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Sensory and Motor Behaviors of Infant Siblings
of Children With and Without Autism
Shelley Mulligan, Barbara Prudhomme White
KEY WORDS
 autistic disorder
 early diagnosis
 motor activity
 sensation
 siblings
Shelley Mulligan, PhD, OTR/L, is Associate Professor
and Chair, Department of Occupational Therapy, Hewitt
Hall, University of New Hampshire, Durham, NH 03824;
shelley.mulligan@unh.edu
Barbara Prudhomme White, PhD, OTR/L, is
Associate Professor, Department of Occupational Therapy,
University of New Hampshire, Durham.
We compared the sensory and motor behaviors of typically developing infants with those of infant siblings of
children with autism spectrum disorders (ASD), who are considered high risk for the disorder, to explore
potential sensory and motor markers for use in early diagnosis of ASD. We compared frequencies of sensory
and motor behaviors during 10-min, videotaped, infant–mother play sessions and during 5 min of spoon-
feeding between groups of 12-mo-old infants. Data from standardized measures of development, sensory
processing, and behaviors commonly associated with ASD were also analyzed descriptively for the
high-risk group. The results indicated that high-risk infants demonstrated fewer movement transitions
(t [23] 5 22.4, p 5 .03) and less object manipulation (t [23] 5 22.4, p 5 .03) than low-risk infants.
The sensory and motor differences found between typical and high-risk infants suggest that early screen-
ings for ASD should include the examination of sensory and motor behaviors.
Mulligan, S.,  White, B. P. (2012). Sensory and motor behaviors of infant siblings of children with and without autism.
American Journal of Occupational Therapy, 66, 556–566. http://dx.doi.org/10.5014/ajot.2012.004077
Autism spectrum disorders (ASD) are developmental disorders characterized by
pervasive deficits in social interaction and communication and a tendency
toward restricted and repetitive behaviors (American Psychiatric Association,
2000). ASD diagnoses have been steadily on the rise, and estimates from the
Centers for Disease Control and Prevention (2012) indicate a prevalence of 1 in
88. Kogan and colleagues (2009) estimated a U.S. ASD prevalence among
3–17-yr-olds of as high as 1.1%. Although the cause remains unknown for most
people with ASD, twin and family studies indicate a strong genetic link.
For children with ASD, early detection is essential for facilitating access
to interventions that can shape neural connections during sensitive periods
of development (Robins, Fein, Barton,  Green, 2001). Early intervention
has been associated with improved developmental and functional outcomes
such as language development, cognitive development, and success at school
(McEachin, Smith,  Lovaas, 1993; Ospina et al., 2008). More specifically for
young children with autism, a combination of play-based and behavioral in-
terventions may be vital in promoting development and adaptive functioning
and minimizing clinical features of the disorder (Dawson, 2008; Johnson 
Myers, 2007).
Unfortunately, children with ASD are often not diagnosed until age 2–4 yr
(Filipek et al., 1999; Ospina et al., 2008; Zwaigenbaum et al., 2007), although
much progress in early detection has been made over the past 5–10 yr. The
American Academy of Pediatrics Council on Children with Disabilities (2006)
recommended that pediatricians incorporate autism screening as part of routine
practice. However, Al-Qabandi, Gorter, and Rosenbaum (2011) reported that
routine screening of all infants may be premature at this time because of a lack
of effective screening tools and evidence-based, efficacious practices and because
of the need for a cost–benefit analysis of conducting universal early screenings.
556 September/October 2012, Volume 66, Number 5
Nonetheless, early identification allows families access to
supports and resources and has the potential to enhance
our understanding of ASD by promoting the study of en-
vironmental, behavioral, and developmental factors that
may influence the course of the disorder.
Retrospective parent interviews (Young, Brewer, 
Pattison, 2003) and studies of home movies of infants
later diagnosed with autism (Baranek, 1999; Osterling 
Dawson, 1994; Werner, Dawson, Osterling,  Dinno,
2000) suggest that abnormalities in development are evi-
dent in the first 2 yr of life. However, the higher incidence
of ASD in infant siblings of children with autism provides
a unique opportunity to look prospectively at the devel-
opment of ASD from infancy and inform us of potential
early signs that may be of diagnostic importance.
Siblings of children with autism have an estimated risk
that is at least 50 times greater than that of the general
population (Liu et al., 2001). Studies using infant siblings
have reported a recurrence risk of 14% (Zwaigenbaum
et al., 2005) to more than 20% (Ozonoff, Young, et al.,
2008; Sullivan et al., 2007; Yirmiya  Charman, 2010).
Several prospective, longitudinal studies of siblings have
been completed or are under way to identify indicators of
ASD in infants and toddlers through ongoing standardized
developmental evaluations and structured observations of
this at-risk population (Bryson et al., 2007; Iverson 
Wozniak, 2007; Ozonoff, Macari, et al., 2008; Yirmiya 
Charman, 2010; Yirmiya et al., 2006; Zwaigenbaum et al.,
2005). Veness et al. (2012), for example, found social
and communication differences between 12-mo-olds with
and without ASD, including the early use of gestures.
Prospective designs enable researchers to examine early
neurodevelopmental signs, behaviors, and developmental
patterns over time under standardized conditions, allowing
greater comparability both within and between individuals
over time. Ultimately, the information can be used in the
development of screening and evaluation tools aimed at
early detection.
Motor disturbances have commonly been noted in
children with autism (Baranek, 2002; Minshew, Sung,
Jones,  Furman, 2004; Molloy, Dietrich,  Bhattacharya,
2003; Provost, Lopez,  Heimerl, 2007; Rinehart,
Bradshaw, Brereton,  Tonge, 2001). Although researchers
have begun to study early motor signs (Loh et al., 2007;
Ozonoff, Young, et al., 2008; Teitelbaum, Teitelbaum,
Nye, Fryman,  Maurer, 1998), few studies have ex-
amined scores from standardized motor assessments in
conjunction with more functional fine and gross motor
behaviors during typical infant activity such as feeding
and play. Studies completed to date have produced in-
consistent results. For example, Teitelbaum et al. (1998)
reported that a sample of infants later diagnosed with
autism showed many abnormal movement patterns,
whereas more recent studies demonstrated no or few
significant motor differences in infants later diagnosed
with autism (Ozonoff, Young, et al., 2008; Provost et al.,
2007). Loh et al. (2007) provided some evidence that
arm waving in the context of standardized test adminis-
tration was more often seen in children who were later
diagnosed with ASD. Motor stereotypies and repetitive
use of objects were found in a sample of high-risk infants
at age 12 mo (Ozonoff, Young, et al., 2008). In a study of
older boys with ASD (6–17 yr), Jansiewicz et al. (2006)
found that children with ASD performed more poorly on
many measures of motor control, including balance and
coordination.
In examining potential early markers for identifying
ASD in infants, Zwaigenbaum and colleagues (2005)
noted that sensory orienting behaviors such as visual at-
tention, orienting to name, visual fixation on certain ob-
jects, and visual tracking may be useful in distinguishing
children with the disorder from typical children. Loh et al.
(2007) found that high-risk infants who were later di-
agnosed with ASD used their hands to cover their ears
(aversive response to sound) in the context of standard-
ized test administration more often than children who
did not have the disorder. Rogers, Hepburn, and Wehner
(2003) found that parents reported sensory differences
in their toddlers who were later diagnosed with ASD.
Baron-Cohen, Ashwin, Ashwin, Tavassoli, and Chakrabarti
(2009) conducted an interesting study examining the
talents of children with autism, concluding that the ex-
cellent attention to detail and pattern recognition abil-
ities of children with the disorder were often rooted in
underlying sensory processing differences such as sensory
hypersensitivities.
In a meta-analysis of 14 studies, Ben-Sasson et al.
(2009) found evidence of significant differences in sen-
sory modulation behaviors between typically developing
children and children with ASD. In an examination of
the sensory modulation patterns of sensory overresponsivity,
sensory underresponsivity, sensory seeking, and total per-
formance, differences were most pronounced from 6–9 yr
of age, with a tendency toward sensory underresponsivity.
More relevant to this study was the finding that children
with ASD birth to age 3 yr were less often categorized
as being sensory seeking than their typically developing
counterparts. In summary, the research indicates that our
understanding of the nature of early infant sensory pro-
cessing and motor development is emerging with this
population and suggests that these are promising areas for
consideration in early screening of ASD.
The American Journal of Occupational Therapy 557
Screening tools such as the Modified Checklist for
Autism in Toddlers (Robins  Dumont-Mathieu, 2006)
emphasize the examination of behaviors associated with
emerging language, social interaction, and play that, if
disrupted, are hallmark features of ASD. Because many of
these types of behavior do not emerge developmentally
until around 12–24 mo of age, however, relying on tools
emphasizing social, language, and play behaviors is lim-
iting at this young age (Baron-Cohen, Allen,  Gillberg,
1992). In contrast, sensory and motor behaviors are easily
captured from birth, and if differences do exist in such
behaviors, they may be able to be detected earlier than
social, play, and language behaviors. Although sensory
and motor behaviors are not core features of ASD, dif-
ferences or abnormalities in sensory processing and motor
skill development have commonly been found, as noted
in the literature described earlier.
