Cardiology in the Young (2012), 22, 574582 r Cambridge University Press, 2012

doi:10.1017/S1047951112000121

Original Article

Gross motor development is delayed following early

cardiac surgery

Suzanne H. Long,1,2 Susan R. Harris,3 Beverley J. Eldridge,4 Mary P. Galea2

1
Heart Research, Critical Care & Neurosciences, Murdoch Childrens Research Institute; 2Physiotherapy, Melbourne
School of Health Sciences, The University of Melbourne, Melbourne, Australia; 3Department of Physical Therapy,
The University of British Columbia, Vancouver, Canada; 4Physiotherapy Department, The Royal Childrens
Hospital, Melbourne, Australia

Abstract Objective: To describe the gross motor development of infants who had undergone cardiac surgery in
the neonatal or early infant period. Methods: Gross motor performance was assessed when infants were 4, 8, 12,
and 16 months of age with the Alberta Infant Motor Scale. This scale is a discriminative gross motor outcome
measure that may be used to assess infants from birth to independent walking. Infants were videotaped during
the assessment and were later evaluated by a senior paediatric physiotherapist who was blinded to each infants
medical history, including previous clinical assessments. Demographic, diagnostic, surgical, critical care, and
medical variables were considered with respect to gross motor outcomes. Results: A total of 50 infants who
underwent elective or emergency cardiac surgery at less than or up to 8 weeks of age, between July 2006 and
January 2008, were recruited to this study and were assessed at 4 months of age. Approximately, 92%, 84%,
and 94% of study participants returned for assessment at 8, 12, and 16 months of age, respectively. Study
participants had delayed gross motor development across all study time points; 62% of study participants did
not have typical gross motor development during the first year of life. Hospital length of stay was associated
with gross motor outcome across infancy. Conclusion: Active gross motor surveillance of all infants undergoing
early cardiac surgery is recommended. Further studies of larger congenital heart disease samples are required,
as are longitudinal studies that determine the significance of these findings at school age and beyond.

Keywords: Congenital heart disease; Alberta Infant Motor Scale; developmental delay

Received: 19 April 2011; Accepted: 18 December 2011; First published online: 29 February 2012

PATIENT- AND MANAGEMENT-SPECIFIC described how motor skills evolve over time following

