow–moderatehyperm

etropiaweremore

common

inCP

(42%

)comparedwith

controls

(9%)(P

,.05)

Ratesofhigh

hyperm

etropiaweremorecommon

inCP

(12%

)comparedwith

controls(2%)(P

,.05)

Ratesofastigmatismweremorecommon

inCP

(36%

)comparedwith

controls(3%)(P

,.05)

Ratesofanisom

etropiaweremorecommon

inCP

(18%

)comparedwith

controls(7%)(P

,.05)

SCPE

(2002)

56CP:Allsubtypes

6502

CPregister:SCPE(73%

of1980–1990

birthcohorts)

111%hadasevere

vision

impairment

Excluded:partd

uplicatecases

with

McM

anus

etal(2006)

52

Shevelletal(2009)41

CP:Allsubtypes

243

CPRegister,Quebec,Canada

(80%

of1999–2002

birthcohort,80%

response

rate)

19%

hadasevere

vision

impairment

Included

2–5y

Ratesofvision

impairmentw

erehigherinthosewith

moresevere

physicaldisability(22%

GMFCS

IVandV)

comparedwith

thosewith

less

physical

disability(3%GM

FCSI–III)(P

,.001)

Sigurdardottiretal

(2008)

42CP:Allsubtypes

148

PopulationCP

register,Iceland

(85%

had

dataavailablefrom

1985–2000

birth

cohort)

112%hadsomevisualimpairment

Excluded:partd

uplicatecases

with

Sigurdardottiran

Vik

(2011)

434–6y

Sigurdardottiretal

(2009)

44CP:Allsubtypes

139

PopulationCP

register,Iceland

(100%of

1990–1996

and1997–2003

birth

cohort)

111%to19%hadavisualimpairment

Excluded:partd

uplicatecases

with


(2011)

43


(2011)

43CP:Allsubtypes

152

PopulationCP

register,Iceland

(100%of

1989–2004

birthcohort)

113%hadsomevisualimpairmentorfunctional

blindness

Included

4–6y

Surm

anetal(2003)

57CP:Allsubtypes

898

Oxford

Register

ofEarlyChildhood

Impairments,UK(1984–1995

CPbirth

cohort)

123%hadavision

impairment

Excluded:partd

uplicatecases

with

Surm

anetal(2006)36

Surm

anetal(2006)

36CP:Allsubtypes

6910

CPregister,UK.UKCP

=5CP

registers,2



birthcohort)(83%

haddata

available)

134%to40%hadavision

impairment

Included:n.500

9%to11%hasasevere

vision

impairment

Surm

anetal(2009)

45CP:Allsubtypes

5019

CPregister,UK.UKCP

=5CP

registers,2



birthcohort)(73%

dataavailable)

143%hadavision

impairment

Excluded:partd

uplicatecases

with

Surm

anetal(2006)36

Wichers

etal(2005)

58CP:Allsubtypes

127

PopulationCP

study,Netherlands(100%

of1977–1988

birthcohort)

125%to42%hasavision

impairment

Included

6–19

y5%

to19%hadasevere

vision

impairment

Activity

limitations

Eating

REVIEW ARTICLE


TABLE1

Continued

Study

Participants

Analyzed

nResearch

Design

Levelof

Evidence

Results

Included/Excluded

Meta-analysis

Andersen

etal(2010)

63CP:Allsubtypes

557

PopulationCP

register,Norway

(97%

of1996–2003

birthcohorts)

124%weretotally

dependentfor

feedingor

were

tube-fed

Excluded:Prevalencedataabout

dependenteatingandtube

feedingcombined

$4y

Himmelmannetal

(2007)

64CP:Bilateralspasticonly

167

PopulationCP

register,W

estern

Sweden

(97%

of1991–1998

birthcohort)

112%wereunderw

eight(.2SDsbelowmeanforB

MI)


included

inTable3

Motionetal(2002)

65CP:Allsubtypes

asan

outcom

eof

earlyfeeding

problems

CP:37

Longitudinalcohortp

opulationstudy

(prevalenceof2.6/1000)

2[Prognostic]

Ratesofweaksuckingat4weeks

ofagewerehigher

inCP

(48%

)comparedwith

norm

alpopulation

(18%

)(P

,.001)

Excluded:Insufficientprevalence

detailbutincluded

inTable3

7–8y

Controls:12332

Ratesoffeedingdifficulties

at6monthswerehigher

with

CP(10%

)comparedwith

norm

alpopulation

(3%)(P

=.017)

Weaksuckingandexhaustionwith

feedingat4

weeks

ofagewas

associated

with

spastic

quadriplegicCP

Parkes

etal(2010)

35CP:Allsubtypes

1119

PopulationCP


(86%

of1980–2001

birthcohort)

121%hadasw

allowingor

chew

ingimpairment

Excluded:Insufficientprevalence

detail

$5y

Shevelletal(2009)41

CP:Allsubtypes

243

CPRegister,Quebec,Canada

(80%

of1999–2002

birthcohort,80%

response

rate)

18%

weretube-fed

Included

2–5y

Ratesoftube

feedingwerehigherinthosewith

more

severe

physicaldisability(79%

GMFCSIVandV)

comparedwith

thosewith

less

physicaldisability

(21%

GMFCSI–III)(P

,.001)


(2011)

43CP:Allsubtypes

152

PopulationCP

register,Iceland

(100%of

1989–2004

birthcohort)

17%

weretube-fed

Included

4–6y

Straussetal(2004)

66CP:Allsubtypes

904

Longitudinalcohortstudy,California

(100%ofavailabledata)

1Ratesoftube

feedingwerecomparableinCP

at20

years(4%)and60

years(2%)

Included:n.500

20,40,and60

yRatesofdependentfeeding

werecomparableat20

years(16%

)and60

years(14%

)Ratesoffinger

feedingwerecomparableinCP

at20

years(9%)and60

years(6%)

Ratesofeatingwith

cutlery

werecomparableinCP

at20

years(71%

)and60

years(78%

)2%

to6%

unabletofeed

self15

yearslater

Wichers

etal(2009)

67CP:Spasticonly

119

Prospectivecross-sectionalpopulation

basedsurvey,Netherlands

(.90%of

1977–1988

birthcohort,100%follow-

up)

123%hadfeedinganddrinking

problemsordependent

forcare

Excluded:Notallsubtypes

6–19

y

Talking

Andersen

etal(2010)

63CP:Allsubtypes

557

PopulationCP

register,Norway

(97%

of1996–2003

birthcohorts)

119%werenonverbal

Included

31%hadsomeimpairment

$4y

48%hadnorm

alspeech

Ofthosewith

speech

impairments,54%

used

AlternativeandAugm

entativeCommunication


TABLE1

Continued

Study

Participants

Analyzed

nResearch

Design

Levelof

Evidence

Results

Included/Excluded

Meta-analysis

Arnaud

etal(2008)

28CP:Allsubtypes

818


8populationCP

Registers(63%

participationrate)

127%werenonverbal

Excluded:partd

uplicatecases

with

Parkes

etal(2010)

358–12

y43%hadsomeimpairment,ofthese16%used

speech

butw

ithdifficulty

AustralianCP

Register

Group(2009)

37CP:Allsubtypes

1897

PopulationCP


Australia

(1993–2003

birthcohort)

127%werenonverbal

Included

33%hadsomeimpairment

Parkes

etal(2010)

