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doi:10.1111/j.1440-1754.2009.01492.

ORIGINAL ARTICLE

Can parents accurately screen children at risk of


developmental delay?
Glenys Dixon,1 Nadia Badawi,1,2 Davina French3 and Jennifer J Kurinczuk1,4
1
Telethon Institute for Child Health Research, Centre for Child Health Research, The University of Western Australia, Subiaco, Western Australia, 2Grace Centre
for Newborn Care, The Childrens Hospital at Westmead, University of Sydney, Sydney, New South Wales, 3School of Psychology, The University of Western
Australia, Nedlands, Western Australia, Australia and 4National Perinatal Epidemiology Unit, University of Oxford Old Road Campus, Oxford, United Kingdom

Aim: To evaluate the validity and potential value of the parent-completed Infant/Child Monitoring Questionnaire (IMQ) as a screening measure
for developmental delay in high-risk infants.
Methods: One hundred and forty-one term infants born with moderate or severe newborn encephalopathy (NE) and 374 randomly selected
comparison infants were administered a Grifths Mental Development Scales (GMDS) assessment and an IMQ concurrently. Concordance of
classications between measures was compared for agreement, sensitivity, specicity, positive predictive value, negative predictive value, false
positives and false negatives.
Results: Overall, sensitivity and specicity of the IMQ for infants with NE averaged across all age groups was 87%, positive predictive value 57%
and negative predictive value 97%. The IMQ did not perform as well for comparison infants with a sensitivity of 50%, specicity 94%, positive
predictive value 15% and negative predictive value 99% averaged across all age groups. Overall under-referral for infants with NE was 13%,
compared with 50% for comparison infants.
Conclusions: Use of the IMQ as an accurate screening measure in infants at risk of developmental delay is supported. The low sensitivity of
the IMQ for the comparison infants indicates a need for caution when considering its application for general population screening.
Key words: developmental delay; parent; questionnaire; screening.

Approximately one-third of children with developmental or


behavioural disorders remain undetected before entering the
education system,1 and although we have no specific Australian
Key Points
data at this point in time, it is probable the numbers would be
1 Children with developmental delay can be difcult to identify
similar. One explanation for the low detection rates of develop-
before school entry. The delay in identication of problems may
mental delay is that identifying children with developmental
be due to the subtlety of symptoms or the costly and time-
and/or behavioural problems is often challenging, particularly
consuming nature of professional assessments. An alternative
when children have more subtle symptoms rather than present-
to professionally administered tests is a parent-completed
ing with clear signs of delay. Developmental problems may also
questionnaire.
remain undetected because of the costly and time-consuming
2 The present study evaluated a parent-completed question-
nature of developmental assessments, which are typically not
naire, the Infant/Child Monitoring Questionnaire (IMQ), com-
readily available to families located in rural and remote com-
pared with a gold standard professionally administered
munities. One alternative to expensive and time-consuming
developmental assessment widely used by Australian paedia-
professionally administered developmental assessments is
tricians, the Grifths Mental Development Scales, in two rela-
screening with parent-completed questionnaires designed to
tively large groups of children with and without risk of delay.
identify children at risk of developmental delay and in need of
3 The ndings from this study support the use of the IMQ for
further evaluation.
screening populations at high risk of developmental delay. Use
Although a number of parent-completed screening instru-
of the IMQ as a screening instrument in the general population
ments have been developed, there are very few that meet the
should be introduced with caution. Further evaluations need to
psychometric properties recommended by National Health and
be undertaken in larger groups of older children.
Medical Research Council (NHMRC) of 70% for specificity
and sensitivity.2 Sensitivity levels of 70% are considered
Correspondence: Ms Glenys Dixon, Telethon Institute for Child Health acceptable3 and specificity levels of 7080% realistic in devel-
Research, Centre for Child Health Research, The University of Western Aus- opmental screening.4,5 These standards are recommended for
tralia, PO Box 855, West Perth, WA 6872, Australia. Fax: +61 8 9489 7700; concurrent outcomes with a developmental test or gold stan-
email: glenysd@ichr.uwa.edu.au
dard as a criterion measure. The Infant/Child Monitoring
Accepted for publication 16 November 2008. Questionnaire (IMQ) was developed as a parent-completed

