FROM BEHAVIOR TO BIOLOGY: EXAMINING OXYTOCIN, SOCIAL
COGNITIVE ABILITY, AND PARENT-CHILD INTERACTIONS IN
PRESCHOOLERS WITH AUTISM SPECTRUM DISORDER
by
OLENA ZYGA, M.A.
Submitted in partial fulfillment of the requirements for the degree of
Doctor of Philosophy
Department of Psychological Sciences
CASE WESTERN RESERVE UNIVERSITY
May 2019 2
CASE WESTERN RESERVE UNIVERSITY
SCHOOL OF GRADUATE STUDIES
We hereby approve the dissertation of
Olena Zyga
candidate for the degree of Doctor of Philosophy
Committee Chair
Anastasia Dimitropoulos, Ph.D.
Committee Member
Sandra Russ, Ph.D.
Committee Member
Elizabeth Short, Ph.D.
Committee Member
Audrey Lynn, Ph.D.
*We also certify that written approval has been obtained for any proprietary material
contained therein. 3
TABLE OF CONTENTS
List of Tables……………………………………………………………………………...6
List of Figures…………………………………………………………………………….7
Abstract …………………………………………………………………………………..8
Introduction …………………………………………………………………………...... 10
The OXT System, Human Sociality, and Autism…………...…………....……...13
Current OXT Intervention Models in ASD…….…………;…………..…...... 20
Early Parent-Child Interactions and the OXT System ……….…………...……. 22
Current Behavioral Intervention Models in ASD…………..……………..……. 26
Aims & Hypotheses ……………………………………………………………………. 29
Method …………………………………………………………………………………. 31
Participants ……………………………………………………………………... 31
Inclusion/Exclusion Criteria …………………………………………...………. 32
Recruitment …………………………………………………………………….. 33
Measures ……………………………………………………………………….. 33
Parent Report of Child …………………………………………………. 33
Parent Report of Self …………………………………………………… 35
Child Assessments ……………………………………………………... 36
PRETEND-ASD Intervention Program ………………………………... 46
Procedure ………………………………………………………………………. 49
Results ………………………………………………………………………………….. 51
Power Analyses ……………………………………………………………...... 51
Data Coding & Interrater Reliability………………………………...…………. 52 4
AIM 1: Baseline Characterization …………………………………………...... 52
Hypothesis 1…………………………………………………………………….. 52
Demographics ………………………………………………………….. 53
Basal peripheral OXT concentration levels across groups …………….. 54
Social cognitive ability across groups ………………………………….. 54
Parent Functioning across groups …………………………………...... 55
Parent-child interaction across groups …………………………………. 57
Hypothesis 2…………………………………………………………………….. 57
Relationship between OXT, social cognitive ability, and PCI ……...... 59
Whole Sample ………………………………………………….. 59
TD Sample ……………………………………………………... 60
ASD Sample ……………………………………………………. 62
AIM 2: Intervention …………………………………………………...……….. 64
Hypothesis 1…………………………………………………………………….. 64
Feasibility ………………………………………………………..……... 64
Efficacy…………………………………………………………………………. 65
Pre-post OXT expression ………………………………………. 65
Pre-post social cognitive ability ……………………………...... 65
Pre-post parent functioning……………………………………... 66
Pre-post parent-child interaction ……………………………….. 67
Discussion ………………………………………………………………………...……. 67
Summary of findings ………………………………………………...…….…… 67
Group Comparisons …………………………………………………...…...... 70 5
Baseline Characterization………………………………………………. 70
Social Cognitive Functioning and OXT Levels ………………….…….. 74
Intervention Effects ……………………………………………………….……. 77
OXT Malleability……………………………………………………….. 77
Behavioral Outcomes………………………………………………...... 79
Refining the PRETEND-ASD Intervention……………….……………. 82
Limitations and Future Directions……………………………………...………. 83
Practical Significance and Conclusion …………………………………………. 85
Tables …………………………………………………………….…………………….. 87
Figures ……………………………………………………………………………….....106
Appendices ……………………………………………………………………………. 108
Appendix A: Linear Regressions ……………………………………….…….. 108
Table 20: Linear Regressions-Baseline OXT across Whole Sample …. 108
Table 21: Linear Regressions-Baseline OXT within TD Sample …….. 109
Table 22: Linear Regressions-Baseline OXT within ASD Sample .…...110
Appendix B: Training Protocol……….……………………………………….. 111
References…………………………………………………………………………...….113
6
LIST OF TABLES
Table 1: Participant Demographics ……………………………………………….……..87
Table 2: Baseline Oxytocin Across Groups ...…………………………………………...88
Table 3: Pretend Play Ability Across Groups (APS-P) ...…………..……………….…. 89
Table 4: Social Cognitive Ability Across Groups ……………………………………... 90
Table 5: Parenting Stress Across Groups (PSI-4) …………………………………...... 91
Table 6: Parenting Functioning Across Groups ………………………………………... 92
Table 7: Parent-Child Interaction Across Groups ………………………………...…...... 93
Table 8: Correlations-Baseline OXT & Social Cognition Across Whole Sample...... 94
Table 9: Hierarchical Regression Predicting Baseline OXT (Whole Sample)………...... 95
Table 10: Correlations-Baseline OXT & Social Cognition within TD Sample...... 96
Table 11: Hierarchical Regression Predicting Baseline OXT (TD Sample)...…………..97
Table 12: Correlations-Baseline OXT & Social Cognition within ASD Sample………..98
Table 13: Hierarchical Regression Predicting Baseline OXT (ASD Sample) ...... 99
Table 14: Change in Oxytocin Level after Intervention in ASD Sample ………..…….100
Table 15: Change in Pretend Play Ability (APS-P) After Intervention in ASD...... 101
Table 16: Change in Social Cognitive Ability After Intervention in ASD …...………..102
Table 17: Change in Parenting Stress After Intervention in ASD ……………………..103
Table 18: Change in Parent Functioning After Intervention in ASD ………...………..104
Table 19: Change in Parent-Child Interaction After Intervention in ASD ……...... 105
7
LIST OF FIGURES
Figure 1: Study Visit Timelines ………………………………………………………..106
Figure 2: PRETEND-ASD Parent Training Program Intervention Schedule ………….107
8
From Behavior to Biology: Examining Oxytocin, Social Cognitive Ability, and Parent-
Child Interactions in Preschoolers with Autism Spectrum Disorder
Abstract
by
OLENA ZYGA, M.A.
Individuals with ASD show pervasive deficits in social cognition. Recently, the
hormone oxytocin (OXT) has been implicated in playing a role in the social difficulties
individuals with ASD face. Given a lack of preliminary research in characterizing OXT
expression, a focus on better understanding the expression of OXT and how malleable
hormone expression is to behavioral intervention alone is warranted. Thus, the current
study aimed to (1) characterize child basal peripheral OXT concentration levels, social
cognitive ability, parent functioning, and parent-child interactions in children with ASD
and typical development (TD) and (2) measure the impact a telehealth parent training
program had on OXT levels, social cognitive ability, parent functioning, and quality of
parent-child engagement in children with ASD. To meet these aims, 18 children with
ASD (ages 3-5 years) and 21 age-matched TD peers were recruited to undergo peripheral
OXT collection via saliva sample and measures of social cognitive ability and parent-
child engagement. 9
Results showed that the two samples did not differ in OXT concentration levels at
baseline and children with ASD had significantly lower scores across all domains as
compared to the TD group. Concerning intervention efficacy, a main finding was that
there was no significant change from baseline to post-intervention in OXT concentration
levels. Participants with ASD did show significant increases in their emotion recognition
and overall mood during the parent child interaction. Parents of children with ASD
showed significant decreases in levels of reported depression and stress.
The findings from this study suggest that the relationship of OXT with social
cognitive ability and disorder category are complex and nuanced. Results do not support
the concept of an “OXT deficit” in children with ASD. Oxytocin may have unique relationships with social cognitive variables across different groups and levels of functioning. A remote parent-training intervention program was shown to be effective in targeting emotional recognition, parental levels of stress and depression, and the overall mood of parent-child engagement. Taken together, these findings suggest that further
investigation of oxytocin in the preschool period is warranted and may have implications
for early identification and treatment.
10
From Behavior to Biology: Examining Oxytocin, Social Cognitive Ability, and Parent-
Child Interactions in Preschoolers with Autism Spectrum Disorder
Social cognition refers to an individual’s stable pattern of processing social information and regulating emotions. The development of social cognition is influenced by a host of biological and environmental factors and relates to a variety of positive outcomes (Olson & Dweck, 2008). Overall, research has shown that typical trajectories of social cognitive development lead children to develop important skills such as joint attention and facial and emotional processing, which leads to new social encounters and heightened awareness and interest in other people (Choudhury, Blakemore, & Charman,
2006). This leads children to seek out social interaction from parents and peers in continuing to develop socioemotional skills (Olson & Dweck, 2008).
It is also well established that individuals with autism spectrum disorder (ASD) show pervasive deficits in social cognition (Geschwind & Levitt, 2007; Hobson, 1993).
These deficits have led to significant functional impairment and overall decreased quality of life, not only for individuals with ASD, but also their family members (Gray, 2006;
Higgins, Bailey, & Pearce, 2005). In particular, a large majority of research within the autism population has focused on areas of social cognition relating to theory of mind, imitation, joint attention, eye gaze processing, pretend play, emotional processing, and social engagement (Geschwind & Levitt, 2007; Hobson, 1993; Senju, 2013). Consistent lines of research have found that individuals with ASD show deficits across the domains listed above that present early in development (Senju, 2013). Specifically, studies have found that preschoolers with ASD show social attention impairments such as social orienting, joint attention, and attention to other’s distress that are more severe than 11
mentally-aged matched children who present with developmental or intellectual delay
alone (Dawson et al., 2004). In terms of social impairment, individuals with ASD have
been characterized as having deficits in understanding and engaging in the pragmatics of
social interactions, such as turn-taking, distancing, greetings, regulating volume of voice,
a tendency to dwell on certain topics, and difficulty understanding and expressing
emotions (White, Keonig, & Scahill, 2007). Taken together, these social deficits make it
difficult for children with ASD, who present at disinterested and unmotivated, to connect
with their parents or typically developing peers. This disconnect in interactions makes
reciprocal engagement difficult in this population, which can negatively impact
development through limited parent-child and peer bonding (Dawson et al., 2004; White
et al., 2007). Deficits in pretend play ability are also another factor that impacts a child
with ASD’s ability to engage with parents or peers. Research has shown that preschoolers
with ASD have significant difficulty in producing pretend play, such as using toys “as if”
they were something else (i.e. a pen as a rocket ship) and also in expressing emotions in
play (Jarrold, 2003; Kasari, Freeman, & Paparella, 2006). Interestingly, research has also shown that structuring play periods for young children with ASD and introduction of
affect and imagination by an adult play partner does indeed lead to increases in symbolic
play ability (Kasari et al., 2006; Zyga et al., 2015). These findings suggest that early intervention, which uses play-based techniques, may be beneficial for this population.
However, overall, children with ASD show consistent and pervasive deficits across the above described domains (Short, Noeder, Gorovoy, Manos, & Lewis, 2011). Given this,
ASD has thus been characterized as a disorder of social communication and interaction with restricted, repetitive patterns of behavior, interest, or activities (APA, 2013). 12
A strong research base has led to the understanding of the behavioral deficits
evident in ASD and how they present across development. An important next step in order to meaningfully target these deficits is to characterize the biological underpinnings of social cognitive impairment in ASD as compared to typical development. Most recently, a burgeoning literature has begun to emerge, which implicates the hormone oxytocin, a nine-amino neuropeptide synthesized in the hypothalamus (OXT), in playing a role in the social difficulties individuals with autism and other disorders face (Carter,
2007; Heinrichs & Domes, 2008). The implication of OXT in social behavior has led to various studies in which OXT is used pharmacologically as an intervention option.
However, in total, these studies show mixed results, which has left some questioning the efficacy and importance of OXT in human social cognitive ability and development
(Young & Barrett, 2015). Given the lack of preliminary research in characterizing oxytocin expression across typical and atypical development and the mixed findings from intranasal intervention studies, it is imperative that the field continues to move forward by first taking a step backward and focusing on better understanding the basal expression of OXT across various ages and developmental trajectories and how malleable hormone expression is to change. This will lead to a better understanding of the unique versus common factors across development, which is vital in understanding the difficulties children with ASD face, current treatment models available to these individuals, and further developing efficacious behavioral and pharmacological interventions to target these sometimes debilitating deficits.
Given this need for further research, the proposed study aims to: (1) characterize peripheral basal concentration levels of OXT, social cognitive ability, parent functioning, 13
and parent-child interactions in preschool children, ages 3-5, who either have ASD or are
typically developing and (2) pilot a telehealth play-based parent training intervention to understand the impact it may have on OXT levels and social cognitive ability in parents and children with ASD. This project intends to extend the field’s current understanding of
OXT expression across typical and atypical development, its relationship to other developmental factors, and also be the first to examine if OXT levels are malleable to behavioral intervention alone.
The OXT System, Human Sociality, and Autism
As a species, humans are innately social beings. Indeed, some of the earliest and most important developmental milestones center on being able to connect and interact with others (Carter, 2014; Hammock, 2015; Donaldson & Young, 2008). On the whole, the origins of human social behavior have been theorized to have multiple genetic and physiological factors that have interacted with the environment over thousands of years
(Carter, 2007, 2014). One such factor that has garnered support in its role in human social development is oxytocin (OXT), a 9 amino acid peptide hormone composed of a 6 amino acid ring and a 3 amino acid tail (Carter, 2014). Interestingly, the genes believed to be responsible for modern day oxytocin peptides are thought to have evolved more than 700 million years ago (Donaldson & Young, 2008). It is theorized that these genes initially regulated processes at the cellular level related to water balance and homeostasis and, in the course of evolution, acquired new functions such as regulating complex social behavior (Carter, 2014). In humans today, OXT has been implicated in social processes such as selective social and pair bonding, social communication, empathy, feelings of reciprocity, expressing emotions and understanding other’s mental and emotional states, 14
modulating social stress, mother-infant interactions, lactation, gestation, and even the biological processes underpinning birth (Carter, 2014; Hammock, 2015; Donaldson &
Young, 2008).
In humans, OXT is synthesized in cells located most predominately in the
hypothalamus, in particular, the paraventricular nucleus (PVN) and supraoptic nuclei of
the hypothalamus (Hammock, 2015). OXT does not act as a classic neurotransmitter in
that its expression is not limited to local actions in the brain by crossing synapses
between an axon and dendrite (Carter, 2014). Instead, research has shown that OXT is released from a neuron’s soma (cell body), axons, and dendrites, implicating it as a neuromodulator. For example, there is evidence that OXT from the PVN can quickly travel to the amygdala and modulate emotional functions and expression (Carter, 2014;
Donaldson & Young, 2008). Further, OXT can be released from the brain into general circulation and the peripheral nervous system (Carter 2014). Given this fact, OXT can be available in high levels in the brain, peripheral nervous system, blood, and saliva. Recent research using mass spectrometry has verified the assumption that OXT is present and measureable in both blood and saliva samples and that these levels have been shown to correlate to central nervous system levels in animal studies (Donaldson & Young, 2008;
Hammock, 2015; Weisman et al., 2012). Specifically, researchers have shown that when rats and mice are given an intranasal dose of OXT, similar increases are shown in peripheral OXT levels as well as in the dorsal hippocampus and amygdala within the brain (Neumann et al., 2013). This suggests that in human studies, peripheral measures of
OXT and intranasal introduction of this hormone may be related to and impact central nervous system functioning (Carter, 2014; Hammock, 2015; Donaldson & Young, 2008). 15
Levels of OXT vary across species and individual differences in hormone expression are common (Carter, 2007, 2014). Most individual variation relates to traits, such as personality factors, or the presence of certain disorders, such as autism, schizophrenia, and genetic syndromes such as Williams syndrome (Carter, 2014; Dai et al., 2012; Modahl et al., 1998). Previous research has found that personality factors, such as extroversion, introversion, and emotional openness impact basal hormone expression
(Hammock, 2015). A study conducted by Luminet et al. (2010) showed that individuals low in emotional expression were more positively impacted by intransal OXT expression while completing a theory of mind task versus those who were rated as more emotional open. Further, Human et al. (2016) found that, in a double-blind randomized placebo controlled study, a significant interaction between OXT administration and extraversion was predictive of prosocial outcomes. Specifically, those high in extraversion expressed elevated levels of OXT so the introduction of the hormone intranasally did not lead to significant improvements in positive behavioral responses. However, those who were characterized as low on extraversion did see significant gains in social behavior when given intranasal OXT (Human et al., 2016). Another factor that can impact OXT expression is time of day. In a seminal study on OXT release, Amico et al. (1983) obtained sequential samples of peripheral and central OXT levels from hospitalized patients at 0600 h, 1200 h, 1800 h, and 2400 h. The researchers found that the highest levels of OXT were expressed at 1200 h (6.41 ± 1.13 μU/ml) and significantly differed from 0600 h (2.50 ± 0.65 μU/ml), 1800 h (2.63 ± 0.61 μU/ml), and 2400 h (2.86 ±
1.13 μU/ml) (Amico et al., 1983). Taken together these results suggest that there are key factors that should be considered when measuring OXT expression to ensure that values 16 reflect endogenous release of the hormone versus other confounding factors. OXT
Characterization Research
To date, few studies on basal expression of OXT and its developmental trajectory have been conducted. Across typically developing individuals, OXT release has been measured in relationship to child rearing in women and effects of OXT expression on anxiety levels (Heinrichs & Domes, 2008). Specifically, studies have shown that in lactating women, OXT levels increase following breast-feeding and this increase is associated with lowering levels of cortisol and other stress hormones (Heinrichs et al.,
2002). Further research has also shown that individuals who maintain a steady release of
OXT while experiencing sadness have lower levels of anxiety in close relationships
(Ditzen et al., 2007). In sum, most work characterizing OXT expression in typical development has been conducted in the adult population and has related to very specific aims, such as better understanding OXT release in regards to child rearing and its potential role in modulating psychosocial stress. Limited work has been done on understanding how OXT release varies as a function of age and developmental stage.
This project will help fill this gap in providing information on how OXT is expressed in a young age group, across both typical and atypical development, and in relation to important domains of development.
In comparing ASD to typical development, only a handful of studies have measured basal levels across childhood and adolescence through varied methods of either saliva and/or blood samples (Alabdabi et al., 2014; Al-Ayadhi et al., 2005; Feldman,
Golan, Hirschler-Guttenberg, Ostfeld-Etzion, & Zagoory-Sharon, 2014; Green et al.,
2001; Miller et al., 2013; Modahl et al., 1998; Parker et al., 2014; Tourines et la., 2014). 17
The earliest study conducted by Modahl et al. (1998) measured midday plasma levels of
OXT from a sample of 29 boys with ASD (mean age: 8.1 years) and 30 typically developing boys (mean age = 8.8 years). Specifically, participants were asked to fast and subsequently had blood draws within 2 hours of noon. It should be noted that the authors reported values of 5 subjects from each group, who did not fast, and included these in the
results, which could have impacted the accuracy of OXT level reported across groups.
