Children With Cerebral Palsy: Racial Disparities in Functional Limitations

Author(s): Matthew J. Maenner, Ruth E. Benedict, Carrie L. Arneson, Marshalyn

Yeargin-Allsopp, Martha S. Wingate, Russell S. Kirby, Kim Van Naarden Braun and
Maureen S. Durkin
Source: Epidemiology , January 2012, Vol. 23, No. 1 (January 2012), pp. 35-43
Published by: Lippincott Williams & Wilkins

Stable URL: https://www.jstor.org/stable/23214174

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 Original Article

Children With Cerebral Palsy

Racial Disparities in Functional Limitations
Matthew J, Maenner,a'h Ruth E. Benedict,a'c Carrie L. Arneson,a Marshalyn Yeargin-Allso
Martha S. Wingate,e Russell S. Kirby,f Kim Van Naarden Braun,d and Maureen S. Durkina

Conclusion: Black children in the United States appear to have a

Background: Previous studies of the frequency of cerebral palsy in
higher prevalence of cerebral palsy overall than white children,
the United States have found excess prevalence in black children
although the excess prevalence of cerebral palsy in black children is
relative to other groups. Whether the severity of cerebral palsy
seen only among those with the most severe limitations. Further
differs between black and white children has not previously been
research is needed to explore reasons for this disparity in functional
investigated.
limitations; potential mechanisms include racial differences in risk
Methods: A population-based surveillance system in 4 regions of
factors, access to interventions, and under-identification of mild
the United States identified 476 children with cerebral palsy among
cerebral palsy in black children.
142,338 8-year-old children in 2006. Motor function was rated by
the Gross Motor Function Classification System and grouped into 3 (Epidemiology 2012;23: 35-43)
categories of severity. We used multiple imputation to account for
missing information on motor function and calculated the race
specific prevalence of each cerebral palsy severity level.
Results: The prevalence of cerebral palsy was 3.7 per 1000 black
children and 3.2 per 1000 white children (prevalence odds ratio
[OR] = 1.2 [95% confidence interval = 1.0-1.4]). When stratified Cerebral
posturepalsy is atolifelong
attributed disorder
nonprogressive of movement
abnormalities in the and
by severity of functional limitation, the racial disparity was present developing brain.1 General population estimates of the prev
only for severe cerebral palsy (black vs. white prevalence OR = 1.7 alence in childhood range from approximately 1 to >4 per
[1.1-2.4]). The excess prevalence of severe cerebral palsy in black
1000 children or live births.2'3 The risk is greatly elevated in
children was evident in term and very preterm birth strata.
children with a history of perinatal complications such as
preterm birth and low birth weight, although these and other
perinatal factors may be in the pathway between antecedent
Submitted 19 February 2011; accepted 9 August 2011; posted 11 November
2011. causes and cerebral palsy rather than root causes themselves.4
From the "Waisman Center, University of Wisconsin-Madison, Madison, Furthermore, many children with cerebral palsy do not have a
WI; "Department of Population Health Sciences, University of Wiscon
history of known perinatal complications.5 Studies in the United
sin School of Medicine and Public Health, Madison, WI; 'Department of
Kinesiology, Organizational Therapy Program, University of Wisconsin States have found the prevalence of cerebral palsy to be
Madison, Madison, WI; dDivision of Birth Defects and Developmental somewhat higher in non-Hispanic black than non-Hispanic
Disabilities, National Center on Birth Defects and Developmental Dis
white children.3'6-9 The reasons for this racial disparity
abilities, Centers for Disease Control and Prevention, Atlanta, GA;
"Department of Health Care Organization and Policy, School of Public
remain unclear.
Health, University of Alabama at Birmingham, Birmingham, AL; De Cerebral palsy is highly complex and heterogeneous in
partment of Community and Family Health, College of Public Health,
its etiology, clinical manifestations, and co-occurrence with
University of South Florida, Tampa, FL; and department of Pediatrics,
University of Wisconsin School of Medicine and Public Health, Madi other developmental disabilities. In addition, the degree of
son, WI. motor impairment can vary widely; the most severe cerebral
Supported by the Centers for Disease Control and Prevention through
Cooperative Agreements UR3/DD000677 and UR3/ DD000078 as part
palsy results in the inability to walk or perform basic daily
of the Autism and Developmental Disabilities Monitoring Network. tasks, such as eating, whereas the mildest forms might be
Additional funding for graduate student support for data analysis was described as "clumsiness."10 The severity of functional lim
provided by the University of Wisconsin-Madison. M J.M. was partially
itation, in turn, is associated with increased use of services
supported by a grant from the Autism Science Foundation. The authors
reported no other financial interests related to this research. and difficulty in accessing care.11 Despite the variability in
|sdc| Supplemental digital content is available through direct URL citations functional limitation with cerebral palsy and the importance
in the HTML and PDF versions of this article (www.epidem.com).
Correspondence: Matthew J. Maenner, Waisman Center, 1500 Highland of severity in determining the need for services, few US
Ave, Rm 529A, Madison, WI 53705. E-mail: mjmaenner@wisc.edu. studies differentiate severity of function when evaluating this
condition. Although the racial disparity in prevalence has been
Copyright © 2011 by Lippincott Williams & Wilkins
ISSN: 1044-3983/12/2301-0035 found in numerous studies,3'6""9 we know of no studies of racial
DOI: 10.1097/EDE.0b013e31823a4205 disparities in the functional severity of cerebral palsy.