Feldman et al. (2012) emphasized the value of ex-
amining child behaviors in the natural context, leading
them to develop the Parent Observation of Early Markers
Scale (POEMS). POEMS includes a large range of pos-
sible early markers or signs of ASD that parents can
observe within the context of their daily routines and
interactions with their child. Feldman et al. found that
differences between typically developing children and
children diagnosed with ASD were more pronounced
at 18–24 mo of age, although subtle differences were
noted as early as 9 mo. The authors concluded that
tools for ongoing surveillance of children by their parents
in natural contexts are helpful additions to performance-
based developmental measures for early detection of
ASD.
The study described in this article examined both
sensory processing and motor behaviors that we thought,
on the basis of prior research, would be useful for the early
detection of autism in 12-mo-old infants at high risk for
developing the disorder. Data collection for this study
included observations and coding of sensory and motor
behaviors within the context of the typical daily routines
of play and feeding via videotape for both typically de-
veloping children and high-risk siblings. Standardized de-
velopmental measures and caregiver questionnaires were
also administered for the high-risk ASD siblings. Details
of these measures are provided in the Methods sec-
tion. The specific aims of this study were to describe the
sensory and motor development and behaviors of infant
siblings of children with ASD at 12 mo of age and to
compare sensory and motor behaviors of high-risk infants
with those of low-risk, typically developing infants at 12 mo
during both a semistructured infant–mother play session
and spoon-feeding.
Method
Research Design
In this study we implemented a two-group exploratory
design to describe and compare data regarding the sensory
and motor behaviors of 13 infant siblings at high risk for
ASD and 12 typically developing infants. The University
of New Hampshire institutional review board for the
protection of human subjects approved the study, and we
obtained informed consent from the parents before data
collection.
Participants
All infants in the study were between age 11 and 13 mo.
Exclusion criteria included known genetic, neuromotor,
or developmental problems or disorders as well as pre-
maturity. High-risk infants were recruited through local
service providers and clinics specializing in young children
and through local organizations for children with special
needs and their families. The high-risk group included 13
infants with a sibling who had a confirmed diagnosis of
ASD determined by a qualified team of professionals that
included a developmental neurologist or pediatrician. All
older siblings were assessed using the Autism Diagnostic
Observation Schedule (Lord, Rutter, DiLavore,  Risi,
2002) either before diagnosis or for confirmation as part
of this study.
We recruited the 12 typically developing infants in the
low-risk group through personal contacts and local day
care and preschool centers. All typically developing infants
had siblings without any developmental concerns. Al-
though the typically developing infants were not formally
tested as part of this study, they were screened by the first
author (Mulligan) and a pediatrician, and only infants for
whom the parents and pediatrician identified no de-
velopmental concerns were recruited to be in the low-risk
group. We made a follow-up phone call to the parents
when the low-risk children reached age 30 mo to verify
that the parents continued to have no concerns regarding
their child’s development.
Procedures
All infants participated with their caregivers in a 10-min
play session and a 5-min feeding session. Each session was
videotaped and subsequently coded for sensory and motor
behaviors. The format of the play and feeding sessions
and the defining and coding of sensory and motor be-
haviors were developed and refined by the primary author
(Mulligan). Sensory and motor behaviors were selected on
the basis of previous research examining potential early
558 September/October 2012, Volume 66, Number 5
markers and on a review of ASD screening tools. Each
sensory and motor behavior was operationally defined and
tested for interrater reliability. In total, we analyzed 16
behaviors for play and 14 behaviors for the feeding session
(see the Appendix for a list of behaviors and definitions).
We used time sampling, recording the presence or absence
of each behavior in 30-s time intervals. To examine in-
terrater reliability, we analyzed data coded from three
trained raters using nine play and feeding sessions. Total
occurrences for each of the 30 variables were tallied and
analyzed using the intraclass correlation coefficient (ICC).
The results yielded an ICC of .97 using average measures,
demonstrating strong consistency across raters.
Infants in the high-risk group were administered
developmental assessments at 11–13 mo of age by the first
author (Mulligan), who was aware that the older sibling
was diagnosed with ASD. Standardized performance-based
measures included the Mullen Scales of Early Learning
(Mullen, 1995) and the Autism Observation Scale for Infants
(AOSI; Bryson, Zwaigenbaum, McDermott, Rombough,
 Brian, 2008; Zwaigenbaum et al., 2005). The stan-
dardized interview format for the Vineland Adaptive
Behavior Scale (VABS; Sparrow, Balla,  Cicchetti, 1984)
was also administered. Test administration followed stan-
dardized protocols and occurred at a university setting with
clinic space and testing rooms for children. In all cases, the
mother was present throughout the testing. A parent
(typically the mother) completed the Infant–Toddler Sen-
sory Profile (Dunn, 2002) before the clinic visit.
The first author and a trained research assistant con-
ducted the play and feeding videotaped sessions in par-
ticipants’ home for both the high-risk and the low-risk
groups. Play sessions occurred in the living room or play
room where the child most often played and included
10 min of the infant playing with the mother on the
floor using a standardized set of common, enticing toys.
The toys included large soft blocks, a mirror, a play
phone, rattles, balls of various sizes and textures, a doll
with a spoon, a bottle and blanket, and a toy hammer–
play structure. The toy hammer–play structure had balls
that, when hammered into holes on the structure, moved
through a maze and out another hole. The mother was
directed to play with her infant and the toys in any way
she liked and in ways that would reflect how the dyad
would typically interact and play with toys. They were
not required to use all the play materials or toys. Feeding
occurred in the family’s kitchen using the child’s high
chair. The mother was asked to support the child in any
way she typically would, and the mother provided a food
item that the child was accustomed to eating (cereal,
applesauce, and yogurt were most common).
Instruments
The VABS assesses adaptive functioning for individuals from
birth to age 90 across four domains—Communication,
Daily Living Skills, Socialization, and Motor Skills—
and provides an overall Adaptive Behavior Composite
score. The VABS was standardized on a national sam-
ple of 3,000 children matched to U.S. census data.
Internal consistency examined by split-half reliability
coefficients is acceptable, ranging from .84 to .88, and
test–retest reliability reported in the manual is also
acceptable, with r values ranging from .78 to .92 for
all areas for the birth to 3 yr age range (Sparrow et al.,
1984).
The Infant–Toddler Sensory Profile (Dunn, 2002) is
a caregiver questionnaire measuring the ability to process
and modulate sensory information, developed with a nor-
mative sample of 589 children from birth to age 36 mo.
The caregiver rates on a scale ranging from 1 to 5 the
frequency with which his or her child exhibits certain
behaviors indicative of sensory processing. Internal con-
sistency estimates across sensory processing sections and
quadrants (representing Low Registration, Sensory Seek-
ing, Sensory Sensitivity, and Sensory Avoiding) varied
with the children aged 7–36 mo and ranged from .42 to
.85, with the strongest values representing the sensory
processing patterns. Test–retest reliability values are ac-
ceptable on the basis of a study of a normative subsample
(n 5 32), with coefficients of .86 for sensory processing
section scores and .74 for quadrant scores. Evidence re-
garding content, construct, and discriminant validity is
also provided in the manual (Dunn, 2002).
The Mullen Scales of Early Learning is a standardized,
norm-referenced, developmental measure for children
from birth to age 68 mo. It was normed on 1,849 children
based on U.S. census data and generates standard
scores for five subscales—Gross Motor, Fine Motor, Vi-
sual Reception, Receptive Language, and Expressive
Language—as well as an Early Learning Composite score.
Split-half internal consistency coefficients are acceptable,
with median values (from the normative data) ranging
from .75 to .91. Test–retest reliability was obtained with
97 children over a 2-wk period and ranged from .71 to
.96 (Mullen, 1995).
The AOSI was developed to assess behaviors specific
to ASD in infants. The clinician administers several play-
based activities to elicit possible behavioral markers for
ASD, such as poor eye contact, lack of orienting to name,
lack of social smiling, deficit in interest in others, and
reactivity to sensory stimuli. The AOSI has demonstrated
strong interrater reliability (.94 with children at 18 mo),
The American Journal of Occupational Therapy 559
fair test–retest reliability (.61 using the total score for
infants 12 mo of age). Predictive validity when admin-
istered at 12 mo has also been found to be strong (Bryson
et al., 2008).
Data Collection and Analysis
Videotaped data from the play and feeding sessions were
transferred to a DVD, and the sessions were viewed to
record the targeted sensory and motor behaviors as being
either present or absent every 30 s. Coding was conducted
by the first author and three research assistants whom
she trained in the use of the measure. Three demonstra-
tion tapes were used for training purposes, and research
assistants achieved 90% agreement with Mulligan across
all cells before coding independently. Training involved
approximately 6 hr of review of the definitions, practice
coding, and discussion with Mulligan to resolve dis-
agreements. The research assistants were blind to group
assignment and coded 70% of the digital recordings;
Mulligan coded the remaining recordings.
The first author or a trained research assistant scored all
standardized tests (i.e., AOSI, Mullen Scales) and parent
questionnaires. Descriptive statistics were used to analyze
data from the standardized developmental measures ad-
ministered to the high-risk infants. One-sample t tests
were used to compare the mean developmental scores of
each of the 13 children in this group with each test’s
normative data to determine whether the group differed
significantly from the normative sample. To examine
sensory and motor differences between the high-risk and
low-risk groups, we coded mean frequencies for each
behavior from the play and feeding sessions and then
compared the groups using independent t tests, with the
a level for determining statistical significance set at .05.
All data were analyzed using PASW Statistics 18, Release
Version 18.0 (SPSS, Inc., Chicago).