35CP:Allsubtypes

1119

PopulationCP


(88%

of1980–2001

birthcohort)

119%werenonverbal

Included

$5y

36%hadsomespeech

impairment

Shevelletal(2009)41

CP:Allsubtypes

243


(80%

of1999–2002

birthcohort,80%

response

rate)

122%werenonverbal

Included

2–5y

Ratesofbeingnonverbalw

erehigher

inthosewith

moreseverephysicaldisability(57%GM

FCSIVand

V)comparedwith

thosewith

less

physical

disability(4%GM

FCSI–III)(P

,.001)


(2011)

43CP:Allsubtypes

152

PopulationCP

register,Iceland

(100%of

1989–2004

birthcohort)

124%werenonverbal

Included

4–6y

Ratesofbeingnonverbalw

erehigher

inthosewith

moresevere

physicaldisability

Straussetal(2004)

66CP:Allsubtypes

904



119%to36%werenonverbal

Included:n

.500

20,40,and60

yWalking

Andersen

etal(2010)

63CP:Allsubtypes

557

PopulationCP

register,Norway

(97%

of1996–2003

birthcohorts)

126%didnotw

alk

Included

$4y

19%walkedusingawalking

fram

eor

device

55%walkedindependently

GMFCSunavailable(collapsed)

Arnaud

etal(2008)

28CP:Allsubtypes

818


8populationCP

Registers(63%

parent

participationrate)

132%didnotw

alk

Excluded:partd

uplicatecases

with

Beckungetal(2008)

688–12

y17%walkedusingawalking

fram

eor

device


GMFCSavailable

AustralianCP

Register

Group(2009)

37CP:Allsubtypes

1195

PopulationCP

registerVictoria,Australia

(1993–2003

birthcohort)

130%didnotw

alk

Included


fram

eor

device


GMFCSavailable

Beckungetal(2008)

68CP:Allsubtypes

9012


ofCerebral

PalsyinEurope

(SCPE)(1976–1996

birthcohorts)

130%didnotw

alk

Included:n

.500

,21


fram

eor

device


Inabilitytowalkmorecommon

with

ID(ID

)(IQ

,50)

(71%

)comparedwith

noID(IQ

.85)(8%)

Inabilitytowalkmorecommon

with

activeepilepsy

(60%

)comparedwith

noepilepsy(25%

)GM

FCSunavailable

REVIEW ARTICLE


TABLE1

Continued

Study

Participants

Analyzed

nResearch

Design

Levelof

Evidence

Results

Included/Excluded

Meta-analysis

Dayetal(2007)

69CP:Allsubtypes,

ambulant

only

CP10

y:7550

RetrospectivecohortCalifornia

116%of10-year-olds

and9%

of25-year-olds

didnot

walk

Excluded:notallseverity

levels.

Included

inTable3

CP25

y:5721

26%of10-year-olds

and19%of25-year-olds

walked

usingawalking

fram

eor

device

58%of10-year-olds

and72%of25-year-olds

walked

independently

Childrenthatcouldwalkandclimbstairs

atage10

yearsonlyhada23%chance

ofdecline15

years

later

Childrenthatcouldwalkwith

difficulty

butd

idnot

need

achairhada33%chance

ofimprovem

ent15

yearslaterandan

11%chance

ofbeing

wheelchairdependent

Childrenthatused

awheelchairwere34%more

likelytolose

theirwalking

ability

GMFCSunavailable

Dolketal(2006)

47CP:Allsubtypes

909

PopulationCP

Register,NorthernIreland

(1981–1987

birthcohorts)

129%didnotw

alk

Excluded:partd

uplicatecases

with

Beckungetal(2008)

68GM

FCSunavailable(partialreporting)

Hannaetal(2008)

70CP:Allsubtypes

657

Longitudinalstudy,OntarioCanada

(73%

consented)

140%didnotw

alk

Included:n.500

$6y


fram

eor

device


GMFCSavailable

Himmelmannetal

(2006)

38CP:Allsubtypes

367

PopulationCP

register,W

estern

Sweden

(89%

of1911-1998birthcohorts)

131%didnotw

alk

Included

4–8y


fram

eor

device


GMFCSavailable

Howardetal(2005)

71CP:Allsubtypes

323

PopulationCP

Register,Victoria,

Australia

(86%

of1990-1993birth

cohort)

134%didnotw

alk

Excluded:partd

uplicatecases

with

ACPR

(2009)


fram

eor

device


GMFCSavailable

Jarvisetal(2005)

51CP:Allsubtypes

3454


ofCerebral

PalsyinEurope

(SCPE)(1976–1990

birthcohorts)

145%didnotw

alkor

walkedusingawalking

fram

eor

device

Excluded:partd

uplicatecases

with

Beckungetal(2008)

68


GMFCSunavailable

McM

anus

etal

(2006)

52CP:Allsubtypes

9128


birth

cohorts)

130%didnotw

alk

Excluded:partd

uplicatecases

with

Beckungetal(2008)

68GM

FCSunavailable

Michelsen

etal(2005)

2CP:Allsubtypes

CP:819

PopulationCP

register,Denmark(n=819

CP,(100%

)of1965–1978

CPbirth

cohort)

2[Prognostic]

16%didnotw

alk

Included

Controls:4406


fram

eor

device

21–35

y62%walkedindependently

GMFCSunavailable

Monganetal(2006)

39CP:Allsubtypes

85PopulationCP


(88%

of1990–1999

birthcohort)

118%didnotw

alk

Included

$5y


fram

eor

device


GMFCSunavailable


TABLE1

Continued

Study

Participants

Analyzed

nResearch

Design

Levelof

Evidence

Results

Included/Excluded

Meta-analysis

Nordmarketal

(2001)

53CP:Allsubtypes

167

PopulationCP

register,South

Sweden

(100%of1990–1993

birthcohort)

127%didnotw

alk

Excluded:partd

uplicatecases

with

Westbom

etal(2007)72

6–9y


fram

eor

device


GMFCSavailable

Parkes

etal(2001)

40CP:Allsubtypes

745

PopulationCP


(95%

of1981–1993

birthcohorts)

129%didnotw

alk

Excluded:partd

uplicatecases

with

Beckungetal(2008)

685y

GMFCSunavailable

Parkes

etal(2010)

35CP:Allsubtypes

1215

PopulationCP


(90%

of1980–2001

birthcohort)

129%didnotw

alk

Excluded:partd

uplicatecases

with

Beckungetal(2008)

68$5y


fram

eor

device


GMFCSavailable

Ravn

etal(2010)

55CP:Allsubtypes

1185

PopulationCP

register,Denmark(.