Findings from the study suggested that the ASD sample had significantly lower plasma
OXT levels than the typically developing group. Further, lower peripheral OXT levels
were associated with lower scores on measures of social cognitive behavior, as measured
by the Vineland Adaptive Behavior Scales (VABS). Green et al. (2001) extended these
findings by measuring plasma OXT and OT-X, a precursor molecule of oxytocin, to
better understand if there were changes in oxytocin peptide formation in children with
ASD. In this study, 28 males with ASD and 31 typically developing males, ages 6-11
years, underwent a single blood draw. Results showed that the ASD group had
significantly lower OXT plasma levels (0.8 pg/mL ± 0.09) and significantly higher
precursor molecule OT-X levels (2.9 pg/mL ± 0.29) as compared to the typical control
group (mean OXT level = 1.4 pg/mL ± 0.10; mean OT-X level = 2.0 ± 0.22). This
suggests that children with ASD may not only express low levels of OXT, but also show
a disruption in the production of OXT from OT-X precursor molecules. Al-Ayadhi et al.
(2005) and Miller et al. (2013) both incorporated female participants into their samples
and found contradictory results in assessing plasma OXT levels. Al-Ayadhi et al. (2005)
measured OXT levels across 77 children with ASD and 77 typically developing children,
ages 3.5 – 14 years. These authors found decreased levels of OXT in the ASD group as 18
compared to the TD control group. Conversely, Miller et al. (2013) collected blood
samples from 40 high-functioning children with ASD (19 girls, 21 boys) and 35 typically
developing children (16 girls, 19 boys), ages 8-18 years. Results showed no significant
effects of diagnosis on OXT levels. However, across all participants, a significant gender
effect was found in that females showed significantly higher levels of OXT as compared
to males across this age range. These studies were pivotal in the inclusion of female
participants, however, the differing ability levels in each sample (lower vs. high
functioning individuals) may have impacted the ability to compare findings between
these two studies and previous research. Parker et al. (2014) extended this work in the
largest characterization study to date with 193 participants, ages 3-12 years, split between
a sample of children with ASD or those who were typically developing. This study found
no significant differences between groups, however oxytocin expression was related to
social cognitive ability across both ASD and TD development. A further examination of
OXT concentration across different diagnostic categories was done by Tourines et al.
(2014), who compared baseline levels in children with ASD, attention deficit hyperactive
disorder (ADHD), and a typical control group, ages 6-17. This study showed that children
with ASD had levels that did not differ from the TD group and were actually higher than
the ADHD group. In regards to the age range being examined in the current study,
Feldman et al. (2014) is the only project to date that measured peripheral OXT levels
through saliva collection in a sample of 40 preschoolers diagnosed with ASD (mean age
= 63.38 months) and 40 typically developing preschoolers (mean age = 53.56). Children
with ASD showed lower baseline OXT levels (40 pg/mL) as compared with the typical control group (70 pg/mL). However, this study extracted OXT levels via saliva versus 19
blood, which provides a barrier to comparing the findings to previous work. As indicated
above, OXT can be extracted via blood draws or saliva collection. Research has shown
that OXT extraction via saliva sample is valid, reliable, and extremely less invasive than
requiring a young child to undergo a needless blood draw (Carson et al., 2015). In
particular, salivary OXT has been found to be associated with plasma OXT and genetic
variability in OXTR receptor gene, which also suggests coordination between central and
peripheral activity on the oxytocin/vasopressin neuropathway (Feldman et al., 2014;
Weisman et al., 2012). Given that OXT extracted via saliva is a reliable way to measure
peripheral expression and that the level of harm and risk is much lower than blood draw,
it has become a standard method in this area of research. Overall, OXT characterization
findings show that children with ASD have either normal or decreased levels of
peripheral OXT, which may vary as a function of gender or disorder severity. These
mixed findings have led some researchers to posit an “OXT deficit hypothesis” in ASD,
which is not fully supported by the field (Young& Barrett, 2015).
Lastly, only one study has investigated the effects of parent-child engagement and synchrony on peripheral OXT expression in ASD (Feldman et al., 2014). This study found that, at baseline, 40 preschool children with ASD (mean age = 63.38 months) expressed significantly lower levels of OXT (40 mg/mL) than age-matched typically-
developing (TD) peers (70 pg/mL). Then, after engaging in 20 minutes of parent-child
interaction, the ASD group’s OXT levels increased (70 pg/mL) and remained elevated for a short period of time, whereas the TD group showed stable high levels of OXT expression from baseline through the parent-child interaction period (70-80 pg/mL). This finding provides initial evidence for the theory that peripheral OXT levels may be 20 malleable to behavioral intervention alone in populations that have altered OXT expression. However, a large limitation in integrating these results, as detailed above, is that studies have used different methodologies in terms of sample characteristics, collection of samples for analysis (blood vs. saliva) and OXT extraction procedures.
Further, across studies, others variables that impact OXT expression, such as time of day or child temperament, have also not been controlled for. Given this variability, studies are needed that use validated measures of OXT, well characterized and representative samples, and control for variables across typical and atypical development.
Current OXT Intervention Models in ASD
Regardless of the field’s limited work in characterizing hormone expression across development, many new OXT intranasal pharmaceutical trials are underway in both children and adults with ASD (Dadds et al., 2013; Einfeld et al., 2014; Guastella et al., 2015; Kuppens et al., 2016). The rationale for these studies comes from findings that, in typically developing individuals, bursts of intranasal OXT produce increases in emotional and facial recognition, feelings of trust, and cooperative behavior and decreases in stress and anxiety (Heinrichs & Domes, 2008). These findings suggest that administering OXT as an intervention to children with social cognitive deficits may naturally increase their ability to attune to caregivers and peers, which could then lead to increased engagement and facilitate social development. In typical developing adults, an early study conducted by Heinrichs et al. (2003) showed that participants who received an intranasal dose of OXT in combination with social support from their best friend showed significantly decreased biological and behavioral stress responses as compared to those participants who received only social support. In regards to social behavior, 21
Kosfeld et al. (2005) gave a group of typically developing adults a single dose of 24 IU intranasal OXT and had them complete a trust game with a group of unfamiliar individuals. In comparison to a control group that did not receive OXT, the treatment group showed a substantial increase in trust, which also significantly impacted their performance in the study’s trust game task. Lastly, two additional studies have shown that single doses of 24 IU OXT improves an individual’s ability to read emotional cues and increases duration of gazes towards the eye region of emotionally neutral human faces (Domes et al., 2007; Guastella et al., 2008). These results suggest that OXT may play a key role in facial processing and interpersonal communication in humans (Domes et al., 2007; Guastella et al., 2008).
In clinical populations, the findings are much more mixed, with some recent studies showing no change in behavior after randomized OXT trials, while others suggest promising results (Hollander et al., 2003; 2007; Anagnostou et al., 2012; Young &
Barrett, 2015). A recent systematic review conducted by Preti et al. (2014) found 7 RCTs, including 101 subjects with ASD ranging in age from adolescence to adulthood. Four of the studies showed improvements in primary outcomes relating to reduction in repetitive behaviors (Hollander et al., 2003), improved comprehension of affective speech
(Hollander et al., 2007), improved performance on a theory of mind task in 60% of participants (Guastella et al., 2010), and eye gaze frequency improvement (Hall et al.,
2012). However, the majority of these studies also had mild to moderate side effects, such as drowsiness, anxiety, depression, headache, backache, feeling tired, and irritability. In three of the studies reported on, there were no statistically significant differences between groups or both the treatment and placebo groups made 22
improvements on measures relating to repetitive behaviors, parent-child social interactions, and theory of mind tasks (Anagnostou et al., 2012; Andari et al., 2010;
Dadds et al., 2013).
Early Parent-Child Interactions and the OXT System
Research has also shown that early parent-child interactions strongly impact the
emergence of skills relating to understanding other’s mental states, empathy, social
cognition, and emotional understanding. A parent’s ability to show emotional support,
synchrony and cohesion, and coordinated joint engagement during early interactions with
their preschool-aged child relates to higher sociability and adaptive functioning later on
in life (Adamson, Bakeman, Deckner, & Nelson, 2012; Haven, Manangan, Sparrow, &
Wilson, 2013). Recent research provides initial support for the concept that these early
parent-child interactions also shape biological systems related to social functioning,
specifically in relation to OXT, which has been implicated in processes related to pair
bonding, increased facial processing, social play, and understanding the emotional and
mental states of others (Carter, 2014; Donaldson & Young, 2008). Specifically, research
has shown that OXT functioning is transferred from parent to child through patterns of
parental care and interactions such that parent’s OXT levels predict child’s OXT levels
(Feldman, Gordon, Influs, Gutbir, & Ebstein, 2013). In addition, a child’s social
reciprocity with a friend is associated with child OXT levels, mother’s OXT related genes
and hormones, and mother-child reciprocity during early interactions (Feldman et al.
2013; Pratt et al., 2015). Specifically, human studies have shown an association between
peripheral levels of OXT and genetic variability of the OXT receptor gene (Oxtr) with
sensitive parenting. Those parents who are not able to provide a warm and attentive early 23 environment have been shown to express lower levels of peripheral OXT during social interactions and are also more likely to express certain allelic variation of the Oxtr gene
(OXTR rs2254298, rs1042778). Further, the children of these parents also express lower peripheral levels of OXT during peer engagement and express similar allelic variations, suggesting both a behavioral and biological synchrony which implicates the oxytocin system across generations (Feldman et al., 2013).
For atypical development, many researchers have theorized a potential link between OXT function and social cognitive deficits in individuals with ASD, given the role OXT plays in human social behaviors (Carter, 2007; Heinrichs & Domes, 2008;
Young et al., 2002). In providing evidence for this theory, a recent focus has shifted towards understanding Magel2 mutations. Specifically, Magel2 is a paternally expressed gene implicated as a potential candidate gene that may contribute to specific phenotypic components of ASD, such as deficits in social cognitive functioning (Schaaf et al., 2013).
Recent animal models suggest that Magel2 mutations induce deficits in social recognition and interactions that are accompanied by anatomical and functional modifications of the
OXT system (Meziane et al., 2015). Extending these findings into human models, studies have begun to provide evidence for the role of the Oxtr gene in the development of ASD
(McCauley et al., 2005; Lauritsen et al., 2006). A study conducted by Lerer et al. (2008) investigated the occurrence of Oxtr gene polymorphisms across a sample of 152 subjects diagnosed with ASD. Results found significant associations with five specific polymorphisms associated with the Oxtr gene that also significantly related to social cognitive ability as measured by the Vineland Adaptive Behavior Scales (Lerer et al.,
2008). These findings have since been replicated by several groups suggesting the 24 implication of certain mutations to the Oxtr gene in individuals with autism (Campbell et al., 2011; Liu et al., 2010; Park et al., 2010) Taken together, OXT may play a causal role in the social difficulties that individuals with ASD display, which may extend to disrupted parent-child interactions evidenced in young children in this population (Carter,
2007).
Indeed, research suggests that a large majority of young children with ASD exhibit abnormal parental or caregiver attachment interactions, ranging from inappropriate over attachment to failure to discriminate between caregivers and strangers
(Adamson et al., 2012; Sigman & Mundy, 1989; Williams, 2003). Most recently,
Adamson et al. (2012) provided evidence for this disrupted parent-child relationship in measuring engagement across parent-child dyads with ASD, Down’s syndrome (DS), or typical development (TD) during a joint play task. Specifically, 23 children with ASD, 29 with DS, and 56 TD children, ages 18 – 30 months, engaged in a video recorded task with their primary caregiver. Video coding found that coordinated joint engagement, defined as the child’s ability to attend to both the play objects and the play partner during the task, was significantly less likely in children with ASD as compared to the other two groups (Adamson et al., 2012). Previous findings match those of Adamson et al. (2012) in showing that during joint interactions, children with autism spend significantly more time focused on objects available to them to the exclusion of adult partners (Lewy &
Dawson, 1992; McAurthur & Adamson, 1996). During face to face interactions, research has shown that, on the whole, toddlers diagnosed with ASD are significantly less likely than typically developing children to make eye contact, use joint attention (pointing and showing), engage in social turn-taking, imitate gestures or communication, and anticipate 25
social routines (such as peek-a-boo) during play sessions with a parent or caregiver
(Bernebei et al., 1998; Dawson et al., 1990; Gillberg et al., 1990; Williams, 2003). The
potential inability children with ASD have to show these behaviors makes it difficult for
parents to engage with them in a meaningful way during day to day activities. Indeed,
research has shown that lack of eye contact or smiling from a child with ASD then causes
a parent to engage in fewer instances of these behaviors as well (Dawson et al., 1990;
Williams, 2003). This causes a negative cycle of decreased social engagement, which
impacts not only the parent-child relationship, but also later social skills of the child and
the health and well-being of the parent as well (Gray, 2006).
Further, certain parental characteristics and behaviors, such as levels of stress,
depression, or anxiety can also impact parent-child interactions and socioemotional development. Recently, Kujawa et al. (2014) found that history of maternal depression and negative parenting behaviors led to decreased emotion recognition ability in a sample of 511 3-year-old typically developing children. In children who have a developmental disorder, research has shown that parents express high levels of stress, depression, worry, and rumination in coping with the diagnosis of a lifelong disorder and managing their child’s care (Higgins, Bailey, & Pearce, 2005; Myers, Mackintosh, & Goin-Kochel,
2009; Singer, Ethridge, & Aldana, 2007; Tehee, Honan, & Hevey, 2009). These
increased levels can impact a parent’s ability to view their child’s diagnosis as
manageable and can lead to difficulty in engaging with their child and providing a warm
and nurturing environment to build socioemotional skills (Davis et al., 2008; Tehee et al.,
2009). Given the importance of early interactions and the impact they may have on later
skill development, the health and well-being of both the child and caregiver, and the 26
expression of key hormones related to social ability, it is imperative to intervene early
and teach parents skills relating to increasing synchrony, engagement, and emotional
understanding in children with ASD. It is also necessary to further characterize how OXT
and parent and child characteristics relate to these early parent child interactions across
this disorder, where joint engagement and synchrony may be limited. However, much is
still unknown regarding the expression, malleability, and developmental trajectory of
OXT across ASD and typical development, which has limited its role in behavioral
intervention work.
Current Behavioral Intervention Models in ASD
In regards to intervening early, parent training intervention models have been
used in various neurodevelopmental disorders, such as ASD, in targeting skill deficits
seen in these populations. Overall, they have become an accepted and effective option for
increasing parent-child engagement and decreasing the severity of children’s autism
characteristics (Scahill et al., 2012; Webster-Stratton, Reid, & Beauchaine, 2011). Mostly recently, a Cochrane Report released on the efficacy of parent-mediated early interventions for young children with ASD reviewed 17 studies from 6 different countries in determining if significant gains are made using this method of treatment (Oono,
Honey, & McConachie, 2013). The review provided findings that supported the ability of parent-mediated early interventions to create positive change in domains relating to patterns of parent-child interaction, parent synchrony, and improvement in child language comprehension in young children with ASD. Further, there is some evidence that this intervention modality also leads to behavior change and a decrease in negative symptoms related to ASD (Oono et al., 2013). 27
Currently, telehealth, or the use of remote videoconferencing, is gaining a larger
presence in the disabilities community, and more specifically in the ASD population.
Specifically, telehealth is being used both in research and clinical practice for assessment
(Harrell, Wilkins, Connor, & Chodosh, 2014), parent training (Meadan & Daczewitz,
2015; Wainer & Ingersoll, 2015), and direct intervention (Duncan, Velasquez, & Nelson,
2014). Numerous studies have found that parental involvement in telehealth interventions
has allowed for successful training in strategies that improve social-communicative
functioning in young children with disorders such as ASD, Fragile X, and Down
syndrome (Ingersoll, Wainer, Berger, Pickard, & Bonter, 2016; Langkamp, McManus, &
Blakemore, 2015; McDuffie et al., 2016; Wainer & Ingersoll, 2015). In Fragile X,
telehealth has been used to deliver parent-implemented language interventions to young boys with the syndrome, either through video coaching strategies or the use of interactive touchscreen technology (Díez-Juan et al., 2014; McDuffie et al., 2016). In ASD, much
intervention work has been conducted in training parents on behavior management and
functional communication techniques in improving problem behaviors and overall
functioning (Vismara, McCormick, Young, Nadhan, & Monlux, 2013; Wacker et al.,
2013).
Given increased use and acceptability, telehealth and related technology-based
methods of treatment delivery have the potential to significantly augment or even replace
traditional service models. Research suggests that there are numerous benefits associated
with the use of telehealth, such as providing cost-effective intervention options, ease of access to services, and possibly increasing provider system coverage relative to traditional in-person service options (Langkamp et al., 2015; Wainer & Ingersoll, 2015). 28
Via telehealth, users are able to interact directly with clinical researchers and
instructional content, which gives providers an ability to track progress and patient
understanding seamlessly through technological applications, such as active learning
tasks and feedback surveys. The benefits of telehealth technology, the ease of access to
this type of treatment, and initial evidence suggesting the feasibility and efficacy of
delivering assessment, parent training, and even direct intervention to older individuals
via this method suggest that telehealth applications may serve as a promising alternative
to traditional in-person treatment (Ingersoll et al., 2016; Meadan & Daczewitz, 2015).
Important next questions in continuing to understand the feasibility and efficacy of telehealth as a treatment method are: (1) whether a parent training intervention can be effectively delivered to parents of children with ASD that aims to target the quality of parent-child interactions, pretend play, emotional understanding, and peer engagement
and (2) if this type of intervention can impact peripheral OXT levels. If OXT levels can
be changed through behavioral intervention alone, pharmacological intervention may not
be necessary, or may be more appropriate for individuals with more significant deficits.
However, to date, no study has measured OXT levels from pre to post engagement in a
behavioral intervention program. This study aims to provide initial support for the theory
that endogenous OXT levels can be impacted by early behavioral intervention efforts.
Overall, it seems as though the field does not yet have a clear understanding of
how OXT relates to social cognitive ability and parent-child interactions early in
development, how it is altered in neurodevelopmental disorders, and how it can best be
targeted through pharmacological or behavioral intervention. It may be that only certain
individuals with ASD, perhaps those with more severe deficits or lower functional 29
abilities, have abnormal levels of OXT expression. Further, children with ASD may show
very different patterns of OXT expression as it relates to social cognitive ability and
parent-child engagement, which would suggest a need for tailored interventions versus a
more universal treatment approach across the entire autism spectrum.
Summary: Aims & Hypotheses
Given the current status of research in understanding how OXT relates to typical
and atypical development, social cognitive ability, parental functioning, and parent-child interactions, it is evident that more research needs to be done. Specifically, studies are needed that better delineate OXT’s role in ASD and its implications as either a target of or treatment intervention option, either through behavioral or pharmacological means. To this end, the current study proposed to investigate two major aims.