268 Journal of Paediatrics and Child Health 45 (2009) 268273


2009 The Authors
Journal compilation 2009 Paediatrics and Child Health Division (Royal Australasian College of Physicians)
G Dixon et al. Screening children at risk for delay

questionnaire6 and was later renamed the Ages and Stages Methods
Questionnaire (ASQ) in 1995 with minimal revision to content.
The change of content was so minimal that the data collected Participants
from the IMQ and the revised ASQ were combined to report
Participants were members of a longitudinal cohort study in
psychometric properties for the revised ASQ, which is essen-
Perth, Western Australia, who were originally recruited in a case
tially the same tool with a new name.7
control study established to investigate the antecedents of
Although there have been a number of comparisons of the
newborn encephalopathy (NE) and subsequently followed to
IMQ and/or ASQ with popular developmental assessments
determine their outcomes. The methods for the Western Aus-
used overseas, the concurrent validity of the IMQ/ASQ with
tralian NE Study have been described in detail previously11 and
criterion measures used in Australia through a population-
are outlined briefly here.
based study has not, as far as we are aware, been evaluated. A
study of 167 infants born prematurely8 and considered at risk Eligibility criteria of children diagnosed with NE
of developmental disability reported the concurrent validity
and test characteristics of the IMQ with the Griffiths Mental Infants with NE were all live infants born at term (37 weeks)
Development Scales (GMDS).9 Based on their results of high in Western Australia between 1 June 1993 and 31 December
negative predictive values, Skellern et al. supported the use of 1996 who fulfilled the criteria in Figure 1. Deaths in the first
the ASQ as a screening tool for cognitive and motor delays in week of life were reviewed to ensure that no baby who fulfilled
the follow-up of ex-premature infants but suggest combining the eligibility criteria died before inclusion in the study.
the screening instrument with other strategies for comprehen- The severity of NE was graded by a neonatologist as moderate
sive follow-up.8 or severe according to the criteria modified from Sarnat and
However, another study examining the test characteristics Sarnat.11,12 In the total sample there were 177 infants with
of the IMQ with high-risk infants from a neonatal intensive care moderate NE and 99 with severe NE. The incidence of moderate
unit reported that the IMQ had a high under-referral rate of or severe NE was 3.80 per 1000 term live births (95% confi-
33% and was therefore considered to have limited value as a dence interval 3.244.43).11
screening instrument of high-risk populations.10 In the absence
of consistent research findings about the utility of this screening Comparison cohort
tool in Australian populations other than ex-premature or very
There were 564 infants in the comparison cohort who were
high-risk infants, the immediate and long-term implications of
randomly selected from the population of term live births
introducing the ASQ (formerly IMQ) into clinical and non-
without NE delivered in metropolitan Perth during the same
clinical populations for comprehensive follow-up programmes
time period as the infants with NE.
remain unclear.
The primary aim of this study was to compare the IMQ with Follow-up procedures
a concurrently administered gold standard measure, the
GMDS, to examine the test characteristics and concurrent valid- The follow-up protocol requested parents to complete the IMQ
ity in two groups of children, one of which is high-risk. when their children were aged 4, 8, 12, 18, 24, 36 and

Fig. 1 Inclusion criteria for cases with moder-


ate or severe newborn encephalopathy.

Journal of Paediatrics and Child Health 45 (2009) 268273 269


2009 The Authors
Journal compilation 2009 Paediatrics and Child Health Division (Royal Australasian College of Physicians)
Screening children at risk for delay G Dixon et al.

48 months. When infants were aged 12 months, the parents the IMQ and GMDS was determined by the concordance
were contacted to have their child assessed by a trained assessor between classifications on the screening measure (IMQ) and
(blind to the case control status of the child) using the GMDS. If criterion measure (GMDS) and was calculated as percentage
families were unavailable for a Griffiths assessment when their agreement using the binary prediction model.19 Measures of
child was 12 months of age, the assessment was arranged for association adjusted for chance agreement (Kappa coefficient)
a later date which resulted in some children being older than were used to measure classification accuracy. Sensitivity, speci-
12 months when the GMDS was completed. ficity, positive and negative predictive values, over-referrals and
under-referrals were calculated in each age group, across all age
Measures groups and for each area of development.

