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J Autism Dev Disord (2008) 38:362372

DOI 10.1007/s10803-007-0403-3

Diagnosing Autism Spectrum Disorders in Pre-school Children


Using Two Standardised Assessment Instruments: The ADI-R and
the ADOS
Ann Le Couteur Gyles Haden Donna Hammal
Helen McConachie

Published online: 29 June 2007


 Springer Science+Business Media, LLC 2007

Abstract The reliable diagnosis of Autism/Autism Introduction


Spectrum Disorder in pre-school children is important for
access to early intervention and for accurate ascertainment Autism is a neurobiological condition, where children
for research. This paper explores the combined use of two experience life-long pervasive difficulties with social
standardised assessment instrumentsthe Autism Diag- interaction and communication, and demonstrate restricted
nostic Interview Revised (ADI-R) and the Autism Diag- and repetitive behaviours. The broader spectrum around
nostic Observation Schedule (ADOS)in a large sample Autism includes individuals across a range of severities,
of pre-school children. The children were recruited to language and intellectual abilities. The prevalence of
research studies, and a best estimate clinical diagnosis Autism Spectrum Disorders (ASD) has been reported to be
reached. The findings show good agreement between the at least 60 per 10,000 children under 6 years of age (e.g.
instruments especially for children with core Autism. The Chakrabarti & Fombonne, 2001, 2005).
instruments appear to have a complementary effect in To make a diagnosis of Autism/ASD according to ICD-
aiding diagnosis and confirm the importance of a multi- 10/DSM-IV-TR (American Psychiatric Association, 2004)
disciplinary assessment process with access to information requires a multidisciplinary assessment procedure that
from different sources and settings. The presence of includes a detailed developmental history and description of
repetitive behaviours during the ADOS appeared of diag- current behaviours, assessment of cognitive and language
nostic significance. abilities, and observations of functioning in a variety of
settings (Le Couteur, 2003; Ozonoff, Goodlin-Jones, &
Keywords Clinical diagnosis  Repetitive behaviours  Solomon, 2005). The information then needs to be combined
Parent report into a consensus opinion. This process can involve the use of
standardised instruments in combination with clinical judg-
ment, usually of at least two or more professionals with ASD
expertise (Baird et al., 2006; de Bildt et al., 2004). Two of
the most widely used diagnostic instruments used for this
purpose are the Autism Diagnostic Interview-Revised (ADI-
R) (Le Couteur, Lord, & Rutter, 2003; Lord, Rutter, & Le
Couteur, 1994) and Autism Diagnostic Observation Sche-
A. Le Couteur (&)  D. Hammal  H. McConachie dule (ADOS) (Lord et al., 2000; Lord, Rutter, Di Lavore, &
School of Clinical Medical Sciences (Child Health), University
Risi, 2001). The ADI-R is an investigator-based interview
of Newcastle, Sir James Spence Institute, Royal Victoria
Infirmary, Queen Victoria Road, Newcastle upon Tyne NE1 designed to provide a framework for the developmental
4LP, UK history for a lifetime differential diagnosis of Pervasive
e-mail: A.S.le-Couteur@newcastle.ac.uk Developmental Disorders and information about current
functioning for individuals (with a mental age of 2 years or
G. Haden
North Durham Transitions Service, Chester Le Street Health above) from early childhood to adult life (American Psy-
Center, Chester Le Street, Durham DH2 3UR, UK chiatric Association, 1994; World Health Organisation,

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J Autism Dev Disord (2008) 38:362372 363