V
ARIOUS
factors have the potential to interact and
manifest as a spectrum of developmental
dysfunction in infants with congenital heart disease.1
Prospective studies to date have revealed cognitive
and, more commonly, motor dysfunction during
infancy in a large proportion of children that have
cardiac surgery in the first 6 months of life.2 Although
these studies have provided valuable quantitative
information on motor performance, they have not
Correspondence to: S. H. Long, PhD, Physiotherapy, Melbourne School of
Health Sciences, The University of Melbourne, 1st Floor, 200 Berkeley Street,
Carlton, Victoria 3010, Australia. Tel: 161 3 8344 4171; Fax: 161 3 8344
4188; E-mail: suzanne.long@me.com
early cardiac surgery. Instead, prospective studies have
typically been limited to motor assessments taking
place at only one time-point during infancy. Results
may therefore be distorted by the natural variability
that occurs throughout infant gross motor develop-
ment, that is, differences in gross motor ability are
observed in typically developing infants over time.3 In
patients with congenital heart disease, this variability
may be magnified by the additional burden of
repeated hospital admissions or frequent medical care,
and as such longitudinal assessment of gross motor
development is preferable.
The aim of this longitudinal study was to
describe gross motor development of infants who
Vol. 22, No. 5 Long et al: Gross motor delay in congenital heart disease
had undergone early cardiac surgery. A secondary
analysis of primary outcome data pertaining to
study participant exposures and potential confoun-
ders was also performed.
Materials and methods
The Human Research and Ethics Committee of the
Royal Childrens Hospital, Melbourne, Australia
(#26030), approved this prospective cohort study.
Neonates and young infants with congenital heart
disease who underwent elective or emergency
cardiac surgery at less than or up to 8 weeks of
age were included. Infants with known chromoso-
mal abnormalities or congenital syndromes such as
the 22q deletion spectrum or Down syndrome were
excluded from participation, as were families in
which the primary caregiver did not speak English,
and families who lived more than 100 kilometres
outside the Melbourne metropolitan area. A senior
paediatric physiotherapist (S.L.) conducted all
recruitment for this study; the primary caregiver
gave signed informed consent to participate.
Gross motor assessment: Alberta Infant Motor Scale
Gross motor performance was assessed at the Royal
Childrens Hospital when infants were 4, 8, 12, and
16 months of age (corrected) with the Alberta
Infant Motor Scale. Where families were unable to
attend scheduled appointments, home visits were
organised. All Alberta Infant Motor Scale assessments
were conducted in accordance with manual guide-
lines.4 The Alberta Infant Motor Scale is a discrimi-
native gross motor outcome measure that may be used
to assess infants from birth to independent walking.
Discriminative tests differentiate between individuals
with or without a specific characteristic or function
and are often norm-referenced.5 Consisting of 58 gross
motor items, each item evaluates the infant in one of
four different positions prone, supine, sitting, and
standing and is scored on a dichotomous scale as
observed (1 point) or not observed (no points)
according to specific criteria relating to three
components of movement: posture, weight-bearing,
and anti-gravity movement. Infants were videotaped
during the assessment and assessments were scored
from digital versatile discs more than 1 month after
the assessment by S.L., and separately by a second
senior paediatric physiotherapist (K.D.) who was
blinded to each infants medical history, including
previous clinical assessments.
A normative data set for the Alberta Infant Motor
Scale was developed by the scale authors through
observations of a birth cohort (19901992) from
Alberta, Canada. The normative sample comprised a
575
random sample of 2202 children, stratified by
geographical region, age, and sex.6 The existence of
normative data allows Alberta Infant Motor Scale
total raw scores to be transformed into age-based
percentile ranks. Extensive reliability testing by the
authors and subsequent scale users has consistently
demonstrated high levels of inter- and intra-rater
reliability.68
Peri-operative data collection
Peri-operative variables were collected prospectively
from Royal Childrens Hospital databases. Categorical
and continuous variables included gestational age;
birth weight; primary diagnosis; age at admission; age
at surgery/surgeries; surgery performed; surgical
variables, including cardiopulmonary bypass time,
cross-clamp time, deep hypothermic circulatory arrest
time, temperature; extra-corporeal membranous oxy-
genation time; ventilation time; respiratory support
time, including ventilation time and non-invasive
ventilation time; intensive care length of stay; and
hospital length of stay. Binary variables included sex,
cardiopulmonary bypass (yes/no), cyanotic or acyanotic
congenital heart disease, palliative or corrective cardiac
surgery, and social risk (high/low). Social risk was
assessed using the Social Risk Index, which is a
composite measure comprising six aspects of social
status including family structure, education of primary
caregiver, occupation of primary income earner, employ-
ment status of primary income earner, language spoken
at home, and maternal age at birth.911
Missing data were retrieved, where possible, by
examination of individual patient unit records.
Statistical methods
Retrospective data review before study commence-
ment suggested that 50 infants would be eligible for
this study over a 24-month period. Precision-based
sample size calculations for 50 study participants