The first aim focused on characterizing child basal peripheral OXT concentration levels, social cognitive ability, parent functioning, and parent-child interactions in children with ASD and typical development (TD). Describing OXT expression and social cognition, as they relate to both behavior and disorder type, will aide in our understanding of the OXT system, its relation to social functioning, and its use as a treatment option. First, it was hypothesized that children with ASD would show significantly lower levels of peripheral OXT (~40 pg/mL) as compared to typically developing preschoolers (~70 pg/mL) as measured through saliva collection at baseline given the only study conducted in the preschool age range (Feldman et al., 2014) showed decreased expression at baseline in the autism group. Further, given previous literature which shows social cognitive deficits in ASD (McGee et al., 1997; Uljarevic & Hamilton,
2012), it was also predicted that the two groups would differ on scores relating to social 30
cognitive ability (APS-P, FACE, social cognitive tasks), parent functioning (PSI-4,
DASS, PAAQ), and a parent-child interaction task. Specifically, the ASD group was
hypothesized to show overall lower scores in these domains. Second, it was hypothesized
that OXT expression would vary by temperament (as measured by the CBQ), social
cognitive ability (as measured by the APS-P, FACE task, and social cognitive tasks) and
quality of parent-child interaction regardless of disorder category. Specifically, those with more severely impacted social cognitive ability and parent-child interactions were hypothesized to show lower levels of OXT expression.
The second aim centered on measuring the impact a telehealth parent training program (Parent-focused Remote Education To ENhance Development in children with
Autism Spectrum Disorder [PRETEND-ASD]) had on OXT levels, social cognitive ability, parent functioning, and quality of parent-child engagement in children with ASD.
Little is known about the effects of behavioral intervention on the quality of parent-child interactions and if OXT levels can change as a function of this treatment modality.
Because such limited work in this area has been conducted, it is imperative to first pilot the effects of parent-training on OXT expression and parent-child engagement before designing a larger intervention trial. Given this, the current study aimed to provide
preliminary evidence for the effects of the PRETEND-ASD program in a treatment group
of children with ASD to see if any positive effects were shown before introducing a
control group. Lack of a control group was a limitation of the present study, however, previous research in children and adolescents with ASD has shown that this population evidences disrupted OXT expression and parent-child interaction, as described above, which do not reverse themselves naturally over time (Adamson et al., 2012; Carter, 31
2014). This suggests that if gains were seen in the ASD group, it would warrant further
research, with stricter control conditions to further clarifying what external factors may
impact change in OXT expression over time. It was hypothesized that children with ASD
who undergo the PRETEND-ASD intervention would show gains in their social
cognitive ability (as measured by the APS-P, FACE task, and social cognitive tasks) and quality of parent-child engagement (as measured by the parent-child interaction task), as
well as a significant increase in OXT expression.
Method
Participants
Thirty-nine children (18 ASD; 21 TD) between the ages of 3-5 years participated
in the current study. Given that ASD encompasses a heterogeneous group of individuals,
who can present with a range of intellectual, language, and behavioral concerns,
recruitment of participants followed a series of matching guidelines to ensure a more
homogenous ASD sample that could be compared to the TD comparison group in terms
of basic demographics. Specifically, all participants were between 3 years 0 months to 5
years 11 months of age. Further, ASD is known to be more common in males than
females, with current estimates suggesting that diagnosis is 4.5 times more common in
boys than in girls (Loomes, Hull, & Mandy, 2017). Given this, the gender split of the
ASD sample reflected this general population difference with 14 male participants
(77.8%) in the current study. Within the TD group, gender was split more evenly to
reflect the general population, with 13 male participants (61.9%). In terms of cognitive
and language ability, participants were assessed using the Visual Reception Subscale of
the Mullen Scales of Early Learning (MSEL) and the Peabody Picture Vocabulary Test 32
(PPVT). Results show that the ASD group scored within the low average range in terms of receptive language ability (M = 82.07; SD = 17.93) and within the below average range for cognitive functioning (M = 31.13; SD = 9.33). This suggests that the ASD sample used in the current study fell within a moderate range of functioning, but were not severely impacted by their diagnosis and other developmental concerns.
Lastly, in terms of both behavior and temperament, parents of both the ASD and
TD groups completed an eligibility survey which asked questions that assessed their child’s level of problem behaviors (i.e. my child is able to sit at a table and participate in tasks; please describe to us any behavior concerns your child has currently or in the past?) and their temperament in social situations (i.e. what is your child like in social situations with others he or she knows? What is your child like in new social situations with strangers?). Any child with severe or problematic behavioral issues was excluded from the current study. Further, a mix of children, with both outgoing and introverted social tendencies across both the ASD and TD groups, as measured by the Child
Behavior Questionnaire, were recruited to participate in the current study.
Inclusion/Exclusion Criteria
To be included in the study, participants must have been minimally verbal, able to sit at a table for short periods of time, and independently partake in the assessment tasks in the current study (i.e. able to point, focus attention towards a task). Participants were excluded from the study if they were currently enrolled in an oxytocin trial, taking medication that altered mood or behavior, and/or were enrolled in another play-based or parent training program. Individuals with ASD were required to provide documentation of a primary diagnosis of ASD from a pediatrician, clinical psychologist, psychiatrist, or 33 pediatric neurologist prior to enrollment. Further, ASD participants and their parents must have had access to a desktop PC, laptop computer, or tablet that had built-in webcams, microphones, and speakers, as well as internet connectivity to have partaken in the telehealth intervention. All children included in the TD group had no previous diagnosis of developmental, behavioral, mental, or learning disorder or disability.
Recruitment
Local recruitment and enrollment of participants occurred on a rolling basis over the course of 12 months. Participants with ASD were recruited through the greater
Cleveland area ASD organizations, local preschools and daycare programs, and assessment clinics associated with UH Developmental Pediatrics and The Cleveland
Clinic Learner Autism Center. Typically developing children were recruited through established relationships with local school districts and preschools and also through posting to the online Case Western Community Board. The TD participants underwent a one-time study visit (approx. 90 min) at the primary advisor’s lab at Case Western
Reserve University (CWRU) whereas the ASD sample underwent two in-person visits at baseline and post-intervention that occurred either at CWRU, the child’s home, or at a location near the child’s home.
Measures
Parent Report of Child
Social Communication Questionnaire (SCQ; Rutter, Bailey, & Lord 2003): brief instrument completed by a parent or caregiver that evaluates communication skills and social functioning in screening for an autism spectrum disorder. The survey provides a global cut-off score of 15 with scores above this value indicating a high probability of 34 autism. It is appropriate for use on children with a mental age over 2.0 years and has been shown to be an efficient, valid, and reliable way to obtain diagnostic information or screen for autistic symptoms. In the current study, this measure was used as a confirmation of elevated ASD concerns within the autism sample as compared to the TD group, with a score above 15 indicated clinical levels of autism symptomology. All participants included in the study met this cut-off criterion.
Child Behavior Questionnaire-Short Form (CBQ-SF; Putnam & Rothbart,
2006): parent report survey designed to measure temperament in children ages 3 to 7 years across 12 dimensions, which included Activity Level (level of gross motor activity including rate and extent of locomotion), Approach (amount of excitement and positive anticipation for expected pleasurable activities), Attentional Focusing (tendency to maintain attentional focus upon task-related channels), Inhibitory Control (the capacity to plan and to suppress inappropriate approach responses under instructions or in novel or uncertain situations), Anger/Frustration (amount of negative affect related to interruption of ongoing tasks or goal blocking), Sadness (amount of negative affect and lowered mood and energy related to exposure to suffering, disappointment, and object loss), Fear
(amount of negative affect, including unease, worry or nervousness related to anticipated pain or distress and/or potentially threatening situations), Shyness (slow or inhibited approach in situations involving novelty or uncertainty), Reactivity & Soothability (rate of recovery from peak distress, excitement, or general arousal), High Intensity Pleasure
(amount of pleasure or enjoyment related to situations involving high stimulus intensity, rate, complexity, novelty, and incongruity), Low Intensity Pleasure (amount of pleasure or enjoyment related to situations involving low stimulus intensity, rate, complexity, 35
novelty, and incongruity), and Perceptual Sensitivity (amount of detection of slight, low
intensity stimuli from the external environment). These domains have been shown to load
onto three factors: negative affectivity, surgency and extraversion, and effortful control.
The CBQ-SF was validated on a representative US sample. Internal consistency (alpha)
ranges from .55 to.80 when mothers are the respondents and from .40 to.74 when fathers
complete the questionnaire.
Parent Self-Report
Depression, Anxiety, and Stress Scale (DASS-21; Lovibond & Lovibond,
1995): a 21 item self-report questionnaire intended to assess the severity of core
symptoms of depression, anxiety, and stress in adult individuals. The self-reporter
indicates the presence of a series of symptoms over the past week. Each item is scored
from 0 (did not apply) to 3 (applied to me very much or most of the time). Individuals are
given a standardized score across all 3 domains ranging from normal, mild, moderate,
severe, and extremely severe. The DASS-21 has been validated on a US sample of 508
adults. Cronbach’s alphas were found to be .90 for Depression, .83 for Anxiety, .86 for
Stress. Factor analysis provided support for construct validity in that maladaptive coping
predicted depression, anxiety, and stress (Mahmoud, Hall, & Staten, 2010).
Parental Stress Inventory (PSI-4; Abidin, 2012): A 120 item self-report
questionnaire that assesses the magnitude of stress in the parent-child system. Items factor into 3 different domains of stress: child characteristics, parent characteristics, and situational/demographic characteristics. The child domain includes 6 subscales relating to distractibility/hyperactivity (DI), adaptability (AD), reinforces parent (RE), demandingness (DE), mood (MO), and acceptability (AC). The parent domain includes 7 36
subscales relating to competence (CO), isolation (IS), attachment (AT), health (HE), role
restriction (RO), depression (DP), and spouse/parenting partner relationship (SP). The
last domain includes 19 dichotomous yes/no items that evaluate overall life stress. The
normative sample for the PSI-4 consisted of 534 mothers and 522 fathers for a total of
1,056 adults from the United States. Researchers attempted to select the most stratified sample that they could in terms of ethnicity and education level. Participants came from
17 states in the South, Northeast, West, and Midwest. The average age of participants was 33.62 (SD = 7.64). In terms of reliability, the coefficient alpha for the Child and
Parent domains were both .96. Construct validity is high and research has shown a strong relationship between the PSI and the Child Abuse Potential Inventory (CAPI), the Center for Epidemiologic Studies Depression Scale (CES-D), and the Beck Depression
Inventory (BDI) (Abidin, 2012).
Parental Acceptance and Action Questionnaire (PAAQ; Cheron, Ehrenreich,
& Pincus, 2009): A 15-item questionnaire that assess experiential avoidance, coping
ability, and control behaviors in the context of parenting a child. Factor analysis of the
PAAQ has yielded two-factors, inaction and unwillingness. Temporal stability is
moderate (r = .68 - .74) and internal consistency is fair (alpha = .64-.65; Cheron,
Ehrenreich, & Pincus, 2009). This questionnaire has been used with parents of children
who are typically developing and those with various developmental or physical
disabilities, including ASD (Barney et al., 2017; Defreitas, 2015).
In-Person Child Assessments
Saliva Collection & OXT Extraction: Two saliva samples from each child
participant were collected during the baseline laboratory visit at the following time 37 points: T1 (baseline – approximately 15 minutes after arrival and after consent process),
T2 (following parent-child interaction – directly following the free play parent-child task and approximately 90 minutes from arrival). ASD participants also underwent one saliva collection during the post-intervention visit approximately 15 minutes after arrival and before engaging in any of the assessment tasks. For both the baseline and post- intervention visit, the T1 saliva sample collection occurred between 8-10am to account for the cyclical nature of peripheral OXT expression. Specifically, it is known that peripheral OXT level peak expression occurs at 1200 hours, and this level of expression significantly differs from 0600, 1800, and 2400 hours (Amico et al., 1983). To control for this factor, participant’s saliva collections did not significantly differ in regards to time of day.
Saliva was collected via cotton swab, placed in Sallivette containers, and subsequently stored at -80 degrees Celsius in Mather Memorial Building on CWRU’s campus until shipment to Emory University for extraction procedures, per validated and replicated procedures (Apter-Levi et al., 2013; Putnam, Lopata, Thomeer, Volker, &
Rodgers, 2015). Extracted values for baseline OXT expression levels were provided specifically through the Emory Biomarkers Core lab. Standard Arbor Assay procedures were used for the extraction process per previous research standards (Apter-Levi et al.,
2013; Putnam et al., 2015). Multiple studies have shown that salivary OXT measured by immunoassay are reliable biomarkers, stable over time, and correlate with hormone related processes. As detailed above, salivary OXT is associated with plasma OXT and genetic variability in OXTR receptor gene, which suggests coordination between central and peripheral activity on the oxytocin/vasopressin neuropathway (Feldman et al., 2014; 38
Weisman et al., 2012). OXT levels in pg/mL (pictogram/milliliter) were be obtained for
each participant at each time point (3 levels for ASD participants; 2 levels for TD
participants) via extraction procedures conducted through laboratory services at Emory
University. As detailed in the introduction, when OXT is extracted via saliva sample,
typically developing preschoolers express a range of 70-80 pg/mL pre and post parent-
child interaction, whereas children with ASD in this age range usually express basal
levels around 40 pg/mL which then increase to as high as 70 pg/mL during parent-child
interaction tasks (Feldman et al., 2013; Feldman et al., 2014).
Peabody Picture Vocabulary Test, Fourth Edition (PPVT-4; Dunn & Dunn,
2007): an individually administered measure of receptive vocabulary for standard
American English for ages 2 years 6 months to 90+ years. The measure has shown good
validity and reliability across both typical and atypical populations (Dunn & Dunn,
2007). Overall receptive language ability is reported as a Standard Score (M = 100; SD =
15).
Mullen Scales of Early Learning – Visual Reception Subscale (MSEL;
Mullen, 1995): an individually administered assessment which measures functioning in
infants and children up to 68 months of age across 5 domains (gross motor, visual
reception, fine motor, expressive language, and receptive language). In the current study,
only the visual reception subscale was administered and T-scores for this scale are reported given that previous research has shown that it is a valid and reliable indicator of early cognitive ability across typical and atypical populations (Bishop, Guthrie, Coffing,
& Lord, 2011). 39
Affect in Play Scale – Preschool Version (APS-P, Kaugars & Russ, 2009;
Russ, 2014): The APS-P is a standardized play task designed to measure various dimensions of children’s pretend play. It has been validated for preschool children ages 4
– 5 years. This study was the second to extend its use to 3-year-old children in the hopes of providing continued validity for use of the scale in a younger age range (Zyga, 2016 master’s thesis). In this task, various toys are laid out on a table (cups, stuffed animals, toy car) and children are provided with a story stem and instructions to play with the toys and talk out loud for a 5-minute period.
The child’s play is scored from the videotape using a criterion-based rating scale.
For this study, a modified version of the APS-P scoring system was used, which included
8 original variables in addition to 7 variables created to better measure a wide range of pretense ability and interpersonal domains in pretend play. The original variables included ones that captured cognitive processes in play, specifically: (1) Imagination, (2)
Organization of the storyline, and (3) Comfort in playing with the toys. These variables were all scored on a 1-5 scale; 1 being the lowest ability in that domain. Original variables that measured affective processes included: (1) Frequency of Affect, a total frequency count of affect units expressed within the play narrative and (2) Variety of
Affect, a total count of the number of affect categories out of 11 possible categories expressed during the play. Further, for each 20-second interval, the rater indicates which of three types of play (No Play; Functional Play; Symbolic Play) was the predominant activity – i.e. occurred for greater than or equal to 10 seconds within each 20-second interval. No Play was defined as the child not moving or interacting with the toys.
Functional Play was coded when a child made simple, repetitive muscle movements with 40
the toys or used them in a functional nature, such as stacking the cups, bouncing the ball,
or pushing the car. Lastly, Symbolic Play was defined as any instance of using toys in an unusual manner, substituting an object for another, or using the object in any way other than how it is intended.
The 7 additional variables included two which aimed to measure more detailed and sensitive levels of pretense and imagination ability in pretend play. The first was a
frequency count of the number of symbolic substitutions a child displayed during the 5-
minute play task. A symbolic substitution was defined as when a child used a toy in a
way that was similar to the properties of the toy, yet still imagined it was something else.
For example, the ball as a sun or the cup as a bowl. This occurred when the object used
for a substitution shared some type of overlap in its shape, size, or consistency with the
imagined object. The second variable measured the frequency count of the number of
symbolic transformations a child made during the 5-minute play period. A symbolic transformation was defined as when a child used a toy in a completely “as-if” manner that did not relate to the original properties of that toy. For example, using a cup as a rocket ship or imagining that there are other objects present (i.e. the table is the road; over here is the house). The other 5 variables all measured aspects relating to interpersonal processes in pretend play, namely: (1) frequency count of personification of the toys (i.e. any instance of when human attributes were ascribed to the toys during the 5-minute play
task), (2) frequency count of the number of cooperative/nurture interactions that occur
between the toys, (3) frequency count of the number of aggressive/negative interactions
that occur between the toys, (4) frequency count of the number of neutral interactions that 41
occur between the toys, and (5) frequency count of the total number of interpersonal acts
included in their play period.
Taken together, the Cognitive Domain encompassed 8 variables – Imagination,
Comfort, Organization, Time in No Play, Time in Functional Play, Time in Symbolic
Play, Frequency of Substitutions, and Frequency of Transformations. The Affective
Domain encompassed 2 variables – Affect Frequency and Affect Variety. Lastly, the
Interpersonal Domain encompassed 5 variables – Frequency of Personifications,
Frequency of Aggressive Interpersonal Acts, Frequency of Nurture/Affectionate
Interpersonal Acts, Frequency of Neutral Acts, and Total Number of Interpersonal Acts.
Kaugars & Russ (2009) have developed a detailed scoring manual for the original
8 variables included in the APS-P. Zyga, Dimitropoulos, and Russ created an additional coding system for the symbolic and interpersonal variables. Interrater reliability across all variables is high, consistently in the .80s and .90s. Internal consistency for the affect scores on the APS-P using the Spearman-Brown split-half reliability is also high (0.85).
The APS-P has a growing body of validity studies demonstrating associations with theoretically relevant criteria (see Kaugars & Russ, 2009; Yates & Marcelo, 2014; Fehr
& Russ, 2013; 2016; Russ, 2014).
Facial Affect Comprehension Evaluation (FACE; Mrakotsky, 2001): The
picture labeling task from the facial affect comprehension evaluation was used in the
current study. This task, created for use in preschool children as young as 3 years of age,
used a total of 18 pictures of adult and child faces with different expressions.
Specifically, 3 pictures depicting happy expressions, 4 sad, 2 angry, 3 scared, 2 surprised,
2 disgusted and 2 ashamed faces are used. The experimenter verbally provided the child 42
with a list of possible feelings (happy, sad, mad, scared, surprised, yucky,
ashamed/guilty) and described the meaning of each (e.g., “Ashamed is when you did
something wrong and your mommy yells at you, you feel ashamed”). Each picture was
then presented to the child and he/she was asked to label each one with an emotion word
by pointing to the list of provided options. No feedback on incorrect trials was provided
for this task. The task is scored as to provide mean ability on each emotion presented
along with total accuracy across all emotions, aggregate scores assessing ability in
regards to negative versus positive emotion identification, and a total ability score. This
task has shown to be a reliable measure of preschool children’s ability to recognize and
label emotions (ages 3-6 years) and significantly correlates with other measures of
emotional understanding, such as emotional listening and learning tasks (Kujawa et al.,
2014).