Results
The study sample included 8 girls and 5 boys in the high-
risk group (mean age 12.6 mo) and 7 girls and 5 boys in
the low-risk group (mean age 12.1 mo). Most participants
were White, and all were from New England. The groups
did not significantly differ with respect to race, gender, and
age. Follow-up at age 30 mo indicated that four of the
high-risk infants met criteria for ASD, and another two
children had received early intervention services for mild
developmental concerns. None of the children received
early intervention services before 12 mo of age, and none
of the infants placed in the low-risk group exhibited
developmental concerns.
The developmental profile of the high-risk group fell
largely within the low end of the average range, although
some relative strengths and weaknesses are noteworthy. A
summary of these results is presented in Table 1. Fine
motor performance and visual reception skills fell in the
average to above-average range, whereas gross motor skills
were within the low-average range. Test scores measuring
social and communication skills were mixed, although
most fell within the low-average to below-average range.
The number of markers noted using the AOSI indicated
that 31% of the children were at high risk for developing
ASD. Sensory processing differences were also found;
scores from the high-risk group reflected less sensory-
seeking behavior than scores of children from the nor-
mative sample and a tendency for more difficulties in the
area of auditory processing.
The results of the one-sample t tests revealed some
significant differences between the scores of this group
and the normative data for each of the tests. On the
Mullen Scales of Early Learning, scores from the high-
risk group were higher on Visual Reception (t [12] 5 2.1,
p 5 .059), approaching significance, and significantly
higher on the Fine Motor Scale (t [12] 5 3.2, p 5 .009).
Performance of the high-risk group was, however, sig-
nificantly poorer on the Expressive Language (t [12] 5
23.6, p 5 .004) and Receptive Language (t [12] 5
23.7, p 5 .004) scales. On the VABS, the Motor Scale
showed significantly lower scores for the high-risk
group than the normative sample (t [12] 5 22.4, p 5
.03); there were no other significant differences.
Videotaped coding of sensory and motor behaviors
during play and feeding revealed that compared with
infants from the low-risk group, the high-risk group
demonstrated significantly fewer movement transitions
(t [23]5 22.4, p 5 .03) and less object manipulation
(t [23] 5 22.4, p 5 .03; see Table 2). Thus, children in
the high-risk group tended to move around less during
the play session, and they manipulated objects in their
hands less frequently (see definitions in the Appendix).
No significant differences were found between groups on
the other sensory and motor behaviors coded during the
play sessions, and no differences between groups were
noted on the behaviors coded from the feeding sessions.
Discussion
The results suggest that contextual, play-based observa-
tions in conjunction with standardized developmental
measures may have a vital role in the early detection of
ASD. In this study, how the infants used their hands to
manipulate toys in play and how much they moved
560 September/October 2012, Volume 66, Number 5
around during play appeared to be important consid-
erations. Although many of the high-risk infants scored
within the normal range on the standardized motor
measures, during more unstructured play, they used less
sophisticated functional fine and gross motor movements
than would be expected.
An examination of the variability and frequency of the
sensory and motor behaviors that were observed and coded
Table 2. Mean Frequencies of Sensory and Motor Behaviors Coded From Play Sessions
Coded Behavior High-Risk Group, Mean (SD) Low-Risk Group, Mean (SD) p
Motor stereotypy 0.2 (0.6) 0.2 (0.6) .95
Object stereotypy 0.85 (1.1) 1.9 (0.5) .19
Mouthing objects 5.7 (5.3) 3.7 (2.9) .27
Movement transition 7.5 (5.5) 12.3 (4.7) .03*
Orients toward visual stimulus 19.8 (1.3) 17.7 (4.1) .09
Orients toward auditory stimulus 15.5 (3.9) 14.9 (4.7) .75
Orients toward tactile stimulus 9.7 (6.5) 6.3 (4.1) .13
Aversive response to visual stimulus 0.23 (0.6) 0 (0) .20
Aversive response to auditory stimulus 0.46 (1.1) 0.2 (0.9) .61
Aversive response to tactile stimulus 0.61 (1.3) 0.2 (0.4) .25
Object manipulation 9.7 (4.4) 13.4 (3.3) .03*
Motor imitation 3.3 (2.7) 2.5 (2.3) .43
Construction play 0.15 (0.5) 0.3 (0.6) .46
Sensorimotor play 13.4 (6.6) 12.1 (6.7) .62
Functional play 10.8 (5.2) 11.2 (3.7) .83
Associative play 9.0 (6.3) 10.3 (3.6) .55
Note. SD 5 standard deviation.
*p  .05.
Table 1. Developmental Standard Scores of the High-Risk Group (n 5 13)
Test Variable Mean (SD)
% Scoring in the
Dysfunctional Range
Autism Observation Scale for Infants 31% received 7 or more markers, which
has been reported to be predictive of ASD.Total score 8.4 (4)
Number of ASD markers 5.2 (2.3)
Mullen Scales of Early Learning 50 (10)
Gross Motor 46.7 23
Fine Motor 57.1a
15
Visual Reception 55.5a
0
Receptive Language 36.9b
69
Expressive Language 40.8b
46
Early Learning Composite 95.9 15
Vineland Adaptive Behavior Scales 100 (15)
Communication 97.1 23
Daily Living Skills 101.5 0
Socialization 94.8 15
Motor Skills 94.3b
15
Adaptive Behavior Composite 97.8 15
Infant–Toddler Sensory Profile Range within 1 SD
Low Registration 47.4 (46–54) 36
Sensory Seeking 35.0 (19–35)a
72
Sensory Sensitivity 47.6 (41–52) 36
Sensory Avoiding 51.2 (45–56) 45
Low Threshold 98.8 (87–107) 45
Auditory Processing 39.7 (35–43) 54
Visual Processing 24.9 (20–27) 45
Tactile Processing 58.1 (48–61) 45
Vestibular Processing 20.1 (18–23) 36
Oral Sensory Processing 24.8 (21–29) 36
Note. ASD 5 autism spectrum disorder; SD 5 standard deviation.
a
Significantly above the norm. b
Significantly below the norm.
The American Journal of Occupational Therapy 561
during the play sessions provides some insight into which
behaviors may be useful for future consideration related
to screening for ASD. For example, behaviors that were
coded as being absent or not observed in almost all
intervals would not be helpful, such as movement ste-
reotypy; aversive responses to visual, auditory, and tactile
sensory input in both feeding and play sessions; con-
struction play; and motor imitation during feeding. Be-
haviors that were seen a moderate amount of time that
appear more promising as possible early markers in young
infants include object stereotypy; mouthing of objects;
motor imitation during play; and spontaneous sensori-
motor, functional, and associative forms of play.
It was somewhat surprising to find no differences
between groups in the frequency of functional, sensori-
motor, and associative forms of play. It may be that dif-
ferences in types of play and other behaviors may not be
clear until after age 12 mo. Christensen et al. (2010), for
example, in a study of infant siblings of children with
ASD at age 14 mo, found that high-risk infants engaged
less often in functional play than did age-matched, typ-
ically developing counterparts.
During the feeding session, the finding of no significant
differences in sensory or motor behaviors between groups
may be explained in part by the nature of the context. First,
the opportunity for movement was limited in the high
chair to largely oral–motor and upper-extremity move-
ment. Second, the task of feeding is familiar and routine
and lends itself to little variation in how it is performed. It
may be that such a familiar task, with little variation in
how it was carried out among the dyads, resulted in similar
sensory and motor behavioral responses from the infants.
The developmental profile of the high-risk group in
this study was consistent with that found in other studies
of young siblings, which have found relative strengths in
visual–perceptual skills, motor performance falling within
the low end of the average range or borderline, and dif-
ficulties in receptive and expressive language (Landa 
Garrett-Mayer, 2006). We also found sensory processing
differences in the high-risk infant siblings, detected
through the use of the Infant–Toddler Sensory Profile
(Dunn, 2002). In particular, the high-risk infants tended
to be less sensory seeking, a finding consistent with that of
Ben-Sasson et al. (2009). It may be that early on, these in-
fants have less capacity or motivation for exploration within
their natural environments. It may also be an adaptive re-
sponse learned by the infants to avoid exposure to sensory
stimulation that they may experience as distressing.
Prospective study designs have been helpful for sup-
porting research in the design of valid screening and eval-
uation instruments for young children before age 2 yr.
Zwaigenbaum et al. (2007) reported that valid instruments
are becoming increasingly available, and clinicians trained
in their use are showing evidence for reliable and stable
early diagnosis (as early as age 2). Although consensus on
the need for universal screening has not yet been reached,
early identification of ASD provides families with impor-
tant information and resources. With very early entry into
effective intervention services, the possibility for significant
reductions in symptoms associated with ASD, or even the
possibility for prevention, is promising.
Limitations and Future Research
A number of limitations must be considered in the in-
terpretation of this study’s findings. First, the sample size
was small, which increases the risk for Type I error, and
because only predominantly White families from New
England were represented, generalizability is limited. Sec-
ond, the high-risk group, as expected, was quite hetero-
geneous in that 4 children ultimately were diagnosed with
ASD, 2 children received early intervention for speech
and language concerns but were not diagnosed with the
disorder, and 7 were not identified as having any neuro-
developmental disorder or delays in development. In ad-
dition, a few of the children who were diagnosed with an
ASD may have represented a broader ASD phenotype
with subtle characteristics of the disorder, but not to the
extent that a diagnosis was made. Therefore, as more data
are collected, analyses using data from only those children
who eventually have confirmed ASD diagnoses would
provide a clearer picture of early markers.