1992). It was originally developed to form part of a multi- tools generally recommended for autism research
disciplinary research based diagnostic assessment in com- (Tanguay, 2000). Despite this, the levels of agreement
bination with the ADOS (see methods section for more between these two assessment tools have only recently
comprehensive description of both these measures). Cur- been considered. A study of 611-year olds (Bishop &
rently the ADI-R provides a summary diagnostic algorithm Norbury, 2002) found that diagnostic categorisation
that distinguishes between Autism and not Autism. The reached by the ADI-R and the Social Communication
ADOS is a play and activity based assessment that provides, Questionnaire (SCQ) (Berument, Rutter, Lord, Pickles, &
through the specification of social presses, standard con- Bailey, 1999) showed good agreement; however agreement
texts for observation of aspects of social behaviour, com- between these tools and the ADOS was less satisfactory.
munication, play and restricted and repetitive behaviours in Noterdaeme, Mildenberger, Sitter, and Amorosa (2002)
individuals (across the ability range) suspected of having a found largely accurate classification of school-age children
possible ASD. There are four different modules for use with with a diagnosis of autism or receptive language disorder,
children or adults of different developmental and language using the ADI-R and ADOS. Discrepancies noted were
levels from no expressive or receptive language through to attributed to factors such as parents under-reporting of
verbally fluent individuals. Each module takes about 30 early difficulties. Recently, Mazefsky and Oswald (2006)
45 min to administer. The ADOS summary diagnostic have examined the diagnostic utility of these two measures
algorithm distinguishes between Autism, ASD and not ASD. in a heterogeneous clinical sample of children (aged from
For a diagnosis of core Autism according to ICD-10/ 22 months to 8 years) referred to a developmental disor-
DSM-IV-TR, there needs to be evidence of delay or ders assessment clinic. The ADI-R and ADOS algorithm
deviation within the first 36 months of life (WHO, 1992). classifications had ~75% agreement with the team diag-
However, parents often suspect difficulties earlier, the noses. Discrepancies were usually false positives on the
average age of first concern being reported as ~19 months two measures. The paper did not report any direct com-
(De Giacomo & Fombonne, 1998; Gray & Tonge, 2001). parisons between the two instruments. For children and
The reported early symptoms of Autism include a lack of adolescents with intellectual disability, de Bildt et al.
joint attention (Charman, 2003), a failure to develop spo- (2004) report that the two instruments provide valuable
ken language (McConachie, Le Couteur, & Honey, 2005) information on social, communicative and stereotyped
and a failure to respond to name (Baranek, 1999). behaviours especially in individuals with mild or moderate
Despite the early features of this disorder becoming intellectual impairment. The authors conclude that,
better recognized, delays in diagnosis still occur, for several although overall the agreement between ADI-R and ADOS
possible reasons. The particular criteria needed for diag- is fair (kappa of 0.67 for autism in 58-year olds but 0.16
nosis may not be apparent at very young ages, the range of for study subjects older than 8 years), the combination of
normal variation is great, and symptoms may change or both instruments is the best way to measure PDD. How-
only appear infrequently. There are only a limited range of ever, they caution both that the diagnosis of PDD remains
assessment tools targeted at the pre-school age range (as difficult in very low-functioning children, and that overall
opposed to screening instruments), potentially impeding the algorithm cut-off scores for both instruments should not
assessment, and a lack of local community based specia- be used as absolute criteria. This observation is in keeping
lised professionals and/or resources may delay the pro- with Robertson, Tanguay, LEcuyer, Sims, and Waltrips
gression of diagnosis (Gray & Tonge, 2001). Despite these (1999) observation that the ADI-R and ADOS measure
potential difficulties, diagnoses in the Autism spectrum slightly different aspects of manifestations of disorder and
made in the pre-school period have been shown to be reli- that the combination provides a conservative approach to
able (Charman & Baird, 2002; Cox et al., 1999; Gillberg, diagnosis. Finally, few studies have considered the use of
Ehlers, Schaumann, & Jakobsson, 1990; Lord, 1995; Moore the two instruments in young children. Ventola et al.
& Goodson 2003; Stone et al., 1999; Volkmar, Charwarska, (2006) considered the efficiency of ADOS and ADI-R, and
& Klin, 2005), and there is emerging evidence that there is found that children screened and assessed at mean age
much to be gained from an early diagnosis, in order to 22 months were unlikely to score above cut-off on the
maximize the benefits of early intervention strategies ADI-R repetitive behaviours domain, despite receiving a
(Bryson, Rogers, & Fombonne, 2003). clinical diagnosis of autism. Given the increasing numbers
of toddlers and young pre-schoolers presenting for diag-
nostic evaluation, it is important to explore the utility of the
Aims of the Paper ADI-R and ADOS in clinical diagnosis.
There were two aims for this study. The first was to
The ADI-R and ADOS were developed to be used in investigate the agreement between the ADI-R and ADOS
combination to contribute to a valid diagnosis, and are the domain scores and the overall diagnostic categorisation