Epidemiology • Volume 23, Number 1, January 2012 www.epidem.com | 35

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 Maenner et al Epidemiology • Volume 23, Number 1, january 2012

We sought to determine whether theclassified

excess at prevalence
levels I or II are able to walk without assistive
of cerebral palsy among US non-Hispanic devices black
(those at children
level II have greater difficulty walking on
persists across all levels of functional limitations,
uneven terrain and after ac
are unable to run or jump). Children at
counting for missing data and considering risk
level factors
III may require such as
trunk support while sitting and are able
birth weight and gestational age. We used to several
walk with approaches
an assistive mobility device. A classification of
for handling missing data to assess the potential
level for bias
IV or V indicates very limited self-mobility with a
created by missing functional information
poweron estimates
wheelchair, of
and children at level V are often unable to
prevalence by severity. maintain antigravity head and trunk postures.13 Although this
system was originally established in clinical settings to iden
METHODS tify appropriate treatments and predict gross motor develop
ment over time,14 the tool has also been found to be a reliable
Study Sample and Design measure in population-based studies.10'15
Children with cerebral palsy were identified through Information
the used to classify gross motor function in
Centers for Disease Control and Prevention Autism and cluded descriptions of functional skills, posture, balance,
Developmental Disabilities Monitoring Cerebral Palsy Net
ambulation, and other forms of mobility. These methods have
work, which conducted surveillance of all 8-year-old children
been described in greater detail elsewhere, and a study dem
with cerebral palsy in 4 geographically defined regions in onstrating the interrater reliability of this approach has been
2006 (birth year 1998). The 4 surveillance regions were
recently published.8'15 Clinician reviewers classified gross
Alabama (32 northern counties, with Birmingham at the motor function into 3 groups as follows. Children classified
southern end of the surveillance region), Georgia (5 countiesas levels I and II were considered able to "walk indepen
in metropolitan Atlanta), Missouri (5 counties in the St. Louis
dently." Those classified at level III were considered to "walk
metropolitan area, including St. Louis), and Wisconsin (10with handheld mobility device," and those classified at levels
counties in southeastern Wisconsin, including Madison and IV or V were considered to have "limited or no walking
Milwaukee). The 2006 NCHS postcensal estimate for the ability." Children who lacked enough information for the
combined population of 8-year-old children across the 5-level
4 classification were classified for walking ability, if
study areas was 142,338. possible. Classifications were based on descriptions of am
bulation and motor function from ages 4 through 8 years
Ascertainment of Children With Cerebral Palsy(functional development trajectories become fairly stable by
and Measures of Motor Limitations age 4).16'17 Reviewers were able to classify gross motor function
For surveillance year 2006, the Network identified for
476 354 (74%) of the 476 case children. The remaining 122
8-year-old children with cerebral palsy. Of these children,
children lacked sufficient information for classification.
97% had documentation of a previous diagnosis of cerebral The overall prevalence of cerebral palsy in this popu
palsy.8 Children suspected of having cerebral palsy were
lation was 3.3 per 1000 8-year-old children (95% confidence
identified by screening medical records at multiple health interval [CI] = 3.1-3.7), with a prevalence of 3.7 (3.2-4.4) in
care settings, including hospitals, clinics, diagnostic centers,
non-Hispanic black and 3.2 (2.8-3.6) in non-Hispanic white