estimated the width of the 95% confidence interval
for mean Alberta Infant Motor Scale total raw scores at
4, 8, 12, and 16 months of age to be 2.4, 3.9, 2.6, and
0.3 items, respectively. This amount of precision was
thought to be clinically meaningful to detect a
difference between the sample studied and Alberta
Infant Motor Scale normative data across the ages
studied.
Linear interpolation of normative data was used
to convert Alberta Infant Motor Scale total raw
scores to an Alberta Infant Motor Scale percentile
rank for each study participant at 4, 8, 12, and 16
months corrected age. Each percentile rank was
categorised according to groupings provided by the
Alberta Infant Motor Scale authors.4 Study partici-
pants with a percentile rank that was less than or
576 Cardiology in the Young
equal to the 10th percentile on three or more
assessment occasions were classified as having
atypical gross motor development (Fig S1). Study
participants with a percentile rank that was less
than or equal to the 10th percentile on two of at
least three assessment occasions were classified as
having suspicious gross motor development. Cases
where the percentile rank was categorised as more
than the 10th to less than or equal to the 25th
percentile were also included in these atypical and
suspicious classifications if they occurred in series
with a lower percentile classification, for example a
study participant that performed on the 8th, 15th,
and 5th percentile at 4, 8, and 12 months,
respectively, would be categorised as having atypical
gross motor development.
Descriptive statistics were used to describe
binary, categorical, and continuous variables col-
lected in the peri-operative period, as well as
Alberta Infant Motor Scale total raw scores for
infants assessed at 4, 8, 12, and 16 months of age.
Bivariate linear regression was used to describe the
association between Alberta Infant Motor Scale total
raw score data, with separate dependent variables for
study participants at 4, 8, 12, and 16 months of age,
and risk factors continuous or categorical variables
collected in the peri-operative period. Hypothesis
tests (two sample t-tests, equal variance not assumed)
were performed to examine differences in Alberta Infant
Motor Scale total raw score data and independent
binary variables.
Intra-class correlation statistics were used to
evaluate intra- and inter-rater reliability. Intra-rater
reliability for K.D. was evaluated by scoring 15
(20%) of the 8-month Alberta Infant Motor Scale
assessment segments on a second occasion after at
least a 3-month interval. Alberta Infant Motor Scale
total raw scores from all assessments were used to
assess inter-rater reliability between S.L. and K.D.
Results
Participants
Eligibility for study participation was assessed in
195 infants who underwent cardiac surgery between
the 5th of July 2006 and the 2nd January 2008. In
all, 63 infants were eligible, and thus our sample
represented 79% of eligible participants (Fig 1).
There were five families that declined participation
in this study, and reasons for non-participation
included not wanting to put their baby through
additional hospital experiences or appointments
(four families) and difficulty committing to the
study schedule of assessments (one family). In
addition, five cases were missed and families of three
October 2012
infants had social circumstances that precluded
recruitment. Of the 55 families approached for
participation, 50 (91%) were recruited.
Of the 50 patients enrolled, there was permanent
attrition of two participants between 4 and 8
months of age: one patient died, and the other did
not attend scheduled appointments. There was
temporary attrition of subjects at each time point,
with 92%, 84%, and 94% returning for assessment
at 8, 12, and 16 months of age, respectively.
Approximately 78% of enrolled subjects completed
all four Alberta Infant Motor Scale assessments.
Study participant characteristics are shown in
Table 1. Study participants had similar characteristics
to non-participants, with the following exceptions:
ineligible cases and deceased patients spent longer in
the paediatric intensive care unit; ventilation and
respiratory support time were much longer in patients
who subsequently died; and ineligible cases and
deceased cases spent more time in hospital. In all,
36 study participants (72%) underwent their first
episode of cardiac surgery with mild to moderate
hypothermic low-flow cardiopulmonary bypass. Low-
flow selective cerebral perfusion was utilised in cases of
circulatory arrest. Gas management was performed
with an alpha-stat strategy. There was one study
participant who required extra-corporeal membranous
oxygenation for 48 hours in the post-operative period
following a cardiac arrest, secondary to spontaneous
blockage of a cardiac shunt; this was the same
participant who died at 213 days of age. There were
21 study participants who underwent a second
surgical procedure during the study period, 16 of
which occurred before 8 months of age; six study
participants underwent a third surgical procedure, and
one study participant a fourth surgical procedure
during the study period.
Gross motor assessment: Alberta Infant Motor Scale
As a group, study participants had delayed gross
motor development with reference to Alberta Infant
Motor Scale normative total raw scores at all study
195 Surgical Exclusion
Cases
18 Deceased
1 Palliative care
72 Interstate/overseas
23 Victorian, >100km from testing centre
63 Eligible
11 Congenital Syndrome
7 English as a second language
5 Declined participation
5 Missed cases
3 Social circumstances precluded recruitment
50 Participants
Figure 1.
Flow chart of individuals assessed for eligibility.
Vol. 22, No. 5 Long et al: Gross motor delay in congenital heart disease 577