Screening measure (IMQ)


Ethical approval
The IMQ is a parent-completed questionnaire designed to
The protocol for this study was approved by the Ethics Com-
screen infants aged from 4 to 48 months with questionnaire
mittees of The University of Western Australia, King Edward
items developed to reflect developmental milestones.7 Each
Memorial and Princess Margaret Hospitals which conforms with
questionnaire has 30 items equally distributed over five sub-
the provision of the Declaration of Helsinki in 1995 (as revised
scales, including gross motor skills, fine motor skills, adaptive
in Edinburgh 2000). Informed consent was obtained from the
skills, communication and personal-social skills. The question-
parents of all children who participated.
naires also include open-ended questions to elicit parental con-
cerns about their childs development.
Results
Criterion measure
Of the original cohort of 840 infants, 36 infants with NE and one
The GMDS was designed to assess the development of babies to comparison infant died before any assessment. One infant with
children from birth to 2 years with an extended version from 2 NE died after the ASQ and GMDS were completed and was
to 8 years.9 The GMDS assesses developmental milestones in included in these results.
gross motor, personal social, speech and hearing (communica- There were 240 surviving infants with NE and 563 surviving
tion), eye and hand co-ordination (fine motor) and perfor- comparison infants eligible for an IMQ and a GMDS. Of the NE
mance (adaptive) (for 02 years) and also practical reasoning group, 203 (84%) returned at least one IMQ, 195 (81%) had a
(for 28 years version). The GMDS is widely used by Australian GMDS and 141 (59%) had concurrent IMQ and GMDS assess-
paediatricians.13,14 ments within a 3-month period, of which 32 (13%) were aged
Traditionally, the use of a General Quotient (GQ) of 100 and more than 24 months. There were 108 infants with moderate
standard deviation (SD) of 12.2 has been used to determine NE and 33 with severe NE that had a concurrent IMQ and a
levels of impairment. However, these scores were based on the GMDS completed. Of the surviving comparison infants, 488
original sample standardised in 1950 (02 years) and 1960 (87%) returned at least one IMQ, 456 (81%) had a GMDS and
(08 years extended version). Results from the GMDS for the 374 (66%) had a concurrent IMQ and GMDS within a 3-month
Western Australian NE Study comparison cohort without NE period, 53 (9%) of which were over the age of 24 months.
revealed a mean GQ of 113 (SD: 9.3), which was in agreement Overall, there were 515 infants assessed concurrently by the
with more recent study results that have utilised the older screening measure (IMQ) and the gold standard criterion
version of the Griffiths scales.1518 We therefore used this mean measure (GMDS) at varying age intervals (Table 1). The mean
and SD to define clinically significant developmental delay (>2 age of GMDS completion was 18 months for comparison infants
SDs below the mean) for this study population. and 19 months for infants with NE.
Overall, 31 infants were classified with delayed development
Statistical analyses on the GMDS, of which 24 (sensitivity 77%) were classified as
being delayed by the concurrent IMQ and seven (23%) were
Statistical analysis was undertaken using SPSS version 13 for false negatives (under-referrals). Among the seven false nega-
Windows (SPSS Inc., Chicago, IL, USA). Concurrent validity of tives on the IMQ, three were infants with moderate NE and four

Table 1 Age of infants with concurrent IMQ and GMDS (N = 515)

Age at concurrent assessments

12 months 18 months 24 months 36 months 48 months


(n = 200) (n = 230) (n = 47) (n = 30) (n = 8)

Comparison infants (n = 374; 202 male, 172 female) 146 175 29 22 2


Infants with NE (n = 141; 81 male, 60 female) 54 55 18 8 6

GMDS, Grifths Mental Development Scales; IMQ, Infant/Child Monitoring Questionnaire; NE, newborn encephalopathy.

270 Journal of Paediatrics and Child Health 45 (2009) 268273


2009 The Authors
Journal compilation 2009 Paediatrics and Child Health Division (Royal Australasian College of Physicians)
G Dixon et al. Screening children at risk for delay

Table 2 Classication indices of concurrent IMQ and GMDS by age at assessment

IMQ age N Sensitivity Specicity Positive Negative Over-referrals Under-referrals Kappa P-value
(%) (%) predictive predictive (false positives) (false negatives)
value (%) value (%) (%) (%)

All ages 515


Comparison infants 374 50 94 15 99 6 50 0.202 <0.001
Infants with NE 141 87 87 57 97 13 13 0.614 <0.001
12-month IMQ 200
Comparison infants 146 100 90 6 100 10 0 0.106 <0.004
Infants with NE 54 100 78 27 100 22 0 0.340 <0.001
18-month IMQ 230
Comparison infants 175 100 97 17 100 3 0 0.279 <0.001
Infants with NE 55 85 98 92 95 6 15 0.845 <0.001
24-month IMQ 47
Comparison infants 29 25 88 25 88 12 75 0.130 0.484
Infants with NE 18 67 87 50 93 13 33 0.471 0.043

GMDS, Grifths Mental Development Scales; IMQ, Infant/Child Monitoring Questionnaire; NE, newborn encephalopathy.