85%

of1983–1998

birthcohortEastern

Denm

ark)

140%didnotw

alkor

walkedusingawalking

fram

eor

device

Excluded:duplicatecaseswith

Michelsen

etal(2005)

2

5–6y


GMFCSunavailable

Reidetal(2010)

73CP:Allsubtypes

singletons

only

CP:1241

PopulationCP

register,Victoria,Australia

(1991–2004

birthcohort)(92%

availabledata)

3[Prognostic]

30%didnotw

alk

Excluded:partd

uplicates

with

ACPR

(2009)

37Controls:2482


fram

eor

device


GMFCSavailable

Rosenbaumetal

(2002)

1CP:Allsubtypes

657

Cross-sectionalstudy,OntarioCanada

(31%

of1986–1996

birthcohort)

141%didnotw

alk

Excluded:partd

uplicatewith

Hannaetal(2008)

701–13


fram

eor

device


GMFCSavailable

Saunders

etal

(2010)

62CP:Allsubtypes

CP:118

PopulationCP


(97%

dataavailable)

135%didnotw

alk

Excluded:partd

uplicatecases

with

Beckungetal(2008)

684–23

yControls:128


fram

eor

device


GMFCSavailable

SCPE

(2002)

46CP:Allsubtypes

6502

CPregister:SCPE(73%

of1980–1990

birthcohorts)

131%didnotw

alk

Excluded:partd

uplicatecases

with

Beckungetal(2008)

68GM

FCSunavailable

Shevelletal(2009)41

CP:Allsubtypes

243


(80%

of1999–2002

birthcohort,80%

response

rate)

134%didnotw

alk

Included


fram

eor

device


GMFCSavailable

Sigurdardottiretal

(2008)

42CP:Allsubtypes

148

PopulationCP

register,Iceland

(85%

had

dataavailablefrom

1985–2000

birth

cohort)

123%didnotw

alk

Excluded:partd

uplicatecases

with

Sigurdardottiretal

(2009)

444–6y


orwalkedusingawalking

fram

eor

device


Sigurdardottiretal

(2009)

44CP:Allsubtypes

139

PopulationCP

register,Iceland

(100%of

1985–2000

birthcohort)

125%didnotw

alk

Included


fram

eor

device




(2011)

43CP:Allsubtypes

152

PopulationCP

register,Iceland

(100%of

1989–2004

birthcohort)

124%didnotw

alk

Excluded:partd

uplicatecases

with

Sigurdardottiretal

(2009)

444–6y


fram

eor

device

orwalkedindependently


REVIEW ARTICLE


TABLE1

Continued

Study

Participants

Analyzed

nResearch

Design

Levelof

Evidence

Results

Included/Excluded

Meta-analysis

Straussetal(2004)

66CP:Allsubtypes

904



135%to40%didnotw

alk

Included:n.500

20,40,and60

y25%to35%walkedusingawalking

fram

eor

device

25%to39%walkedindependently

GMFCSunavailable

Surm

anetal(2006)

36CP:Allsubtypes

6910

UKCP

database

of5CP

registers,2were

population-based(NorthernIreland

andScotland)(1960–1997

birth

cohort)

129%to31%didnotw

alk

Included:n.500

GMFCSunavailable

Westbom

etal(2007)

72CP:Allsubtypes

343

PopulationCP

register,SouthernSw

eden

(100%of1990–1997

birthcohort)

122%didnotw

alk

Included

4–11


fram

eor

device


GMFCSavailable

Wichers

etal(2005)

58CP:Allsubtypes

127

Populationstudy,theNetherlands100%

of1977–1988

birthcohort)

139%didnotw

alk

Excluded:partd

uplicatecases

with

Wichers

etal(2009)

676–19

y61%walkedindependently

orusingawalking

fram

e/device

GMFCSunavailable

Wichers

etal(2009)

67CP:Spasticonly

119


survey,the

Netherlands(.

90%of

1977–1988


up)

127%didnotw

alk

Included

6–19

y8%

walkedusingawalking

fram

eor

device


GMFCSavailable

Wuetal(2004)

74CP:Allsubtypes

2295

Retrospectivecohort,California(43%

follow-up,1987–1999

birthcohort)

1In2-year-olds,prognosisoffutuream

bulationby

age

6yearsisp

redicted

by:(1)motormilestonesatage

2years(including:rolling[OR:4.6(95%

CI:2.2–

9.6)];sitting[OR:12.5(95%

CI:5.8–27.2)];and

pulling

tostand[OR:28.5(95%

CI:13.4–60.4)];(2)

type

ofCP

(spasticquadriplegiahashighestrisk

fornotw

alking);and(3)blindness

Excluded:partd

uplicatecases

with

Straussetal(2004)

66

Included

inTable3

Nonambulant

atGM

FCSunavailable(collapsed)

2–3.5y

Co-occurring

diseases

Epilepsy

Arnaud

etal(2008)

28CP:Allsubtypes

818


8populationCP

registers(63%

consented)

130%hadactiveepilepsy

Excluded:partd

uplicatecases

with

Surm

anetal(2006)36

8–12

y

AustralianCP

Register

Group(2009)

37CP:Allsubtypes

1897

PopulationCP


Australia

(1993–2003

birthcohort)


Included

2%hadresolved

epilepsy

Carlsson

etal(2003)

75CP:Allsubtypes

146

PopulationCP

register,GoteborgSw

eden

(100%1987–1994

birthcohort)

138%hadepilepsy

Included

6–14

yRatesofepilepsyincreasedwith

severityofmotor

disability(P

,.001).

Ratesofepilepsyweresignificantlyhigher

forthe

spastic

type

born

atterm

(48%

)comparedwith

preterminfants(28%

),P=.04

Ratesofepilepsyweresignificantlyhigher

for

childrenwith

CPplus

ID(61%

)comparedwith

CPwith

norm

alintelligence(19%

),P,

.001


TABLE1

Continued

Study

Participants

Analyzed

nResearch

Design

Levelof

Evidence

Results

Included/Excluded

Meta-analysis

El-Tallawyetal(2011)

49CP:Allsubtypes

52Populationsurvey,w

ithdoor

knocking,

Egypt(100%

of1990–2007

birth

cohort)

152%hadepilepsy

Included

Himmelmannetal

(2006)

38CP:Allsubtypes

367

PopulationCP

register,W

estern

Sweden

(89%

of1991–1998

birthcohort)

133%hadepilepsy

Included

4–8y

Ratesofepilepsywerehigher

inthosewith

more

severe

orbilateralm

otor

involvem

ent(87%

tetraplegia;52%dyskinesia;34%

diplegia)

comparedwith

thosewith

hemiplegic/unilateral

motor

involvem

ent(23%)

Michelsen

etal(2005)

2CP:Allsubtypes

CP:819

PopulationCP

register,Denmark(100%

of1965–1978

birthcohort)

3[Prognostic]

17%hadepilepsy

Included

21–35

yControls:4406

Epilepsywith

CPwas

asignificant

predictorofnot

beingcompetitivelyem

ployed

inadulthood(OR:

3.69

[95%

CI:1.46–9.36],P=.0439)

Monganetal(2006)

39CP:Allsubtypes

75PopulationCP


(88%

of1990–1999

birthcohort)

146%hadepilepsyatsometim

eIncluded

$5y


Ofthosewith

epilepsy,58%hadan

IDand44%had

spastic

quadriplegia

Nordmarketal

(2001)

53CP:Allsubtypes

167

PopulationCP

register,South

Sweden

(100%of1990–1993

birthcohort)

136%hadepilepsy

Included

6–9y

Parkes

etal(2001)

40CP:Allsubtypes

745

PopulationCP


(95%

of1981–1993

birthcohorts)


Excluded:partd

uplicatecases

with

Parkes

etal(2010)

355y

Parkes

etal(2010)

35CP:Allsubtypes

1119

PopulationCP


(91%

of1980–2001

birthcohort)

143%hadepilepsyatsometim

eExcluded:partd

uplicatecases

with

Surm

anetal(2006)36

$5y


Ravn

etal(2010)