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for autism or ASD in a well defined cohort of pre-school Diagnostic Assessment Tools
children. The second was to consider how a BECD could
be reached when the diagnostic algorithm cut off scores The ADI-R is an investigator-based semi-structured diag-
for these two standardised instruments do not apparently nostic interview which was designed to provide a devel-
agree. opmental history framework for a lifetime differential
diagnosis of Pervasive Developmental Disorders and
information about current functioning (over previous
Methods 3 months), for individuals with a mental age of 2 years or
above) from early childhood to adult life. It is usually
Study Sample undertaken with the parents/main carer of the child, has
111 questions and usually takes about 23 hours to com-
One hundred and one pre-school children aged 24 plete. Interviewers are intensively trained in its adminis-
49 months were assessed with both the ADI-R and ADOS. tration, with inter-rater reliability on individual algorithm
The cohort was recruited from two previous studies, one an items ranging from r = 0.63 to 0.89. Most items are scored
evaluation of a group parent training intervention, and the from zero (no impairment with respect to the behavioural
other a study of the relationship between executive func- definition for each item) to three (severe impairment for the
tion and autistic symptomatology in very young children individual and their family), relying on the interviewer to
(McConachie et al., 2005; Shearer, 2001). The Northern make judgements on the childs behaviour based on the
and Yorkshire Multi-Centre Regional Ethical Committee recall of information from parents/carers. The scoring
and all relevant Local Ethics Committees approved both algorithms generated draw on items relating to social
studies. Informed consent was obtained from parents for interaction, communication and repetitive behaviours, with
each study. All children were initially identified from a scoring cut-off for Autism. Scores are transformed fol-
within the North East of England by local speech and lowing the protocol in the manual (e.g. 3s become 2s).
language therapists and paediatricians as having speech or Lord et al. (1994) reported internal consistency (alpha
communication difficulties, or suspected ASD. At the time coefficients) for the domains from 0.69 to 0.95. The ADI-R
of recruitment, not all had a firm clinical diagnosis (Honey, algorithm diagnosis is not a clinical diagnosis but the result
McConachie, Randle, Shearer, & Le Couteur, 2006). All of of combining the coded information from the interview. To
those for whom the question of autism had been raised had date there is no algorithm cut-off within the ADI-R for
either recently concluded or were still undergoing diag- ASD, and the published interview (Le Couteur et al., 2003)
nostic assessment. has not been modified for use with young pre-school
A best-estimate clinical diagnosis (BECD) was made children. For this study of pre-school children the current
by the senior authors (ALC, HM) based on all available behaviour scores were used for the algorithm.
clinical information across settings, along with the ADI-R, The ADOS is a standardized semi-structured observa-
ADOS and all other research assessment information (as tional play and activity assessment of the child, and usually
reported in McConachie et al., 2005). This procedure is in lasts about 40 min. As with the ADI-R, intensive training
line with accepted best practice for research assessments in the administration of the ADOS is required. Inter-rater
(Dunn, 2000). The senior authors are clinicians in the reliability of items is good (j 0.6). The exception is the
Regional Childrens PDD service, and thus had access to coding of some items such as some repetitive behaviours
additional clinical information and reports about many of and sensory abnormalities (Lord et al., 2000). The module
the children. Where there was uncertainty, the ADI-R for administration is chosen according to the develop-
interview schedule was consulted and the ADOS assess- mental and language level of the child. Modules 1 and 2 are
ment videotape watched to reach a consensus judgement. appropriate for young pre-school children. There are ten
Forty-nine children were grouped as showing Autism sets of materials and play activities in Module one appro-
where they met all ICD-10 criteria for a corresponding priate for children with no speech or single words, and 14
diagnosis, and 28 were classified as having an ASD activities in Module two suitable for more fluent young
where the diagnosis was more likely to be under another children with phrase speech, from which around 30
heading of pervasive developmental disorder (PDD-NOS, behaviours are coded on a 3 or 4 point scale (as for the
APA, 1994). All children had significant social, commu- ADI-R). Selected algorithm items relating to social inter-
nication or behaviour abnormalities identified in their action and communication are then entered into an algo-
development before 36 months of age. Children defined as rithm. These scores are transformed following the protocol
Other had all been clinically referred for specific speech in the manual (e.g. 3s become 2s) for the diagnostic
or language difficulties, and did not have social commu- algorithms. To obtain an ADOS classification of Autism or
nication difficulties consistent with an ASD diagnosis (24). ASD, an individuals scores must meet the separate