Social Cognitive Tasks (Wade, Hoffmann, Wigg, & Jenkins, 2014): Three
domains were measured as part of the social cognitive observation tasks. All tasks were
video recorded for later coding analysis.
Joint attention: measured a child’s ability to follow the gaze of an adult examiner using a gaze-following task (Carpenter et al., 1998). A child’s ability to redirect attention to a focal object was scored along a 4-point scale for 8 consecutive trials:
1. Immediately Follows Point: The child gets credit if he/she turns his/her eyes or head sufficiently to indicate that he/she is looking in the correct direction and beyond the end of the index finger of the tester, immediately after the examiner points and before the examiner says the child’s name. 2. After Name: The child gets credit if he/she turns his/her eyes or head sufficiently to indicate that he/she is looking in the correct direction and beyond the end of the index finger of the tester, after the examiner says the child’s name. 3. Delayed/After label: If the child looks to the target after the tester’s point has ended (tester put finger or hand down), or after the poster is labeled, but before 43
the next poster is pointed to, give the child credit for the look but note that the response was ‘delayed’. 4. Did not follow: The child is engaged but did not look in the direction of the interviewer’s point at any time during the trial.
A composite score was created based on mean scores across the 8 trials for each participant.
Empathy: measured a child’s responsiveness to the feigned distress of an adult examiner. At standard points during the task, the examiner would pretend to hurt her knee and finger, as well as drop and ostensibly break her favorite toy. A frequency count of the number of times the child engaged in each of the 6 domains below was recorded for each task (hurt finger, hurt knee, and broken toy). Then, a score was given based on each task’s frequency counts on 5-point scale (“very untrue” of the child’s behavior to “very true” of the child’s behavior). The six items scored on each task included:
Comfort - will try to comfort or reassure another in distress (provide comforting statements) Offer - offers helpful statements or suggestions Glance - based on facial expression or gaze, child can tell how the examiner is feeling/if they are in distress Ask - child asks what’s wrong or what happened when seeing the examiner distressed Sorry – child expresses feeling sorry for the examiner when she/he is hurt or unhappy Not upset – child does not become upset when the examiner gets hurt or is unhappy
The 5-point likert scale included:
1 = very untrue (never engages in the behavior) 2 = untrue (partial attempt to engage; looks over) 3 = somewhat untrue (attempts to engage such as moving towards examiner, making some type of action) 4 = somewhat true (at least one clear example) 5 = true (2-3 examples of the behavior) 6 = very true (consistently shows the behavior/understanding the examiner is hurt/needs comforting)
44
Cooperation: measured the child’s engagement in helping an adult experimenter complete a sequence of actions. First, the child was asked to help the experimenter hold
up a napkin as to act as a “trampoline” for a stuffed animal rabbit to jump on. Second, the
child was invited to help the experimenter complete a sequence of actions in which she
rolled a ball down a tube and asked the child to help her hold the tube and catch the ball
at the bottom. For the child to be successful, they could not just imitate the experimenter,
as in task one, but had to have engaged in a complementary behavior to achieve the goal.
Each task was administered and scored over 2 trials.
For the trampoline task, ratings from 1-4 were assigned based on the following criteria:
1 = no success (child does not lift or hold the trampoline) 2 = low engagement (child engages but lots of stopping, low excitement, and child needs a lot of encouragement) 3 = medium engagement (some stopping, but expresses some excitement or desire to try the activity again) 4 = high engagement (continuously interacts with the toys; initiates taking another turn)
For the tube task, ratings from 1-4 were assigned based on the following criteria:
1 = child makes no attempt (child does not touch the tube/roll the ball/catch the ball or engage with the objects) 2 = no success (child tries to engage but does not understand how to help – examiner needs to give multiple prompts and may have to show the child how to do the actions) 3 = some success (child successfully holds the tube or rolls the ball down but only after multiple prompts) 4 = complete success (the child successfully holds the tube, can roll the ball down when it’s their turn – both with minimal/no prompting) The tasks have been validated within toddler to preschool age samples and shown
mean inter-rater reliability of α = .86 (ranging from .68 to .96). All of the items loaded
significantly onto the same factor, explaining 47% of the variance, with item loadings
ranging from .54 to .76, suggesting construct validity (Wade, Hoffmann, Wigg, &
Jenkins, 2014). 45
Parent-Child Interaction Task (Hudson & Rapee, 2001; NICHD): Parent-
child dyads were asked to play with a set of toys (i.e. blocks, small figurines, and cars)
any way they liked for 5 minutes. This interaction was recorded and later coded based on
a modified tangram task coding system (Hudson & Rapee, 2001; Wallace, 2017) and
Mother-Child Structured Interaction Rating Scales (adapted from NICHD Study of Early
Child Care Research Network). Variables which measured aspects of the parent’s
engagement included: (1) Degree of Parental Involvement (2) Unsolicited Help, and (3)
Response to Child. Variables that measured the child’s ability to engage in the task included: (1) Social Interest and (2) Social Competence. Lastly, variables that measured the dyad’s overall interaction included: (1) Overall Mood and (2) Mutual Engagement.
Degree of Parental Involvement, Unsolicited Help, Response to Child, and Overall Mood
were scored on an 8-point scale and Mutual Engagement was scored on a 5-point scale.
For degree of parental involvement and unsolicited help, a score of 1 indicated no help or involvement where a score of 8 indicated over involvement or unnecessary help. For response to child, overall mood, and mutual engagement, a score of 1 indicated a positive response or interaction whereas higher scores indicated a negative response or interaction. Scales that measured Social Interest and Social Competence were taken from the Mother-Child Structured Interaction Rating Scale (NICHD) and were coded on a 7- point scale, where a score of 1 indicated low interest or competence. These measures have been used as valid and reliable measures of the variables above across diverse child populations, including those with ASD (McDonald et al., 2016). Further, detailed coding manuals have been developed and validated and coded variables have shown good internal consistency (intraclass correlation coefficient= 0.85). Intraclass correlations 46 revealed good reliability on all scales (0.80) across all measures coded (Hudson & Rapee,
2001; McDonald et al., 2016).
Intervention
PRETEND-ASD Intervention Program (Dimitropoulos, Zyga, Russ, 2015).
The Parent-focused Remote Education To ENhance Development in children with
Autism Spectrum Disorder [PRETEND-ASD] Program is an 8-week intervention administered remotely via a videoconferencing software to parents of children with ASD.
Overall, the intervention focuses on building 4 core skill areas – (1) engagement and play, (2) improving problem behaviors, (3) emotional understanding and coping skills, and (4) social skills and peer interactions. The PRETEND-ASD program is an adaptation of a validated pretend play based intervention (Moore & Russ, 2008) and a parent- training intervention (PEMB; Tonge et al., 2014). The Russ Intervention has been shown to produce gains in emotion expression abilities and access to coping strategies through increases in divergent thinking abilities. Further, the PEMB intervention has been used within populations with developmental disorders, such as ASD, in targeting emotion regulation abilities, parent-child interactions, and behavioral strategies in diminishing externalizing behaviors. The use of telehealth allowed the interventionist to model actions and concepts for the parents. Further, a similar protocol has been found to be feasible in a preschool population with Prader-Willi Syndrome (PWS) (Zyga, Russ, Dimitropoulos, in press).
Specific modifications were made to the PRETEND-ASD program for use within the ASD population. In particular, session content was tailored to focus on the specific deficits seen across ASD in relation to play ability and social engagement. Sessions 2 and 47
3 focused on psychoeducation on how children with ASD may not naturally find
engaging in play or interactions with others as reinforcing, which in turn makes it
difficult for others to continue to engage with them if there is not this reciprocal
enjoyment. Further, an emphasis was placed on building imaginative play skills versus
more basic skills surrounding functional play, given that previous research has shown
that children with ASD do not show deficits in functional play (Jarrold, 2003; Zyga et al.,
2015). During the middle sessions of the program, a stronger emphasis was placed on
helping parents understand the antecedents and consequences of their child’s behavior in
realizing how this may maintain problem behaviors. Examples and worksheets for these
lessons were taken from the ASD parent training literature (Scahill et al., 2012; Webster-
Stratton, Reid, & Beauchaine, 2011). A stronger emphasis was also placed on developing
an emotion vocabulary for children with ASD and how this could be worked on through
play, given that previous research has shown that emotional expression is difficult for
children with ASD to engage in alone but is malleable to change during adult-supported
play periods (Zyga et al., 2015). Lastly, in building social skills, psychoeducation was
provided specific to this population regarding expectations of interactions and building in
breaks from engaging with others given the sometimes overstimulating nature of social
interactions for children with ASD (White et al., 2007).
The program took place over 8 weeks, with one session a week, each 45-60
minutes in length. The first session focused on describing each child’s social cognitive
profile to the parent. This included results on the various assessment tasks given during
the baseline visit, specifically the APS-P, the FACE task, the social cognitive tasks, and the parent-child interaction task. From these baseline results, the basic structure of the 48
program was tailored to meet the child’s needs. Sessions 2 and 3 focused on how to be a
good play partner, where key concepts relating to joint engagement, joint attention, and
skills such as turning taking and symbolic transformations were reviewed and practiced.
Session 4 focused on the ABC’s of behaviors, specifically how they are formed, what
their function may be, and how to begin to work on changing behaviors through altering
antecedents and consequences surrounding maladaptive behaviors. Session 5 and 6 built
off of behavior change by discussing emotional understanding and how to begin to build
early emotion regulation skills. Lastly, sessions 7 and 8 focused on identifying skills to
work on in building social communication and interaction skills, such as greeting, turn-
taking, joining group play, awareness of personal space and regulating volume of speech
in different situations. Each session began with a “check-in” of how well the parent was
able to implement the skills from the last session. In addition, parents were asked to
complete either worksheet or play-based homework assignments with their child that reinforced concepts such as engaging in play, positive behavior change, emotion expression and labeling, building social communication skills, and the various coping techniques and strategies presented during the sessions. Play-based homework assignments were assigned on weeks 2, 3, 5, 6, and 7 during the program and were video recorded via Zoom to review during session. Worksheet-based homework assignments were assigned on weeks 1 and 4. Further, in-vivo coaching of certain skills relating to play and emotional expression occurred during sessions 2 and 3 of the intervention period via Zoom (see Figure 2 for full details). Specifically, parent and child were asked to engage in an area of difficulty together (i.e. floor time) and the interventionist coached
the parent through any difficulties that arose in real time. 49
Videoteleconferencing (VTC) was used to deliver real-time parent training sessions to participants. Administration of the intervention was in accordance with the
American Telemedicine Association’s current practice guidelines (ATA, 2009). Specific hardware used include: computing device in both the researcher’s office and the participant’s home (desktop PC, laptop Computer, or tablet), webcams, microphones, speakers, and internet connectivity at both sites. Sessions were administered over easy-to- use web conferencing software (Zoom), which is compliant with existing standards and practice guidelines.
Procedure
Baseline Visit. Eligible participants were contacted to schedule an in-person
baseline visit. Before this study visit, parents were mailed the Social Communication
Questionnaire (SCQ; Rutter, Bailey, & Lord 2003), Child Behavior Questionnaire-Short
Form (CBQ; Rothbart, 1997) to assess their child’s temperament, the Depression,
Anxiety, and Stress Scale (DASS-21; Lovibond & Lovibond, 1995) to assess parental
mental health, the Parental Stress Inventory (PSI-4; Abidin, 2012) to assess parenting
related stress, and the Parental Acceptance and Action Questionnaire (PAAQ; Cheron,
Ehrenreich, & Pincus, 2009) to assess parent’s experiential avoidance and ability to cope
with stressors.
Scheduled visits began between 8-10am, to account for time of day and the
cyclical nature of OXT expression. To obtain a baseline sample of OXT, the child was
asked to chew on a cotton swab (Salivette) (T1: approx. 15 minutes after arrival and
consent process). Then the child underwent cognitive and social cognitive assessments,
including the Peabody Picture Vocabulary Test, Fourth Edition (PPVT-4; Dunn & Dunn, 50
2007) to assess receptive language ability, the Mullen Scales of Early Learning – Visual
Reception Subscale (MSEL; Mullen, 1995) to assess cognitive ability, the Affect in Play
Scale – Preschool Version (APS-P; Kaugars & Russ, 2009) to assess pretend play ability,
Facial Affect Comprehension Evaluation (FACE; Mrakotsky, 2001) to assess emotional understanding and recognition, and a series of Social Cognitive Tasks (Wade, Hoffmann,
Wigg, & Jenkins, 2014) that measure a child’s ability to respond to cues of joint attention, empathy, and cooperation. Following assessments, the child engaged in a
Parent-Child Interaction Task (McDonald et al., 2016) with their primary caregiver.
Within 5 minutes after the interaction task, a post-interaction measure of OXT (T2) was
obtained from the child. See Figure 1 for full details.
Intervention. Given the preliminary nature of the current study with the main aim
of establishing feasibility, there was no control comparison group. Thus, all parent-child dyads in the ASD group completed the telehealth PRETEND-ASD program with one session a week for 8 weeks. As outlined above, the sessions were structured around building skills relating to structuring play scenarios, engaging children in play, creating strategies to target problem behaviors, and understanding and building emotional competence, language skills, social communication, and imagination. See Figure 2 for full details.
Post-Intervention Assessment. Participating ASD parent-child dyads were seen
for a face-to-face visit within 2 weeks of intervention completion and underwent the
same set of social cognitive and parent-child interaction assessments, excluding the
MSEL. At the post-intervention visit, OXT was only collected at the start of the study visit (T1). See Figure 1 for full details. 51
Results
Power Analyses
Previous research in OXT characterization has ranged from 5 to 40 participants across developmental disorder samples, including ASD, and typical development
(Feldman et al., 2014; Green et al., 2001; Miller et al., 2013; Modahl et al., 1998). Pilot intervention research in the domain of OXT intranasal administration and telehealth intervention have ranged from samples of 4 to 50 participants across typical and atypical sample groups (Anagnostou et al., 2012; Andari et al., 2010; Dadds et al., 2013). Further, average effects sizes (ES) have ranged from small (0.1) to moderate (0.3) with alpha values less than 0.05 across both characterization and pilot intervention studies (Feldman et al., 2013; Preti et al., 2014).
Given these parameters, a set of power analyses were conducted to ensure proper sample size estimation given the proposed sample (ASD = 15; TD = 15; Total = 30) and statistics for the current study. Power threshold was set at 0.80, which is a standard within the field (Cohen, 1988). In terms of proposed group comparison analyses, with an alpha =
0.05 and a medium effect size (0.5), a sample size of 15 per group (n = 30) gives a power of 0.81, suggesting adequate sample size for these analyses. To conduct correlational analyses, a sample size of 13 with an alpha = 0.05 and a medium effect size r = 0.5 yields a power of 0.83. This provided accurate power for the proposed sample size and also suggests that the obtained sample of 18 children with ASD and 21 TD children was adequate for the analyses conducted in the current study. Further, given the number of correlations conducted, a Bonferroni correction was calculated to adjust the p-value from
0.05 by the number of comparisons made. Lastly, for regression analysis, a total sample 52
of 29 with an alpha = 0.05, a medium effect size (0.30), and 2 predictors in the model
yields a power of 0.81, suggesting that the sample recruited in the current study allowed
for regression models that include either 1 or 2 predictors.
Data Coding & Interrater Reliability
Parent surveys, PTI-2, FACE, and videos of the APS-P, social cognitive tasks, and parent-child interaction task were scored based on standardized manuals by undergraduate and graduate research assistants who were trained in the coding systems by the author (Zyga) and secondary mentor (Russ). Interrater reliability for all video coded tasks (APS-P; social cognitive tasks; parent-child interaction task) was assessed.
Independent coders, blind to conditions, rated 20% of each of these video recorded tasks.
Intra-class correlations (ICC) were calculated based on a single measures, absolute
agreement, two-way mixed effects model to assess the degree of consistency between
coders for each task. The resulting ICCs for the APS-P, social cognitive tasks, and
parent-child interaction task were Excellent (IRR = 0.75-0.85) (Cicchetti, 1994). Raters
had overall high levels of agreement and rated behaviors consistently across participants.
Aim 1
Hypothesis 1
At baseline, children with ASD were predicted to show significantly lower levels
of peripheral OXT (~40 pg/mL) as compared to typically developing preschoolers (~70
pg/mL) as measured through saliva collection. Further, the two groups would differ on
scores relating to the APS-P, FACE, social cognitive tasks, parent functioning, and
parent-child interaction task. Specifically, the ASD group was hypothesized to show
overall lower scores in these domains. 53
Data Analysis Plan. To meet Aim 1, hypothesis 1, demographics including age,
gender, receptive language (PPVT), cognitive ability (MSEL), autism severity (SCQ) and
temperament (CBQ), baseline OXT levels, social cognitive ability (APS-P, FACE, social
cognitive tasks), parent functioning (PSI-4, DASS-21, PAAQ), and parent-child interaction measures were organized by group. Descriptive statistics and a between- subjects ANOVAs were conducted to (1) better characterize demographic factors, OXT expression, social cognitive ability, parent functioning, and parent-child interaction by group and (2) understand if group membership significantly impacted peripheral OXT concentration levels, social cognitive abilities, parent functioning, and the quality of parent-child interactions.
Demographics
Participants across the 2 groups did not differ on age (M = 4.38; SD = 0.85).
However, there was a significant difference in receptive language ability (F = 52.42; p =
0.000) across the 2 groups as measured by the PPVT-4 and early cognitive ability (F =
15.00; p = 0.000) as measured by the visual reception subscale on the MSEL, with the
ASD group scoring lower than the TD group. Further, the TD (61.9% male) sample had a more even split on gender whereas the ASD group (77.8% male) had a higher percentage of male participants which reflects the gender distribution in this disorder. The groups did not differ on race (majority Caucasian). One outlier was identified in the ASD group based on scores across language, cognitive, and social cognitive ability. This participant was excluded from all further analyses, leaving the total sample to include 17 children with ASD and 21 TD children. Lastly, given that language and cognitive ability differed between groups, all subsequent analyses controlled for cognitive ability. 54
In regards to level of autism symptom severity, the ASD group scored significantly higher than the TD group (F = 120.6; p = 0.000) and fell above the cut-off score of 15, suggesting the presence of autism. Child temperament as measured by the
Child Behavior Questionnaire (CBQ) showed that the ASD group scored significantly higher on domains relating to Activity Level (F = 7.11; p = 0.01) and Shyness (F = 13.65; p = 0.001), whereas the TD group scored higher on domains relating to Attentional
Focusing (F = 13.34; p = 0.001), Inhibitory Control (F = 25.83; p = 0.000),
Reactivity/Soothability (F = 8.03; p = 0.02), Low Intensity Pleasure (F = 11.61; p =
0.002) and Perceptual Sensitivity (F = 7.45; p = 0.01). See Table 1 for full details.
Baseline OXT Concentration Levels
Within the ASD group, one value was identified as an outlier (pg/ml > 149) and within the TD group, one sample was deemed corrupted and provided an invalid result.