55CP:Allsubtypes

1185

PopulationCP

register,Denmark(.85%

of1983–1998

birthcohortEastern

Denm

ark)

129%hadepilepsy

Included

5–6y

SCPE

(2002)

56CP:Allsubtypes

6502

CPregister:SCPE(73%

of1980–1990

birthcohorts)


Excluded:partd

uplicatecases

with

Surm

anetal(2006)36

Shevelletal(2009)41

CP:Allsubtypes

243


(80%

of1999–2002

birthcohort,80%

response

rate)


Included

2–5y

Ratesofactiveepilepsyweresignificantlyhigher

inthosewith

less

grossmotor

function(32%

inGM

FCSIVandV)comparedwith

thosewith

better

grossmotor

ability(9%inGM

FCSI–III)(P

,.001)

Sigurdardottiretal

(2008)

42CP:Allsubtypes

148

PopulationCP

register,Iceland

(85%

had

dataavailablefrom

1985–2000

birth

cohort)

127%hadepilepsy

Excluded:partd

uplicatecases

with

Sigurdardottiretal

(2010)31

4–6y

Sigurdardottiretal

(2009)

44CP:Allsubtypes

139

PopulationCP

register,Iceland

(100%of

1997–2003

birthcohort)

115%to38%hadepilepsy(15%

in1997–2003

birth

cohorts,38%in1990–1996

birthcohorts)

Excluded:partd

uplicatecases

with

Sigurdardottiretal

(2010)

31Ratesofepilepsyweresignificantlydecreasing

over

time(P

=.002)


(2011)

43CP:Allsubtypes

152

PopulationCP

register,Iceland

(100%of

1989–2004

birthcohort)


Included

4–6y

REVIEW ARTICLE


TABLE1

Continued

Study

Participants

Analyzed

nResearch

Design

Levelof

Evidence

Results

Included/Excluded

Meta-analysis

Surm

anetal(2006)

36CP:Allsubtypes

6910

CPregister,UK.UKCP

=5CP

registers,2



CPbirthcohort)(52%

haddata

available)

118%to33%hadepilepsy

Included:n.500

Wichers

etal(2005)

58CP:Allsubtypes

127

Populationstudy,theNetherlands(100%

of1977–1988

birthcohort)

140%hadactiveor

previouslytreatedepilepsy

Included

6–19

yRatesofactiveor

previouslytreatedepilepsywere

higher

inthosewith

bilateralspastic(42%

)or

ataxic/dyskinetic

(63%

)motor

typescompared

with

thosewith

unilateralm

otor

involvem

ent

(33%

).Zafeiriouetal(1999)

76CP:Allsubtypes

CP+Epilepsy:

178

Longitudinalprospectivecohortstudy,

Greece

(36%

ofCP

population)

3[Prognostic]

70%hadtheirfirstseizurewithintheirfirstyearoflife

Excluded:,

80%ofpopulation

butincludedinTable3

5yand$5yfollow-up

Epilepsy

Controls:150

Ratesofbecomingseizure-free

after3yearsof

antiepilepticmedicationwerelower

inCP

(75%

),comparedwith

thosewith

epilepsyonly(81%

)(P

,.05)

Hipdisplacementand

spinedeform

ities

Hagglund

etal(2005)

77CP:Allsubtypes

CP:258

Southern

Sweden

cerebralpalsy

populationregister

(89%

of1991–

1997

birthcohort)

18%

ofcontrolshaddislocated

hips

Included

Control:103

0%ofhipsurveillancegrouphaddislocated

hips

(P,

.001)

21%ofthehipsurveillancegrouphaddisplacedhips

andreceived

treatm

ent,includingorthopedic

surgery

After10

yearsoffollow-up,2childreninsouthern

Sweden

haddislocated

hips,neither

ofwhom

wereinthehipsurveillanceprogram

Hagglund

etal(2005)

78CP:Allsubtypes

CP:221

PopulationCP

register,South

Sweden

(95%

of1991–1995

birthcohort)

1Ratesoforthopedicsurgerytocorrectd

eformity,

contracture,andsalvageofdislocated

hips

were

lower

inthecohortsregularlytreatedwith

SDR,

ITB,andbotulinum

andprovided

active

surveillancewerelower(15%

)com

paredwith

the

earlycontrolcohortinwhomthesetreatm

entsand

activesurveillancewerenotp

rovided(40%

)

Excluded:partd

uplicatecases

with

Hagglund

etal(2005)

77Control:74

Hagglund

etal(2007)

79CP:Allsubtypes

212

PopulationCP

register,South

Sweden

(97%

of1992–1997

birthcohort)

127%haddisplacedhips

Excluded:partd

uplicatecases

with

Hagglund

etal(2005)

77Hipdisplacementw

asrelatedtoseverityofphysical

disability(0%GM

FCSI;64%GM

FCSV)

Loetersetal(2010)

80CP:GMFCSIV–V

NASystem

aticreview

4Low-levelevidenceprecludedfirm

recommendations

onrisk

factorsforscoliosis

Excluded:Asperconventionfor

review

sPersson-Bunkeetal

(2006)

81CP:Allsubtypes

CP:207

PopulationCP

register,South

Sweden

(100%of1991–1995

birthcohort)

112%ofcontrolshadwindsweptd

eformity

Excluded:partd

uplicatecases

with

Hagglund

etal(2005)

77Control:68

7%ofhipsurveillancegrouphadwindswept

deform

ity


26–42) had epilepsy at some point, and24% (95% CI: 17–29) had active epilepsy;(6) hearing (9492 cases for analysis):4% (95% CI: 2–6) had a severe hearingimpairment or were deaf; (7) hips andspine (632 cases for analysis): 28% (95%CI: 21–34) had displaced hips (ie, mi-gration .30%) and 7.5% (95% CI: 7–8)had dislocated hips if they had not re-ceived hip surveillance, but this wasreduced to 0% dislocation with surveil-lance; (8) intellectual function (12 053cases for analysis): 49% had an in-tellectual disability (ID) (IQ ,70) (95%CI: 34–64) and 28% had a severe ID(IQ,50) (95% CI: 21–35); (9) pain (1224cases for analysis): 75% were in pain(95% CI: 72–78); (10) sleep (173 casesfor analysis): 23% had a pathologicsleep disorder (95% CI unable to becalculated because only 1 study wasincluded); (11) talking (4872 cases foranalysis): 23% (95% CI: 19–27) werenonverbal; (12) walking (21 350 casesfor analysis): 28% (95% CI: 16–40) couldnot walk, 16% walked with assistance(95% CI: 8–24), and 56% walked in-dependently (95% CI: 32–80), withGMFCS (2924 cases for analysis) levelI at 36% (95% CI: 26–46), level II at 22%(95%CI: 9–35), level III at 11% (95%CI: 8–14), level IV at 14% (95% CI: 8–20), andlevel V at 17% (95% CI: 13–21); and (13)vision (19 076 cases for analysis): 11%(95% CI: 5–17) were functionally blind.

The data regarding average rates ofimpairments were combined withprognostic data from the includedstudies regarding the likelihood of un-wanted events occurring in the future.From these data, we developed a seriesof simple, plain English clinical mes-sages that could be communicated topeople with cerebral palsy and theirfamilies about the prognosis of thecondition (Table 3).