Table 1. Study participant characteristics.

Participants (n 5 50)

n % p25 Med p75

Male 28 56
Gestational age (weeks) 50 100 38 40 40
Birth weight (g)* 49 98 3000 3390 3660
High social risk** 21 44
CHD classification
Acyanotic, closed 7 14
Acyanotic, open 13 26
Cyanotic, corrected 15 30
Cyanotic, complex 15 30
Surgical classification
Biventricular repair 26 52
Biventricular repair, arch repair 8 16
Single ventricle procedure 11 22
Single ventricle procedure, arch repair 5 10
Primary surgery
Age (days) 50 100 4 9 21
CPB Time (mins) 36 72 99.5 147 182.5
CCT (min) 34 68 47 79.5 104
CAT (min) 4 8 1.5 16 39.5
Temperature nadir (degrees) 36 72 24.5 29 32
Haematocrit 36 72 29.2 29.65 30.4
Primary admission
PICU length of stay (days) 50 100 2 3.5 7
Age at PICU admission (days) 50 100 3 8 21
Ventilation time, invasive (h) 50 100 32.58 61.08 103.58
Respiratory support time (h) 50 100 36.67 66 108.5
RCH length of stay (days) 50 100 12 18 31
CAT 5circulatory arrest time; CCT 5cross clamp time; CPB 5 cardiopulmonary bypass; Med 5 median; PICU 5 Paediatric
Intensive Care Unit, RCH 5 Royal Childrens Hospital
n 5 sample size; p25 5 25th percentile; p75 5 75th percentile
*Missing data (n 5 1)
**High social risk 5 social risk index >2, missing data (n 5 2)

Table 2. Descriptive statistics AIMS total raw scores for study participants at 4, 8, 12, and 16 months of age.

AIMS total raw scores

Study participants AIMS normative data

Age (months) n Mean SD 95% CI Age (months) n Mean SD 95% CI

4.2 6 0.4 50 11.76 3.03 10.90 12.62 3 to ,4 90 12.6 3.29 11.9 13.2
4 to ,5 122 17.9 4.15 17.2 18.6
8.2 6 0.3 45 27.07 8.66 24.46 29.67 7 to ,8 222 32.3 6.85 31.4 33.2
8 to ,9 220 39.8 8.69 38.6 41
12.1 6 0.4 41 45.49* 10.05 42.32 48.66 11 to ,12 155 51.3 7.11 50.2 52.4
12 to ,13 124 54.6 4.52 53.8 55.4
16.0 6 0.5 46 52.79* 8.19 50.36 55.22 15 to ,16 40 57.8 0.45 57.7 57.9
16 to ,17 49 57.8 0.55 57.6 58
AIMS 5 Alberta Infant Motor Scale; CI 5 confidence interval; SD 5 standard deviation
n 5 sample size
*Data not normally distributed. At 12 months of age, median AIMS total raw score 5 49, inter-quartile range 4051. At 16 months of age,
median AIMS total raw score 5 56, and inter-quartile range 5 5158

time points (Table 2). The greatest inter-individual Alberta Infant Motor Scale percentile rank
variation in Alberta Infant Motor Scale total raw categorisations are shown in Table 3 and Figure 2.
scores occurred at 12 months of age (Table 2). Ceiling scores were achieved on the prone (66%),
578 Cardiology in the Young October 2012

