DEVELOPMENTAL MEDICINE & CHILD NEUROLOGY ORIGINAL ARTICLE

Global developmental delay and its relationship to cognitive skills

1
EMILIE M RIOU MD FRCPC | SHUVO GHOSH MD FRCPC 2 | EMMETT FRANCOEUR MD CM FRCPC 2 | MICHAEL I
1
SHEVELL MD CM FRCPC

1 Division of Pediatric Neurology Montreal Children's Hospital. McGill University Health Center, Montreal Canada. 2 Child Development Program; Montreal Children's
Hospital – McGill University Health Center, Montreal, Canada.

Correspondence to Dr Michael Shevell at Division of Child Neurology, Montreal Children's Hospital, 2300 Tupper Street, Montreal, Quebec, H3H 1P3, Canada. E-mail:
michael.shevell@muhc.mcgill.ca

PUBLICATION DATA
Accepted for publication 3rd October 2008.
Published online 6th January 2009.
ABBREVIATION
GDD Global developmental delay
ACKNOWLEDGMENTS
The authors are grateful to Sbastien Dub
for assistance with statistical analysis and
Alba Rinaldi for secretarial assistance.
Michael Shevell is grateful for the support of
the MCH Foundation during the preparation of
this manuscript.
Global developmental delay (GDD) is defined as evidence of significant delays
in two or more developmental domains. Our study determined the cognitive
skills of a cohort of young children with GDD. A retrospective chart review of all
children diagnosed with GDD within a single developmental clinic was carried
out. Scores on fine motor (Peabody Developmental Motor Scale 2), expressive
language (Expressive One Word Picture Vocabulary Test) and receptive
language (Reynell Developmental Language Scales or Clinical Evaluation of
Language Fundamentals – Preschool 2) testing, and cognitive performance
(Wechsler Preschool and Primary Scale of Intelligence, Third Edition) were
obtained. A multiple regression analysis was performed and correlations
obtained. Results from a total of 93 patients (86 males, seven females) were
retained for analysis. Mean age was 3 years 8 months (SD 10mo, range
2.5–4.75y). Cognitive scores were widely distributed, with 73% of participants
displaying a global IQ score of 70 or more, despite concurrent global delay.
Significant correlation was present for fine motor and expressive language
scores, when isolated and compared with cognitive performance (p values of
0.04 and 0.05 respectively). The conclusion was made that an initial diagnosis
of GDD is not necessarily associated with objective cognitive impairment.

Developmental delay is common in pediatric practice.
Although some diagnosed delays are benign, certain pre-
sentations are more worrisome. It is generally recognized
that global developmental delay (GDD) in a child portends
a worse prognosis than isolated single-domain delay and is
more likely to be associated with an underlying, presum-
ably causative, pathology.1–4 Adequate identification of
GDD versus isolated delay in a child allows better selection
of necessary rehabilitation resources, which has been dem-
onstrated to allow better long-term functional outcome.5–7
Delay in development can be defined as a difficulty in
achieving specific developmental milestones compared
with chronological peers. Significant delay is captured by
performance that is two standard deviations or more below
the mean on age-appropriate, standardized, norm-refer-
enced testing. Shevell et al.8 defined GDD as evidence of
significant delay in two or more of the following develop-
mental domains: gross ⁄ fine motor, speech ⁄ language,
social ⁄ personal, cognition, and activities of daily living.8,9
Typically, it is assumed that delay in two developmental
domains is associated with delay across all domains
evaluated.
Significant controversy still surrounds the symptom
complex of GDD. One reason for this lies in the epidemio-
logy of the condition, mainly diagnosed in children under
the age of 5 years. Since formal psychometric testing
often cannot be reliably undertaken in the young
child,10,11 a clear-cut, objective threshold for diagnosis, as
in the case of cognitive delay, often cannot be clearly
established. Therefore, GDD diagnosis relies on the sum-
mation of clinical findings in several developmental areas.
Another cause for controversy is the apparent wide vari-
ability of clinical presentations that fall under the GDD
diagnostic umbrella. Effectively, GDD is a symptom com-
plex analogous to cerebral palsy with heterogeneous pre-
sentations, causes, associated conditions, and evolution