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cut-offs for both the communication and social domains recorded as before 36 months and if two of the three
and the cut-off for the summation of the two. Repetitive domain cut-offs (social interaction, communication or
behaviours are recorded and coded as part of the clinical repetitive behaviours) were reached, an ASD study
observations but do not contribute to the ADOS summary descriptor was applied, as has been used in a previous study
algorithm. Internal consistency is high: Alpha coefficients (Bishop & Norbury, 2002). Agreement with the two
are 0.860.91 for the social domain (across modules), diagnostic tools was examined separately in relation to the
0.740.84 for communication, and 0.630.65 for repetitive three categories of BECD.
behaviours (modules 1 and 2) (Lord et al., 2000).

Assessment of Ability Results

The Mullen Scales of Early Learning (Mullen, 1995) are a Table 1 shows data on age, gender, age equivalents for
standardised assessment of young childrens ability, used three Mullen scales (Visual Reception, Receptive and
in many studies of children with ASD (Lord et al., 1994). Expressive Language) together with the profile of scores
Age equivalents are reported for Visual Reception (a non- from the ADI-R and ADOS for the 101 study subjects. All
verbal scale), Receptive Language and Expressive Lan- subjects have been grouped according to the best-estimate
guage (McConachie, Randle, Hammal, & Le Couteur, clinical diagnosis (BECD) of Autism, ASD or Other (i.e. a
2005). non-ASD diagnosis). All but two subjects received Module
1 of the ADOS.
Procedure The social interaction total scores in the ADI-R and
ADOS had a correlation coefficient of r = 0.71, indicating
Children were usually seen in their own homes. The that as the total social domain score in the ADI-R increased
informant(s) for the ADI-R was one or both of the childs the total social interaction score in the ADOS was also
biological parent(s), usually the mother. All the assess- likely to increase. The correlation between the communi-
ments (apart from 3 of the ADOS assessments) used in this cation domains was also relatively strong, with a coeffi-
study were undertaken by one of two research associates cient of r = 0.64. There was a weaker association between
trained to the accepted standards (Le Couteur et al., 2003; the ADI-R and ADOS repetitive behaviour scores with a
Lord et al, 2000) and blind to other clinical information correlation coefficient of r = 0.51.
about the children. The ADOS was video-recorded for later The levels of agreement between the ADI-R and ADOS
rating. Throughout both previous studies regular inter-rater (Table 2) for above both the social and the communication
reliability checks were made in joint group coding sessions. algorithm cut-offs scores for Autism are moderate. The
Both research associates maintained a consistent minimum kappa level for a best estimate diagnosis of Autism is good,
level of 75% agreement across all ADOS items and scale but moderate for a spectrum diagnosis.
points. In order to explore the relationship between BECD and
the instrument classifications, the distribution of subjects
Statistical Analysis according to their diagnosis on the different instruments is
presented in Table 3, and illustrative individuals presented
In order to examine the degree of agreement, correlations in Tables 4 and 5.
were calculated between the ADI-R and ADOS social
interaction and communication algorithm domain scores Best Estimate Clinical Diagnosis of Autism
using Pearsons correlation coefficients. The total score for
the four ADOS items relating to repetitive behaviours was The joint agreement of the ADI-R and ADOS with a BECD
correlated with the repetitive behaviours domain score of Autism was 67% (33/49). Six children met criteria on
from the ADI-R algorithm. the ADI-R but not the ADOS. However, of these, five did
The levels of agreement between the ADI-R and ADOS meet criteria for ASD on the ADOS. The sixth child met
were also compared (by calculating Kappa statistics; criteria for ASD separately for communication (2) and
Cohen, 1988) with respect to overall diagnostic categori- social interaction (4); however, the joint algorithm cut-off
sation of Autism, and when looking at attainment of cut-off score is 7, which is why the child appears below spectrum
for each domain of the algorithm. cut-off on ADOS on Table 3 (marked a).
In order to incorporate the ADI-R into contributing to Eight children met the criteria for Autism on the ADOS
the diagnostic category of ASD, a revised use of the ADI-R but not on all domains of the ADI-R. The one child
algorithm was employed, i.e. if the age of onset was (Table 3, marked b) who appears below spectrum cut-off