Table 3. AIMS percentile rank classification for study were assessed at all three remaining study time
participants at 4, 8,12, and 16 months of age. points. Linear interpolation data revealed that 38%
(15 of 39) of study participants had atypical motor
AIMS Assessment development and a further 23% (9 of 39) had
Percentile suspicious motor development, as measured by the
category 4-month 8-month 12-month 16-month Alberta Infant Motor Scale over three assessments.
Inter-rater reliability for Alberta Infant Motor
<5th 8 16 12 32 Scale total raw scores was high at 4 months of age
5th to <10th 10 9 3 0
10th to <25th 15 8 11 0
(Intra-class correlation coefficient 5 0.84) and very
25th to <50th 14 7 9 0 high at 8, 12, and 16 months of age (Intra-class
50th to <75th 3 3 5 0 correlation coefficient 5 0.95, 0.97, 0.97, respec-
75th to <90th 0 2 1 0 tively). Intra-rater reliability for 20% of Alberta
.90th 0 0 0 15 Infant Motor Scale assessments at 8 months of age was
Missing* 5 9 3
very high (Intra-class correlation coefficient 5 0.98).
AIMS 5 Alberta Infant Motor Scale
*Missing due to permanent or temporary attrition of study subjects
Gross motor outcome and peri-operative variables
Length of stay in hospital for the initial surgical
procedure, as well as cumulative length of stay in
hospital, was predictive of Alberta Infant Motor
Scale total raw scores at 4, 8, 12, and 16 months of
age (Table 4). Other independent variables pre-
dictive of Alberta Infant Motor Scale total raw
scores included: at 4 months of age, length of stay in
the paediatric intensive care unit following primary
surgery, cumulative length of stay in paediatric
intensive care, post-operative respiratory support
time, and age at surgery; at 8 months of age, post-
operative respiratory support time; and at 16
months of age, cumulative length of stay in
paediatric intensive care, and cumulative respiratory
support time (Table 4). No other categorical or
continuous variables were predictive of Alberta
Infant Motor Scale total raw scores at 4, 8, 12, or 16
months of age.
Figure 2. At 4 months of age, a statistically significant
Trajectory of Alberta Infant Motor Scale total raw scores versus difference was found between Alberta Infant Motor
Alberta Infant Motor Scale percentile ranks.4 Scale total raw scores for infants who had undergone
Notes: Each black dot represents a single study participants Alberta palliative surgery and those who had had corrective
Infant Motor Scale total raw score at their corrected age at assessment surgery (p 5 0.004); this difference was not sig-
across the four study time points. nificant at other study time points. No difference
was found for other binary independent variables at
supine (62%), sitting (91%), and standing subscales 4, 8, 12, or 16 months of age.
(40%) at 16 months of age. Approximately 32%
of study participants achieved the maximum
Alberta Infant Motor Scale total raw score of 58 at
Discussion
16 months of age. An additional 17% of study This is the first study to describe longitudinal
participants achieved a score of 57, that is, 51% of Alberta Infant Motor Scale assessment data for
study participants were walking independently at infants with congenital heart disease. Our data
16 months of age (versus 100% of the normative suggest that more than half (62%) of study
sample being able to walk independently). participants did not have typical gross motor
Alberta Infant Motor Scale percentile ranks were development during the first year of life. The
not sensitive in distinguishing the motor abilities validity of study findings was strengthened by high
of study participants at 16 months of age (Table 3). participation rates and serial data collection. Inter-
These data were not considered suitable for and intra-rater reliability were high across the time
additional discriminative analyses. In all, 39 infants points of this study.
Vol. 22, No. 5 Long et al: Gross motor delay in congenital heart disease 579

Table 4. Variables associated with AIMS total raw scores.