Table 3 Concordance classication indices of concurrent IMQ and GMDS according to area of development

Development Sample (N) Sensitivity Specicity Positive Negative Over-referrals Under-referrals Kappa P-value
(%) (%) predictive predictive (false (false negatives)
value (%) value (%) positives) (%) (%)

Communication Total (515) 44 97 52 96 3 56 0.443 <0.001


Comparison infants (374) 11 98 11 98 2 89 0.089 0.085
Infants with NE (141) 57 96 72 92 4 4 0.573 <0.001
Gross motor Total (515) 83 98 61 99 2 17 0.688 <0.001
Comparison infants (374) 50 99 38 99 1 50 0.418 <0.001
Infants with NE (141) 94 94 70 99 6 6 0.768 <0.001
Fine motor Total (515) 15 99 92 95 0 68 0.449 <0.001
Comparison infants (374) 15 99 67 97 0 85 0.240 <0.001
Infants with NE (141) 41 100 100 90 0 59 0.539 <0.001
Adaptive Total (515) 55 97 39 98 3 45 0.433 <0.001
Comparison infants (374) 17 98 12 99 2 83 0.127 0.013
Infants with NE (141) 71 92 50 97 8 28 0.579 <0.001
Personal social Total (515) 48 99 69 98 1 52 0.548 <0.001
Comparison infants (374) 25 99 33 99 0.5 75 0.279 <0.001
Infants with NE (141) 53 97 77 93 2 47 0.534 <0.001

GMDS, Grifths Mental Development Scales; IMQ, Infant/Child Monitoring Questionnaire; NE, newborn encephalopathy.

were comparison infants. The characteristics of the validity P = 0.001, n = 54), almost perfect at 18 months (Kappa = 0.845,
measures according to age are given in Table 2. Because of the P < 0.001, n = 55) and moderate at 24 months (Kappa = 0.471,
small numbers, validity measures are not provided beyond age P = 0.043, n = 18). The association of agreement for the
24 months. comparison group showed slight agreement at 12 months
For infants with NE, across all ages, the sensitivity and speci- (Kappa = 0.106, P = 0.004, n = 146), was fair at 18 months (Kappa
ficity of the IMQ was 87%, positive predictive value 57% and = 0.279, P < 0.001, n = 175) and slight at 24 months (Kappa =
negative predictive value 97%. For comparison infants across 0.130, P < 0.484, n = 29).
all ages, the sensitivity of the IMQ was 50%, specificity 94%, Classifications of at-risk of delay on the IMQ and being
positive predictive value 15% and negative predictive value delayed on the GMDS were also compared according to cor-
99%. Overall under-referral for infants with NE was 13%, com- responding areas of development to determine if parents and
pared with 50% for comparison infants. professionals had parallel agreement in detecting delay in spe-
For infants with NE the agreement between the IMQ and cific areas of development (Table 3). Overall, the sensitivity was
GMDS measured by Kappa was fair at 12 months (Kappa = 0.340, lower for corresponding areas of development compared with

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2009 The Authors
Journal compilation 2009 Paediatrics and Child Health Division (Royal Australasian College of Physicians)
Screening children at risk for delay G Dixon et al.