Overall, in the ASD sample, two values were thus excluded from any subsequent baseline analyses including OXT (1 outlier due to oxytocin level; 1 previously identified outlier) to bring the sample size to 16. In the TD sample, one sample was excluded from any subsequent baseline analyses including OXT, bringing the sample size to 20. A one-way, between-subjects ANOVA showed that the TD and ASD samples did not differ on their baseline levels of OXT expression (F = 2.87; p = 0.11). See Table 2 for full details.
Social Cognitive Ability
Pretend Play (APS-P). A series of one-way, between-subjects ANCOVAs, controlling for cognitive ability, were conducted to better understand how global scores reflecting cognitive and affective ability in pretend play differed between the TD and
ASD groups based on the standardized play task administered as part of the current study. 55
Results showed that the two groups significantly differed on Imagination (F = 7.91; p =
0.008), Organization (F = 10.07; p = 0.003), Time Spent in Functional Play (F = 6.84; p =
0.01), Time Spent in Symbolic Play (F = 5.25; p = 0.03), number of Divergent Themes (F
= 8.03; p = 0.008), Affect Frequency (F = 5.41; p = 0.03), and number of Personifications
(F = 11.55; p = 0.002). For all variables except for Time Spent in Functional Play, the
ASD group performed significantly lower than the TD group. See Table 3 for full details
Emotion Recognition (FACE). A series of one-way, between-subjects
ANCOVAs, controlling for cognitive ability, were conducted to better understand how emotion recognition and understanding differed between the TD and ASD groups.
Results showed that the two groups significantly differed in their ability to recognize
Happy (F = 11.07; p = 0.002), Angry (F = 4.97; p = 0.03), Surprised (F = 9.60; p =
0.004), Positive Emotions (F = 8.75; p = 0.006), Negative Emotions (F = 8.98; p =
0.006), Total Accuracy by each emotion (F = 16.39; p = 0.000), and Total Ability across positive and negative emotional categories (F = 17.57; p = 0.000). For all variables, the
ASD sample scored significantly lower than the TD sample. See Table 4 for details.
Joint Attention, Empathy, and Cooperation (Social Cognitive Tasks). A series of one-way, between-subjects ANCOVAs, controlling for cognitive ability, were conducted to better understand how social cognitive ability relating to joint attention, empathy, and cooperation differed between the TD and ASD groups. Results showed that the two groups significantly differed on Joint Attention (F = 4.12; p = 0.05), Empathy (F = 5.51; p 0.03), and Cooperation (F = 18.33; p = 0.000). For all variables, the ASD sample scored significantly lower than the TD sample. See Table 4 for details.
Parent Functioning 56
Parenting Stress Index (PSI-4). A series of one-way, between-subjects
ANCOVAs, controlling for cognitive ability, were conducted to better understand how
the TD and ASD groups differed in levels of parenting stress. Within the child domain,
parents of children with ASD reported experiencing significantly more stress as it related
to their child as a whole (F = 19.09; p = 0.000) and also in domains relating to the child’s
Distractibility/Hyperactivity (F = 26.16; p = 0.000), Adaptability (F = 10.66; p = 0.003),
Reinforces Parent (F = 4.80; p = 0.04), Demandingness (F = 18.35; p = 0.000), and
Acceptability (F = 15.16; p = 0.001) as compared to parents of TD children. Within the parenting characteristics domain, stress was significantly higher in the ASD group in regards to parent Health (F = 10.98; p = 0.002) and Role Restriction (F = 16.69; p =
0.000). Parents of children with ASD also showed significantly more Life Stress (F =
4.28; p = 0.05) and Total Stress (F = 11.18; p = 0.002) than the TD parents. See Table 5 for full details.
Depression, Anxiety and Stress (DASS). A series of one-way, between-subjects
ANCOVAs, controlling for cognitive ability, were conducted to better understand parent
levels of depression, anxiety, and stress between the TD and ASD groups. Results
suggest no significant differences across these domains. Stress between ASD and TD
parents trended towards significance (F = 3.88; p = 0.06). See Table 6 for full details.
Parental Acceptance and Action Questionnaire (PAAQ). A series of one-way,
between-subjects ANCOVAs, controlling for cognitive ability, were conducted to better
understand experiential avoidance, coping ability, and control behaviors in the context of
parenting a child between the TD and ASD groups. Results showed that parents of
children with ASD reported significantly higher levels of unwillingness to accept their 57
child’s ability and level of functioning (F = 8.09; p = 0.008) and a significantly higher
Total Score (F = 5.18; p = 0.03) suggesting more impairment in regards to avoidance,
coping, and control behaviors as compared to the TD group. See Table 6 for full details.
Parent-Child Interaction Results
Parent-Child Free Play Task. A series of one-way, between-subjects ANCOVAs
were conducted, controlling for cognitive ability, to better understand how the TD and
ASD groups differed on the quality of parent-child interaction. All variables significantly differed between the TD and ASD groups except for Response to Child. For parent’s engagement, Degree of Parental Involvement (F = 46.12; p = 0.000) and Unsolicited
Help (F = 25.18; p = 0.000) were significantly higher indicating that ASD parents were over involved and more intrusive during the play task than the TD parents. For the child characteristics, children with ASD had significantly lower Social Interest (F = 4.17; p =
0.05) and Social Competence (F = 15.07; p = 0.000) during the interaction that TD peers.
Lastly, Mutual Engagement (F = 5.06; p = 0.03) and Overall Mood (F = 2.41; p = 0.02) were significantly more disrupted in the ASD group. See Table 7 for full details.
Hypothesis 2
When controlling for group membership (whole sample comparison), it was
hypothesized that: (1) children characterized as more extroverted (higher activity,
approach, reactivity, and high intensity pleasure; lower low intensity pleasure, perceptual
sensitivity, shyness, and fear) by the CBQ would express higher levels of OXT; (2)
scores on imagination, organization, affect frequency, functional play, symbolic play,
divergent storylines, transformation frequency, aggressive interpersonal acts, and
personifications in play as measured by the APS-P would significantly positively 58
correlate with basal OXT concentration levels; (3) scores on the FACE task (emotion
recognition) would significantly positively correlate with basal OXT concentration
levels; (4) higher scores on the joint attention, empathy, and cooperation tasks would
significantly positively correlate with basal OXT concentration levels; (5) scores on
measures of general mood, mutual engagement, parent involvement, unsolicited help
from the parent, parent’s response to child, child social interest, and child social
competence child from the parent-child free play task would significantly positively correlate with basal OXT concentration levels. It was also hypothesized that the above relationships between baseline OXT concentration levels and measures of temperament, social cognitive ability, and parent-child interaction would vary between the two groups in that the TD and ASD groups would show significant relationships between variables of interest that were unique to group membership such as stronger relationships between social cognitive factors in the ASD group versus parent-child interaction quality and temperament in the TD group. Lastly, it was predicted that extroversion, pretend play ability, social cognitive ability, and parent involvement in the joint play task would account for a significant portion of the variance in predicting peripheral OXT concentration levels if placed into a regression model.
Data Analysis Plan. To meet Aim 1, hypothesis 2, temperament (CBQ), social cognitive ability (APS-P, FACE, social cognitive task), and parent-child interaction variables were correlated with basal OXT concentration levels across the whole sample and within the TD and ASD samples, respectively. If significant correlations (p < 0.05) were found at r ≥ 0.30, the variables of interest were placed into a linear regression model to better understand their potential predictive power on peripheral OXT expression. 59
Whole Sample Comparison
Correlational analyses were used to examine the relationship between variables
relating to child temperament (CBQ), pretend play ability (APS-P), emotional understanding (FACE), social cognitive ability, and parent-child interaction across the entire sample. As predicted, for child temperament, results indicate a significant positive correlation between baseline OXT concentration level and Activity (r = 0.45; p = 0.01) and a significant negative correlation between baseline OXT concentration level and Low
Intensity Pleasure (r = -0.36; p = 0.05). Further, significant positive correlations were found between baseline OXT concentration levels and Level of Parental Involvement (r =
0.37; p = 0.04) and Unsolicited Help from the Parent (r = 0.36; p = 0.05) in reference to the PCI task. No significant correlations were found between baseline OXT levels and variables on the APS-P, FACE, or social cognitive tasks. See Table 8 for full details.
Given that Activity and Low Intensity Pleasure on the CBQ and Level of Parental
Involvement and Unsolicited Help from the Parent in the PCI task significantly correlated with baseline OXT concentration levels at r ≥ 0.30, p < 0.05, a four step hierarchical multiple regression was conducted with baseline OXT concentration level as the dependent variable. Activity level (CBQ) was entered at Step One of the regression given
that it was shown to account for the most variance in regards to predicting baseline OXT
levels (see linear regression analysis in Appendix A). Subsequent variables were entered
based on variance predicted, with later variables predicting less variance (See Appendix
A for full details). Specifically, Parental Involvement (PCI) was entered at Step Two,
Unsolicited Help (PCI) was entered at Step Three, and Low Pleasure Intensity (CBQ) was entered at Step Four. The hierarchical regression revealed that at Step One, Activity 60
Level contributed significantly to the regression model (t = 2.66, p = 0.01) and accounted
for 17.4% of the variation in baseline OXT concentration level. Introducing Parental
Involvement (PCI) explained 7.7% of variation in baseline OXT levels and this change in
R2 trended towards significance (t = 1.69, p = 0.10). Adding Unsolicited Help (PCI) to the
regression model explained an additional 0.8% of variation in baseline OXT
concentration level, which was a nonsignificant change in R2 (t = -0.533, p 0.60). Lastly, adding Low Pleasure Intensity (CBQ) to the regression model accounted for 0.2% of variation in baseline OXT levels, which was also a nonsignificant change in R2 (t = 0.28, p = 0.78). Thus, Step One was found to be the model of best fit with Activity Level
(CBQ) accounting for the most significant amount of variance in baseline OXT concentration level across the whole sample.
TD Sample Comparison
Correlational analyses were used to examine the relationship between variables relating to child temperament (CBQ), pretend play ability (APS-P), emotional understanding (FACE), social cognitive ability, and parent-child interaction within the
TD group. First, for child temperament, results indicate a significant positive relationship between baseline OXT concentration level and Perceptual Sensitivity (r = 0.45; p = 0.05).
Second, in regards to pretend play, results show a significant positive correlation between baseline OXT concentration level and Functional Play (r = 0.53; p = 0.02), which was predicted, and significant negative correlations between baseline OXT and Symbolic Play
(r = -0.53; p = 0.02) and Aggressive Interpersonal Acts (r = -0.54; p = 0.02). Third, a significant positive correlation was found between baseline OXT concentration level and
Sad emotion recognition (r = 0.58; p = 0.009). Fourth, a significant positive correlation 61
was found between baseline OXT concentration level and Joint Attention (r = 0.53; p =
0.02). Lastly, in regards to parent-child interaction, a significant positive correlation was
found between baseline OXT concertation level and General Mood (r = 0.43; p = 0.05) and a significant negative correlation was found between baseline OXT and Social
Competence (r = -0.53; p = 0.02). See Table 10 for full details.
Given that Functional Play, Symbolic Play, and Aggressive Interpersonal Acts on the APS-P, Sad emotion recognition on the FACE, Joint Attention on the Social
Cognitive Task, and Social Competence in the PCI task significantly correlated with baseline OXT concentration levels at r ≥ 0.30, p < 0.05, a six step hierarchical multiple regression was conducted with baseline OXT concentration level as the dependent variable. Sad (FACE) was entered at Step One of the regression given that it was shown to account for the most variance in regards to predicting baseline OXT levels (see linear regression analysis in Appendix A). Subsequent variables were entered based on variance predicted, with later variables predicting less variance (See Appendix A for full details).
Specifically, Aggressive Interpersonal Acts (APS-P) was entered at Step Two, Functional
Play (APS-P) was entered at Step Three, Symbolic Play (APS-P) was entered at Step
Four, Social Competence (PCI) was entered at Step Five, and Joint Attention was entered at Step Six. The hierarchical regression revealed that at Step One, Sad (FACE) contributed significantly to the regression model (t = 2.97, p = 0.009) and accounted for
30.2% of the variation in baseline OXT concentration level. Introducing Aggressive
Interpersonal Acts (APS-P) explained 18.4% of variation in baseline OXT levels and this change in R2 was significant (t = -2.490, p = 0.02). Adding Functional Play (APS-P) to
the regression model explained an additional 9.5% of the variation in baseline OXT level 62
and this change in R2 trended towards significance (t = 1.94, p = 0.07). Lastly, adding
Symbolic Play (APS-P) explained 2.3% variance (nonsignificant change; t = -0.96, p =
0.35), Social Competence (PCI) explained 2.8% variance (nonsignificant change; t = -
1.05, p 0.31), and Joint Attention explained 1.9% variance (nonsignificant change; t =
0.86, p = 0.41) in baseline OXT concentration level. Given this, Step Two was found to
be the model of best fit with Sad (FACE) and Aggressive Interpersonal Acts (APS-P)
serving as unique predictors which accounted for the most significant amount of variance
in baseline OXT concentration level within the TD sample.
ASD Sample Comparison
Correlational analyses were used to examine the relationship between variables
relating to child temperament (CBQ), pretend play ability (APS-P), emotional understanding (FACE), social cognitive ability, and parent-child interaction within the
ASD group. In pretend play, significant positive correlations were found between Affect
Frequency (r = 0.62; p = 0.03), Divergent Storylines (r = 0.60; p = 0.04), Transformation
Frequency (r = 0.73; p = 0.007), Aggressive Interpersonal Acts (r = 0.72; p = 0.009), and
Personifications (r = 0.61; p = 0.03) and baseline OXT concentration levels. No other significant correlations were found between baseline OXT concentration levels and variables relating to child temperament (CBQ), emotion recognition (FACE), social cognitive ability, or parent-child interaction in the ASD sample. See Table 12 for full details.
Given that Affect Frequency, Divergent Storylines, Transformation Frequency,
Aggressive Interpersonal Acts, and Personifications on the APS-P significantly correlated with baseline OXT concentration levels at r ≥ 0.30, p < 0.05, a five step hierchical 63
regression was conducted with baseline OXT concentration level as the dependent
variable. Transformation Frequency (APS-P) was entered at Step One of the regression given that it was shown to account for the most variance in regards to predicting baseline
OXT levels (see linear regression analysis in Appendix A). However, when
Transformations was plotted against baseline OXT levels, it became apparent that there were two data points that acted as influential points, those that have disproportionate effects on the slope of the regression equation if they are included in the model versus
excluded (James, 1984). Specifically, when these two data points were included in the
model, β = 2.25 and when excluded, β = 33.00. Given this, Transformations was
removed from the hierarchical regression analysis. Subsequent variables were then entered based on variance predicted, with Interpersonal Aggressive Acts (APS-P) as the most significant predictor once Transformations was excluded from analysis, which led to a four step hierarchical regression. Later variables entered into the model predicted less variance (See Appendix A for full details). Specifically, Aggressive Interpersonal Acts
(APS-P) was entered at Step One, Personifications (APS-P) was entered at Step Two,
Affect Frequency (APS-P) was entered at Step Three, and Divergent Storylines (APS-P) was entered at Step Four. The hierarchical regression revealed that at Step One,
Aggressive Interpersonal Acts (APS-P) contributed significantly to the regression model
(t = 3.25, p = 0.009) and accounted for 46.5% of the variation in baseline OXT concentration level. Introducing Personifications (APS-P) accounted for 8.1% variance
(nonsignificant change; t = 1.34, p = 0.21), Affect Frequency (APS-P) accounted for
1.0% variance (nonsignificant change; t = -0.47, p = 0.67), and Divergent Storylines accounted for 9.3% variance (nonsignificant change; t = 1.47, p =0.19) in baseline OXT 64
concentration level. These findings suggest that Step One is the model of best fit with
Aggressive Interpersonal Acts (APS-P) accounting for the most significant amount of
variance in baseline OXT concentration level within the ASD sample.
Aim 2
Hypothesis 1
Children with ASD who engaged in the PRETEND-ASD intervention program
would show significant positive gains in: (1) expression of OXT concentration levels, (2)
pretend play ability specifically relating to organization, divergent storylines,
imagination/symbolic play, affect expression, and interpersonal processes as measured by
the APS-P, (3) emotion recognition as measured by the FACE task, and (4) joint
attention, empathy, and cooperation as measured by the social cognitive tasks. Parents of
children who participated in the intervention were predicted to show decreased levels of
stress, depression, and anxiety and increased feelings of action and willingness to engage in treatment (PAAQ). Lastly, engagement in the PRETEND-ASD program was hypothesized to significantly increase measures of general mood, mutual engagement, degree of parental involvement, parent’s response to the child, and child’s social interest and competence in the parent-child free play task.
Data Analysis Plan. To meet Aim 2, hypothesis 1, paired samples t-tests were conducted to better understand if changes in peripheral OXT concentration levels, social cognitive ability (as measured by the FACE, APS-P, social cognitive observation tasks), parent functioning (PSI-4, DASS-21, PAAQ), and parent-child interaction occurred from baseline to post-intervention within the ASD group.
Intervention Feasibility 65
In regards to feasibility, 16 out of the 18 ASD participant families were able to
complete the intervention program. Two of the families were lost to intervention follow-
up after the baseline assessment visit. All families who participated in the intervention
completed the total eight sessions. However, most participants deviated from the 8-week
intervention timeline due to family vacations, holidays, and unexpected events such as
illness. Average length of participation trended towards 10 weeks.
Intervention Efficacy
OXT Concentration Levels. From pre to post intervention, 2 participants did not
provide samples at the final visit due to dropping out of the study and 1 sample from the
post-intervention visit was unable to be used for extraction. Further, the 2 baseline
outliers described above were also excluded from the analysis. In total, this excluded 5
participants from the OXT pre-post level analysis, leaving an n = 13. A paired samples t-
test showed that the ASD sample’s OXT concentration levels did not significantly change
from pre-post intervention (t = -0.67; p = 0.52). See Table 14 for full details.
Social Cognitive Ability
Pretend Play (APS-P). A series of paired samples t-tests were conducted to better
understand how global scores reflecting cognitive and affective ability in pretend play differed from pre to post intervention in ASD group based on the APS-P task
administered as part of the current study. Results showed that Divergent Themes in play
trended towards significance (t = -1.806; p = 0.09). No other significant changes in play
ability were observed. See Table 15 for full details.
Emotion Recognition (FACE). A series of paired samples t-tests were conducted
to better understand how emotion recognition and understanding differed from pre to post 66 intervention in the ASD group. Results showed that there were significant increases in ability in recognizing Sad (t = -4.78; p = 0.001), Negative Emotions (t = -2.12; p = 0.05),
Total Accuracy (t = -4.55; p = 0.001), and Total Ability (t = -2.661; p 0.02). Further, there were trending increases in ability to recognize Happy (t = -2.02; p = 0.07) and
Ashamed (t = -1.77; p = 0.10). No other significant changes were observed. See Table 16 for full details.
Joint Attention, Empathy, and Cooperation (Social Cognitive Tasks). A series of paired samples t-tests were conducted to better understand how social cognitive ability relating to joint attention, empathy, and cooperation differed from pre to post intervention in the ASD group. Results showed that ability in Joint Attention (t = 1.80; p = 0.09) trended towards significance. No other significant changes were observed. See Table 16 for full details.