Variable R-squared Coefficient 95% confidence interval p-value

4-month AIMS total raw scores

*PICU length of stay (days) primary admission 0.166 21.645 22.714 20.576 0.003
*PICU length of stay (days) cumulative 0.137 21.230 22.126 20.334 0.008
*Respiratory support time primary admission 0.134 21.292 22.244 20.340 0.009
*RCH length of stay (days) primary admission 0.259 22.036 23.035 21.036 0.000
*RCH length of stay (days) cumulative 0.253 21.946 22.916 20.976 0.000
*Age at primary surgery 0.144 1.091 0.320 1.862 0.007
8-month AIMS total raw scores
*Respiratory support time primary admission 0.154 23.845 26.619 21.070 0.008
*RCH length of stay (days) primary admission 0.249 25.897 29.045 22.750 0.000
*RCH length of stay (days) cumulative 0.203 25.044 28.117 21.970 0.002
12-month AIMS total raw scores
*Respiratory support time primary admission 0.166 24.604 27.951 21.257 0.008
*RCH length of stay (days) primary admission 0.226 26.697 210.709 22.684 0.002
*RCH length of stay (days) cumulative 0.201 25.952 29.801 22.103 0.003
16-month AIMS total raw scores
*Respiratory support time cumulative 0.169 23.928 26.546 21.310 0.004
*PICU length of stay (days) cumulative 0.143 23.369 25.844 20.894 0.009
*RCH length of stay (days) primary admission 0.152 24.275 27.307 21.244 0.007
*RCH length of stay (days) cumulative 0.218 24.734 27.422 22.045 0.001
PICU 5 Paediatric Intensive Care Unit, RCH 5 Royal Childrens Hospital