health care providers, and state public health and rehabilita
children.8 Table 1 shows the distribution of cases and cate
tion agencies. In Georgia, potential cases also were identified
gorization of motor function by sex, cerebral palsy subtype (4
through public-school special-education programs. Case categories
sta based on clinical manifestation), co-occurring ep
tus was determined by clinician reviewers based on diagnos
ilepsy, and race/ethnicity.
tic information and physical findings in a child's records.
Imputation and Analysis
These physical findings included impairments in muscle tone,
reflexes, and abnormal movement patterns at age 2 or older. Information on gross motor functioning was missing
Children with physical findings that could be explainedfor
by 26% of cases (Table 1). A number of variables were
documented genetic disorders or other apparent noncerebral
associated with both severity of gross motor limitations and
palsy etiologies, such as progressive disorders, were ex
whether or not this information was missing (eAppendix,
cluded. Cerebral palsy of postneonatal origin (>28 days) http://links.lww.com/EDE/A524).
was These associations formed
noted when indicated in the records. Detailed methods onthe
thebasis of multiple imputation models, which included
Network methodology have been previously published.3'8
covariates of interest to this analysis as well as other variables
There is no consensus on how to classify functional
previously identified in the literature as being related to
limitations in children with cerebral palsy12; however, functional
the limitations among children with cerebral
Gross Motor Function Classification System is increasingly
palsy.18'19 Our final imputation model included the following
becoming a standard tool. This freely available tool classifies
variables: cerebral palsy subtype (clinical manifestation cat
gross motor abilities and limitations into 5 levels, ranging
egory), age at last known evaluation, number of total evalu
from mildest (level I) to most severe (level V).13 Children
ations available in the record, surveillance site, co-occurring

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Epidemiology • Volume 23, Number 1, January 2012 Racial Disparities in Functional Severity of Cerebral Palsy

epilepsy or intellectual disability, race, sex, and census-

fs «I «a o o o o
measures of income.
o e «a
PS <
Multiple imputation and statistical analysis were per
« »
formed with SAS 9.1.3 (SAS Institute, Cary NC) and the
V <u
*£ OD o o o o SAS-callable implementation of IVEware.20 Additional de
2 <
co tails about the robustness of the imputation model can be
found in the eAppendix (http://links.lww.com/EDE/A524).
r£ Confidence intervals for prevalence estimates and proportions
>. w?»5
•2 CI.-S of cases were calculated following the standard rules for
2a»E
t-- os os in
o o m

-ls«
« w -, S
ce
m «-< o o o o
combining results across multiply imputed data sets. Preva
Ih
^ - - § ^ lence odds ratios (ORs) using imputed data were performed
« o ■*■' >*
>- S u o in SAS via PROC LOGISTIC and PROC MIANALYZE.
« € «
u£l
Subanalysis of Maternal and Birth
t-- as <n
o i- -
Characteristics and Their Relationship With
dod Severity of Functional Limitations
a We assessed the associations of selected maternal and
w
birth characteristics with level of function, using the subset of
os oo o 366 (77%) case children with birth-certificate information. Case
Tf M P CJ 't
© dodo children who were missing birth-certificate information are as
sumed to have been born in another state or country. An
additional imputation was performed using only the cases with
available birth-certificate data. Birth characteristics included in
<N
« d o o o o the model were gestational age, birth weight, plurality, and
» 5-minute Apgar score. We examined whether the distribution of
e
o
Z motor limitations differed by birth characteristics and whether
racial differences persisted across levels of these factors.