The inconsistency in scores across the measures of
social and communication skills is also a weakness of the
study and may be attributed in part to the specific
characteristics of the tests used and the differences in the
measures’ sensitivity for detecting differences in children
at age 12 mo. For example, the expectation of word ut-
terances at 12 mo of age is very low for even typically
developing infants. Therefore, this type of test item is not
as discriminating as it would be for children at age 18 mo.
The study design would also have been strengthened if all
data had been coded by researchers blind to group assign-
ment; although we attempted to blind researchers, occa-
sionally they were able to hear siblings on the videos in the
background, and because of limited resources, only about
70% videotapes were coded blindly. Therefore, researcher
bias may have affected the coding, although the operational
definitions of the behaviors and strong interrater reliability
data addressed the risk of researcher bias to some degree.
Ongoing research is needed to further develop and
validate screening and assessment measures for early
562 September/October 2012, Volume 66, Number 5
diagnosis. Larger studies that systematically collect data
longitudinally from at-risk infants from more diverse
populations at multiple time points, such as at 6, 12, 18,
and 24 mo of age, would provide a more in-depth un-
derstanding of potential markers and increase generaliz-
ability. In addition, an analysis of data from only those
children who eventually develop ASD would provide more
clarity with respect to potential markers for early screening
and diagnostics.
Implications for Occupational
Therapy Practice
The results of this study have the following implications
for clinical occupational therapy practice:
• Occupational therapists and other team members who
are involved in the initial diagnosis of children with
ASD should be aware of reliable and valid evaluation
and screening tools, receive training in their use, and
apply them in the diagnostic process.
• Occupational therapists are and should continue to be
involved as valuable contributors in the evaluation
process for determining ASD diagnoses, in light of
their expertise in the areas of occupational perfor-
mance, sensory processing, play behaviors, and motor
development of infants and toddlers.
• Contextual observations of mother–infant play should
be considered for inclusion in the diagnostic process.
Not only do they have the potential to elicit sensory and
motor behaviors that may be important for diagnostic
purposes, but they can enhance our understanding of
how such behaviors may influence a young child’s abil-
ity to play, learn, and perform daily activities. s
Acknowledgments
The authors would like to express their gratitude to the
occupational therapy students and Maternal Child and
Health Leadership Program for Working With Children
With Neurodevelopmental Disabilities and Their Families
(MCH LEND) trainees who were involved as research
assistants in multiple phases of the project and to the
children and families who agreed to participate in the
study. We also would like to thank the University of New
Hampshire, Office of the Provost and Vice President, for
partial funding, and Anne Dillon, Michelle Sullivan, and
Rae Sonnenmeier for all of their work with the Early
Markers Project.
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Note. Appendix begins on next page.
The American Journal of Occupational Therapy 565
Appendix.
Operational Definitions for Coding Sensory and Motor Behaviors
Behaviors During Play
Motor stereotypy—Repetitive body movement. Child may engage in wiggling, flapping, or flicking of the fingers or hands; alternate flexion and extension of
the fingers; alternate pronation and supination of the forearm; or head rolling, head banging, body rocking, or body swaying back and forth.
Object stereotypy—Repetitive movement with an object. Child makes the same movement with an object more than 10 times in a 30-s interval.
Mouthing objects—Child puts a toy or object partially or completely in mouth for any length of time, or an object touches child’s lips.
Movement transition—Child transitions from one position to another or physically moves from a location. Examples of movement transitions include moving
in or out of sitting or to and from kneeling; side lying, supine, or prone lying positions; crawling; scooting; pivoting in sitting; or walking. Repositioning self in
sitting—for example, from side sitting to cross-legged or a long sit—does not qualify as a movement transition if child remains in the same spot.
Orients toward visual—Child responds positively to visual stimuli, such as mirror, mom’s face, moving objects, or bright colors, by turning head toward,
moving body toward, reaching for, or looking toward the stimulus.
Orients toward auditory—Child responds positively to auditory stimuli, such as squeaky ball, ringing phone, rattle, banging of the mirror, mom’s voice, or
other environmental sounds, by turning head toward, moving body toward, reaching for, or looking toward the stimulus.
Orients toward tactile—Child responds positively to tactile stimuli, such as parent’s touch, baby blanket, bumpy ball, or fuzzy blocks, by turning head
toward; moving body toward; repeatedly touching, rubbing, or patting; or mouthing the stimulus.
Aversive response to visual—Child reacts negatively to visual stimuli, such as mirror, moving objects, or bright colors, by closing eyes, screaming, crying,
or turning head or moving away from the stimulus.
Aversive response to auditory—Child reacts negatively to auditory stimuli, such as squeaky ball, ringing phone, rattles, or banging, by covering ears,
screaming, pushing toys away, crying, or turning head or moving away from the stimulus.
Aversive response to tactile—Child reacts negatively to tactile stimuli, such as baby doll, blanket, or bumpy ball, by withdrawing hands after touching
object, pushing object away, or moving body away from the object. Child cries or sounds distressed following a tactile stimulus.
Object manipulation—Child transfers object from one hand to the other; repositions object in the hand, perhaps using the floor or body part to help
reposition object in the hand; or manipulates object in the hands, which does not include simple picking up, grasping, holding, throwing, and shaking.
Motor imitation—Child imitates hand motions (pushing, rubbing, tapping, banging hammer, squeezing, turning, pointing, shaking rattle), facial movements
(blowing, kissing, sticking out tongue), or other body movements that he or she has seen the caregiver perform; includes movements performed after
verbal prompting if the movement was also demonstrated. The imitated behavior must occur within the same time segment as the stimulus movement.
Types of Play
All forms of play may be seen in the same segment.
Construction play—Child builds or attempts to build with soft blocks.
Sensorimotor play—Child examines sensory features of play objects by feeling, looking at, touching, listening to, or exploring the objects but does not
need to display any functional use of the objects.
Functional play—Child uses objects functionally for their intended purposes, such as covers baby doll with blanket, puts bottle to baby doll’s mouth,
hammers ball into hole, rolls ball, talks or babbles on phone, brings phone to ear, presses phone buttons, or builds with blocks.
Associative play—Child demonstrates at least one instance of social play or engagement with the parent during the segment. Examples include turn taking,
performing 3-point eye gaze between parent and object (parent–object–parent or object–parent–object), imitating, giving, showing, following a verbal
direction, and responding appropriately to gestural communication.
Behaviors During Feeding
Motor stereotypy— Repetitive body movement. Child may engage in wiggling, flapping, or flicking of the fingers or hands; alternate flexion and extension of
the fingers; alternate pronation and supination of the forearm; or head rolling, head banging, body rocking, or body swaying back and forth.
Object stereotypy—Repetitive movement with an object. Child makes the same movement with an object, such as banging the spoon or spinning the bowl,
more than 10 times.
Independently scoops—Child uses spoon to scoop food on his or her own.
Spoon to mouth—Child brings food to mouth using spoon and places food in mouth.
Hand to mouth without spoon—Child brings hand to mouth in the context of feeding, such as putting food item in mouth, pushing food into mouth,
stopping food loss, wiping food, or sucking fingers.
Plays with food—Child uses food nonfunctionally (not trying to eat it) by, for example, pushing food around tray, throwing food, or mashing food between
fingers.
Orients toward visual—Child responds positively to visual stimuli, such as mom’s face or moving objects, by moving head or body toward or reaching for
stimulus; child visually tracks spoon as it is brought to mouth.
Orients toward auditory—Child responds positively to auditory stimuli, such as mom’s voice or other environmental sounds, by turning head toward,
moving body toward, reaching for, or looking toward the stimulus.
Orients toward tactile—Child responds positively to tactile stimuli, such as parent’s touch or contact with food, by turning head toward, moving body
toward, or repeatedly touching the stimulus; child plays with food or licks food off lips or hands.
Aversive response to visual—Child reacts negatively to visual stimuli, such as caregiver’s face or moving spoon or bowl, by closing eyes, screaming,
crying, or turning head or moving away from the stimulus.
Aversive response to auditory—Child reacts negatively to auditory stimuli, such as mom’s voice or other environmental sounds, by covering ears,
screaming, pushing stimulus away, crying, or turning head or moving away from the stimulus.
Aversive response to tactile—Child reacts negatively to tactile stimuli, such as holding spoon or feeling food on hands, by withdrawing hands; shaking
hands to remove food; pushing food, bowl, or spoon away; or moving body away from the stimulus.
Sits upright—Child sits upright in high chair without slouching or needing repositioning by the parent.
Motor imitation—Child imitates hand motions (pushing, rubbing, tapping, pointing, scooping) or facial movements (blowing, kissing, sticking out tongue)
that he or she has seen the caregiver perform; includes movements performed after verbal prompting as long as the movement was also demonstrated in
the same segment.
566 September/October 2012, Volume 66, Number 5
Reproduced with permission of the copyright owner. Further reproduction prohibited without permission.