Parent Functioning
Parenting Stress Index (PSI-4). A series of paired samples t-tests were conducted to better understand how levels of parenting stress differed from pre to post intervention in the ASD group. Results showed a significant decrease in stress as it related to the
Demandingness of the child (t = 2.73; p = 0.02). No other significant changes were observed. See Table 17 for full details.
Depression, Anxiety and Stress (DASS). A series of paired samples t-tests were conducted to better understand how parent levels of depression, anxiety, and stress changed from pre to post intervention in the ASD group. Results showed a significant decrease in Depression (t = 2.91; p = 0.01) and Stress (t = 2.18; p = 0.05). No other significant changes were observed. See Table 18 for full details. 67
Parental Acceptance and Action Questionnaire (PAAQ). A series of paired
samples t-tests were conducted to better understand how experiential avoidance, coping
ability, and control behaviors in the context of parenting a child changes from pre to post
intervention in the ASD group. No significant changes were observed across these
domains. See Table 18 for full details.
Parent-Child Interaction Results
Parent-Child Free Play Task. A series of paired samples t-tests were conducted to
better understand how the quality of parent-child interaction differed from pre to post intervention in the ASD group. Results showed a significant increase in Overall Mood (t
= -2.52; p = 0.03). No other significant changes were observed. See Table 19 for full details.
Discussion
Summary of Findings
The current study had two major aims relating to (1) characterizing child basal
peripheral OXT concentration levels, social cognitive ability, parent functioning, and
parent-child interactions in children with ASD and typical development (TD) and
understanding how OXT levels relate to these factors across diagnostic categories and (2)
measuring the impact a telehealth parent training program (Parent-focused Remote
Education To ENhance Development in children with Autism Spectrum Disorder
[PRETEND-ASD]) had on OXT levels, social cognitive ability, parent functioning, and
quality of parent-child engagement in children with ASD.
For the first aim, a main finding was that, at baseline, the two samples did not
differ in OXT concentration levels, which did not support the hypothesis that ASD would 68
show lower values as compared to the TD group. Other main findings in regards to Aim 1
indicated that children with ASD had significantly lower receptive language, cognitive ability, and lower scores in pretend play, emotion recognition, joint attention, cooperation, and empathy. Parents of children with ASD reported increased stress and
significantly more unwillingness to witness their child experience negative emotions,
which could impact their ability to engage in treatment. Lastly, in terms of parent-child
interaction, parents of children with ASD had higher degrees of parental overinvolvement and unsolicited help, lower mood and mutual engagement, and children in this sample showed lower social interest and social competence. Conversely, parents in both groups
responded in a warm and noncritical manner during the joint play interaction. Overall,
these findings confirmed the hypothesis that children with ASD would score significantly lower than the TD sample across the various social cognitive and parent functioning measures.
When evaluating how social cognitive factors relate to baseline OXT concentration levels, analyses showed that, across the whole sample, baseline OXT significantly correlated with temperament factors relating to activity (positive relationship) and low intensity pleasure (negative relationship) along with significant positive correlations with parental involvement and unsolicited help from the parent during parent-child interactions. When these predictors were entered into a hierarchical regression, activity accounted for the largest portion of variance and was a unique significant predictor of baseline OXT concentration. Parental involvement trended towards significantly predicting baseline OXT concentration levels but was not a unique predictor. Within the TD sample, baseline OXT concentration levels significantly 69
positively correlated with sad emotion recognition, functional play, and joint attention ability and negatively correlated with symbolic play aggressive interpersonal acts in play, and child social competence during the parent-child interaction. When these predictors were entered into a hierarchical regression, sad emotion recognition was the largest significant predictor of baseline OXT concentration levels. Aggressive interpersonal acts also uniquely predicted a portion of the variance in baseline OXT concentration levels.
Functional play trended towards significance but was not a unique predictor in predicting
OXT levels in the TD sample. Within the ASD sample, baseline OXT concentration levels significantly correlated with affect frequency, divergent storylines, transformation frequency, aggressive interpersonal acts, and personifications in play. When these predictors were entered into a hierarchical regression, aggressive interpersonal acts accounted for the largest portion of variance and was a unique significant predictor of baseline OXT concentration. No other variables significantly predicted OXT levels above and beyond aggressive interpersonal acts. These findings confirmed the hypothesis that relationships between baseline OXT concentration levels and measures of temperament, social cognitive ability, and parent-child interaction would vary based on group membership. Specifically, the TD and ASD groups showed significant relationships between variables of interest that were unique to group membership.
Concerning the second aim and intervention efficacy, a main finding was that there was no significant change from baseline to post-intervention in OXT concentration levels, which did not support the hypothesis that children with ASD would show significant positive gains in OXT expression after engaging in the PRETEND-ASD program. Participants with ASD did show significant increases in their ability to 70
recognize sad and negatively valenced emotions and increased in their total accuracy and
ability on the FACE emotion task. In regards to play and social cognitive skill, the ASD
sample had trending increases in divergent storylines and joint attention ability. Parents
of children with ASD showed significant decreases in stress relating to the
demandingness of the child (PSI-4) and levels of reported depression and stress (DASS).
Lastly, there was a significant increase in the overall mood of parent-child interactions from pre to post intervention. These results confirm some of the hypothesized outcomes in regards to intervention efficacy although also suggest that the PRETEND-ASD intervention may have targeted more base level abilities, such as setting up conducive environments for parent-child engagement and building emotion recognition instead of targeting higher level skills, such as pretend play ability and overall social interest or competence.
Group Comparison
Baseline Characterization. A first major aim of this study was to better understand baseline OXT concentration levels in preschoolers with ASD and how these levels compared with typical developing children of the same age. It was predicted that children with ASD would show decreased levels given previous findings in the literature
within the preschool age range (Feldman et al., 2014) posited an “OXT deficit”
hypothesis in regards to individuals with autism (Feldman et al., 2014; Green et al., 2001;
Modahl et al., 1998). Contradictory to this hypothesis, the current study found that
preschoolers with ASD did not have lower OXT concentration levels as compared to the
TD group. This finding lends evidence towards the potential complex relationship
between OXT and social cognitive functioning and its potential use as an indicator across 71
diagnostic categories or populations rather than being solely specific to ASD. Building
off of this concept of complexity, more recent studies point to mixed findings that are
beginning to question the “OXT deficit” hypothesis in ASD. Whereas older studies found
lower levels of baseline OXT in children with ASD (Al-Ayadhi, 2005; Green et al., 2001;
Modahl et al., 1998) more recent studies are showing no difference between diagnostic
groups (Miller et al., 2013; Parker et al., 2014; Taurines et al., 2014) or differences that
are dependent on levels of cognitive and social impairment (Alabdabi et al., 2014). Miller
et al. (2014) measured baseline OXT levels in 40 males and females with high
functioning autism (HFA) and 35 TD children, ages 8-18, and found no difference in
OXT expression between the two groups. Females with HFA actually trended towards
having higher levels of OXT expression than the other groups (Miller et al., 2014). Parker et al. (2014) conducted the largest study to date and found no difference between ASD and TD samples in OXT concentration levels, again providing no evidence to support the
“OXT deficit” hypothesis in ASD. The authors noted that OXT biology did not seem to
be uniquely related to autism, but may instead enact independent and accumulative
influences on individual differences in human social functioning, which would also
include the social deficits evidenced in ASD (Parker et al., 2014). Lastly, Tourines et al.
(2014) compared OXT concentration levels between males, ages 6-17, diagnosed with
either ASD, ADHD, or were typically developing. Results of the study showed that OXT
levels in ASD were actually significantly higher than the ADHD group and did not
significantly differ from the TD group. Further, the authors found a strong positive
correlation (r = 0.60; p = 0.01) between OXT levels and ADOS severity scores in the
ASD sample, suggesting that higher OXT actually related to more severe autism 72
symptomology. The authors noted that proposing an OXT deficit or excess model for
ASD seemed to be inadequate in fully describing findings from the literature and previous studies. Tourines et al. (2014) posited that perhaps high OXT could potentially be understood as a secondary physiological effect from negative moods or stressors with a potential aim to moderate anxiety given that one of OXT functions is the regulation of social anxiety. This assumption that different levels of OXT can be impacted by factors outside of diagnostic category, such as comorbid conditions, levels of stress, and social cognitive ability, falls in line with the concept that the role OXT may play in ASD or in an individual is more complex and further study of its expression across ages, levels of functioning, and diagnostic categories is warranted.
This study further characterized social cognitive abilities between preschoolers with ASD and typically developing peers across domains relating to pretend play, emotion recognition, joint attention, empathy, cooperation, parent functioning as it relates to stress, anxiety, depression, and ability to accept and act on their child’s difficulties, and also parent-child interactions. Previous research has shown that children with ASD as young as 2 years old show a decreased ability to produce or engage in pretend or symbolic play (Lam & Young, 2011; Rutherford & Rogers, 2003; Jarrold, 2003; Zyga et al., 2015), show a preference for non-speech versus motherese sounds when toddlers
(Kuhl et al., 2005), visually attend less to social faces when circumscribed objects of interests are shown in tandem with these faces (Sasson & Touchstone, 2014), show decreased skill in social interactions relating to less social bids and focus of engagement on other children (McGee et al., 1997), and decreased emotional understanding and recognition (Uljarevic & Hamilton, 2012). A large majority of young children with ASD 73 also exhibit abnormal parental or caregiver attachment interactions. Toddlers diagnosed with ASD are significantly less likely than typically developing children to make eye contact, use joint attention, engage in social turn-taking, imitate gestures or communication, and anticipate social routines during play sessions with a parent or caregiver (Adamson et al., 2012; Bernebei et al., 1998; Dawson et al., 1990; Gillberg et al., 1990; Williams, 2003). Lastly, higher parenting stress and psychological distress is usually evident in parents of children with ASD versus other developmental disorders and typical development (Estes et al., 2009). Overall, in the current study, these previous findings were replicated in that the ASD group scored significantly lower than the TD sample across all social cognitive measures as predicted. Further, parents of children with
ASD expressed higher levels of stress as it related to their child and their own functioning. Parent-child interactions were also significantly impacted in the ASD sample, with parents showing high levels of overinvolvement and unsolicited help and the overall interaction was less cohesive, integrated, and positive than TD parent-child dyads. These findings provide further support that social cognitive deficits are evident in children with ASD and in their interactions with primary caregivers even in this early age range (Estes et al., 2009; Jarrold 2003; Warreyn et al., 2005). These findings add to the literature in that many previous studies have characterized socioemotional deficits in preschoolers through the use of parent report or interviews. The current study’s use of behavioral assessments in capturing skills relating to various domains such as pretend play, joint attention, cooperation, empathy, emotion recognition, and parent-child interaction provides more objective evidence of the difficulties young children with autism may face. These findings then also provide more evidence for a need for early 74
intervention, not only for the child but also for the parent and child, who show disrupted
interactions as well. Further, given the more objective and quantitative nature of
behavioral assessments, it may be important to use these baseline findings to better tailor
the needs of the intervention or use as stronger outcome measures than parent report
alone. These implications will be discussed further in regards to the intervention efficacy
in the current study.
Social Cognitive Functioning and OXT Levels. Results showed that the relation
between baseline OXT levels and child factors such as temperament and social cognitive
ability did vary by group membership. Across the whole sample, factors relating to
temperament and parent involvement in interactions accounted for the highest level of
variance in predicting OXT levels. Within the TD sample, emotion recognition and use of
aggressive interpersonal acts in play accounted for the highest variance in predicting
oxytocin. Lastly, in the ASD sample, aggressive interpersonal acts also accounted for the
highest variance in predicting baseline OXT, however in the opposite direction as that
found in the TD sample. These findings fall in line with previous results in two main
ways: (1) previous findings suggest a relationship between OXT and factors such as
temperament, parental involvement, and emotional understanding and expression and (2) the theory that OXT may not be uniquely related to ASD or one diagnostic category but rather related to individual differences across groups in a way that is nuanced and complex.
To the first point, multiple lines of research have shown the potential role of OXT in personality, trust, altruism, social bonding, and human’s ability to infer the emotional
states of others (Donaldson & Young, 2008). Tost et al. (2010) found that decreased 75
hypothalamus volumes, which co-occurred with the expression of certain OXTR allelic variations, predicted lower prosocial temperament scores in participants. Further, variations of the OXTR gene may affect anxiety-related temperament traits surrounding harm avoidance in females (Wang et al., 2013). Bendix et al. (2015) found that personality factors relating to impulsiveness and negative emotionality were significant independent predictors of plasma oxytocin in individuals diagnosed with psychiatric conditions such as anxiety or depression. Other research findings have shown that the
OXTR rs53576 allelic variation relates to deficits in empathy, attachment, and sensitive parenting (Strathearn et al., 2012). Mothers who express this gene showed a reduced maternal awareness of the needs and subtle emotional signals of their child (Strathearn et al., 2012). This study also found that maternal oxytocin response was negatively related to effortful control and positively related to orienting sensitivity temperament styles in their infants. Lastly, in regards to emotion recognition and expressions, studies have shown that higher OXT expression is related to better classification of emotions displayed on faces and increased gaze to the eye region in human faces (Donaldson &
Young, 2008).
To the second point, current research suggests that the question should not be whether or not OXT relates to social cognitive factors but rather under what circumstances in that the effects of OXT are most often found to be moderated by contextual factors or by stable characteristics of the individual (Bartz et al., 2011). As shown in the results of this study, factors such as group membership, developmental trajectory, and severity of socioemotional deficits led to differences in how OXT related to various domains and measures. An interesting finding in the current study that has also 76
been shown in previous research is the opposite nature of relationships between OXT and
areas of functioning when comparing a TD sample against individuals with ASD. In the
current study, it was found that aggressive interpersonal acts in play negatively correlated with baseline OXT levels in TD children but positively correlated with OXT in children with ASD. That is, for TD children, higher levels of aggressive interpersonal acts in their play related to lower OXT expression whereas, in ASD, higher interpersonal aggression related to higher OXT levels. This reversal of relationships provides evidence to the concept that the effects of oxytocin and its relationship with social cognition are nuanced in that for typically developing children, an expression of aggressive acts may be less prosocial given higher overall social ability (McGee et al., 1997) which then would lead this factor to be negatively associated with OXT expression. However, in ASD, where social deficits are evident, the ability to express any type of interpersonal act, be it aggressive or otherwise, may show a true prosocial ability, which could lead to a positive relationship with OXT. A previous study conducted by Modahl et al. (1998) also showed this reverse relationship in that elevated OXT was associated with higher scores on social and developmental measures in a typically developing sample of male children, ages 6-11
years. However, in the ASD sample of males ages 6-11, elevated OXT was associated
with lower scores on these same measures. Other studies have shown these reverse
relational patterns in regards to factors such as gender (Miller et al., 2013) and severity of
diagnosis (Alabdabi et al., 2014). Taken together, the findings of this and previous
studies suggest that more research needs to be done in better understanding the various
circumstances and relationships that may be present between OXT and other domains of
functioning, how context and person-dependent factors impact these relationships, and 77
the thought that there may be distinct mechanisms of action for OXT within these various
relationships or group differences.
Intervention Effects
OXT Malleability. A second major aim of the current study was to better
understand the efficacy of a remote parenting training intervention (PRETEND-ASD) on child OXT concentration levels, social cognitive ability, parent functioning, and parent- child interaction quality. Contrary to the hypothesized results, there were no significant changes to OXT levels from pre to post intervention in the ASD group. The rationale for the hypothesis that behavioral intervention may positively impact OXT levels was that previous research findings suggested early interactions between parents and children can shape later biological systems and also behavioral outcomes of the child (Baran, 2017;
Meaney, 2011). For example, it is known that human OXTR expression is high at birth and for the first 3 years of life in the neocortex (Kang et al., 2011) and the development of adult social skills stems from early life events that bring children into contact with social stimuli. This then leads to the hypothesis that experience-dependent oxytocin activity during this early age range, which may present as a critical window in shaping the OXT system, could shape development in both a biological and behavioral context
(Hammock, 2015). Since children with ASD and their parents have disrupted early parent-child relationships, past theories have posited that an improvement in these interactions early in life, perhaps during critical periods of hormonal development, may then positively impact neuropeptide systems, such as OXT, and subsequent behavior as well. However, current findings in this area mostly stem from animal models. For example, Veenema (2012) found that differences in the quality of the early social 78
environment in rats was linked to brain-region specific changes in oxytocin and
vasopressin expression and receptor binding. These hormonal changes were associated
with actual alterations in social behaviors, including maternal care, play-fighting, and social recognition. This study was a first attempt in extending these findings from animal models to human subject research.
Particularly, the current study aimed to test this theory in measuring if behavioral intervention between a parent and child with ASD would then also show an alteration in
OXT concentration levels, perhaps suggesting malleability in this hormones expression early in development. This could then also potentially lead to lasting biological changes in OXT system development along with behavioral outcomes. However, there may be a few potential explanations as to why this change was not observed in the current study.
The first lends itself to the previous discussion of the complexity of OXT and its relationship with social cognitive factors. It was originally hypothesized that the ASD group would show deficits in OXT expression as compared to the TD group. Yet, some research findings suggest that there may not be a true “OXT deficit” in individuals with
ASD (Young & Barrett, 2015). This then leads to the question of directionality of change in OXT levels when being targeted by behavioral intervention. Should levels continue to increase as a sign of positive change? Or would another relationship of change be expected? More work into the trajectory of OXT expression and development will help delineate if children with ASD do indeed show lower or altered levels and what increases in these levels may mean. A second reason as to why change may not have occurred centers on the age of the current sample. As stated earlier, the critical window for OXT system development may only extend up to 3 years of age (Kang et al., 2011). The 79 current sample was comprised of preschoolers ages 3-5 years. Given this older age range, perhaps any changes made in OXT expression were more activational (i.e. occurred for a short period of time thus didn’t show from pre to post intervention) than organizational in nature. It will be important to extend this work into younger populations to see if malleability in expression is more evident given the potential overlap with a critical window of development. This could be done with prospective studies which measured
OXT concentration levels in children at risk for developing ASD, such as those who have a sibling or family member with the diagnosis. A third reason, which will be discussed below in more detail, relates to the design of the current intervention program and level of functioning of the participants. It may have been that the intervention was not long enough or delivered at a high enough intensity to make an impact beyond the behavioral changes reported. Relatedly, OXT malleability may relate to the severity of social cognitive deficits. Participants in the current study were within the moderate range of functioning but showed significant difficulty in being able to engage in even basic levels of play. It may be that a population where higher level skills could be targeted may then have showed changes to OXT expression.
Behavioral Outcomes. In regards to child and parent outcome measures, the current study did find significant increases in the child’s emotion recognition ability, the overall mood of parent-child interactions, and decreases in parental stress and depressive symptoms. Further there were trending increases in divergent themes in play and joint attention ability. These findings suggest that the current intervention and modality of delivery (telehealth) were effective in enacting change in at least some of the hypothesized areas of functioning. This falls in line with previous research studies, which 80
have showed parent training models to be an accepted and effective option for increasing
parent-child engagement and decreasing the severity of children’s autism characteristics
(Scahill et al., 2012; Webster-Stratton, Reid, & Beauchaine, 2011).