Comparison With Other Population Studies

— o o o o

To compare our findings with other published studies,

we searched the literature for all population-based studies of
cerebral palsy that classified functional limitations using the
o o o o
Gross Motor Function Classification System or a comparable
«8 «
Q-S
3-category variable.21-29 We compared the overall prevalence
of cerebral palsy, the proportion of case children at each level
o Os *n vo of motor function, and the proportion of case children for
O CN <N
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the proportion of all cases with known gross motor function
p •t
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d d
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<N m o o

unknown functional limitations as a separate category.

?3 CN OO Tf O
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a o o o o'
J O c c s

Proportion and Prevalence of Cerebral Palsy

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TABLE1.SelctdChareistcandProptinfChldreWitCbralPsy SevritofGsMtorLimans,AutimandDevlopmnta DisabiltesMonitoringNetwork,20 6a u sified as mild or higher percentage classified as severe (Table 2).

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Maenner et al Epidemiology » Volume 23, Number 1, January 2012

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Epidemiology • Volume 23, Number 1, January 2012 Racial Disparities in Functional Severity of Cerebral Palsy

Using the multiple-imputation approach, the total pro
portion of all cases in the mild category increased to 60% and
remained higher in non-Hispanic white (66%) than non
Hispanic black children (53%) (Table 2). There was a corre
sponding decrease after imputation in the proportion of se
vere cases (29% overall, 35% among non-Hispanic black
cases, and 25% among non-Hispanic white cases). Overall,
cases with imputed functional information were more likely
than cases with complete information to be in the mild
category.
After imputation, the overall prevalence of mild, mod
erate, and severe motor limitations was estimated to be 2.0,
0.4, and 1.0 per 1000, respectively (Table 2). The prevalence
of mild cerebral palsy was similar for black and white
children, whereas the prevalence of severe cerebral palsy was
0.9 to 1.8 per 1000, whereas the prevalence of severe cerebral
palsy ranged from 0.5 to 0.7 per 1000 (Table 4). The preva
lence of mild cerebral palsy tended to decrease as the pro
portion with missing functional information increased (Table
4, in which studies are listed in order of increasing percent of
missing functional information).
DISCUSSION
Our study extends previous research by examining
racial differences in the severity of functional limitations
among children with cerebral palsy. Consistent with previous
reports, the overall prevalence of cerebral palsy was modestly
higher in black than white children. Furthermore, black chil
dren with cerebral palsy had more severe functional limita
tions than white children. It is difficult to determine whether