DISCUSSION

In this systematic literature reviewregarding the rates of co-occurringTA

BLE1

Continued

Study

Participants

Analyzed

nResearch

Design

Levelof

Evidence

Results

Included/Excluded

Meta-analysis

Sooetal(2006)

82CP:Allsubtypes

374

Retrospectivestudyfrom

VictorianCP

populationregister,Australia(86%

ofCP

population)

135%hadhipdisplacementw

ithamigration

percentage

.30°

Included

7%hadhipdislocation

Ratesofhipdisplacementw

ererelatedtoGM

FCS

severitylevel(P,

.001)andmotor

type

with

spastic

quadriplegiabeingthebiggestriskfactor

(RR:4.3[95%

CI:2.7–6.8])

Wichers

etal(2009)

58CP:Spasticonly

119


survey,the

Netherlands(.

90%of

1977–1988


up)

13%

haddislocated

hips


included

inTable3

6–19

y8%

hadfixedscoliosis;6%

hadfixedkyphosis

Hipandspinedeform

ities

wereall(100%

)seen

inchildrenwith

bilateralspasticity

Excluded:Sam

ple,80%ofpopulationOR

notallsubtypes

ORduplicatedataANDdatadidnotfurther

contributetoclinicalmessages.Behavior:Carlssonetal(2008)

83;Kennesetal(2002)

29;Bladder

andbowelcontrol:Brossetal(2007)

84;Karam

anetal

(2005)

85;Ozturketal(2006)

86;Parkesetal(2010)

87;Veugelersetal(2010)

88;Eating:Andersen

etal(2008)

89;Himmelmannetal(2007)

90;Parkesetal(2010)

87;Sullivan

etal(2000)

34;Epilepsy:Andersen

etal(2008)


64;Himmelmann

etal(2009)

50;Hum

phreys

etal(2007)

91;Parkesetal(2008)

22(duplicatedatawith

Arnaud

etal

28);Parkes

etal(2010)

87;Sigurdardottir

etal(2010)

31;W

anigasingheetal(2010)

92;Hearing:Andersenetal(2008)


50;Kennesetal

(2002)

29;Parkesetal(2008)


Arnaud

etal28);Parkes

etal(2010)

87Sigurdardottiretal(2010)

31;Hipsandspine:Robinetal(2008)

93;Scruttonetal(2001)

94;Intelligence:Andersenetal(2008)


90;Himmelmann

etal(2009)

50;Parkesetal(2008)


Arnaud

etal28);Parkes

etal(2010)

87;Sigurdardottir

etal(2010)

31;PAIN:Parkinsonetal(2010)


Arnaud

etal

28);Sleeping:none;Talking:Andersen

etal(2008)

89;Kennesetal

(2002)

29;Parkesetal(2008)


Arnaud

etal28);Walking:Andersenetal(2008)

89;Beainoetal(2010)

96;Benedictetal(2011)97 ;Gorter

etal(2004)

98(duplicatecaseswith

Rosenbaumetal[2002]

1 );Hanna

etal(2009)

99;Himmelmannetal

(2007)



50;Kennesetal(2002)

29;M

cCormicketal(2007)

100 ;Palisanoetal(2010)

101(duplicatecaseswith

Rosenbaumetal[2002]

1 );Parkesetal(2010)

87;Riceetal(2009)

102 ;Shevelletal(2009)12

(duplicatecaseswith

Shevelletal[2009]41 );Sigurdardottir

etal(2010)

31;Surman

etal(2003)

57(datacouldnotbeseparated);W

anigasingheetal(2010)

92;Vision:Andersen

etal(2008)


50;Kennesetal(2002)

29;M

cClelland

etal

(2006)

103 ;Parkes

etal(2008)


Arnaud

etal

28);Parkes

etal(2010)

87;Pennefather

andTin(2000)

104 ;Sigurdardottiretal(2010).31

CP,cerebralpalsy;OR,odds

ratio,RR,relativerisk;SCPE,SurveillanceofCerebralPalsyinEurope;UKCP,

UnitedKingdomCerebralPalsyregister.ACPR,AustralianCerebralPalsyRegister;ID,intellectualdisability;ITB,IntrathecalBaclofen;NA,notapplicable;SDR

,SelectiveDorsalRhizotom

y;WA,Western

Australia.

aDidnotmeetcriteriaforclassificationas

alevels1through4study,thereforecodedas

level5

evidence.

REVIEW ARTICLE


impairments, diseases, and functionallimitations with cerebral palsy, wefound that large volumes of either high-quality or moderate-quality evidenceexisted to provide guidance to parentsabout “how bad is it?” Included preva-lence studies were nearly all rated aslevel 1 evidence on the Oxford Scalebecause they were cerebral palsypopulation registry studies. Further-more, the methodologic quality of al-most all studies was very high with lowlevels of bias, providing high degree ofcertainty about the plain English mes-sages that were developed from themeta-analysis. The exception were dataregarding behavior, bladder control,dribbling, pain, and sleep, in whichmore research, including that ofa higher quality, was needed.

From the evidence appraised, we foundthat among children who have cerebralpalsy: 3 in 4were inpain; 1 in 2hadan ID;1 in 3 could not walk; 1 in 3 had a hipdisplacement; 1 in 4 could not talk; 1 in 4had epilepsy; 1 in 4 had a behaviordisorder; 1 in 4 had bladder controlproblems; 1 in 5 had a sleep disorder; 1in 5 dribbled; 1 in 10 were blind; 1 in 15were tube-fed; and 1 in 25 were deaf.Rates of co-occurring impairments,

diseases, and functional limitationswere strongly linked to the severity ofthe motor impairment, with the ex-ception of pain and behavior disorders.Pain was very likely to be present atall levels of physical disability. In-terestingly, behavior disorders weremore common in children with milderlevels of physical disability. In general,findings could potentially be explainedto parents by saying, “All children withcerebral palsy will have physical chal-lenges. The bigger the child’s brain in-jury, the more likely the child is to haveother co-occurring impairments, dis-eases, and functional limitations ac-companying the physical disability,except for pain and behavior, which arecommon regardless of the level ofphysical disability.” If these objectiveand simple messages are given toparents, this information may help al-leviate the stress parents experiencewhile trying to envisage the future atthe time of diagnosis4 and in planningrealistic and achievable services.3

The clinical messages we developedare consistent with cerebral palsymessages published elsewhere in theliterature based on smaller, single-country, nonaggregated samples; forexample, the Swedish register,23 theDanish register,2 and the Canadianregister.12 However, to the best of ourknowledge, this is the first review toaggregate data from all the publishedinternational registries on a multitudeof clinical problems associated withcerebral palsy to produce a suite ofparent-friendly clinical messages.