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Sensory and Motor Differences Between Infant Siblings of Children With and Without Autism

  • 1. Sensory and Motor Behaviors of Infant Siblings of Children With and Without Autism Shelley Mulligan, Barbara Prudhomme White KEY WORDS autistic disorder early diagnosis motor activity sensation siblings Shelley Mulligan, PhD, OTR/L, is Associate Professor and Chair, Department of Occupational Therapy, Hewitt Hall, University of New Hampshire, Durham, NH 03824; shelley.mulligan@unh.edu Barbara Prudhomme White, PhD, OTR/L, is Associate Professor, Department of Occupational Therapy, University of New Hampshire, Durham. We compared the sensory and motor behaviors of typically developing infants with those of infant siblings of children with autism spectrum disorders (ASD), who are considered high risk for the disorder, to explore potential sensory and motor markers for use in early diagnosis of ASD. We compared frequencies of sensory and motor behaviors during 10-min, videotaped, infant–mother play sessions and during 5 min of spoon- feeding between groups of 12-mo-old infants. Data from standardized measures of development, sensory processing, and behaviors commonly associated with ASD were also analyzed descriptively for the high-risk group. The results indicated that high-risk infants demonstrated fewer movement transitions (t [23] 5 22.4, p 5 .03) and less object manipulation (t [23] 5 22.4, p 5 .03) than low-risk infants. The sensory and motor differences found between typical and high-risk infants suggest that early screen- ings for ASD should include the examination of sensory and motor behaviors. Mulligan, S., White, B. P. (2012). Sensory and motor behaviors of infant siblings of children with and without autism. American Journal of Occupational Therapy, 66, 556–566. http://dx.doi.org/10.5014/ajot.2012.004077 Autism spectrum disorders (ASD) are developmental disorders characterized by pervasive deficits in social interaction and communication and a tendency toward restricted and repetitive behaviors (American Psychiatric Association, 2000). ASD diagnoses have been steadily on the rise, and estimates from the Centers for Disease Control and Prevention (2012) indicate a prevalence of 1 in 88. Kogan and colleagues (2009) estimated a U.S. ASD prevalence among 3–17-yr-olds of as high as 1.1%. Although the cause remains unknown for most people with ASD, twin and family studies indicate a strong genetic link. For children with ASD, early detection is essential for facilitating access to interventions that can shape neural connections during sensitive periods of development (Robins, Fein, Barton, Green, 2001). Early intervention has been associated with improved developmental and functional outcomes such as language development, cognitive development, and success at school (McEachin, Smith, Lovaas, 1993; Ospina et al., 2008). More specifically for young children with autism, a combination of play-based and behavioral in- terventions may be vital in promoting development and adaptive functioning and minimizing clinical features of the disorder (Dawson, 2008; Johnson Myers, 2007). Unfortunately, children with ASD are often not diagnosed until age 2–4 yr (Filipek et al., 1999; Ospina et al., 2008; Zwaigenbaum et al., 2007), although much progress in early detection has been made over the past 5–10 yr. The American Academy of Pediatrics Council on Children with Disabilities (2006) recommended that pediatricians incorporate autism screening as part of routine practice. However, Al-Qabandi, Gorter, and Rosenbaum (2011) reported that routine screening of all infants may be premature at this time because of a lack of effective screening tools and evidence-based, efficacious practices and because of the need for a cost–benefit analysis of conducting universal early screenings. 556 September/October 2012, Volume 66, Number 5
  • 2. Nonetheless, early identification allows families access to supports and resources and has the potential to enhance our understanding of ASD by promoting the study of en- vironmental, behavioral, and developmental factors that may influence the course of the disorder. Retrospective parent interviews (Young, Brewer, Pattison, 2003) and studies of home movies of infants later diagnosed with autism (Baranek, 1999; Osterling Dawson, 1994; Werner, Dawson, Osterling, Dinno, 2000) suggest that abnormalities in development are evi- dent in the first 2 yr of life. However, the higher incidence of ASD in infant siblings of children with autism provides a unique opportunity to look prospectively at the devel- opment of ASD from infancy and inform us of potential early signs that may be of diagnostic importance. Siblings of children with autism have an estimated risk that is at least 50 times greater than that of the general population (Liu et al., 2001). Studies using infant siblings have reported a recurrence risk of 14% (Zwaigenbaum et al., 2005) to more than 20% (Ozonoff, Young, et al., 2008; Sullivan et al., 2007; Yirmiya Charman, 2010). Several prospective, longitudinal studies of siblings have been completed or are under way to identify indicators of ASD in infants and toddlers through ongoing standardized developmental evaluations and structured observations of this at-risk population (Bryson et al., 2007; Iverson Wozniak, 2007; Ozonoff, Macari, et al., 2008; Yirmiya Charman, 2010; Yirmiya et al., 2006; Zwaigenbaum et al., 2005). Veness et al. (2012), for example, found social and communication differences between 12-mo-olds with and without ASD, including the early use of gestures. Prospective designs enable researchers to examine early neurodevelopmental signs, behaviors, and developmental patterns over time under standardized conditions, allowing greater comparability both within and between individuals over time. Ultimately, the information can be used in the development of screening and evaluation tools aimed at early detection. Motor disturbances have commonly been noted in children with autism (Baranek, 2002; Minshew, Sung, Jones, Furman, 2004; Molloy, Dietrich, Bhattacharya, 2003; Provost, Lopez, Heimerl, 2007; Rinehart, Bradshaw, Brereton, Tonge, 2001). Although researchers have begun to study early motor signs (Loh et al., 2007; Ozonoff, Young, et al., 2008; Teitelbaum, Teitelbaum, Nye, Fryman, Maurer, 1998), few studies have ex- amined scores from standardized motor assessments in conjunction with more functional fine and gross motor behaviors during typical infant activity such as feeding and play. Studies completed to date have produced in- consistent results. For example, Teitelbaum et al. (1998) reported that a sample of infants later diagnosed with autism showed many abnormal movement patterns, whereas more recent studies demonstrated no or few significant motor differences in infants later diagnosed with autism (Ozonoff, Young, et al., 2008; Provost et al., 2007). Loh et al. (2007) provided some evidence that arm waving in the context of standardized test adminis- tration was more often seen in children who were later diagnosed with ASD. Motor stereotypies and repetitive use of objects were found in a sample of high-risk infants at age 12 mo (Ozonoff, Young, et al., 2008). In a study of older boys with ASD (6–17 yr), Jansiewicz et al. (2006) found that children with ASD performed more poorly on many measures of motor control, including balance and coordination. In examining potential early markers for identifying ASD in infants, Zwaigenbaum and colleagues (2005) noted that sensory orienting behaviors such as visual at- tention, orienting to name, visual fixation on certain ob- jects, and visual tracking may be useful in distinguishing children with the disorder from typical children. Loh et al. (2007) found that high-risk infants who were later di- agnosed with ASD used their hands to cover their ears (aversive response to sound) in the context of standard- ized test administration more often than children who did not have the disorder. Rogers, Hepburn, and Wehner (2003) found that parents reported sensory differences in their toddlers who were later diagnosed with ASD. Baron-Cohen, Ashwin, Ashwin, Tavassoli, and Chakrabarti (2009) conducted an interesting study examining the talents of children with autism, concluding that the ex- cellent attention to detail and pattern recognition abil- ities of children with the disorder were often rooted in underlying sensory processing differences such as sensory hypersensitivities. In a meta-analysis of 14 studies, Ben-Sasson et al. (2009) found evidence of significant differences in sen- sory modulation behaviors between typically developing children and children with ASD. In an examination of the sensory modulation patterns of sensory overresponsivity, sensory underresponsivity, sensory seeking, and total per- formance, differences were most pronounced from 6–9 yr of age, with a tendency toward sensory underresponsivity. More relevant to this study was the finding that children with ASD birth to age 3 yr were less often categorized as being sensory seeking than their typically developing counterparts. In summary, the research indicates that our understanding of the nature of early infant sensory pro- cessing and motor development is emerging with this population and suggests that these are promising areas for consideration in early screening of ASD. The American Journal of Occupational Therapy 557
  • 3. Screening tools such as the Modified Checklist for Autism in Toddlers (Robins Dumont-Mathieu, 2006) emphasize the examination of behaviors associated with emerging language, social interaction, and play that, if disrupted, are hallmark features of ASD. Because many of these types of behavior do not emerge developmentally until around 12–24 mo of age, however, relying on tools emphasizing social, language, and play behaviors is lim- iting at this young age (Baron-Cohen, Allen, Gillberg, 1992). In contrast, sensory and motor behaviors are easily captured from birth, and if differences do exist in such behaviors, they may be able to be detected earlier than social, play, and language behaviors. Although sensory and motor behaviors are not core features of ASD, dif- ferences or abnormalities in sensory processing and motor skill development have commonly been found, as noted in the literature described earlier. Feldman et al. (2012) emphasized the value of ex- amining child behaviors in the natural context, leading them to develop the Parent Observation of Early Markers Scale (POEMS). POEMS includes a large range of pos- sible early markers or signs of ASD that parents can observe within the context of their daily routines and interactions with their child. Feldman et al. found that differences between typically developing children and children diagnosed with ASD were more pronounced at 18–24 mo of age, although subtle differences were noted as early as 9 mo. The authors concluded that tools for ongoing surveillance of children by their parents in natural contexts are helpful additions to performance- based developmental measures for early detection of ASD. The study described in this article examined both sensory processing and motor behaviors that we thought, on the basis of prior research, would be useful for the early detection of autism