70% higher in black children than white children (OR = 1.7 racial differences in access to care might lead to different
[95% CI = 1.1-2.4]; Table 2). functional outcomes, or whether these results are due to
differential ascertainment of cerebral palsy by racial group.
Associations Between Selected Maternal and Children with cerebral palsy who have mild functional limi
tations are thought to be less likely to be ascertained in
Birth Characteristics and Severity of Functional
Limitations population-based studies, as they likely have fewer health
Table 3 shows the distribution of selected maternal and care needs and less interaction with the services through
birth characteristics among non-Hispanic black and non which cases are typically identified.10'25'30'31
Hispanic white births in the overall surveillance study area inAmong college-educated women, the somewhat ele
1998 and among cases categorized by severity. Although the vated odds of a black mother having a child with mild
relative frequency of cerebral palsy varies widely among cerebral palsy is consistent with more educated black mothers
strata of some factors, the proportion of mild, moderate, and having more resources and greater ability to access services
severe cerebral palsy is fairly consistent across strata. Non than black mothers with less education. It is possible that ou
Hispanic black children displayed similar odds for severe surveillance system may not have captured an excess risk o
limitations in both very preterm (<32 weeks gestation) and mild cerebral palsy among black children. If true, this under
term (>37 weeks gestation) categories (OR = 1.7 in both). ascertainment would be further evidence of disparities in
Among infants born at gestational age 32 to 36 weeks, there access to services, as the surveillance system is based on
were only 7 non-Hispanic black cerebral palsy cases, and a records of children receiving services. Thus, the magnitude of
higher odds for severe limitations was not observed. The the racial disparities in total prevalence could be even greater
gestational age findings were comparable to the findings than what we observed.
across birth-weight categories. The excess risk of severe We found no evidence that the increased severity of
cerebral palsy among black children rose with maternal motor limitations among black children was associated with
education. There was also black excess of mild cerebral palsy selected maternal or birth characteristics. The racial dispari
among the most educated women, although statistical power ties in level of function persisted within both term and very
was limited (OR = 1.4 [95% CI = 0.9-2.2]). preterm births and within normal and very-low-birth-weight
categories, and the overall proportion of case children with
Comparison With Other Population-based severe limitations was generally similar across strata. It was
Studies of Cerebral Palsy beyond the scope of this study to determine causal relation
Compared with other recent population-based studies, ships between perinatal factors and motor outcomes. The
non-Hispanic black and non-Hispanic white children with observed associations likely represent complex causal path
cerebral palsy in this study tended to have similar or higher ways involving unmeasured factors. It is also possible that
prevalence of each category of motor limitation (Table 4). some characteristics, such as low Apgar score, are indicators
Notably, the prevalence of severe cerebral palsy among of an early presence of cerebral palsy. Other factors, such as
non-Hispanic black children in the United States was more disparities in access to care, maternal age, substance abuse, or
than double that in other countries, whereas the prevalence ofother pathologic processes, may contribute to the associations
severe cerebral palsy among non-Hispanic whites was only of preterm delivery or low birth weight with cerebral
slightly higher. palsy.32'33
The prevalence of cerebral palsy with mild functional The literature describing the relationship between birth
limitations was more variable across studies, ranging from weight or prematurity and functional limitations is mixed,

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Maenner et al Epidemiology • Volume 23, Number 1, January 2012

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Epidemiology • Volume 23, Number 1, January 2012 Racial Disparities in Functional Severity of Cerebral Palsy

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Maenner et al Epidemiology « Volume 23, Number 1, jariuary 2012