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Table 1 Sample characteristics


BECDb
by BECD group
Autism (n = 49) ASD (n = 28) Other (n = 24)

Gender 41 M/8 F 22 M/6 F 18 M/6 F


AGE (mths) 35.6 (7.0) 37.8 (6.0) 37.6 (5.7)
MULLEN Visual reception 21.5 (7.6) 25.9 (7.7) 30.9 (7.2)
Age equiv (mths) Receptive language 15.8 (8.7) 22.2 (6.5) 28.5 (6.1)
Expressive language 16.2 (8.9) 19.8 (6.9) 24.7 (6.9)
ADI-R Social 17.5 (4.6) 7.9 (5.2) 2.8 (3.5)
Mean (SD) Communication 11.1 (2.8) 6.2 (3.3) 3.5 (3.0)
Repetitive 5.1 (1.4) 3.7 (2.2) 1.3 (1.3)
Totala 33.7 (6.6) 17.8 (7.8) 7.6 (5.5)
a ADOS Social 10.4 (2.7) 4.7 (3.3) 0.3 (0.6)
Social, communication and
repetitive behaviours Mean (SD) Communication 5.6 (1.5) 2.4 (1.9) 0.7 (0.9)
b
There are significant Repetitive 3.8 (1.4) 3.3 (1.3) 0.6 (0.8)
differences between the BECD Totala 19.8 (4.2) 10.4 (5.2) 1.6 (1.7)
groups for all assessment scores

Table 2 Levels of agreement and kappa statistics between the ADI- Subject 2 had age appropriate language skills, though
R and ADOS Subject 1 used less language on formal testing than reported
Outcome Agreement ADI-R Kappa by parents. Both children used language effectively for
versus ADOS (%) statistic communication and to establish joint attention at the time of
assessment, which may have affected how parents reported
Autism social interaction cut- 78 0.56
their skills. The BECD of Autism was arrived at because of
off
the degree of impairment shown by the children in everyday
Autism communication cut-off 74 0.48
settings such as playgroup or nursery.
Above Autism cut-off/below 81 0.62
Autism cut-off In summary, the agreement between ADI-R and ADOS
Above spectrum cut-off/below 78 0.54 ratings, and a BECD of Autism appeared generally sound
spectrum cut-off and for those individuals where there were differences
between the BECD and the instruments classifications, the
differences in scores were marginal and there was consis-
on the ADI-R was below on both social interaction and tent clinical evidence of difficulties across a range of set-
repetitive behaviours by only one point. tings and over time.
Only two subjects were found to score below criteria for
Autism on both the ADI-R and ADOS when the BECD Best Estimate Clinical Diagnosis of ASD
indicated a diagnosis of Autism. However, both of these had
scores above cut-off for ASD on the ADOS, and had scores The joint agreement of the ADI-R and ADOS with a BECD
above cut-off on Social Interaction and on Repetitive of ASD was only 14% (4/28). Four children met criteria for
Behaviours on ADI-R (Table 3, marked c; see Table 4). Autism on the ADOS but were not above cut-off on the

Table 3 Diagnosis based on


BECD ADI-R ADOS
BECD, ADI-R and ADOS
Below spectrum cut-off Above ASD cut-off Above Autism cut-off

Autism Below spectrum cut-off 0 0 1b


c
Above ASD cut-off 0 2 7
Above autism cut-off 1a 5 33
a, b, c, d, e, f, g = see text for ASD Below spectrum cut-off 6f 8 4d
explanation Above ASD cut-off 5 4 0
c = see also Table 4 Above autism cut-off 1e 0 0
d = see also Table 5 Other Below spectrum cut-off 22 0 0
Bold text indicates the expected Above ASD cut-off 2g 0 0
area of joint agreement for each Above autism cut-off 0 0 0
BECD

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Table 4 Vignettes of two children with a best estimate clinical diagnosis of Autism, but not meeting criteria on all domains of ADI-R and
ADOS
ADI-R domain scoresa Subject 1 scores Subject 2 scores