in 12-mo-old infants at high risk for developing the disorder. Data collection for this study included observations and coding of sensory and motor behaviors within the context of the typical daily routines of play and feeding via videotape for both typically de- veloping children and high-risk siblings. Standardized de- velopmental measures and caregiver questionnaires were also administered for the high-risk ASD siblings. Details of these measures are provided in the Methods sec- tion. The specific aims of this study were to describe the sensory and motor development and behaviors of infant siblings of children with ASD at 12 mo of age and to compare sensory and motor behaviors of high-risk infants with those of low-risk, typically developing infants at 12 mo during both a semistructured infant–mother play session and spoon-feeding. Method Research Design In this study we implemented a two-group exploratory design to describe and compare data regarding the sensory and motor behaviors of 13 infant siblings at high risk for ASD and 12 typically developing infants. The University of New Hampshire institutional review board for the protection of human subjects approved the study, and we obtained informed consent from the parents before data collection. Participants All infants in the study were between age 11 and 13 mo. Exclusion criteria included known genetic, neuromotor, or developmental problems or disorders as well as pre- maturity. High-risk infants were recruited through local service providers and clinics specializing in young children and through local organizations for children with special needs and their families. The high-risk group included 13 infants with a sibling who had a confirmed diagnosis of ASD determined by a qualified team of professionals that included a developmental neurologist or pediatrician. All older siblings were assessed using the Autism Diagnostic Observation Schedule (Lord, Rutter, DiLavore, Risi, 2002) either before diagnosis or for confirmation as part of this study. We recruited the 12 typically developing infants in the low-risk group through personal contacts and local day care and preschool centers. All typically developing infants had siblings without any developmental concerns. Al- though the typically developing infants were not formally tested as part of this study, they were screened by the first author (Mulligan) and a pediatrician, and only infants for whom the parents and pediatrician identified no de- velopmental concerns were recruited to be in the low-risk group. We made a follow-up phone call to the parents when the low-risk children reached age 30 mo to verify that the parents continued to have no concerns regarding their child’s development. Procedures All infants participated with their caregivers in a 10-min play session and a 5-min feeding session. Each session was videotaped and subsequently coded for sensory and motor behaviors. The format of the play and feeding sessions and the defining and coding of sensory and motor be- haviors were developed and refined by the primary author (Mulligan). Sensory and motor behaviors were selected on the basis of previous research examining potential early 558 September/October 2012, Volume 66, Number 5
  • 4. markers and on a review of ASD screening tools. Each sensory and motor behavior was operationally defined and tested for interrater reliability. In total, we analyzed 16 behaviors for play and 14 behaviors for the feeding session (see the Appendix for a list of behaviors and definitions). We used time sampling, recording the presence or absence of each behavior in 30-s time intervals. To examine in- terrater reliability, we analyzed data coded from three trained raters using nine play and feeding sessions. Total occurrences for each of the 30 variables were tallied and analyzed using the intraclass correlation coefficient (ICC). The results yielded an ICC of .97 using average measures, demonstrating strong consistency across raters. Infants in the high-risk group were administered developmental assessments at 11–13 mo of age by the first author (Mulligan), who was aware that the older sibling was diagnosed with ASD. Standardized performance-based measures included the Mullen Scales of Early Learning (Mullen, 1995) and the Autism Observation Scale for Infants (AOSI; Bryson, Zwaigenbaum, McDermott, Rombough, Brian, 2008; Zwaigenbaum et al., 2005). The stan- dardized interview format for the Vineland Adaptive Behavior Scale (VABS; Sparrow, Balla, Cicchetti, 1984) was also administered. Test administration followed stan- dardized protocols and occurred at a university setting with clinic space and testing rooms for children. In all cases, the mother was present throughout the testing. A parent (typically the mother) completed the Infant–Toddler Sen- sory Profile (Dunn, 2002) before the clinic visit. The first author and a trained research assistant con- ducted the play and feeding videotaped sessions in par- ticipants’ home for both the high-risk and the low-risk groups. Play sessions occurred in the living room or play room where the child most often played and included 10 min of the infant playing with the mother on the floor using a standardized set of common, enticing toys. The toys included large soft blocks, a mirror, a play phone, rattles, balls of various sizes and textures, a doll with a spoon, a bottle and blanket, and a toy hammer– play structure. The toy hammer–play structure had balls that, when hammered into holes on the structure, moved through a maze and out another hole. The mother was directed to play with her infant and the toys in any way she liked and in ways that would reflect how the dyad would typically interact and play with toys. They were not required to use all the play materials or toys. Feeding occurred in the family’s kitchen using the child’s high chair. The mother was asked to support the child in any way she typically would, and the mother provided a food item that the child was accustomed to eating (cereal, applesauce, and yogurt were most common). Instruments The VABS assesses adaptive functioning for individuals from birth to age 90 across four domains—Communication, Daily Living Skills, Socialization, and Motor Skills— and provides an overall Adaptive Behavior Composite score. The VABS was standardized on a national sam- ple of 3,000 children matched to U.S. census data. Internal consistency examined by split-half reliability coefficients is acceptable, ranging from .84 to .88, and test–retest reliability reported in the manual is also acceptable, with r values ranging from .78 to .92 for all areas for the birth to 3 yr age range (Sparrow et al., 1984). The Infant–Toddler Sensory Profile (Dunn, 2002) is a caregiver questionnaire measuring the ability to process and modulate sensory information, developed with a nor- mative sample of 589 children from birth to age 36 mo. The caregiver rates on a scale ranging from 1 to 5 the frequency with which his or her child exhibits certain behaviors indicative of sensory processing. Internal con- sistency estimates across sensory processing sections and quadrants (representing Low Registration, Sensory Seek- ing, Sensory Sensitivity, and Sensory Avoiding) varied with the children aged 7–36 mo and ranged from .42 to .85, with the strongest values representing the sensory processing patterns. Test–retest reliability values are ac- ceptable on the basis of a study of a normative subsample (n 5 32), with coefficients of .86 for sensory processing section scores and .74 for quadrant scores. Evidence re- garding content, construct, and discriminant validity is also provided in the manual (Dunn, 2002). The Mullen Scales of Early Learning is a standardized, norm-referenced, developmental measure for children from birth to age 68 mo. It was normed on 1,849 children based on U.S. census data and generates standard scores for five subscales—Gross Motor, Fine Motor, Vi- sual Reception, Receptive Language, and Expressive Language—as well as an Early Learning Composite score. Split-half internal consistency coefficients are acceptable, with median values (from the normative data) ranging from .75 to .91. Test–retest reliability was obtained with 97 children over a 2-wk period and ranged from .71 to .96 (Mullen, 1995). The AOSI was developed to assess behaviors specific to ASD in infants. The clinician administers several play- based activities to elicit possible behavioral markers for ASD, such as poor eye contact, lack of orienting to name, lack of social smiling, deficit in interest in others, and reactivity to sensory stimuli. The AOSI has demonstrated strong interrater reliability (.94 with children at 18 mo), The American Journal of Occupational Therapy 559
  • 5. fair test–retest reliability (.61 using the total score for infants 12 mo of age). Predictive validity when admin- istered at 12 mo has also been found to be strong (Bryson et al., 2008). Data Collection and Analysis Videotaped data from the play and feeding sessions were transferred to a DVD, and the sessions were viewed to record the targeted sensory and motor behaviors as being either present or absent every 30 s. Coding was conducted by the first author and three research assistants whom she trained in the use of the measure. Three demonstra- tion tapes were used for training purposes, and research assistants achieved 90% agreement with Mulligan across all cells before coding independently. Training involved approximately 6 hr of review of the definitions, practice coding, and discussion with Mulligan to resolve dis- agreements. The research assistants were blind to group assignment and coded 70% of the digital recordings; Mulligan coded the remaining recordings. The first author or a trained research assistant scored all standardized tests (i.e., AOSI, Mullen Scales) and parent questionnaires. Descriptive statistics were used to analyze data from the standardized developmental measures ad- ministered to the high-risk infants. One-sample t tests were used to compare the mean developmental scores of each of the 13 children in this group with each test’s normative data to determine whether the group differed significantly from the normative sample. To examine sensory and motor differences between the high-risk and low-risk groups, we coded mean frequencies for each behavior from the play and feeding sessions and then compared the groups using independent t tests, with the a level for determining statistical significance set at .05. All data were analyzed using PASW Statistics 18, Release Version 18.0 (SPSS, Inc., Chicago). Results The study sample included 8 girls and 5 boys in the high- risk group (mean age 12.6 mo) and 7 girls and 5 boys in the low-risk group (mean age 12.1 mo). Most participants were White, and all were from New England. The groups did not significantly differ with respect to race, gender, and age. Follow-up at age 30 mo indicated that four of the high-risk infants met criteria for ASD, and another two children had received early intervention services for mild developmental concerns. None of the children received early intervention services before 12 mo of age, and none of the infants placed in the low-risk group exhibited developmental concerns. The developmental profile of the high-risk group fell largely within