ª The Authors. Journal compilation ª Mac Keith Press 2008

600 DOI: 10.1111/j.1469-8749.2008.03197.x
over time. Although it is presumed that children with
GDD are or will eventually be cognitively impaired, this
has not yet been demonstrated formally. Indeed, if we
consider the strict current definition of GDD, it appears
to allow inclusion of children with combined delays whose
profile could include intact cognitive skills despite delays
in other domains. For lack of effective psychometric test-
ing in very young children, GDD is sometimes considered
as equivalent to cognitive delay, and children with GDD
often receive similar rehabilitation measures, interven-
tions, and programmatic follow-up. In view of these
potential limitations, it is important to examine this
widely-held assumption of equivalence between GDD
and cognitive delay.
The rationale for this study lies in rigorously evaluating
this assumption: that GDD in young children is equivalent
to intellectual delay. By analyzing cognitive testing results
of children diagnosed with GDD in an ambulatory setting
based on performance in other developmental domains, we
hoped to clarify the potential correlation between elements
of GDD and cognitive performance. To address these
questions, we sought to determine the relationship
between results on formal psychometric cognitive testing
and a combination of developmental scores for language
and motor performance in pre-school-age children identi-
fied as being globally delayed in a multidisciplinary devel-
opmental clinic.
METHOD
A detailed retrospective chart review of all children diag-
nosed with GDD in the Montreal Children’s Hospital
Ambulatory Developmental Progress Clinic between Janu-
ary 2002 and December 2004 was undertaken (i.e. a 3-year
inclusive interval). Permission was obtained from the hos-
pital’s Director of Professional Services. Children were
identified by review of the charts of all patients assessed in
the Developmental Progress Clinic during the time period
studied. Referrals to the clinic derive mainly from commu-
nity pediatricians or family physicians in the greater Mon-
treal area. The population seen reflects a wide range of
different ethnic and socio-economic backgrounds. A previ-
ous survey of referring physicians disclosed a local ten-
dency of community physicians to refer children with
suspected developmental delays for specialty assessment.12
Thus the cohort can be viewed as generalizable to commu-
nity pediatric settings.
All children referred to the clinic undergo a formal
multidisciplinary developmental assessment (occupational
therapy, speech ⁄ language pathology, complete history, and
developmental pediatrics examination). A diagnostic con-
clusion from these evaluations is drawn after the first or
second visit to the clinic.
A diagnosis of GDD was ascribed to all children pre-
senting with significant delays (defined as performance
that is two SDs below the mean on age-appropriate,
standardized, norm-referenced testing) in two or more
developmental domains. As part of standard clinical pro-
tocol, all children found to have GDD undergo a for-
mal cognitive assessment with a trained pediatric
neuropsychologist or clinical psychologist between 4 and
5 years of age.
Developmental assessment
Language development and fine motor development were
chosen as study variables because generally accepted norms
are used in the evaluation of these two domains in young
children. Furthermore, early language skills are often clini-
cally used as a proxy for cognitive capabilities. Motor per-
formance was assessed by a trained occupational therapist
using the standardized and validated Peabody Develop-
mental Motor Scale 2 (PDMS-2).13–15 Fine motor perfor-
mance was compiled as the overall score given after the
PDMS-2, then transferred into a centile using the
age-appropriate norms given by this scale.
Language performance was assessed in either English or
French (depending on the child’s better language) by a
trained speech ⁄ language pathologist (fluent in both lan-
guages) using the following measures. First, expressive
vocabulary was evaluated by way of the Expressive One-
Word Picture Vocabulary Test (third edition), a standard-
ized and validated test of expressive vocabulary in
pre-school children.16,17 Scores were compared with
age-appropriate norms and converted into centiles. Form
and use (pragmatics) of language, although assessed in the
clinic, were not scored objectively and thus could not be
retained in the analysis. Second, receptive language was
evaluated using the Reynell Developmental Language
Scales for Children (English and French, first edition) in
children 2 to 4 years of age and the Clinical Evaluation of
Language Fundamentals – Preschool 2 in children 4 years
and older. Both tests have been validated and standardized
in the pre-school population.18,19 (French versions have
been validated and a working standardization is used; final