Communication (8) 5 4
First word apple at 22.5 years. Currently uses a Single words from age 10 months. Currently uses a
few two-word and three-word phrases. Attempts wide vocabulary and is able to point successfully
pointing to express interest. No pretend play to express interest. Said to have a fabulous
imagination
Social interaction (10) 10 13
No eye-contact until 3 years 3 months. Finds Described as affectionate but does not like to mix
everything funny. Sometimes mixes with others. with other children. Continues to have difficulty
Prefers older children, especially girls with direct eye contact. Tantrums if he cannot do
as he wants
Repetitive behaviours (3) 4 6
Upset by one particular advert on TV, but now Shows an unusual fear of hats. Is very sensitive to
watches warily. Repetitive drumming behaviour noise and puts his hands over his ears.
on all flat surfaces Compulsively touches objects in his surroundings
ADOS
Communication (4) 3 2
Babbled jargon to mother, with gestures. A few Used lots of appropriate language
single words
Social interaction (7) 7 7
Made lots of requests. Eye contact not well Showed some appropriate eye contact but very
integrated but did initiate joint attention active and easily distressed
Socialcommunication (12) 10 9
Repetitive behaviours 3 2
Distracted during ADOS by repetitive interest in Hands nearly always moving
videos on shelf
Mullen scales of early learning
Chronological age 3 years 6 months 3 years 10 months
Visual reception age equiv. 1 year 10 months 3 years 5 months
Receptive Language age eq. 1 year 6 months 3 years 11 months
Expressive Language age eq 1 year 3 months 3 years 0 month
Notes Attends specialist language unit. Parents first Attends mainstream school. Teachers describe
concerned when he was 22.5 years because of difficulties coping with his behaviour especially
slow development of speech during lunch break.
Parents first concerned at 9 months because of
constant crying and lack of interest in toys
a
Autism cut-off score in brackets

ADI-R (Table 3, marked d; see Table 5). Three attended a partial explanation for the ADI scores not meeting
mainstream school and one a specialist language class. algorithm cut-off in any domain. For subject 4, the ADI-R
Subject 3 illustrates the complexity of the diagnostic pro- ratings may again represent under-reporting by parents;
cess, and clinical judgements to be made. During the their child was only 2 years old at the time of the assess-
ADOS, carried out at home, he repeatedly requested that ment, and they stated the belief that his lack of spoken
the television be switched back on. This fixed idea may language is his only problem. Subjects 5 and 6 are both
have increased (i.e. made more abnormal) the scoring of girls. For both families the early social developmental
the ADOS on social interaction on that occasion (when histories did not alert professionals to the girls complex
assessed again 4 months later, his socialcommunication developmental difficulties. Subject 5 was described by her
algorithm score was below the Autism cut-off and above parents as placid and compliant whilst Subject 6 showed
the ASD cut-off, in line with his BECD). He is an only definite behavioural difficulties described as severe temper
child of relatively elderly parents, and described by them tantrums. For both these girls the diagnostic picture became
as a happy lad with no behaviour problems. His easy clearer over the next year, having perhaps been masked by
temperament and his parents lack of comparison may offer their particular temperamental characteristics.

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Table 5 Vignettes of four children with a best estimate clinical diagnosis of ASD, but meeting criteria for Autism on ADOS
368

ADI-R domain Subject 3 scores Subject 4 scores Subject 5 scores Subject 6 scores
scoresa