The PRETEND-ASD intervention program did significantly impact emotion
recognition in the ASD sample, however, other areas, such as pretend play, did not show
significant increases in performance. These results suggest that the intervention may have
targeted more base level abilities, such as setting up conducive environments for parent-
child engagement and building emotion recognition instead of targeting higher level
skills, such as pretend play ability and overall social interest or competence. Jarrold
(2003) reviewed evidence that play can be impacted in children with ASD through
appropriately structured intervention sessions with parent or adult modeling. Relatedly,
Zyga et al. (2015) also found that the addition of an adult play partner helped children
with ASD express higher affect frequency and variety. However, Jarrold (2003) and
Short et al., (2011) point to the importance of assessment and, in order to see significant
change, it is important to evaluate the level and comprehension of pretense of the child before intervening. This is an interesting point and relates to both the level of functioning of the sample in the current study and also a discussion of how the baseline measures obtained could be used in a more effective way to determine an intervention protocol
with more flexibility in teaching to various skills levels.
For example, the ASD sample in the current study had difficulty simply engaging
with others, such as the adult examiners or their own parents, and also displayed
decreased pretend play ability at baseline. Given this, the intervention focus was tailored
to building on simple interactions between parent and child, where the parent learned 81
how to have the child face them and engage in activities such as singing along together, or more foundational play abilities, such as stacking blocks or cause and effect toys (i.e. functional play). Particular attention was paid in modeling to the parent how to set up a conducive play environment by clearing preferred items, only have a select group of toys out for the child to engage in, always having the child face them while engaging, and being positive and reinforcing in their interactions. An aim of the intervention was also to help parents understand how long they should expect to be able to have a quality interaction with their child (i.e. aim for 5 minutes of direct interaction versus 15 minutes and feel disheartened when this did not go well). The goal was to build on this baseline interaction time to help both the parent and child feel more comfortable and successful in the time spent together. During actual interactions, parents were coached on how to pick out a limited set of toys and model a simple story for the child or, if needed, how to functionally play with the toys (i.e. stack cups or blocks, sing songs with dolls, push cars). Another main focus in the intervention was building emotion recognition and understanding through the use of songs, coloring, and picture labeling. Thus, the changes seen from pre to post intervention in the current study fit with the skills and techniques used by the interventionist. Relatedly, the lower baseline ability level of the current sample could also explain why changes weren’t seen in higher level skills, such as pretend play ability or social cognitive skills such as joint attention, empathy, cooperation, or social interest and competence in parent-child interactions. It may be that the outcome measures given were capturing an ability level above and beyond the level of change that could be made with the current sample in the defined intervention timeline. Next steps could include recoding the baseline and post-intervention 82 assessments for more basic level skills to better understand the changes made during the program that may be more commensurate with the ASD sample’s level of functioning in this study.
Refining the PRETEND-ASD Intervention. The results of the current study suggest that key factors relating to emotional understanding, general mood of parent- child interactions, and areas of parental functioning, such as stress and depressive symptoms, can be impacted by an 8-week remote parent training program with once a week sessions. However, significant changes in pretend play skill, social cognitive abilities, and parent and child factors during joint interactions were not observed in the current study. In moving forward, certain changes to the intervention protocol may allow for more targeted changes based on level of functioning. For example, creating a more flexible manual based on ability level at the baseline assessment may be warranted. As
Jarrold (2003) noted, it is important to assess the level of pretense comprehension in order to know the child’s ability for change. Given this, a future version of the intervention program could include multiple levels of skill building based on certain assessment outcomes. For example, children who scored below a certain point on measures of interest (such as imagination or level of symbolic play on the APS-P) may benefit more from a program that focuses on building simple interactions with their caregivers and peers. Conversely, those participants who scored above a certain score on these same measures could perhaps benefit more from undergoing an intervention program where play and social skills are targeted.
Outside of tailoring the protocol to child baseline ability level, another important change may be more involvement of the child in the intervention sessions in general. The 83
current protocol included both recorded play practices and live coaching sessions
between the parent, child, and interventionists across the 8-week period, however direct
child involvement was more sporadic and the majority of the sessions were between the
interventionist and parent. Moving forward, it may be important to ensure that the child is
engaging with the parent and practicing a skill during each session so as to (1) give the
parent time to practice the skill and (2) provide immediate feedback and ways to keep
working on the skill moving forward. Logistically, this may mean that the individual
intervention sessions may increase in length to fit both time for parent-child practice and covering new material or the overall intervention program may benefit from a longer time course. Extending the program from 8 to 10 or 12 weeks would increase the ability to work on more skills over a longer period of time, which would help with skill formation and generalization but would also require more commitment from families. Piloting longer intervention sessions versus a longer intervention period would be an important next step. Lastly, another important change would be to include follow-up sessions after
the intervention has been completed. These “booster” sessions could provide parents a
chance to check in with the interventionist, discuss any new problems that have come up,
and gain more knowledge on how to continue to work on building skills and generalizing
them to different environments and situations after the intervention period has come to a
close.
Limitations and Future Directions
A few limitations should be noted in the present study when interpreting the
results and determining future directions. One limitation is the small sample size, which
yields low statistical power for detecting group differences. It is important to note, given 84
the small sample size, that the description of the intervention effects is preliminary and
should be interpreted with caution. However, given significant findings in key areas,
implementation of the intervention with a larger sample size may be warranted. Further,
given that it is not clear as to which components of the intervention effected change, a
larger sample size would allow for a more thorough examination of the intervention,
which techniques were most impactful on parent and child outcomes, and the opportunity
to make changes to the intervention protocol and structure as discussed above. Relatedly,
a large number of analyses were conducted given the small sample size. Bonferroni
corrections were used when appropriate, however, it will be important for future studies,
with larger samples sizes, to be conducted in order to replicate the current findings.
Another limitation to note is the cross-sectional nature of the current study which does not allow conclusions to be drawn about actual developmental change or change
through time. This makes it difficult to understand the trajectory of OXT expression and
how it may relate and change with social cognitive ability. Future studies should extend
the findings of the current study in characterizing OXT expression across developmental
stages (i.e. infancy, toddler, preschool, and school-age periods) in a longitudinal design to obtain a better understanding of the course of OXT development. Further, the lack of an intervention control group makes it difficult to understand if changes observed occurred
as a result of the intervention or simply through the passage of time and development.
However, previous work has demonstrated that the social and emotional skill deficits
observed in children developmental disabilities such as autism do not generally resolve
unaided over time (Begeer et al., 2008; Sappok et al., 2014), providing more evidence
that the gains observed in this study could have occurred due to participating in the 85
PRETEND-ASD intervention program. Nonetheless, future work should include a randomized control group to ensure intervention efficacy.
A last limitation to note is a potential bias effect with the parent report measures used in this study. Parents of children with ASD knew that they were enrolled in and completing the intervention program which could have naturally led them to report higher scores at the end of the intervention period. However, the observed results in the current study speak to the mechanisms of change employed during the intervention and are specific to certain parental variables versus across all parental report measures, suggesting specificity to the reported results. Future work should continue to include behavioral assessments when possible instead of parent report to ensure an accurate representation of child ability level and any change that occurs as a result of participating in the intervention program.
Practical Significance and Conclusion
The prevalence rate of ASD continues to rise and the deficits associated with this disorder severely impact not only the affected child but the family system as a whole.
Research into the molecular basis of ASD has begun to show a potentially important link between ASD and the OXT system. However, current treatment models, both pharmacologically and behaviorally, show limited efficacy in targeting functioning across individuals with ASD. Vital next steps in this area include exploring the biology behind observable deficits, which will provide clarity on the nature of this disorder and begin to reveal what underlying mechanisms may be implicated in disorder development or as treatment targets. The findings from this study suggest that the relationship of OXT with social cognitive ability and disorder category are complex and nuanced. Specifically, 86
results do not support the concept of an “OXT deficit” in children with ASD but rather show that oxytocin may have unique relationships with social cognitive variables across different groups and levels of functioning. Results of the study also suggest that preschoolers with ASD show deficits in social cognitive ability, parents show increased psychological distress, and parent-child dyads have disrupted interactions. A remote parent-training intervention program was shown to be effective in targeting emotional recognition, parental levels of stress and depression, and the overall mood of parent-child
engagement. Taken together, these findings suggest that further investigation of oxytocin
and its relationship to social cognitive ability in the preschool period is warranted and
may have large implications for early identification and treatment. Specifically,
additional work may delineate that OXT expression does indeed vary as a function of the
presence of disorder, implicating its measurement as a potential biomarker. In relation to
the parent-child relationship, future investigations may clarify if it is possible to use early
interactions in molding OXT system development in children with autism. Ultimately,
continued research will aid in better understanding deficits evidenced in ASD and
knowledge as to how to target these deficits earlier and more effectively, potentially
through the use of both biological and behavioral techniques.
87
Table 1: Participant Demographics TD (n = 21) ASD (n = 17) F-Value p-value Age 4.47 (0.90) 4.27 (0.81) 0.483 0.49 Gender (%male) 13 (61.9%) 14 (77.8%) 0.896 0.35 PPVT 118.86 82.07 (17.93) 52.42 0.000** (12.61) MSEL-VRS 47.57 (14.39) 31.13 (9.33) 15.00 0.000** Autism Severity (SCQ) 4.10 (1.89) 17.88 (5.21) 120.6 0.000** Child Temperament (CBQ) Activity Level 4.54 (0.83) 5.22 (0.68) 7.113 0.01* Approach 5.21 (0.66) 5.10 (0.72) 0.245 0.62 Attentional Focusing 4.67 (1.12) 3.30 (1.14) 13.34 0.001** Inhibitory Control 4.77 (1.11) 3.07 (0.88) 25.83 0.000** Anger/Frustration 4.81 (0.68) 5.26 (1.17) 2.196 0.15 Sadness 3.75 (0.67) 3.79 (0.88) 0.019 0.89 Fear 3.25 (1.16) 3.57 (1.41) 0.565 0.46 Shyness 2.66 (0.67) 4.24 (1.54) 13.65 0.001** Reactivity/Soothability 5.00 (0.94) 4.11 (0.95) 8.030 0.02* High Intensity Pleasure 5.25 (0.75) 4.99 (0.82) 1.055 0.31 Low Intensity Pleasure 6.11 (0.58) 5.47 (0.56) 11.61 0.002** Perceptual Sensitivity 5.25 (0.50) 4.67 (0.77) 7.446 0.01* PPVT: Peabody Picture Vocabulary Test; MSEL-VRS: Mullen Scales of Early Learning-Visual Reception Subscale; SCQ: Social Communication Questionnaire; †trending; *p < 0.05; **p < 0.01
88
Table 2: Baseline Oxytocin Across Groups
TD (n = 20) ASD (n = 16) F-Value p-value
OXT Concentration 54.38 (18.19) 65.62 (15.73) 2.87 0.11 (pg/ml) †trending; *p < 0.05; **p < 0.01
89
Table 3: Pretend Play Ability Across Groups (APS-P)
TD (n = 21) ASD (n = 17) F-Value p-value Imagination 3.95 (0.74) 2.60 (1.18) 7.909 0.008** Organization 3.67 (0.66) 2.33 (1.11) 10.07 0.003** Comfort 3.57 (0.87) 3.33 (0.72) 0.552 0.46 No Play (%time) 3.19 (6.59) 4.80 (9.13) 0.001 0.97 Functional Play (%time) 31.33 (24.7) 61.87 (25.4) 6.840 0.01* Symbolic Play (%time) 64.62 (27.5) 33.33 (26.7) 5.249 0.03* Divergent Themes 3.05 (2.00) 0.80 (1.08) 8.034 0.008** Transformation Frequency 0.52 (0.68) 0.20 (0.56) 0.714 0.40 Substitution Frequency 1.43 (0.81) 0.73 (0.70) 1.630 0.21
Affect Frequency 14.43 (6.74) 7.33 (5.43) 5.408 0.03* Affect Variety 4.29 (1.85) 2.47 (1.77) 3.577 0.07†
Neutral Interpersonal Acts 1.10 (1.76) 0.47 (0.74) 2.760 0.11 Aggressive Interpersonal 2.43 (2.71) 0.80 (1.82) 2.743 0.11 Acts Nurturing Interpersonal 0.43 (0.75) 0.00 (0.00) 3.170 0.08† Acts Personifications 4.19 (2.34) 1.13 (1.51) 11.55 0.002** Number of Characters 5.38 (2.13) 3.40 (2.17) 2.097 0.16 †trending; *p < 0.05; **p < 0.01
90
Table 4: Social Cognitive Ability Across Groups
TD (n = 21) ASD (n = 17) F-Value p-value Facial Affect Comprehension Evaluation (FACE) Happy 93.71 (13.3) 53.30 (45.0) 11.07 0.002** Sad 72.62 (26.1) 47.50 (36.2) 2.703 0.11 Angry 81.00 (33.5) 50.00 (47.1) 4.973 0.03* Scared 39.62 (34.4) 26.60 (26.4) 0.405 0.53 Surprised 61.90 (41.6) 20.00 (35.0) 9.595 0.004** Disgusted 64.30 (35.9) 30.00 (35.0) 3.698 0.07† Ashamed 9.50 (25.6) 0.00 (0.00) 1.232 0.28 Total Accuracy 62.57 (11.9) 36.10 (19.4) 16.39 0.000** Positive Emotions 81.00 (20.5) 50.00 (35.6) 8.752 0.006** Negative Emotions 91.19 (6.42) 78.50 (13.3) 8.981 0.006** Total Ability 88.19 (5.50) 70.50 (15.4) 17.57 0.000** Social Cognitive Tasks Joint Attentiona 1.02 (0.06) 1.56 (0.85) 4.122 0.05* Empathy 2.81 (0.46) 2.28 (0.62) 5.513 0.03* Cooperation 3.86 (0.20) 3.07 (0.60) 18.33 0.000** areverse scored; †trending; *p < 0.05; **p < 0.01
91
Table 5: Parenting Stress Across Groups (PSI-4)
TD (n = 21) ASD (n = 17) F-Value p-value Child Domain Distractibility/Hyperactivity (DI) 45.20 (7.17) 62.62 (9.45) 26.16 0.000** Adaptability (AD) 43.00 (6.81) 55.85 (10.0) 10.66 0.003** Reinforces Parent (RE) 42.15 (4.25) 48.62 (9.42) 4.801 0.04* Demandingness (DE) 46.55 (6.65) 61.46 (8.33) 18.35 0.000** Mood (MO) 48.00 (7.75) 54.08 (15.5) 1.316 0.26 Acceptability 42.65 (4.04) 54.38 (8.25) 15.16 0.001** Parent Domain Competence (CO) 43.45 (7.07) 50.62 (9.45) 3.183 0.09† Isolation (IS) 48.70 (8.95) 57.31 (10.1) 2.662 0.11 Attachment (AT) 44.85 (5.44) 46.92 (5.74) 1.416 0.24 Health (HE) 44.35 (7.39) 56.15 (11.6) 10.98 0.002** Role Restriction (RO) 44.75 (6.71) 57.92 (8.43) 16.69 0.000** Depression (DP) 48.95 (10.7) 52.23 (10.2) 0.702 0.41 Spouse/Parenting Partner 51.55 (11.9) 53.69 (10.2) 0.002 0.97 Relationship (SP) Overall Stress Scores Child Domain Stress 43.55 (5.72) 57.69 (8.38) 19.09 0.000** Parent Domain Stress 46.00 (7.96) 53.31 (9.53) 3.343 0.08† Life Stress 44.90 (7.43) 50.31 (6.60) 4.275 0.05* Total Stress 44.55 (6.51) 55.65 (7.65) 11.18 0.002** †trending; *p < 0.05; **p < 0.01
92
Table 6: Parent Functioning Across Groups
TD (n = 21) ASD (n = 17) F-Value p-value Depression, Anxiety, and Stress Scale (DASS-21) Depression 1.50 (2.01) 2.92 (3.73) 1.743 0.20 Anxiety 1.05 (1.70) 2.54 (2.30) 1.654 0.21 Stress 3.75 (3.24) 5.92 (2.81) 3.875 0.06† Parental Acceptance and Action Questionnaire (PAAQ) Action 21.70 (6.77) 24.36 (6.82) 0.990 0.33 Unwillingness 20.90 (6.15) 26.64 (6.96) 8.094 0.008** Total Score 42.60 (11.1) 51.00 (10.8) 5.180 0.03* †trending; *p < 0.05; **p < 0.01
93
Table 7: Parent-Child Interaction Across Groups TD (n = 21) ASD (n = 17) F-Value p-value Parent Variables Degree of Parental Involvementa 3.62 (0.92) 6.47 (1.13) 46.12 0.000** Unsolicited Helpa 2.86 (1.24) 5.87 (1.51) 25.18 0.000** Response to Child 2.57 (0.87) 2.67 (1.23) 0.004 0.95 Child Variables Social Interest 5.38 (0.74) 4.33 (1.59) 4.173 0.05* Social Competence 6.43 (0.98) 4.40 (1.64) 15.07 0.000** Dyad Variables Overall Mooda 1.67 (1.20) 2.89 (1.94) 2.41 0.02* Mutual Engagementa 2.24 (0.89) 3.13 (1.19) 5.063 0.03* areverse scored; †trending; *p < 0.05; **p < 0.01
94
Table 8: Correlations Between Baseline OXT & Social Cognition Across Whole Sample (N = 36)
CBQ Activity Approach Reac High Low Perceptua Shy Fear tive Intensity Intensity l Pleasure Pleasure Sensitivit y OXT 0.45* -0.05 -0.20 -0.16 -0.36* -0.16 0.28 0.02
APS-P
Imagine Organize Affec Function Symbolic Divergent Tran Aggressiv Personify t Play Play Storylines Freq interper Freq acts OXT -0.27 -0.21 -0.22 0.32 -0.33 -0.29 0.11 -0.24 -0.04 FACE Happy Sad Angr Scared Positive Negative Total Total Emotions Emotions Ability Accuracy OXT -0.01 0.15 -0.10 -0.08 -0.12 -0.08 -0.06 -0.10 Social Cognitive Tasks Joint Coop Emp Atten ath OXT 0.17 -0.31 -0.26 PCI Gen Parent Unso Response Social Social Mutual Mood Involve licit to child Interest comp. Eng. Help
OXT 0.16 0.37* 0.36* 0.10 -0.25 -0.30 0.15 †trending; *p < 0.05; **p < 0.01
95
Table 9: Hierarchical Regression Analysis for Predicting Baseline OXT across Whole Sample (N = 36)
Variable B t-value p-value R2 Adjusted ΔR2 Sig. R2 ΔF
Step 1b Activity 13.512 2.663 0.013* 0.202 0.174 0.202 0.013*
Step 2 Activity 11.621 2.306 0.029* 0.202 0.174 0.202 0.013* Parental Involvement 4.311 1.694 0.102† 0.279 0.225 0.077 0.102†
Step 3 Activity 12.052 2.331 0.028* 0.202 0.174 0.202 0.013* Parental Involvement 8.408 1.037 0.309 0.279 0.225 0.077 0.102† Unsolicited Help -3.742 -0.533 0.599 0.287 0.204 0.008 0.599
Step 4 Activity 13.553 1.808 0.083 0.202 0.174 0.202 0.013* Parental Involvement 9.184 1.055 0.301 0.279 0.225 0.077 0.102† Unsolicited Help -4.232 -0.575 0.570 0.287 0.204 0.008 0.599 Low Pleasure 2.923 0.281 0.781 0.289 0.175 0.002 0.781 Intensity bmodel of best fit; †trending; *p < 0.05; **p < 0.01
96
Table 10: Correlations Between Baseline OXT & Social Cognition within TD Sample (N = 20)
CBQ Activity Approach Reactive High Low Percept Shyness Fear Intense Intense Sensitive Pleasure Pleasure OXT 0.27 0.08 -0.02 -0.08 -0.09 0.45 0.17 0.11 APS-P Img. Org. Affect Function Symbolic Divergent Tran Agg. Personify Freq. Play Play Storylines Freq. interper acts OXT -0.31 -0.22 -0.34 0.53* -0.53* -0.37 -0.05 -0.54* 0.10 FACE Happy Sad Angry Scared Positive Negative Total Total Emotion Emotions Ability Accur OXT -0.06 0.58** 0.16 -0.13 -0.17 -0.13 0.24 -0.29 Social Cognitive Tasks Joint Cooperation Empathy Attention OXT 0.53* -0.19 -0.42 PCI General Parent Unsolicited Response Social Social Mutual Mood Involvement Help to child Interest comp. Eng.