the low end of the average range, although some relative strengths and weaknesses are noteworthy. A summary of these results is presented in Table 1. Fine motor performance and visual reception skills fell in the average to above-average range, whereas gross motor skills were within the low-average range. Test scores measuring social and communication skills were mixed, although most fell within the low-average to below-average range. The number of markers noted using the AOSI indicated that 31% of the children were at high risk for developing ASD. Sensory processing differences were also found; scores from the high-risk group reflected less sensory- seeking behavior than scores of children from the nor- mative sample and a tendency for more difficulties in the area of auditory processing. The results of the one-sample t tests revealed some significant differences between the scores of this group and the normative data for each of the tests. On the Mullen Scales of Early Learning, scores from the high- risk group were higher on Visual Reception (t [12] 5 2.1, p 5 .059), approaching significance, and significantly higher on the Fine Motor Scale (t [12] 5 3.2, p 5 .009). Performance of the high-risk group was, however, sig- nificantly poorer on the Expressive Language (t [12] 5 23.6, p 5 .004) and Receptive Language (t [12] 5 23.7, p 5 .004) scales. On the VABS, the Motor Scale showed significantly lower scores for the high-risk group than the normative sample (t [12] 5 22.4, p 5 .03); there were no other significant differences. Videotaped coding of sensory and motor behaviors during play and feeding revealed that compared with infants from the low-risk group, the high-risk group demonstrated significantly fewer movement transitions (t [23]5 22.4, p 5 .03) and less object manipulation (t [23] 5 22.4, p 5 .03; see Table 2). Thus, children in the high-risk group tended to move around less during the play session, and they manipulated objects in their hands less frequently (see definitions in the Appendix). No significant differences were found between groups on the other sensory and motor behaviors coded during the play sessions, and no differences between groups were noted on the behaviors coded from the feeding sessions. Discussion The results suggest that contextual, play-based observa- tions in conjunction with standardized developmental measures may have a vital role in the early detection of ASD. In this study, how the infants used their hands to manipulate toys in play and how much they moved 560 September/October 2012, Volume 66, Number 5
  • 6. around during play appeared to be important consid- erations. Although many of the high-risk infants scored within the normal range on the standardized motor measures, during more unstructured play, they used less sophisticated functional fine and gross motor movements than would be expected. An examination of the variability and frequency of the sensory and motor behaviors that were observed and coded Table 2. Mean Frequencies of Sensory and Motor Behaviors Coded From Play Sessions Coded Behavior High-Risk Group, Mean (SD) Low-Risk Group, Mean (SD) p Motor stereotypy 0.2 (0.6) 0.2 (0.6) .95 Object stereotypy 0.85 (1.1) 1.9 (0.5) .19 Mouthing objects 5.7 (5.3) 3.7 (2.9) .27 Movement transition 7.5 (5.5) 12.3 (4.7) .03* Orients toward visual stimulus 19.8 (1.3) 17.7 (4.1) .09 Orients toward auditory stimulus 15.5 (3.9) 14.9 (4.7) .75 Orients toward tactile stimulus 9.7 (6.5) 6.3 (4.1) .13 Aversive response to visual stimulus 0.23 (0.6) 0 (0) .20 Aversive response to auditory stimulus 0.46 (1.1) 0.2 (0.9) .61 Aversive response to tactile stimulus 0.61 (1.3) 0.2 (0.4) .25 Object manipulation 9.7 (4.4) 13.4 (3.3) .03* Motor imitation 3.3 (2.7) 2.5 (2.3) .43 Construction play 0.15 (0.5) 0.3 (0.6) .46 Sensorimotor play 13.4 (6.6) 12.1 (6.7) .62 Functional play 10.8 (5.2) 11.2 (3.7) .83 Associative play 9.0 (6.3) 10.3 (3.6) .55 Note. SD 5 standard deviation. *p .05. Table 1. Developmental Standard Scores of the High-Risk Group (n 5 13) Test Variable Mean (SD) % Scoring in the Dysfunctional Range Autism Observation Scale for Infants 31% received 7 or more markers, which has been reported to be predictive of ASD.Total score 8.4 (4) Number of ASD markers 5.2 (2.3) Mullen Scales of Early Learning 50 (10) Gross Motor 46.7 23 Fine Motor 57.1a 15 Visual Reception 55.5a 0 Receptive Language 36.9b 69 Expressive Language 40.8b 46 Early Learning Composite 95.9 15 Vineland Adaptive Behavior Scales 100 (15) Communication 97.1 23 Daily Living Skills 101.5 0 Socialization 94.8 15 Motor Skills 94.3b 15 Adaptive Behavior Composite 97.8 15 Infant–Toddler Sensory Profile Range within 1 SD Low Registration 47.4 (46–54) 36 Sensory Seeking 35.0 (19–35)a 72 Sensory Sensitivity 47.6 (41–52) 36 Sensory Avoiding 51.2 (45–56) 45 Low Threshold 98.8 (87–107) 45 Auditory Processing 39.7 (35–43) 54 Visual Processing 24.9 (20–27) 45 Tactile Processing 58.1 (48–61) 45 Vestibular Processing 20.1 (18–23) 36 Oral Sensory Processing 24.8 (21–29) 36 Note. ASD 5 autism spectrum disorder; SD 5 standard deviation. a Significantly above the norm. b Significantly below the norm. The American Journal of Occupational Therapy 561
  • 7. during the play sessions provides some insight into which behaviors may be useful for future consideration related to screening for ASD. For example, behaviors that were coded as being absent or not observed in almost all intervals would not be helpful, such as movement ste- reotypy; aversive responses to visual, auditory, and tactile sensory input in both feeding and play sessions; con- struction play; and motor imitation during feeding. Be- haviors that were seen a moderate amount of time that appear more promising as possible early markers in young infants include object stereotypy; mouthing of objects; motor imitation during play; and spontaneous sensori- motor, functional, and associative forms of play. It was somewhat surprising to find no differences between groups in the frequency of functional, sensori- motor, and associative forms of play. It may be that dif- ferences in types of play and other behaviors may not be clear until after age 12 mo. Christensen et al. (2010), for example, in a study of infant siblings of children with ASD at age 14 mo, found that high-risk infants engaged less often in functional play than did age-matched, typ- ically developing counterparts. During the feeding session, the finding of no significant differences in sensory or motor behaviors between groups may be explained in part by the nature of the context. First, the opportunity for movement was limited in the high chair to largely oral–motor and upper-extremity move- ment. Second, the task of feeding is familiar and routine and lends itself to little variation in how it is performed. It may be that such a familiar task, with little variation in how it was carried out among the dyads, resulted in similar sensory and motor behavioral responses from the infants. The developmental profile of the high-risk group in this study was consistent with that found in other studies of young siblings, which have found relative strengths in visual–perceptual skills, motor performance falling within the low end of the average range or borderline, and dif- ficulties in receptive and expressive language (Landa Garrett-Mayer, 2006). We also found sensory processing differences in the high-risk infant siblings, detected through the use of the Infant–Toddler Sensory Profile (Dunn, 2002). In particular, the high-risk infants tended to be less sensory seeking, a finding consistent with that of Ben-Sasson et al. (2009). It may be that early on, these in- fants have less capacity or motivation for exploration within their natural environments. It may also be an adaptive re- sponse learned by the infants to avoid exposure to sensory stimulation that they may experience as distressing. Prospective study designs have been helpful for sup- porting research in the design of valid screening and eval- uation instruments for young children before age 2 yr. Zwaigenbaum et al. (2007) reported that valid instruments are becoming increasingly available, and clinicians trained in their use are showing evidence for reliable and stable early diagnosis (as early as age 2). Although consensus on the need for universal screening has not yet been reached, early identification of ASD provides families with impor- tant information and resources. With very early entry into effective intervention services, the possibility for significant reductions in symptoms associated with ASD, or even the possibility for prevention, is promising. Limitations and Future Research A number of limitations must be considered in the in- terpretation of this study’s findings. First, the sample size was small, which increases the risk for Type I error, and because only predominantly White families from New England were represented, generalizability is limited. Sec- ond, the high-risk group, as expected, was quite hetero- geneous in that 4 children ultimately were diagnosed with ASD, 2 children received early intervention for speech and language concerns but were not diagnosed with the disorder, and 7 were not identified as having any neuro- developmental disorder or delays in development. In ad- dition, a few of the children who were diagnosed with an ASD may have represented a broader ASD phenotype with subtle characteristics of the disorder, but not to the extent that a diagnosis was made. Therefore, as more data are collected, analyses using data from only those children who eventually have confirmed ASD diagnoses would provide a clearer picture of early markers. The inconsistency in scores across the measures of social and communication skills is also a weakness of the study and may be attributed in part to the specific characteristics of the tests used and the differences in the measures’ sensitivity for detecting differences in children at age 12 mo. For example, the expectation of word ut- terances at 12 mo of age is very low for even typically developing infants. Therefore, this type of test item is not as discriminating as it would be for children at age 18 mo. The study design would also have been strengthened if all data had been coded by researchers blind to group assign- ment; although we attempted to blind researchers, occa- sionally they were able to hear siblings on the videos in the background, and because of limited resources, only about 70% videotapes were coded blindly. Therefore, researcher bias may have affected the coding, although the operational definitions of the behaviors and strong interrater reliability data addressed the risk of researcher bias to some degree. Ongoing research is needed to further develop and validate screening and assessment measures for early 562 September/October 2012, Volume 66, Number 5