123
Communication 3 5 6 6
(8) Despite using single words from age Had single words from age 10 months, Placid child. Delayed use of single No initial speech delay but limited use.
12 months he then lost vocabulary of over then stopped using these from words. Phrase speech Tends to be demanding with odd
20 words. Useful speech returned aged 20 months. Uses words now and again 39 months.No protodeclarative intonation and repetitive questions &
3 years. At age 2 years was able to point pointing, few gestures.unable to statements
to express interest nod head for yes
Social interaction 5 4 5 7
(10) Has always been able to use brief eye Eye contact and social smiling Cheerful demeanour. Brief eye Always on the go. Difficult with other
contact appropriate. Shy but interested in other contact but no understanding of children. Parents report behaviour
children others distress difficulties and temper tantrums
Repetitive 2 2 2 4
behaviours (3) Some mild unusual sensory interests Difficulties with changes to routine at
and brief hand mannerisms home and with routes to school/shops.
reported Some complex whole body
mannerisms
ADOS domain scoresb
Communication 6 6 6 4
(2) Speech consisted mainly of babbled phrases Few vocalisations, no pointing or Used single words but not clearly Little spontaneous speech and poor co-
and echoing gestures directed. Intonation showed little operation- tends to try to refuse
variation. Pushed adults hand to activities
object
Social interaction 8 10 8 10
(4) Showed occasional eye-contact Difficult to get eye contact. Gave objects Limited eye contact. No requests, Put Tantrums and disruptive repetitive
but did not request her finger in her ears but compliant behaviour interfered with tasks
Social- 14 16 14
communication 14
(7)
Repetitive 5 3 4 4
behaviours Showed overreaction to sounds and Held on to one toy throughout Definite hand mannerisms and some Resistance to change and other repetitive
repeatedly requested TV on sensory exploration of toys interests affected task performance in
addition to poor concentration
Notes Boy aged 4 years 1 month. Attends Boy aged 2 years 8 months. Attends Girl aged 3 years 3 months. Attends Girl aged 3 years 0 months. Attends
mainstream school with support teacher. specialist language group at Child mainstream school with full-time mainstream school with support. No
Parents first concerned when he was over Development Centre. Parents first support. Parents concerned at speech delay; parents concerned about
2 years of age because of loss of speech concerned around 20 months of age 18 months because of delay in unco-operative behaviour and lack of
because of loss of speech speech and walking social use of speech
a
Autism cut-off score in brackets
b
ASD cut-off score in brackets
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J Autism Dev Disord (2008) 38:362372 369

One child (Table 3, marked e) showed markedly dis- demonstrated a similar level of agreement between the
crepant ADI-R and ADOS ratings. The assessor com- instruments in pre-school children to that reported in
mented that he was somewhat of a contradiction. During school aged children for autism (7475%: Bishop & Nor-
the ADOS he seemed able to engage and to be social, but bury, 2002; Conti-Ramsden, Simkin, & Botting, 2006; de
without a definite social press he did not take any ini- Bildt et al., 2004), The correlation co-efficient for total
tiative. Social difficulties were reported in a variety of domain scores across the two instruments was greatest for
situations and thus scored on the ADI-R items. In addition the social domain. For these young children the develop-
his ADOS repetitive behaviour score was 3. When assessed mental history would have been relatively fresh for parents,
1 year later, he met the ADOS criterion for autism on so- rather than having to recall information about the past, as
cial interaction, for ASD on communication, and had a would be the case for parents of older individuals. This
total rating of four on repetitive behaviours. This probably meant that the social behaviours as reported by parents and
means that the interactions with the experimenter during as observed during the ADOS and other assessments were
the ADOS, on the first occasion, were unrepresentative. contemporaneous. Parental report of communication was
There were six children below spectrum cut-off on both also strongly related to rating of observed communication,
ADI-R and ADOS, with a BECD of ASD (Table 3, marked but the association was weaker for repetitive behaviours.
f). It was noticeable that all these children had significant This may have been as a consequence of the limited time
history of repetitive behaviours as reported by parents period for observation during the administration of the
during the ADI-R developmental history and as rated on ADOS, or that the algorithm threshold for repetitive
ADOS (2 or more), and/or an ADI-R social interaction behaviours in young pre-school children may need to be
score of 8 or 9, i.e. just below the cut-off score of 10. In one reviewed (Ventola et al., 2006).
case the clinical diagnosis had been influenced strongly by Further, the findings demonstrated that the ADI-R and
additional observations made in a nursery setting, where ADOS have a complementary effect in aiding diagnosis, i.e.
the childs social difficulties and self-absorbed and ste- when their results were taken together they provided a
reotyped behaviours were particularly evident. greater level of diagnostic clarity but used as a single
In summary, there is evidence that some of the apparent assessment instrument in isolation each could over or under
disagreement between the instruments and between the two score particular behaviours. This study of pre-school chil-
instrument classifications and BECD arises from scores dren confirms the importance of a multidisciplinary
that are just below cut-off. assessment and the need to consider the developmental
history alongside direct assessments of current level of
Best Estimate Clinical Diagnosis of Other functioning and evidence of co-morbidity. However, there
is a complexity to the diagnostic process especially for the
Those subjects given a BECD of Other all had scores just subthreshold cases. The best agreement between ADI-
below algorithm cut-offs on both instruments, with the R, ADOS and BECD was for the non-ASD or other group.
exception of two, giving a joint agreement of 92%. The two Turning to the disagreement between ADI-R and
children (Table 3, marked g) both scored above cut-off on ADOS algorithm scores: These were greatest for children
two out of three ADI-R domains. with a BECD of ASD. For the 14 children with none or
Finally, we examined the repetitive behaviours scores for only one above algorithm cut-off score for ASD on ADI-R,
the children with BECD of ASD and Other (n = 52). Using and/or ADOS algorithm scores, the clinical information
a cut-off value of 2 or 3 on the ADI-R repetitive behaviours available in addition to the research assessments suggested
domain did not significantly predict a BECD of ASD patterns of three common influences. First, there could
(respectively, v2 = 1.77, p = 0.18; v2 = 1.30, p = 0.25). have been a tendency for some parents/carers to underre-
However, a value of 2 or more for ADOS repetitive port unusual features in communication in young children
behaviours was highly predictive of ASD (v2 = 13.52, with good language skills i.e. useful expressive speech
p = 0.00). That is, all but one of the children with ADOS (Noterdaeme et al., 2002). Second, the clinicians BECD
social/communication scores above ASD cut-off had a was influenced by the findings from the direct assessments
rating of 2 or more on ADOS repetitive behaviours. of a childs skills and difficulties and other clinical obser-
vations in settings such as nursery, thus taking into account
the impact of potentially unpredictable, noisy and
Discussion unstructured social environments upon childrens function
and behaviour. Third, the analysis showed that the presence
The findings from this study of pre-school children of repetitive behaviours observed during the course of the
demonstrated good agreement between the ADI-R and ADOS, contributed to the consideration of an ASD
ADOS, especially in relation to core Autism. The results diagnosis.