agreement for overall failure by age groups. Sensitivity for the 12-month age group for both the infants with NE and the
infants with NE ranged from 41% (fine motor) to 94% (gross comparison infants. The question is whether fewer false posi-
motor) and only met the acceptable level in gross motor devel- tives are more optimal in terms of costs than higher rates of false
opment, whereas sensitivity for comparison infants was lower negatives. The initial financial costs of further professional
in all developmental areas. testing could be high and would seem to defeat the purpose
of screening; however, the longer-term cost of under-referral
Parent comments could be significant when a child potentially misses out on early
intervention for a problem harder to habilitate at a later age.
Parent comments to the open-ended unscored questions on However, it should be acknowledged that over-inclusion (over-
the IMQ were also examined. Overall, 182 (35%) had parental referral) could provoke unnecessary anxiety in parents of chil-
comments on the IMQ with infants with NE (53%) having more dren referred for further diagnostic testing yet subsequently not
parental comments than comparison infants (29%). When shown to have a problem, which in itself is not without cost.
further examined according to infants who passed or failed the Among the failures on the IMQ for both groups, parents made
concurrent GMDS, there were more comments on the IMQ of comments at almost twice the rate they did when the child
failed infants (96% of infants with NE and 50% of comparison passed their IMQ. The parents comments on the open-ended
infants) compared with infants who passed (45% of infants with questions in the overall section of the questionnaires may offer
NE and 28% of comparison infants). Of the seven false nega- a way to augment quantitative data and increase early detection
tives on the IMQ, all infants with NE (3) had parental concerns of more subtle problems. The true value of parent comments to
noted and none of the four comparison infants had parental the open-ended questions should be more thoroughly exam-
concerns noted. ined to facilitate ways to quantitatively process these concerns
and accurately assess their value. Previous research has shown
Discussion that screening measures which accurately elicit parental con-
cerns are as predictive as other screening measures.21 Such
We examined the screening characteristics of the IMQ in two screening measures also serve to actively involve parents in
relatively large groups of children, the encephalopathy survi- their childs health care which could facilitate advice and infor-
vors born at term who represent a group at high risk of devel- mation in response to parental concerns to be delivered within
opmental delay and the comparison cohort who represent the the broader historical and family context.22
general population of children born at term in Western Austra- Parent-completed screening measures which meet criteria for
lia. We compared the IMQ results with the professionally adequate psychometric properties (sensitivity and specificity
administered Griffiths test and our findings support the use of >70%) may be appropriate for follow-up programmes designed
the IMQ as an accurate and potentially cost-effective alternative to detect certain conditions and as such, should be confined to
to professional screening of infants at risk of developmental these conditions. However, even with evidence of the effective-
delay aged 018 months. These results for the high-risk group ness of such screening, it is recommended that the quality of the
were similar to those reported by Skellern et al.8 However, test be just part of an overall process which will include the test,
agreement between parents of comparison infants and profes- the interpretation, and training of testers, tracking, referral,
sionals is only slight or fair which may be a function of the reporting, support and overall management (NHMRC Report).
general population being at low risk for developmental delay. The notion of one-off screening in itself should not be the
The results from this sample of infants indicate exercising central focus for a pass/fail result as these tests are not diag-
caution before any widespread introduction of the IMQ as a nostic22 but alternatively could provide reinforcement for a
single method to monitor or screen the general population. surveillance system which is family-centred in practice to effec-
Overall, the IMQ performed better for infants with NE com- tively monitor the health of the whole community of children
pared with the comparison infants when individual areas of over time.23
development were examined for agreement with the gold stan- In summary, our findings support the potential value of using
dard assessment. These results could indicate that the IMQ is the parent-completed IMQ (now ASQ) for infants at risk of
sensitive to developmentally at-risk populations whereby developmental delay in Australian populations. The test char-
the most obvious and severe problems are detected more easily acteristics of the IMQ make it useful for the follow-up surveil-
than the more subtle problems or it could also reflect that the lance of infants up to the age of 18 months with wider coverage
parents of infants with NE were more anxious and therefore of rural and remote locations at potentially less cost compared
more vigilant about signs of delay in their children compared with a professional assessment. On the basis of the results from
with parents of comparison infants. The heightened anxiety of this study, the widespread use of the IMQ as a screening instru-
parents of infants who have experienced a serious illness or ment in the general population where the risk of developmental
accident early in life resulting in perceived child vulnerability delay was perceived to be minimal is not recommended in
has been well researched and supported in previous studies20 isolation or without caution when interpreting results, and
and it is thus plausible that parents of children with NE could further research is needed with larger numbers of children aged
have a perceived child vulnerability resulting in the differences more than 18 months to examine the efficacy of such measures.
in parent responses in the two groups. Finally, the value of parents comments on the IMQ in closing
When the sensitivity of an instrument is low and specificity is the gap on the under-referrals in all groups needs to be further
high, there will be fewer false positives (over-referrals), but evaluated. Initial examination appears to suggest that parents
there will be more false negatives (under-referrals) as is seen in have a valuable contribution to make in detecting the early signs

272 Journal of Paediatrics and Child Health 45 (2009) 268273


2009 The Authors
Journal compilation 2009 Paediatrics and Child Health Division (Royal Australasian College of Physicians)
G Dixon et al. Screening children at risk for delay

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