standardization is pending.) Scores were converted into
centiles by comparison with age-appropriate norms.
Cognitive performance was routinely evaluated accord-
ing to clinic protocol at 4 to 5 years of age by a clinic-affili-
ated psychologist, using the Wechsler Preschool and
Primary Scale of Intelligence (third edition, WPPSI-III),
in French or English. The WPPSI-III is a well-validated
scale in the pediatric population.20 Scores are given for
verbal, performance, and global IQ. Results were compiled
as rank categories in the chart, corresponding to a range
of centiles, as determined by the WPPSI. (Numerical
Global Developmental Delay and Cognitive Skills Emilie M Riou et al. 601
results are not compiled in the chart for ethical [i.e. local
privacy] reasons.)
For children with significant delays in speech and lan-
guage that prevented appropriate administration of the
WPPSI-III, the Griffiths Mental Developmental Scales
were used. The Griffiths is a standardized test of develop-
mental performance in preschool children.21,22 Language,
performance, and global scores were retained and
expressed in rank categories. We reviewed the charts of all
children with GDD on initial assessment who underwent
psychological testing. Children with incomplete evalua-
tions were excluded from the final analysis. Sociodemo-
graphic characteristics were, however, retained and
compiled for general comparison with the study group.
A value of 33% or higher was used as evidence of signifi-
cant correlation for calculation of sample size. Considering
an a error of 5%, a minimum of 70 participants was
required to reach 80% power in this study. Regression
analysis was undertaken by least squares.
Data analysis
Developmental scores were recorded as centiles for the
following variables fine motor performance, expressive
vocabulary, and receptive language. Cognitive scores were
mainly reported as rank categories in patients’ charts. Such
rankings on the Griffiths and WPPSI scales were accepted
as concordant (rank categories correspond to the same cen-
tile ranges) and, for the purpose of this study, were taken
to be equivalent.
All data gathered were analyzed using SPSS software
version 11.0 (SPSS Inc., Chicago). Fine motor, expressive
vocabulary, and receptive language scores were used and
correlated by multiple regression analysis with perfor-
mance, Verbal, and Global IQ scores of the WPSSI, and
language, performance and global scores of the Griffiths
scales. A p value of 0.05 or less was considered to be statis-
tically significant (p values did not undergo Bonferroni cor-
rection as all outcomes were considered as independent
hypotheses).23 Approval for chart review and information
retrieved was obtained from the hospital’s Director of
Professional Services.
RESULTS
A total of 140 children initially diagnosed with GDD were
identified over the study inclusion interval. Of these, 93
children (86 males, seven females) underwent detailed cog-
ilies of children tested (53, 23, and 31% respectively). Very
few children had evidence of an underlying genetic or
neurological disorder at time of initial assessment. Twenty
six per cent of the cohort were subsequently found to meet
criteria for an autistic spectrum disorder, as assessed by
standardized testing in a specialized autism clinic.
Forty-seven children with GDD did not undergo later
psychological testing (either because of loss to follow-up
[38 patients] or refusal from the parents [nine patients])
and therefore were not analyzed. No significant difference
was noted in the demographics of these patients (age at
assessment, sex, family history, and associated conditions)
compared with those undergoing later cognitive testing.
The results of the cognitive scores are shown in Figures 1
and 2. Performance, Verbal, and Global IQ scores were
widely distributed within the cohort. Mean categories were
in the mild deficiency, low average, and borderline ranges
for Performance, Verbal, and Global IQ, respectively.
Table II summarizes the results of the multiple regres-
sion analysis. Expressive vocabulary scores appeared to be
associated with Verbal and Global IQ scores ( p=0.006 and
0.05). Fine motor performance correlated significantly
with Performance IQ ( p=0.027) and Global IQ scores
( p=0.037).
A combination of fine motor, expressive vocabulary, and
receptive language scores showed no statistically significant
correlation with all modalities of IQ scores, as evidenced
by non-significant p values. Further, r2 values confirmed
the poor correlation value of the combined scores for IQ
determination.
Table I: Baseline characteristics of study cohort
Males, n (%) 86 (93)
Family history, n (%):
Neurological disorders 25 (23)
Genetic disorders 2 (2)
Learning disorders 57 (53)
Psychiatric disorders 34 (31)
Consanguinity )
Associated conditions, n (%):
Sensory deficit 4 (4)
Chromosomal anomaly )
Genetic syndrome 2 (2)
Neurological disorder 3 (3)