Parents generally maintain a re-markably optimistic view of the futuredespite receiving bad news about theirchild’s prognosis.24 Research suggeststhat parental recall about diagnosticinformation shared does not matchwhat professionals claim to have com-municated. Parents are thought to activelyblock the recall of bad news as a copingstrategy. It is therefore paramount that

professionals understand that whencommunicating bad news with fami-lies, “truth disclosure is a process, notan event.”25 The literature providesguidance about how professionalsmight communicate these cerebralpalsy prognostic clinical messages.Parents recommend and prefer thefollowing communication strategieswhen receiving bad news: (1) use of anhonest, upfront, specific, and trans-parent communication style to enableparents to seek their right supportsafter receiving the news; (2) discus-sion about the child’s strengths toconvey hope and reframe the future;(3) person-centered and respectfultreatment of the child, so that thenews feels personalized; (4) provisionof a list of frequently asked questionsto help parents prepare their ownquestions; (5) delivery of news to bothparents simultaneously, or to oneparent accompanied by a supportperson, to assist with information re-call; (6) provision of a follow-up in-terview to assist with informationrecall, understanding, and accep-tance; (7) provision of key informationin writing; and (8) involvement of anadvocate/keyworker/case managerto help with planning after the receiptof news.5,24,26,27 In light of this parentalcounsel about preference fora strengths-based approach, pro-fessionals could positively reinforcethat the level of physical disabilityassociated with cerebral palsy doesnot predetermine a child’s “happi-ness” and quality of life (high-qualityGRADE).28,29

Study Limitations

Our review was limited to evaluation ofrates of co-occurring impairments,diseases, and functional limitations instudies published in English. Never-theless, in most topic areas, pub-lications were sourced from a widerange of developed countries acrossEurope, the United Kingdom, North

FIGURE 1Flow diagram of eligible studies.


TABLE2

MethodologicQuality

ofPrevalence

StudiesRetrieved

Study

Was

theTargetPopulation

Definedby

ClearInclusion

andExclusionCriteria?

Was

Probability

SamplingUsed

toIdentifyPotential

Respondents(orthe

WholePopulation

Approached)?

DidCharacteristicsof

RespondentsMatch

theTargetPopulation,

(ie,Was

theResponse

Rate$80%or

AppropriateAnalysis

Included

Comparing

Responders

and

Non-Responders?

WereData

Collection

Methods

Standardized?

Were

Measures

Used

Valid?

Were

Measures

Used

Reliable?

WereFeatures

ofSampling

Design

Accountedforinthe

Analysis,Through

Appropriate

WeightingoftheData,orthe

WholePopulationApproached?

TotalScore

Outof7

Andersen

etal(2010)

63Y

YY

YP

PY

6Andersen

etal(2008)

89Y

YY

YP

PY

6Arnaud

etal(2008)

28Y

PN

PP

PP

3.5

AustralianCP

Register

Group(2009)

37Y

YY

PP

PY

5.5

Bairdetal(2000)

4Y

NN

YN

NN

2Beckungetal(2008)

68Y

YY

PP

PY

5.5

Carlsson

etal(2003)

75Y

YY

PP

PY

5.5

Dayetal(2007)

69Y

NY

PY

YN

4.5

Dolketal(2006)

47Y

YY

PY

YY

6.5

Dolketal(2010)

48Y

YP

PP

PY

5El-Tallawyetal(2011)

49Y

YP

YY

YY

6.5

Hagglund

etal(2005)

77Y

YY

YY

YY

7Hagglund

etal(2005)

78Y

YY

YY

YY

7Hagglund

etal(2007)

79Y

YY

YY

YY

7Hannaetal(2008)

70Y

NN

PY

YN

3.5

Himmelmannetal(2006)

38Y

YY

PP

PY

5.5

Howardetal(2005)

71Y

YY

PP

PY

5.5

Jarvisetal(2005)

51Y

YY

PP

PY

5.5

McM

anus

etal(2006)

52Y

YP

PP

PY

5Monganetal(2006)

39Y

YY

PP

PY

5.5

Newman

etal(2006)

61Y

NN

PY

YN

3.5

Nordmarketal(2001)

53Y

YY

PP

PY

5.5

Parkes

etal(2001)

40Y

YY

PN

NY

4.5

Parkes

etal(2010)

35Y

YY

PP

PY

5.5

Parkes

etal(2008)

22Y

YY

PY

YY

6.5

Persson-Bunkeetal(2006)

81Y

YY

YY

YY

7Rankinetal(2009)

54Y

YY

PN

NY

4.5

Ravn

etal(2010)

55Y

YY

PP

PY

5.5

Roijenetal(2001)

32Y

NY

PN

NN

2.5

Rosenbaumetal(2002)

1Y

NN

PY

YN

3.5

Saunders

etal(2010)

62Y

NN

PP

PN

2.5

Shevelletal(2009a)41

YY

YP

PP

Y5.5

Sigurdardottiretal(2008)

42Y

YY

YY

YY

7Sigurdardottiretal(2009)

44Y

YY

YP

PY

6Sigurdardottir&Vik(2011)

43Y

YY

YP

PY

6Sooetal(2006)

82Y

YY

YY

YY

7Straussetal(2004)

66Y

NY

PY

YN

4.5

Surm

anetal(2006)

36Y

YP

PN

NY

4

REVIEW ARTICLE


America, and Australia using cerebralpalsy register population data. How-ever, accurate cerebral palsy pop-ulation data from developing countriesare difficult to ascertain, and the issuesmaynotbecomparable. In thecontextofdeveloping countries, public healthstrategies such as rubella vaccines andrhesis management are often unavail-able, thus changing the profile of ce-rebral palsy. Publication bias is anotherpotential study limitation. Studiesmay have evaluated the interaction ofimpairments and prognostic outcomesbut published only those that werestatistically significant. For vision andhearing impairments, there was a lackof consistency in codingof impairmentsbetween authors and registries, assome authors used the term “someimpairment.” To improve the rigor ofthe estimates calculated in the meta-analysis, only severe hearing and se-vere vision impairments were includedbecause these impairments are for-mally tested. Rates were calculatedwith a CI, and the body of supportingevidence was graded as moderate tohigh, indicating that more research islikely to change our understanding andthe precision of these estimates.

Implications for Research

There is a vital need for populationstudies that more precisely investigatethe rates of behavioral problems,bladder control problems, dribbling,feeding problems, pain, and sleepdisorders in cerebral palsy. These 6problem areas have not received suf-ficient attention in the literature,and more data of a higher quality arelikely to change and enhance our un-derstanding of these problems. More-over, more data would inform thedesign of future intervention studies,which may create the possibility forimproved outcomes. The findings ofthis study suggest an underresearchedlink between chronic pain, sleep dis-orders, and behavioral problems. TheTA

BLE2

Continued

Study

Was

theTargetPopulation

Definedby

ClearInclusion

andExclusionCriteria?

Was

Probability

SamplingUsed

toIdentifyPotential

Respondents(orthe

WholePopulation

Approached)?

DidCharacteristicsof

RespondentsMatch

theTargetPopulation,

(ie,Was

theResponse

Rate$80%or

AppropriateAnalysis

Included

Comparing

Responders

and

Non-Responders?

WereData

Collection

Methods

Standardized?

Were

Measures

Used

Valid?

Were

Measures

Used

Reliable?

WereFeatures

ofSampling

Design

Accountedforinthe

Analysis,Through

Appropriate

WeightingoftheData,orthe

WholePopulationApproached?

TotalScore

Outof7

Surm

anetal(2009)

45Y

YP

PN

NY

4Surm

anetal(2003)

57Y

YY

PN

NY

4.5

SCPE

(2000)

46Y

YY

PN

NY

4.5

SCPE

(2002)

56Y

YP

PP

PY

5Westbom

etal(2007)

72Y

YY

PY

YY

6.5

Wichers

etal(2009)

67Y

YY

YP

PY

6Wichers

etal(2005)

58Y

YY

PN

NY

4.5

Wuetal(2004)

74Y

NY

PY

YN

4.5

N,No;P,Partially;Y,Yes.A

scoreof1was

givenfor“yes”;ascoreof0.5was

givenfor“partially”;andascoreof0was

givenfor“no.”Prognosticstudieswerenotratedformethodologicquality

onthisscale.