OXT 0.43 0.30 0.29 0.15 -0.26 -0.53* 0.31 †trending; *p < 0.05; **p < 0.01 97 Table 11: Summary of Hierarchical Regression Analysis for Predicting Baseline OXT within TD Sample (N = 20)
Variable B t-value p-value R2 Adjusted R2 ΔR2 Sig. ΔF Step 1 Sad 44.819 2.967 0.009** 0.341 0.302 0.341 0.009**
Step 2b Sad 37.612 2.780 0.013* 0.341 0.302 0.341 0.009** Aggressive Interpersonal Acts -3.315 -2.490 0.024* 0.525 0.466 0.184 0.024*
Step 3 Sad 33.710 2.665 0.018* 0.341 0.302 0.341 0.009** Aggressive Interpersonal Acts -2.619 -2.046 0.059† 0.525 0.466 0.184 0.024* Functional Play 28.176 1.943 0.071† 0.621 0.545 0.095 0.071†
Step 4 Sad 32.762 2.575 0.022* 0.341 0.302 0.341 0.009** Aggressive Interpersonal Acts -2.994 -2.231 0.043* 0.525 0.466 0.184 0.024* Functional Play -18.232 -0.361 0.723 0.621 0.545 0.095 0.071† Symbolic Play -41.566 -0.960 0.353 0.644 0.542 0.023 0.353
Step 5 Sad 26.651 1.911 0.078† 0.341 0.302 0.341 0.009** Aggressive Interpersonal Acts -3.283 -2.405 0.032* 0.525 0.466 0.184 0.024* Functional Play -49.126 -0.843 0.415 0.621 0.545 0.095 0.071† Symbolic Play -57.776 -1.261 0.230 0.644 0.542 0.023 0.353 Social Competence -5.200 -1.050 0.313 0.672 0.546 0.028 0.313
Step 6 Sad 25.095 1.767 0.103† 0.341 0.302 0.341 0.009** Aggressive Interpersonal Acts -2.718 -1.780 0.100† 0.525 0.466 0.184 0.024* Functional Play -8.118 -0.107 0.916 0.621 0.545 0.095 0.071† Symbolic Play -22.210 -0.358 0.726 0.644 0.542 0.023 0.353 Social Competence -3.662 -0.689 0.504 0.672 0.546 0.028 0.313 Joint Attention 63.477 0.862 0.406 0.691 0.536 0.019 0.406 bmodel of best fit; †trending; *p < 0.05; **p < 0.01 98
Table 12: Correlations Between Baseline OXT & Social Cognition within ASD Sample (N = 16)
CBQ Activity Approach Reactive High Low Percept Shy Fear Intensity Intense Sensitive Pleasure Pleasure OXT 0.49 -0.03 -0.04 -0.05 -0.24 -0.27 -0.04 -0.23 APS-P Img. Org. Affect Function Symbol Divergent Tran Aggressive Personify Freq. Play Play Storylines Freq. interperson acts OXT 0.30 0.49 0.62* -0.51 0.53 0.60* 0.73** 0.72** 0.61* FACE Happy Sad Angry Scared Positive Negative Total Total Emotion Emotion Ability Accuracy OXT 0.55 0.31 0.14 0.12 0.37 0.32 0.34 0.39 Social Cognitive Tasks Joint Cooperate Empathy Attention OXT -0.03 -0.05 0.13 PCI General Parent Unsolicit Response Social Social Mut. Mood Involve Help to child Interest comp. Eng.
OXT -0.39 -0.14 -0.09 -0.06 0.04 0.31 -0.39 †trending; *p < 0.05; **p < 0.01
99
Table 13: Summary of Hierarchical Regression Analysis for Predicting Baseline OXT within ASD Sample (N = 16)
Variable B t-value p-value R2 Adjusted R2 ΔR2 Sig. ΔF
Step 1b Aggressive Interpersonal 12.134 3.252 0.009** 0.514 0.465 0.514 0.009** Acts
Step 2 Aggressive Interpersonal 9.239 2.207 0.055* 0.514 0.465 0.514 0.009** Acts Personifications 5.805 1.344 0.212 0.595 0.505 0.081 0.212
Step 3 Aggressive Interpersonal 11.083 1.841 0.103 0.514 0.465 0.514 0.009** Acts Personifications 7.340 1.292 0.232 0.595 0.505 0.081 0.212 Affect Frequency -1.166 -0.447 0.667 0.605 0.457 0.010 0.667
Step 4 Aggressive Interpersonal 8.907 1.530 0.170 0.514 0.465 0.514 0.009** Acts Personifications 13.445 1.993 0.086 0.595 0.505 0.081 0.212 Affect Frequency -5.442 -1.432 0.195 0.605 0.457 0.010 0.667 Divergent Storylines 20.579 1.468 0.186 0.698 0.526 0.093 0.186 bmodel of best fit; †trending; *p < 0.05; **p < 0.01
100
Table 14: Changes in Oxytocin Level after Intervention in ASD Sample (N = 13)
Baseline Post-Intervention F-Value p-value
OXT Concentration 62.91 (16.15) 69.39 (27.17) -0.669 0.52 (pg/ml) †trending; *p < 0.05; **p < 0.01
101
Table 15: Change in Pretend Play Ability After Intervention in ASD Sample (N = 15)
Baseline Post-Intervention t-value p-value Imagination 2.69 (1.18) 2.85 (1.07) -0.519 0.61 Organization 2.46 (1.13) 2.46 (0.88) 0.000 1.00 Comfort 3.38 (0.77) 3.31 (0.86) 0.365 0.72 No Play (%time) 5.53 (9.63) 4.69 (11.4) 0.215 0.83 Functional Play (%time) 58.5 (24.9) 53.5 (0.28) 0.753 0.47 Symbolic Play (%time) 35.9 (27.0) 41.8 (0.29) -0.844 0.42 Divergent Themes 0.85 (1.14) 1.23 (1.30) -1.806 0.09† Transformation Frequency 0.23 (0.60) 0.15 (0.38) 0.365 0.72 Substitution Frequency 0.69 (0.63) 1.15 (1.46) -1.148 0.27
Affect Frequency 7.46 (5.22) 8.08 (7.22) -0.300 0.77 Affect Variety 2.69 (1.75) 2.38 (1.90) 0.693 0.50
Neutral Interpersonal Acts 0.54 (0.78) 0.31 (0.48) 1.148 0.27 Aggressive Interpersonal 0.92 (1.94) 1.00 (2.00) -0.267 0.79 Acts Nurturing Interpersonal 0.00 (0.00) 0.00 (0.00) 0.000 1.000 Acts Personifications 1.31 (1.55) 1.23 (1.17) 0.210 0.84 Number of Characters 3.69 (2.10) 3.38 (2.50) 0.417 0.68 †trending; *p < 0.05; **p < 0.01 102
Table 16: Change in Social Cognitive Ability After Intervention in ASD Sample (N = 15)
Baseline Post-Intervention t-value p-value Facial Affect Comprehension Evaluation (FACE)a Happy 52.75 (46.0) 77.83 (32.8) -2.024 0.07† Sad 39.58 (37.6) 77.08 (24.9) -4.780 0.001** Angry 41.70 (46.9) 70.80 (45.0) -1.629 0.13 Scared 22.17 (26.0) 47.00 (36.2) -1.607 0.14 Surprised 16.70 (32.6) 37.50 (37.7) -1.603 0.14 Disgusted 22.70 (34.4) 45.50 (35.0) -1.242 0.24 Ashamed 0.00 (0.00) 16.70 (32.6) -1.773 0.10† Total Accuracy 31.00 (21.6) 56.42 (21.3) -4.554 0.001** Positive Emotions 50.00 (38.6) 66.70 (23.1) -1.520 0.16 Negative Emotions 60.25 (37.9) 86.33 (21.1) -2.115 0.05* Total Ability 57.42 (31.8) 81.42 (16.0) -2.661 0.02* Social Cognitive Tasks Joint Attentionb 1.54 (0.83) 1.17 (0.24) 1.802 0.09† Empathy 2.21 (0.66) 2.02 (0.18) 1.238 0.24 Cooperation 2.83 (0.90) 2.83 (1.09) 0.000 1.00 areverse scored; †trending; *p < 0.05; **p < 0.01
103
Table 17: Change in Parenting Stress After Intervention in ASD Sample (N = 15)
Baseline Post-Intervention t-value p- value Child Domain Distractibility/Hyperactivity (DI) 62.46 (8.84) 59.69 (6.55) 1.443 0.18 Adaptability (AD) 55.08 (9.30) 52.08 (9.18) 1.601 0.14 Reinforces Parent (RE) 50.31 (10.2) 48.23 (9.08) 0.633 0.54 Demandingness (DE) 61.23 (8.98) 56.77 (9.12) 2.728 0.02* Mood (MO) 50.23 (13.9) 51.85 (12.3) -0.850 0.41 Acceptability 53.85 (7.76) 53.15 (9.42) 0.478 0.64 Parent Domain Competence (CO) 52.00 (9.02) 49.08 (10.1) 1.651 0.13 Isolation (IS) 55.69 (10.9) 54.85 (12.4) 0.683 0.51 Attachment (AT) 46.31 (4.50) 51.08 (13.8) -1.317 0.21 Health (HE) 57.15 (11.1) 53.46 (12.0) 1.287 0.22 Role Restriction (RO) 58.92 (8.52) 58.31 (10.9) 0.266 0.80 Depression (DP) 52.62 (9.86) 52.15 (9.02) 0.428 0.68 Spouse/Parenting Partner 53.00 (10.2) 56.00 (10.2) -1.860 0.09† Relationship (SP) Overall Stress Scores Child Domain Stress 57.08 (8.09) 54.77 (8.37) 1.481 0.16 Parent Domain Stress 53.46 (9.25) 53.23 (9.57) 0.175 0.86 Life Stress 50.08 (6.52) 48.00 (7.26) 0.758 0.46 Total Stress 55.38 (7.51) 54.23 (8.76) 1.030 0.32 †trending; *p < 0.05; **p < 0.01
104
Table 18: Change in Parent Functioning After Intervention in ASD Sample (N = 15)
Baseline Post-Intervention t-value p- value Depression, Anxiety, and Stress Scale (DASS-21) Depression 3.75 (3.72) 2.25 (3.28) 2.913 0.01* Anxiety 2.08 (2.58) 1.75 (2.09) 0.374 0.72 Stress 6.25 (2.86) 4.17 (2.59) 2.177 0.05* Parental Acceptance and Action Questionnaire (PAAQ) Action 24.50 (6.35) 22.42 (8.98) 1.332 0.21 Unwillingness 26.58 (7.44) 26.17 (8.03) 0.271 0.79 Total Score 51.08 (11.3) 48.58 (12.2) 1.228 0.25 †trending; *p < 0.05; **p < 0.01
105
Table 19: Change in Parent-Child Interaction After Intervention in ASD Sample (N = 15)
Baseline Post- t-value p-value Intervention Parent Variables Degree of Parental Involvementa 6.29 (1.07) 6.64 (1.08) -0.924 0.37 Unsolicited Helpa 5.64 (1.45) 6.14 (1.29) -0.959 0.36 Response to Child 2.93 (1.21) 3.00 (1.18) -0.268 0.79 Child Variables Social Interest 4.36 (1.69) 3.93 (1.39) 1.385 0.19 Social Competence 4.43 (1.74) 3.79 (1.53) 1.605 0.13 Dyad Variables Overall Mood 2.29 (1.33) 3.36 (1.74) -2.519 0.03* Mutual Engagementa 3.07 (1.21) 3.43 (1.09) -1.439 0.17 areverse scored; †trending; *p < 0.05; **p < 0.01
106
Figure 1: STUDY VISIT TIMELINES
Figure note: ASD participants (n = 15) will immediate enroll and begin the PRETEND intervention program after the baseline visit; post-intervention visit includes only 1 saliva sample collection and does not include a re-administration of the PTI-2 (cognitive assessment)
107
Figure 2: PRETEND Parent Training Program Intervention Schedule Time Points Events Enrollment Baseline Assessment (in-person)
Week 1 Intervention Session 1: The Social Cognitive Profile Homework Worksheet
Week 2 Intervention Session 2: How to be a good play partner [live coaching session] Play-based Homework
Week 3 Intervention Session 3: Building skills in play [live coaching session] Play-based Homework
Week 4 Intervention Session 4: The ABC’s of Behavior Homework Worksheet
Week 5 Intervention Session 5: All about Emotions Play-based Homework
Week 6 Intervention Session 6: Behavior change & emotion regulation Play-based Homework
Week 7 Intervention Session 7: Improving Social communication and interaction Play-based Homework
Week 8 Intervention Session 8: Putting it all together
Study Termination Post‐Intervention Assessment (in‐person visit within two weeks of session completion)
108
Table 20: Linear Regressions for Predicting Baseline OXT across Whole Sample (N = 36)
Child Temperament (CBQ)
Variable B S.E. B Beta t-value p-value R2 Adjusted R2 Activity 13.512 5.075 0.45 2.663 0.01* 0.20 0.17
Low Intensity Pleasure -14.499 7.087 -0.36 -2.046 0.05* 0.13 0.10
Parent Child Interaction
Variable B S.E. B Beta t-value p-value R2 Adjusted R2 Parental Involvement 5.776 2.729 0.366 2.117 0.04* 0.13 0.10 Unsolicited Help 4.796 2.341 0.356 2.048 0.05* 0.13 0.10
109
Table 21: Linear Regressions for Predicting Baseline OXT within TD Sample (N = 20)
Child Temperament (CBQ) Variable B S.E. B Beta t-value p-value R2 Adjusted R2 Perceptual Sensitivity 16.884 8.290 0.454 2.037 0.06 0.21 0.16
Pretend Play Ability (APS-P) Variable B S.E. B Beta t-value p-value R2 Adjusted R2 Functional Play 45.503 17.562 0.532 2.591 0.02* 0.28 0.24 Symbolic Play -39.680 15.467 -0.528 -2.565 0.02* 0.28 0.24 Aggressive -4.107 1.537 -0.544 -2.673 0.02* 0.30 0.25 Interpersonal Acts
Emotion Recognition (FACE) Variable B S.E. B Beta t-value p-value R2 Adjusted R2 Sad 44.819 15.103 0.584 2.967 0.009** 0.34 0.30
Social Cognitive Ability Variable B S.E. B Beta t-value p-value R2 Adjusted R2 Joint Attention 165.578 65.104 0.525 2.543 0.02* 0.28 0.23
Parent Child Interaction Variable B S.E. B Beta t-value p-value R2 Adjusted R2 General Mood 7.699 3.766 0.434 2.044 0.06 0.19 0.14 Social Competence -11.020 4.296 -0.528 -2.565 0.02* 0.28 0.24
110
Table 22: Linear Regressions for Predicting Baseline OXT within ASD Sample (N = 16)
Pretend Play Ability (APS-P)
Variable B S.E. B Beta t-value p-value R2 Adjusted R2 Affect Frequency 3.623 1.465 0.616 2.474 0.03* 0.38 0.32
Personifications 10.711 4.361 0.613 2.456 0.03* 0.38 0.31 Aggressive 12.134 3.731 0.717 3.252 0.009** 0.51 0.47 Interpersonal Acts Divergent Storylines 17.570 7.449 0.598 2.359 0.04* 0.36 0.29 Transformation 36.906 10.996 0.728 3.356 0.007** 0.53 0.48 Frequency
111
ICARE Training Protocol Recruitment
• Interested families will be sent the eligibility link to complete: https://cwru.az1.qualtrics.com/jfe/form/SV_cGAQ4A32Qk4m1yR
• Families who have a child, either TD or ASD, that is between 3-5 years, is verbal, able to sit at a table, minimal behavioral problems, and average IQ will be contacted to participant o Follow-up with Olena if any questions about eligibility
Enrollment
• Eligible families will be emailed to schedule a date and time to come into the lab • Study visits must begin between 8-10am and will last approximately 2 hours • When confirming with the family, ask for their address to mail the survey packets to • Send information on location and parking once date is set
Before the Study Visit
• Mail survey packets to family o Include letter, Demographics, SCQ, CBQ, SSiS, PSI-4, DASS-21, and PAAQ • Follow-up with the family to confirm the visit and ask if any questions about location or time
Baseline Visit
• Complete the consent and PHI forms once the family arrives • Next, collect the baseline saliva sample [follow procedures in regulatory binder] o Store sample in subzero freezer immediately after collection • After saliva collection, administer the following assessments: o PPVT o Mullens (visual reception subscale) o PTI-2 o MSFM o KAI-R o FACE o Social Cognitive Behavioral Tasks o APS-P o Parent-Child Free Play Task • End the visit with the post parent-child engagement saliva sample collection o Store sample in subzero freezer immediately after collection
Date: May 15, 2017 Version 1.0 112
Intervention (ASD ONLY)
• Children with ASD and their parents will automatically be enrolled into the PRETEND telehealth intervention program o The program will be explained to families at the baseline visit o A tech check to test Adobe Connect will also be schedule at the baseline visit o Lastly, the manual and intervention materials [bag of toys] will be given to the family at baseline visit • After the tech check is complete, the family will complete once a week sessions for 8 weeks following the manualized schedule
Post-Intervention Visit (ASD ONLY)
• This visit should be scheduled between 8-10am and will last 60-90 minutes • The visit will begin with a baseline saliva sample [follow procedures in regulatory binder] o Store sample in subzero freezer immediately after collection • After saliva collection, administer the following assessments: o MSFM o KAI-R o FACE o Social Cognitive Behavioral Tasks o APS-P o Parent-Child Free Play Task • There is NO post parent-child engagement saliva collection sample for this visit • After completing the visit, email the parent the link to complete the BIRs survey
Date: May 15, 2017 Version 1.0 113
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