nitive assessment and their scores were retained for analy- Psychosocial deprivation 3 (3)
Autistic spectrum disorder 28 (26)
sis. The demographic characteristics of the cohort are
Mean age at initial assessment, mo 44.3 (SD 9.6)
outlined in Table I. The mean age of patients at initial
Mean age at cognitive assessment, mo 54 (SD 9.9)
evaluation was 3 years 8 months (SD 10mo, range 2.5–
Mean delay between initial and 9.6 (SD 9.0)
4.75y). Specific learning disorders, psychiatric disorders, cognitive assessments, mo
and neurological disorders were fairly common in the fam-

602 Developmental Medicine & Child Neurology 2009, 51; 600–606

DISCUSSION order to provide early beneficial interventions to the child
Recent guidelines strongly recommend the systematic and family.24 In light of this, it appears relevant to question
screening of developmental delay in young children, in the accuracy of diagnoses of those identified with develop-

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Global Developmental Delay and Cognitive Skills Emilie M Riou et al. 603
 cognitive impairment. This would be better assessed in a
Table II: Multiple regression analysis
larger prospective cohort of children with GDD.
The poor predictive value of the combined scores may
Predictor(s) IQ modality r2 p
also be related to an inherent defect of our definition of
Combined score (FM, EV, RL) GDD. It appears that evidence of significant delay in at least
Verbal IQ 0.07 0.056 two areas of fine motor, expressive vocabulary, or receptive
Performance IQ 0.066 0.07 language is not predictive of cognitive performance in our
Global IQ 0.069 0.06 population. This seems contrary to what was found with
Receptive language
respect to the developmental and functional outcomes of
Verbal IQ 0.01 0.25
children with GDD.27 The answer to this apparent conun-
Performance IQ 0 0.86
drum may be that cognitive performance is not equivalent
Global IQ 0.012 0.26
to developmental performance or functionality in a child,
Expressive vocabulary
Verbal IQ 0.07 0.006a
and thus all three should be considered as distinct, and thus
Performance IQ 0.025 0.099 potentially complementary, outcome measures.
Global IQ 0.036 0.05a Although we were unable to assess personal ⁄ social, gross
Fine motor motor, and activities of daily living domains in a more
Verbal IQ 0.004 0.542 comprehensive standardized fashion, significant delays in
Performance IQ 0.045 0.027a these areas were found during the initial evaluation and
Global IQ 0.04 0.037a were included in the diagnostic process. Hence, most of
a
our participants met more than the present minimum cri-
Significant correlations. FM, fine motor; EV, expressive
teria for impairment in at least two domains.
vocabulary; RL, receptive language.
Scores on cognitive testing were somewhat widely dis-
mental concerns. This is especially so if a diagnosis of a tributed. However, most children fell within the low aver-
GDD is put forward. age to mild deficiency range. This is in keeping with recent
The paucity of studies assessing this particular issue estimates establishing that 90% of individuals with mental
probably reflects the relative novelty of the concept of retardation* ⁄ intellectual disability function within the mild
GDD.8 Retrospective studies have detected evidence of range of impairment.10,28–30 Surprisingly, a small propor-
early childhood developmental delays in children affected tion of our participants scored in the high average range.
with specific learning disabilities.25,26 However, the specific This is again in contradiction to the common assumption
subject of cognitive delay has not yet been addressed in the of equivalence between GDD and cognitive limitation. It
GDD population as a whole. appears that a non-trivial proportion of children with GDD
Shevell et al.27 have prospectively assessed the functional have average intelligence (e.g. 20% of our cohort). This
and developmental outcomes of a cohort of GDD patients, finding may not be completely foreign to the developmental
using the Battelle Developmental Inventory and the Vine- and functional outcomes found by Shevell et al.27 Indeed,
land Adaptive Behavior scale. On follow-up evaluation, one may hypothesize that the subgroup of their cohort
96% of their cohort still met criteria for GDD and 70% attending regular classes without aid (17%) may have had
showed functional impairments in at least two domains. somewhat close to average overall intellectual performance.
Only 17% of the children attended a regular class without Thus there may be two subsets of children with GDD;
an aid. Interestingly, the degree of initial delay was not those with and without cognitive impairment.
found to be predictive of later developmental outcome. Another issue pertains to the significant number of par-
Our study addressed the specific topic of cognitive delay in ticipants with autistic features in our subject population. It
children meriting a concurrent diagnosis of GDD. Our aims may be that children with autistic spectrum disorders are
were twofold: (1) to explore the relationship between initial less amenable to valid developmental testing as toddlers,
language and fine motor scores and cognitive performance; increasing the risk of overestimating delay initially. Chil-
and (2) to determine the overall distribution of cognitive dren with autistic spectrum disorder may perform better at
scores in a cohort of young children with confirmed GDD. later cognitive testing, especially with nonverbal cognitive
We found no statistically significant correlation between subcategories. This could be an explanation for the wide
a combination of fine motor, expressive vocabulary, and distribution of IQ scores obtained. It also raises the ques-
receptive language scores and the results of cognitive test- tion whether children with autistic features should be
ing. However, a trend was evident towards a possible cor- consistently excluded from studies involving GDD
relation (p value of 0.06) and thus we would not completely
exclude this combination in the evaluation of possible *UK usage: learning disability.