  • 8. diagnosis. Larger studies that systematically collect data longitudinally from at-risk infants from more diverse populations at multiple time points, such as at 6, 12, 18, and 24 mo of age, would provide a more in-depth un- derstanding of potential markers and increase generaliz- ability. In addition, an analysis of data from only those children who eventually develop ASD would provide more clarity with respect to potential markers for early screening and diagnostics. Implications for Occupational Therapy Practice The results of this study have the following implications for clinical occupational therapy practice: • Occupational therapists and other team members who are involved in the initial diagnosis of children with ASD should be aware of reliable and valid evaluation and screening tools, receive training in their use, and apply them in the diagnostic process. • Occupational therapists are and should continue to be involved as valuable contributors in the evaluation process for determining ASD diagnoses, in light of their expertise in the areas of occupational perfor- mance, sensory processing, play behaviors, and motor development of infants and toddlers. • Contextual observations of mother–infant play should be considered for inclusion in the diagnostic process. Not only do they have the potential to elicit sensory and motor behaviors that may be important for diagnostic purposes, but they can enhance our understanding of how such behaviors may influence a young child’s abil- ity to play, learn, and perform daily activities. s Acknowledgments The authors would like to express their gratitude to the occupational therapy students and Maternal Child and Health Leadership Program for Working With Children With Neurodevelopmental Disabilities and Their Families (MCH LEND) trainees who were involved as research assistants in multiple phases of the project and to the children and families who agreed to participate in the study. We also would like to thank the University of New Hampshire, Office of the Provost and Vice President, for partial funding, and Anne Dillon, Michelle Sullivan, and Rae Sonnenmeier for all of their work with the Early Markers Project. References Al-Qabandi, M., Gorter, J. W., Rosenbaum, P. (2011). 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  • 10. may be useful for early diagnosis of autism. Proceedings of the National Academy of Sciences of the United States of America, 95, 13982–13987. http://dx.doi.org/10.1073/pnas.95.23.13982 Veness, C., Prior, M. R., Bavin, E., Eadie, P., Cini, E., Reilly, S. (2012). Early indicators of autism spectrum dis- orders at 12 and 24 months of age: A prospective, longi- tudinal comparative study. Autism, 16, 163–177. http:// dx.doi.org/10.1177/1362361311399936 Werner, E., Dawson, G., Osterling, J., Dinno, N. (2000). Brief report: Recognition of autism spectrum disorder before one year of age: A retrospective study based on home videotapes. Journal of Autism and Developmental Disorders, 30, 157–162. http://dx.doi.org/10.1023/A:1005463707029 Yirmiya, N., Charman, T. (2010). The prodrome of autism: Early behavioral and biological signs, regression, peri- and post-natal development and genetics. Journal of Child Psy- chology and Psychiatry, and Allied Disciplines, 51, 432–458. http://dx.doi.org/10.1111/j.1469-7610.2010.02214.x Yirmiya, N., Gamliel, I., Pilowsky, T., Feldman, R., Baron- Cohen, S., Sigman, M. (2006). The development of siblings of children with autism at 4 and 14 months: Social engagement, communication, and cognition. Journal of Child Psychology and Psychiatry, and Allied Disciplines, 47, 511–523. http://dx.doi.org/10.1111/j.1469-7610.2005. 01528.x Young, R. L., Brewer, N., Pattison, C. (2003). Parental identification of early behavioural abnormalities in chil- dren with autistic disorder. Autism, 7, 125–143. http:// dx.doi.org/10.1177/1362361303007002002 Zwaigenbaum, L., Bryson, S., Rogers, T., Roberts, W., Brian, J., Szatmari, P. (2005). Behavioral manifestations of autism in the first year of life. International Journal of Developmental Neuroscience, 23, 143–152. http://dx.doi. org/10.1016/j.ijdevneu.2004.05.001 Zwaigenbaum, L., Thurm, A., Stone, W., Baranek, G., Bryson, S., Iverson, J., et al. (2007). Studying the emergence of autism spectrum disorders in high-risk infants: Methodo- logical and practical issues. Journal of Autism and Devel- opmental Disorders, 37, 466–480. http://dx.doi.org/10. 1007/s10803-006-0179-x Note. Appendix begins on next page. The American Journal of Occupational Therapy 565
  • 11. Appendix. Operational Definitions for Coding Sensory and Motor Behaviors Behaviors During Play Motor stereotypy—Repetitive body movement. Child may engage in wiggling, flapping, or flicking of the fingers or hands; alternate flexion and extension of the fingers; alternate pronation and supination of the forearm; or head rolling, head banging, body rocking, or body swaying back and forth. Object stereotypy—Repetitive movement with an object. Child makes the same movement with an object more than 10 times in a 30-s interval. Mouthing objects—Child puts a toy or object partially or completely in mouth for any length of time, or an object touches child’s lips. Movement transition—Child transitions from one position to another or physically moves from a location. Examples of movement transitions include moving in or out of sitting or to and from kneeling; side lying, supine, or prone lying positions; crawling; scooting; pivoting in sitting; or walking. Repositioning self in sitting—for example, from side sitting to cross-legged or a long sit—does not qualify as a movement transition if child remains in the same spot. Orients toward visual—Child responds positively to visual stimuli, such as mirror, mom’s face, moving objects, or bright colors, by turning head toward, moving body toward, reaching for, or looking toward the stimulus. Orients toward auditory—Child responds positively to auditory stimuli, such as squeaky ball, ringing phone, rattle, banging of the mirror, mom’s voice, or other environmental sounds, by turning head toward, moving body toward, reaching for, or looking toward the stimulus. Orients toward tactile—Child responds positively to tactile stimuli, such as parent’s touch, baby blanket, bumpy ball, or fuzzy blocks, by turning head toward; moving body toward; repeatedly touching, rubbing, or patting; or mouthing the stimulus. Aversive response to visual—Child reacts negatively to visual stimuli, such as mirror, moving objects, or bright colors, by closing eyes, screaming, crying, or turning head or moving away from the stimulus. Aversive response to auditory—Child reacts negatively to auditory stimuli, such as squeaky ball, ringing phone, rattles, or banging, by covering ears, screaming, pushing toys away, crying, or turning head or moving away from the stimulus. Aversive response to tactile—Child reacts negatively to tactile stimuli, such as baby doll, blanket, or bumpy ball, by withdrawing hands after touching object, pushing object away, or moving body away from the object. Child cries or sounds distressed following a tactile stimulus. Object manipulation—Child transfers object from one hand to the other; repositions object in the hand, perhaps using the floor or body part to help reposition object in the hand; or manipulates object in the hands, which does not include simple picking up, grasping, holding, throwing, and shaking. Motor imitation—Child imitates hand motions (pushing, rubbing, tapping, banging hammer, squeezing, turning, pointing, shaking rattle), facial movements (blowing, kissing, sticking out tongue), or other body movements that he or she has seen the caregiver perform; includes movements performed after verbal prompting if the movement was also demonstrated. The imitated behavior must occur within the same time segment as the stimulus movement. Types of Play All forms of play may be seen in the same segment. Construction play—Child builds or attempts to build with soft blocks. Sensorimotor play—Child examines sensory features of play objects by feeling, looking at, touching, listening to, or exploring the objects but does not need to display any functional use of the objects. Functional play—Child uses objects functionally for their intended purposes, such as covers baby doll with blanket, puts bottle to baby doll’s mouth, hammers ball into hole, rolls ball, talks or babbles on phone, brings phone to ear, presses phone buttons, or builds with blocks. Associative play—Child demonstrates at least one instance of social play or engagement with the parent during the segment. Examples include turn taking, performing 3-point eye gaze between parent and object (parent–object–parent or object–parent–object), imitating, giving, showing, following a verbal direction, and responding appropriately to gestural communication. Behaviors During Feeding Motor stereotypy— Repetitive body movement. Child may engage in wiggling, flapping, or flicking of the fingers or hands; alternate flexion and extension of the fingers; alternate pronation and supination of the forearm; or head rolling, head banging, body rocking, or body swaying back and forth. Object stereotypy—Repetitive movement with an object. Child makes the same movement with an object, such as banging the spoon or spinning the bowl, more than 10 times. Independently scoops—Child uses spoon to scoop food on his or her own. Spoon to mouth—Child brings food to mouth using spoon and places food in mouth. Hand to mouth without spoon—Child brings hand to mouth in the context of feeding, such as putting food item in mouth, pushing food into mouth, stopping food loss, wiping food, or sucking fingers. Plays with food—Child uses food nonfunctionally (not trying to eat it) by, for example, pushing food around tray, throwing food, or mashing food between fingers. Orients toward visual—Child responds positively to visual stimuli, such as mom’s face or moving objects, by moving head or body toward or reaching for stimulus; child visually tracks spoon as it is brought to mouth. Orients toward auditory—Child responds positively to auditory stimuli, such as mom’s voice or other environmental sounds, by turning head toward, moving body toward, reaching for, or looking toward the stimulus. Orients toward tactile—Child responds positively to tactile stimuli, such as parent’s touch or contact with food, by turning head toward, moving body toward, or repeatedly touching the stimulus; child plays with food or licks food off lips or hands. Aversive response to visual—Child reacts negatively to visual stimuli, such as caregiver’s face or moving spoon or bowl, by closing eyes, screaming, crying, or turning head or moving away from the stimulus. Aversive response to auditory—Child reacts negatively to auditory stimuli, such as mom’s voice or other environmental sounds, by covering ears, screaming, pushing stimulus away, crying, or turning head or moving away from the stimulus. Aversive response to tactile—Child reacts negatively to tactile stimuli, such as holding spoon or feeling food on hands, by withdrawing hands; shaking hands to remove food; pushing food, bowl, or spoon away; or moving body away from the stimulus. Sits upright—Child sits upright in high chair without slouching or needing repositioning by the parent. Motor imitation—Child imitates hand motions (pushing, rubbing, tapping, pointing, scooping) or facial movements (blowing, kissing, sticking out tongue) that he or she has seen the caregiver perform; includes movements performed after verbal prompting as long as the movement was also demonstrated in the same segment. 566 September/October 2012, Volume 66, Number 5
  • 12. Reproduced with permission of the copyright owner. Further reproduction prohibited without permission.