TABLE3

Clinical

Messages

Problem

HowCommon

IsThisProblem?

Who

Isat

Risk?

Long-term

Implications?

ClinicalRecommendations

Behavior

1in4childrenwith

cerebralpalsyhaveabehavior

disorder

(moderate-quality

GRADE)

Childrenwith

cerebralpalsyandan

IDaremore

likelyto

have

behavioralproblems(high-

quality

GRADE)

Unknow

nThorough

assessmentofb

ehavioris

recommended.Also

apain

assessmentisessentialinthe

presence

ofbehavioralproblems,

even

forchildrenwith

mild

physical

impairments.

Therateofabnorm

albehavior

inchildrenwith

cerebralpalsyis2to4tim

eshigher

than

the

population(m

oderate-quality

GRADE)

Childrenwith

cerebralpalsyandepilepsyare

morelikelytohavebehavioralproblems;these

childrenarealso

morelikelytohave

anintellectualimpairment(moderate-quality

GRADE)

Paincontrolm

ayremediateor

minimize

thebehavioralproblem.

Childrenwith

cerebralpalsyandseverepainare

morelikelytohave

behavioralproblems

(high-quality

GRADE)

Standard

psychometricIQassessmentis

also

recommendedinthepresence

ofbehavioralproblemstoenablethe

family

tounderstand

theprognosisof

thebehavioralproblem.

Childrenwith

cerebralpalsyandmilder

physical

disabilityaremorelikelytohave

behavioral

problemsthan

childrenwith

severe

physical

disability(high-quality

GRADE)

Bladderandbowel

control

1in4childrenwith

cerebralpalsydo

noth

ave

bladdercontrol(moderate-quality

GRADE)

Therisk

ofbladderandbowelcontrolproblem

sincreaseswith

severityofphysicaldisability

(moderate-quality

GRADE)

Unknow

nMedicalinvestigations

arewarranted

asabnorm

alanatom

icfindings

are

common

Therateofbladdercontrolproblem

sinchildren

with

cerebralpalsy,4yearsoldis2to

3tim

eshigher

than

thepopulation(low

-quality

GRADE)

Childrenwith

cerebralpalsywho

areunableto

walkor

havean

IDaremostatriskforbladder

andbowelcontrolproblem

s(m

oderate-

quality

GRADE)

Childrenwith

cerebralpalsyshouldbe

offeredstandard

toilettrainingbut

over

alonger

period

oftim

e

1in3to4childrenwith

cerebralpalsyhave

constipation(low

-qualityGR

ADE)

Prescriptionofincontinenceaidesw

illbe

required

for1

in3-4andthiswillbe

for

longer

periodsoftim

ethatchildren

withoutp

hysicaldisabilities

Dribbling

1in5childrenwith

cerebralpalsydribble

(moderate-quality

GRADE)

Childrenwith

severephysicaldisabilityaremore

likelytodribble(m

oderate-quality

GRADE)

Unknow

nSocialstigmaisamajor

problemarising

from

dribblingandeffective

treatm

entssuch

asBotulinum

toxinA

orsurgicalinterventions

shouldbe

explored.

Eating

1in15

childrenwith

cerebralpalsyaretube-fed

(moderate-quality

GRADE)

Childrenwith

ahistoryofpoor

suckingduring

infancyaremorelikelytohave

feeding

problems(m

oderate-quality

GRADE)

Eatingskillsremainstableinadulthood

(high-quality

GRADE)

Infantswith

cerebralpalsyandpoor

suckingshouldhave

theireating

comprehensivelymonitored.

Childrenwith

cerebralpalsyare3tim

esmore

likelytohave

feedingproblemsat6monthsof

age(m

oderate-quality

GRADE)

Childrenwith


likelytoneed

someone

tofeed

them

(moderate-quality

GRADE)andaremorelikely

toneed

tube

feeding(m

oderate-quality

GRADE).

Swallowingsafetyshouldbe

comprehensivelyassessed

ifconcerns

arereported.

Childrenwho

arenonverbalare

morelikelyto

have

difficulty

feeding(high-quality

GRADE)

Weightshouldalso

bemeasured

regularlyas

thosewith

moresevere

physicaldisabilityh

avehigherrisk

for

malnutrition.

REVIEW ARTICLE


TABLE3

Continued

Problem

HowCommon

IsThisProblem?

Who

Isat

Risk?

Long-term

Implications?


Epilepsy

1in4childrenwith

cerebralpalsyhave

active

epilepsy(high-quality

GRADE)

Theriskofepilepsyw

ithcerebralpalsyincreases

with

severityofphysicaldisability(high-

quality

GRADE)

Adultswith

cerebralpalsyandepilepsy

areless

likelytowork(high-quality

GRADE)

Anti-epilepticmedications

areusually

effectiveformanagingseizures

and

areconsidered

standard

practicefor

managingepilepsyinchildrenwith

cerebralpalsy

1in3childrenwith

cerebralpalsyhave

had

epilepsyatsometim

e(high-quality

GRADE)

Childrenwith

bothsidesofthebody

affected

are

morelikelytohave


GRADE)

Childrenwith

cerebralpalsyareless

likelyto

becomeseizure-free

(low

-quality

GRADE)

Childrenwith

cerebralpalsyandan

IDaremore

likelytohave


GRADE)

Hearing

1in25

childrenwith

cerebralpalsyhave

severe

hearingimpairmentoraredeaf(high-quality

GRADE)

Childrenwith

moreseverephysicaldisabilityare

morelikelytohave

ahearingimpairment

(moderate-quality

GRADE)

Unknow

nEarlyscreening,assessment,and

accommodationforhearing

impairmentisrecommended

Hips

andspine

1in3childrenwith

cerebralpalsyhave

hip

displacement(high-qualityGR

ADE)

Childrenwith

bothsidesofthebody

affected

and

who

cannotwalkareatthegreatestrisk

ofhip

problems(high-quality

GRADE)

andscoliosis

(low

-qualityGR

ADE)

Long-term

activehipsurveillance

reducesthelikelihoodofprogression

from

hipdisplacementtohip

dislocation(m

oderate-quality

GRADE)

6-to12-month

hipsurveillanceis

recommendedandiseffectivefor

ensuring

access

toearlytreatm

ent.