The Alberta Infant Motor Scale is an excellent
test for discriminating between typical and atypical
gross motor development for infants with congeni-
tal heart disease who are at risk for motor
dysfunction in the first 12 months of life. However,
the Alberta Infant Motor Scale did not appear to
discriminate between typical and atypical gross
motor development in older infants, as seen by the
classification of percentile ranks of 16-month-old
infants in this study, and as supported by previous
studies.12,13 Although not the only tool for assessing
gross motor function in infancy, the Alberta Infant
Motor Scale is the only single-domain, gross motor
assessment tool available for use with infants between
birth and independent walking. Owing to the fact
that the test is observational in nature, infants who are
medically fragile or irritable need not be excluded
from assessment. This was important in choosing the
Alberta Infant Motor Scale for use in the congenital
heart disease population. Similarly, because the
Alberta Infant Motor Scale can be performed in as
few as 15 minutes, it is ideal for infants with
endurance limitations related to cardiac dysfunction.
The majority of outcome studies of infants with
congenital heart disease have used multi-dimensional
developmental assessments such as the Bayley Scales of
Infant Development. Peri-operative variables signifi-
cantly associated with these outcomes have included
specific congenital heart disease diagnoses/surgical
procedures, that is, hypoplastic left heart syndrome,14
Norwood,15 complex congenital heart disease,16
acyanotic heart disease (compared with cyanotic heart
disease),17 and transposition of the great arteries with
ventricular septal defect (compared with the great
arteries with intact ventricular septum);18 electroence-
phalogram abnormalities;14,18,19 microcephaly;14 low
birth weight or weight at surgery;20,21 markers of
peri-operative illness severity;14,15 deep hypothermic
circulatory arrest temperature22 or duration;15,17,18
length of hospital stay;20,23 apolipoprotein E poly-
morphisms;24 and the need for cardiopulmonary
resuscitation.23 However, the association of these
variables with developmental outcomes has been
observed to alter over time. For example, peri-
operative factors that have been associated with
infant motor ability, such as illness severity,
pre-operative ventilation, and weight at surgery,
have no longer been relevant at preschool age.15,25
In the present study, those who had palliative
(versus corrective) surgery had lower Alberta Infant
Motor Scale total raw scores at 4 months of age;
however, there was no association between type of
surgery and Alberta Infant Motor Scale total raw
scores at the other study time points. In a similar
vein, age at surgery was associated with Alberta
Infant Motor Scale total raw scores at 4 months of
age, yet there was no association between age at
surgery and Alberta Infant Motor Scale total raw
scores at later study time points. The dilution of
significance of certain variables over time is likely
because other factors have greater influence on gross
motor development at later times. Importantly,
which risk factors are important in relation to short-
and long-term gross motor outcomes is unclear.
580 Cardiology in the Young
There are some known entities. The risks of
impaired delivery of oxygen and glucose to the
brain during cardiopulmonary bypass secondary
to embolic events, systemic inflammatory response,
or induced cerebral ischaemia in addition to the
risks associated with intra-operative management,
that is, pH management, haemodilution strategies,
and surgical technique, leading to brain injury and
long-lasting developmental dysfunction are very
real. The landmark Boston Circulatory Arrest Trial
was the first to systematically study the effects of
deep hypothermic circulatory arrest on a subgroup
of patients with congenital heart disease, conclud-
ing that a deep hypothermic circulatory arrest time
of more than 41 minutes is associated with lower
psychomotor developmental index scores at 1 year
of age.18 Other studies have also shown that the
risk of brain injury or developmental dysfunction in
the first 2 years of life increases with duration of
circulatory arrest.15,17,2628 In our exploratory
analysis, intra-operative variables were not related
to Alberta Infant Motor Scale scores. This is not
unexpected only four study participants under-
went circulatory arrest during their first surgical
procedure, and none underwent deep hypothermic
circulatory arrest; this reaffirms that generalisation
or comparison of findings of different studies, or
this study, with other samples must take into
consideration the characteristics of the target group.
Research examining specific congenital heart
disease diagnoses as risk factors for developmental
dysfunction has been inconclusive. Studies that have
considered brain injury as a surrogate measure for
developmental outcome have shown that the overall
risk has not differed by anatomical classifica-
tion.29,30 The exception has been infants with
hypoplastic left heart syndrome, who are thought to
be at a higher risk of acquired brain injury in the
peri-operative period when compared with infants
with diagnoses requiring other types of surgery.29
Those with hypoplastic left heart syndrome have
been shown to have global developmental delay at
12 months of age, whereas infants diagnosed with
other forms of single-ventricle congenital heart
disease have more specific gross motor delay at
this age.14 We could not explore the relationship
between specific congenital heart disease diagnoses
and gross motor outcome in this study. Even with
relatively strict inclusion criteria, the categorised
subgroups of congenital heart disease each repre-
sented a spectrum of conditions, and the character-
istics of participants varied accordingly.
Post-operative respiratory support time was
predictive of Alberta Infant Motor Scale total raw
scores at 4, 8, and 12 months of age, and cumulative
respiratory support time at 16 months of age was
October 2012
associated with 16-month Alberta Infant Motor
Scale total raw scores. Interpretation of these data is
difficult because of the association between respira-
tory support time and length of stay in both the
intensive care unit and the hospital. Study