604 Developmental Medicine & Child Neurology 2009, 51; 600–606

cohorts, as their cognitive profile is likely to differ from
children with GDD alone.
Taken in isolation, expressive vocabulary and fine motor
scores accurately predicted verbal IQ scores and perfor-
mance IQ scores, respectively. This further supports the
validity and inference of the expressive vocabulary and fine
motor scales that we used. Since cognitive testing relies
heavily on language-associated tasks, it is not surprising to
find a significant association between expressive vocabulary
deficits and global IQ scores. This has interesting implica-
tions for prognosis in GDD. It may be that children with
GDD with higher expressive vocabulary scores are more
likely to have a favorable cognitive outcome. This remains
to be studied further in a systematic prospective fashion.
Fine motor performance was also predictive of global IQ,
which probably relates to the important overlap between
fine motor performance and cognition in the pre-school
child as assessed by present developmental assessments.
Surprisingly, we found no correlation between receptive
language scores and verbal IQ, which contradicts the gen-
eral assumption that language comprehension predicts
cognition, specifically verbal IQ. This could be partly
related to the scales used for receptive language assess-
ment. However, this is unlikely as both the Reynell and the
Clinical Evaluation of Language Fundamentals – Pre-
school 2 (CELF) tests have been validated for assessment
of receptive and expressive language in a population of pre-
school children. However, it may be that the receptive lan-
guage function assessed through these scales is not directly
implicated in verbal IQ function, a hypothesis that has
never been explored. Thus, verbal IQ may depend on
aspects of cognition other than language comprehension
alone and could not be predicted with these scales.
Inherent to our study was the possibility of variations
in the status of delay in our participants between the
initial evaluation and slightly later cognitive testing.
Some children may have improved significantly, while
others may have plateaued or worsened (i.e. gap widen-
ing) relative to age-equivalent norms. Since our aim was
to determine the nosologic value of our initial diagnosis,
we do not consider this variation as a bias, but as evi-
dence of the wide variety of developmental profiles of
children with GDD. A corollary to this is the possibility
of the child’s benefiting from rehabilitation resources
between the initial assessment and cognitive evaluation.
This is likely to have had no impact on the overall
results, because of the lengthy average waiting time for
rehabilitation resources in our community (most chil-
dren would have been assessed cognitively before any
actual rehabilitation intervention being provided).
One limitation of the study is its retrospective design.
However, we believe that the overall structure of the
Developmental Progress Clinic, the clinical source of all
our study participants, limits potential biases, as all patients
undergo the same standardized evaluations, recorded in a
predetermined uniform format. We reviewed the charts of
all participants presenting to the Developmental Progress
Clinic within a selected defined time interval. A review of
the 47 children with GDD not analyzed because of lack of
cognitive assessment revealed comparable demographics
and developmental profiles with our study group: hence,
potential selection biases are likely to be limited.
Most of the children assessed had no identifiable cause
for their GDD and few had associated medical conditions.
This is probably representative of the majority of patients
seen in developmental pediatrics and community pediatrics
settings. Locally, children with associated neurologic
impairment appear to be preferentially referred to hospi-
tal-based child neurology clinics. Patients with obvious
causes for delay or significant associated medical condi-
tions may have very different developmental and cognitive
profiles, with a different referral pattern (e.g. child neuro-
logy clinic evaluation), which we could not assess because
of the nature of recruitment in this particular study.
GDD remains a symptom complex with an as yet un-
defined nosologic profile and implications. Our study
found no correlation between a combination of develop-
mental scores and cognitive scores in children with GDD.
However, fine motor and expressive vocabulary scores
taken in isolation were predictive of global cognitive
scores, which could have important implications for accu-
rate diagnosis and eventual prognosis.
A previous study has shown persistence of develop-
mental and functional impairments at age 7 in children
diagnosed with GDD as young children.27 According to
recent consensus, mental retardation ⁄ intellectual disability
is defined as significantly sub-average functioning existing
concurrently with related limitations in at least two appli-
cable adaptive skill areas.10 Adaptive skills were not
assessed in our cohort; we cannot, therefore, comment on
the proportion of our participants meeting current criteria
for diagnosing mental retardation ⁄ intellectual disability.
Even so, we believe our results shed some doubts on the
validity of the assumption of GDD–mental retarda-
tion ⁄ intellectual disability equivalence.
Prediction of outcome of GDD should take into consid-
eration intellectual performance, developmental perfor-
mance, and, most importantly, functional performance.
The current definition of GDD likely needs to be revisited,
to better embrace the now well-established variety of pro-
files of children with several areas of delay apparent at a
young age and an apparent impairment dichotomy in
children with GDD with reference to actual associated
cognitive profile.
Global Developmental Delay and Cognitive Skills Emilie M Riou et al. 605
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