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To date, the diagnostic algorithm for the ADOS has not clinical and research practice, with the increasing emphasis
included repetitive behaviours, i.e. it is a composite of on early detection and early intervention, and as the diag-
communication and social interaction ratings. This was nostic criteria have broadened from a narrow definition of
because the assessment is a snapshot in time, and repetitive autism to the broader Autism spectrum, it is important that
behaviours may not be triggered in a one-to-one play ses- a clear assessment framework is used. Standardised struc-
sion with an attentive adult. However, the examples pre- tured instruments (interviews and observations) have both
sented in this paper suggest that where repetitive advantages and limitations when used in clinical practice.
behaviours are noted, they may be considered significant. However, one of the principal advantages is the opportu-
This recommendation is consistent with the recently pub- nity to gather clinically relevant information in a system-
lished revised ADOS algorithms (Gotham, Risi, Pickles, & atic and comparable fashion. For example, change over
Lord, 2006). Further, with respect to the ADI-R our find- time is hard to predict, but where a query of possible ASD
ings concur with other recent publications (Mazefsky & is raised in a child with language delay, there is good
Oswald, 2006; Risi et al., 2006) that further research is evidence that the characteristics may become clearer in
needed, to address the potential use of the ADI-R for middle childhood (Michelotti, Charman, Slonims, & Baird,
identifying ASD and to generate joint revised algorithms 2002; Miniscalco, Nygren, Hagberg, Kadesjo, & Gillberg,
for pre-school children and across the lifespan. 2006). Therefore, in complex cases, especially where there
is diagnostic uncertainty, detailed recording of behaviour
Limitations with specific instruments such as ADI-R and ADOS is
valuable in the diagnostic process.
This cohort has been carefully described in previous papers
(Honey et al., 2006; McConachie et al., 2005). The chil- Acknowledgments We thank the parents and children who took
part in the research studies, and the local district clinicians and col-
dren were recruited to two research studies and as such
leagues for supporting the studies. We also thank Nicola Burton for
may not be representative of all pre-school children with her help with the manuscript.
suspected ASD from the local population. The research
group may for example be more advantaged economically
than the families that declined to take part. In the light of References
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