participants spent a median of 18 days as inpatients
for their first surgical procedure, and length of stay
was predictive of Alberta Infant Motor Scale total
raw scores at all study time points. Previous studies
have linked hospital length of stay to cognitive
ability in infancy31 and at school age.32 In addition,
more recent studies have linked the Psychomotor
Developmental Index, the Bayley Scales of Infant
Development, Second Edition, and hospital length
of stay. Gaynor et al20 reported lower psychomotor
development scores in 1-year-old infants who had
undergone biventricular repair and had longer
hospital stays. Atallah et al23 reported lower psycho-
motor development scores in 2-year-old infants who
had undergone a Norwood procedure and had longer
hospital stays. What is thought provoking about these
two studies is that longer length of stay is indicative of
unfavourable outcome following both corrective and
palliative surgery. This makes obvious the fact that a
multitude of factors coexist to determine length of
stay, and may render both groups subsequent gross
motor trajectory either low or very high risk.33 Factors
such as the need for pre-operative intubation, lower
birth weight, low cardiac output, sepsis, arrhythmia,
pulmonary hypertension, seizures, chylothoracies, and
neuromuscular weakness are implicated in the
requirement for prolonged ventilation and admission.
It was not possible, nor valid, to collect such a large
number of variables in this study. Both prolonged
ventilation and hospital admission are thus taken as
markers of a more complex medical course, and likely
identify the most compromised infants.15
Typical motor development is characterised by
intra- and inter-individual variability, which is
reflected as peaks and plateaus in the acquisition of
new motor skills. Results of our study showed
that inter-individual variability was greatest at 12
months of age. In a longitudinal study of pre-term
infants, Van Haastert et al13 also reported a gradual
increase in the width of the standard deviation of
Alberta Infant Motor Scale total raw scores up to 11
and 12 months of age, followed by a subsequent
decrease to the end of the scale (18 months). This
contrasts with the standard deviation peak seen
between 8 and 9 months of age in the Alberta Infant
Motor Scale normative sample,4 but is in line with
results by Sarajuuri et al14 who found that a sample
of 12-month-old infants with congenital heart
disease had a wider standard deviation compared
with a sample of healthy 12-month-old term infants.
These results suggest that, as a group, infants with
Vol. 22, No. 5 Long et al: Gross motor delay in congenital heart disease
congenital heart disease acquire certain gross motor
skills several months later than typically develop-
ing infants. Furthermore, it suggests that these
infants lack the readiness required for the same level
of environmental exploration as their typically
developing peers.
Conclusions
This study has shown that the Alberta Infant Motor
Scale is valid for identifying infants with gross
motor delay following early cardiac surgery. The
ability to objectively identify infants progressing on
an atypical gross motor trajectory is essential to
ensure that infants with congenital heart disease are
offered timely intervention services. A plethora of
risk factors for gross motor delay exist and interact
in the pre-, peri-, and post-operative periods;
therefore, identifying associative and causal rela-
tionships is challenging. Given that more than half
of the study participants showed delayed gross
motor development, which persisted across infancy,
active developmental surveillance of all infants
undergoing early cardiac surgery is recommended.
Further studies of larger congenital heart disease
samples, as well as longitudinal studies that
determine the significance of these findings at
school age and beyond, are needed. Studies evaluat-
ing early intervention services designed specifically
for infants with congenital heart disease and their
families are also warranted.
Limitations
Approximately 42% of infants who underwent
cardiac surgery during the recruitment period were
ineligible for participation in this study because of
residency outside Victoria. This limited the sample
size available for this study; however, given that
only sicker, more vulnerable infants are transferred
interstate for surgery, inclusion of these infants
without the inclusion of all interstate infants
undergoing early cardiac surgery would have biased
the sample, and made generalisation of findings
more difficult.
Although the importance of serial developmental
assessment is appreciated,3 there is no existing
consensus on how to interpret or report serial
Alberta Infant Motor Scale scores. We defined atypical
and suspicious gross motor development categories by
incorporating previously suggested cut-off scores13,34
with our individual experiences with interpreting
serial Alberta Infant Motor Scale data.
The analysis of risk factors was exploratory in
nature; the sample was not adequate to perform
multivariate analysis and more definitively investigate
581
the contribution of risk factors on gross motor
outcome. Finally, variables were predominantly
collected in the pre- and peri-operative periods.
Consideration of later risk factors for gross motor
dysfunction, such as additional measures of environ-
mental conditions, would greatly benefit the inter-
pretation of data.
Acknowledgements
The authors wish to thank A/Prof Susan Donath for
statistical consultancy, Katy DeValle for her assess-
ment of digital versatile disc footage, and the families
and infants that willingly participated in this study.
Financial Support
Dr Long received a Trust Fund Scholarship from the
Faculty of Medicine, Dentistry and Health Sciences,
University of Melbourne for the duration of her PhD
studies in 2009. Philanthropic financial support was
received to perform this study, including the Janina &
Bill Amiet Foundation (2006) and the Marion & EH
Flack Nominees (2007). The MCRI is supported
through a Federal Government infrastructure grant.
Supplementary materials
For supplementary material referred to in this
article, please visit http://dx.doi.org/doi:10.1017/
S1047951112000121.
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