Strengths
and measures of "severity" of motor limitations and
also Weaknesses
vary
across studies. A study from Northern Ireland22
Thisfound
study that
was the
subject to several limi
proportion of children with cerebral palsy functioning
sample at important
sizes for level subgroups, suc
V (indicating severe motor limitation) wasethnic
other greater among
groups or children with specific
normal birth weight births than those palsy,
with low birth
were too weight.
small for analysis. Another li
Similar findings were noted in 2 studies from the
our measure of United
functional limitation is focu
Kingdom (including Northern Ireland),34,35 although
and gross motor a differ
function and does not encom
ent measure of motor function was used. In contrast, a studypalsy "severity." Other
construct of cerebral
from Northern England36 reported a considered
higher proportion
in this of
study include the natur
severe motor limitations among children with cerebral palsy
neurologic impairment, the presence of co-o
bom at low birth weights than those born at normal birth
ities, fine motor and cognitive skills, man
weights. Norwegian26 and Icelandic29studies found no differ
care, and an individual's participation in a
ences in motor functioning in term versus preterm births. A
activities. Intellectual disability has sometim
study by Pharoah et al37 suggested that the proportion of
part of the definition of "severe cerebral pals
children with severe cerebral palsy was nearly identical in
does not have information on all associated conditions and
low-birth-weight and normal-birth-weight children. The Sur
impairments (such as cognitive impairment) that might con
veillance of Cerebral Palsy in Europe group30 also found that
tribute to functional limitations.
gestational age and birth weight were not strongly associated
A strength of this study is its reliance on population
with walking ability. Although some birth characteristics are
based surveillance methodology, which incorporates infor
clearly related to the overall prevalence of cerebral palsy,
mation from
their association with level of motor function is multiple
unclear. medical and education sources and
does not depend on children receiving services specific to
Imputation and Missing Data their disability. Nearly all (97%) of the cases have a
documented
The results of the imputation analysis largely cerebral palsy diagnosis from a qualified ex
supported
aminer, suggesting
the notion that children with missing functional informationthe surveillance case definition is not
more inclusive
are more likely to have milder limitations. than the actual diagnoses given to children
Under-ascertain
in this population.
ment of cerebral palsy with mild limitations is thoughtAs functional
to be measures (such as the
Gross
more likely than under-ascertainment of Motor Function
children Classification System) become
with severe
more routinely on
limitations when ascertainment is dependent recorded in clinical settings, imputation
service
should cerebral
use.22'25'31 It follows that if children with be less necessary
palsy in who
future surveillance and moni
toring efforts. excluded from
have milder limitations are disproportionately
analysis and reports, the proportion of cases with more severe
limitations will be overrepresented.
Conclusion
Missing information complicates comparisons across
This population-based study observed an excess prev
studies of cerebral palsy severity. Variability in the inclusion
alence of severe cerebral palsy among non-Hispanic black
of children with mild motor limitations in population-based
children when compared with non-Hispanic white children in
registers or surveillance programs has been considered a
the United States, whereas the prevalence of mild cerebral
likely contributor to overall differences in the prevalence of
palsy was similar in the 2 groups. It is not clear whether the
cerebral palsy across systems.30 Because prevalence varies
somewhat among studies, it is moreobserved racial disparity
meaningful is present
to use an only for the severe cate
gory or whether black children with mild motor limitations
absolute measure, such as prevalence, rather than the propor
are less likely
tion of cases in different severity categories, when thancomparing
white children to be ascertained by the
surveillance
severity among studies. As illustrated in Table 4, system.
it is difficult
The
to make meaningful comparisons across populations results of our
by imputed
con analysis suggest that, com
pared
sidering only the proportion of children withwithcerebral
other published
palsystudies,
at the relatively higher
prevalence of cerebral
a given functional level when prevalence and completeness palsy in US-based studies is not due
entirely
fluctuate. As expected, previous studies tendedto a greater inclusion
to report a of milder cases, because in
creased prevalence
lower prevalence of children with cerebral palsy iswho
seen in all 3 severity categories. The
had
presence
mild motor limitations as the proportion of (and
of cases approach
with to) missing information may
missing
functional information increased. Only influence
a how
few information about severity is interpreted in
population
population-based
based studies have directly reported the prevalence studies. Multiple imputation is a feasible
of cere
bral palsy severity using the Gross Motor Function
and useful approach Classifi
and affords meaningful comparisons
cation System or a compatible 3-category ambulation
across studies when the prevalence and completeness of data
classification 26-28 vary.

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Epidemiology • Volume 23, Number "1, January 2012 Racial Disparities in Functional Severity of Cerebral Palsy

ACKNOWLEDGMENTS 18. Shevell MI, Dagenais L, Hall N, REPACQ Consortium. Comorbidities

in cerebral palsy and their relationship to neurologic subtype and
We gratefully acknowledge the project coordinators,
GMFCS level. Neurology. 2009;72:2090-2096.
clinician reviewers, abstractors, and investigators19.who con
Beckung E, Hagberg G, Uldall P, Cans C, for Surveillance of Cerebral
tributed to the surveillance project and data collection. We
Palsy in Europe. Probability of walking in children with cerebral palsy
in Europe. Pediatrics. 2008;121:el87-el92.
also thank anonymous reviewers for their helpful suggestions
20. Raghunathan TE, Solenberger PW, Van Hoewyk J. IVEware: Imputa
to improve this manuscript. tion and Variance Estimation Software. User Guide. Ann Arbor, MI:
University of Michigan; 2002.
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