Radiograph

andclinicalassessment

shouldcommence

very

early.For

thosewho

receivehipsurveillancethe

rateofsalvageorthopedicsurgeryis

lower

1in10

childrenwith

cerebralpalsyhave

hip

dislocationwithouth

ipsurveillance(high-

quality

GRADE)

Therisk

ofhipabnorm

alities

with

cerebralpalsy

increaseswith


(high-quality

GRADE)

Therisk

ofassociated

spinaldeform

ityincreaseswith


(low

-qualityGR

ADE)

Intellect

1in2childrenwith

cerebralpalsyhave

anID

(moderate-quality

GRADE)

Childrenwith

moreseverephysicaldisabilityare

morelikelytohavean

intellectualimpairment

(moderate-quality

GRADE)

Unknow

nForm

alassessmentand

diagnosisofan

IDisan

importantp

rognostic

indicatorforwalking,bladdercontrol,

schoolperformance,and

likelihoodof

independentliving

1in4childrenwith

cerebralpalsyhave

asevere

ID(m

oderate-quality

GRADE)

Childrenwith

dyskineticcerebralpalsywho

have

anIDaremorelikelytohave

asevere

IDthan

thosewith

spastic

cerebralpalsy(m

oderate-

quality

GRADE)

Ifmultipleimpairmentsexist,

psychometricscreeningof

intelligenceishighlyrecommended

forinterventionandschoolplanning

Pain

3in4childrenwith

cerebralpalsyareinpain

(moderate-quality

GRADE)

Childrenandadultswith

cerebralpalsy

regardless

oflevelofdisabilityareatrisk

for

pain(high-quality

GRADE)

Painislinkedtohigher

ratesof

behavioralproblemsandlower

participation(high-quality

GRADE)

Parentsandchildrenreportlevelsof

paindifferentlyandthereforethe

child’sperceptions

shouldalwaysbe

sought

Forthosewho

canwalk,neck,back,andfeetare

high-riskpainsites(low

-qualityGR

ADE)

Painincreaseswith

age(m

oderate-

quality

GRADE)

Investigateawiderangeofpainorigins

(eg,dental,gastrointestinal,

muscular,neuropathic,

rheumatology,skeletal,tonal)

Childrenandadultswith

contractureareat

higher

risk

ofdeveloping

pain(m

oderate-

quality

GRADE)

Comprehensive

painmanagem

ent

shouldbe

instigated

tominimizethe

likelihoodofsecondarybehavioral

problemsfrom

developing


TABLE3

Continued

Problem

HowCommon

IsThisProblem?

Who

Isat

Risk?

Long-term

Implications?


Sleeping

1in5childrenwith

cerebralpalsyhave

asleep

disorder

(low

-qualityGR

ADE)

Childrenwith

cerebralpalsyandactiveepilepsy

aremostatriskforsleepdisorders(low

-quality

GRADE)

Unknow

nThroughandspecialistassessmentof

sleepproblemsarerecommended

Therateofsleepdisordersinchildrenwith

cerebralpalsyis5tim

eshigher

than

the

population(low

-qualityGR

ADE)

Childrenwith

spastic

quadriplegiaor

dyskinesia

orasevere

visualimpairmentare

morelikely

tohave

difficulty

initiatingandmaintaining

sleep(low

-qualityGR

ADE)

Earlytreatm

entofsleep

problems(both

medicalandbehavioral)isadvisable

before

secondaryacadem

icand

behavioralproblemsem

erge

orare

established

Talking

1in4childrenwith

cerebralpalsycannottalk

(high-quality

GRADE)

Childrenwith

moresevere

physicaldisability/

nonambulatory

aremorelikelytohave

aspeech

impairment(high-qualityGR

ADE)

Unknow

nEarlyassessm

entand

recommendations

ofaugm

entativeandalternative

communicationoptions

forspeech

impairmentisrecommended

1in3childrenwith

cerebralpalsyhave

some

speech

impairment(high-quality

GRADE)

Childrenwith

dyskinesiaaremorelikelytohave

speech

problems(high-quality

GRADE)

Walking

1in3childrenwith

cerebralpalsycannotwalk

(high-quality

GRADE)

Childrenwith

cerebralpalsywho

have

4lim

bsaffected

and/or

IDand/or

epilepsyand/or

avision

impairmenthaveahigherriskofbeing

unabletowalk(high-quality

GRADE)

Childrenwho

walkusingaids

orcannot

walklose

walking

functionduring

adolescence(m

oderate-quality

GRADE)

Childrenwho

walkusingaidesandtheir

families

shouldbe

emotionally

prepared

forpotentiallossofmotor

functioninadolescence

1in6childrenwith

cerebralpalsywalkusing

aides(high-quality

GRADE)

Atage2years,childrenwith

cerebralpalsywho

areunabletoroll,sit,or

pulltostandhave

averyhigh

risk

ofbeingunabletowalk(high-

quality

GRADE)

Abilitytowalkfurtherdeclines

during

lateradulthood.(high-quality

GRADE)

Childrenwho

walkusingaidesrequire

mobilityassessmentsatthe

commencementofadolescenceto

enableprescriptionofappropriate

mobilitydevicestoaccommodate

decliningmotor

function

1in2childrenwith

cerebralpalsywalk

independently

(high-quality

GRADE)

Achild’swalking

abilityatage12

yearsis

predictiveoftheirwalking

abilityas

anadult.

(high-quality

GRADE)

Vision

1in10

childrenwith

cerebralpalsyhaveasevere

visualimpairmentorareblind(m

oderate-

quality

GRADE)

Childrenwith


likelytohaveavisualimpairment(high-quality

GRADE).Amongthosewith

severe

physical

disability,severe

visualimpairmentoccurs

morefrequentlywith

spasticity

than

dyskinesia(high-quality

GRADE)

Unknow

nEarlyscreening,assessment,and

treatm

entofvisionimpairmentis

recommended

1in4childrenwith

cerebralpalsyhave

avision

impairment(moderate-quality

GRADE)

Childrenborn

prem

aturelywith

cerebralpalsy

aremorelikelythan

childrenwithoutcerebral

palsytohavevisualimpairments(high-quality

GRADE)

GRADEsystem

(Guyattetal[2008])21was

asfollows:high-quality:furtherresearch

isveryunlikelytochange

ourconfidenceintheestim

ateofeffect;m

oderate-quality:furtherresearch

islikelytohavean

importantimpacton


ate

ofeffectandmay

change

theestim

ate;low-quality:furtherresearch

isverylikelytohave

animportantimpacton


ateofeffectandislikelytochange

theestim

ate;andvery-low-quality:anyestim

ateofeffectisveryuncertain.

REVIEW ARTICLE


relationship between pain, sleep, andbehavior warrants urgent attention, aseffective evidence-based interventionstrategies exist in other diagnosticgroups that may offer great promise toindividuals with cerebral palsy.

Future research and cerebral palsyregister data sets collecting informa-tion on vision and hearing impairmentsassociated with cerebral palsy shoulduse agreed data dictionary terminologyto code impairments and therefore

improve data quality and ease of in-terpretation.

CONCLUSIONS

There is high-quality grade evidencethat among children with cerebralpalsy: 1 in 3 cannot walk; 1 in 4 cannottalk; 1 in 4 hadepilepsy; and 1 in 25weredeaf. There is moderate-quality evi-dence that 3 in 4 were in pain; 1 in 2 hadan ID; 1 in3hadahipdisplacement; 1 in4had a behavior disorder; 1 in 4 had

bladder control problems; 1 in 5 drib-bled; 1 in 10 were blind; 1 in 15 weretube-fed. There is low-quality evidencethat 1 in 5hada sleepdisorder. Childrenand adults unable to walk are morelikely to experience these accompany-ing impairments. The risk for pain andbehavioral problems occurs equally atall levels of physical disability. There isinsufficientevidence tobecertainaboutthe rates of sleep disorders, and moreresearch is warranted.

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Clinical Prognostic Messages From a Systematic Review on … · Clinical Prognostic Messages From a Systematic Review on Cerebral Palsy abstract OBJECTIVE: To summarize evidence on