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Journal of Autism and Developmental Disorders (2021) 51:4644–4654

https://doi.org/10.1007/s10803-021-04905-0

ORIGINAL PAPER

Factors Associated with Restricted, Repetitive Behaviors and Interests


and Diagnostic Severity Level Ratings in Young Children with Autism
Spectrum Disorder
Esther Hong1 · Johnny L. Matson1

Accepted: 29 January 2021 / Published online: 12 February 2021


© The Author(s), under exclusive licence to Springer Science+Business Media, LLC part of Springer Nature 2021

Abstract
Autism spectrum disorder (ASD) is a neurodevelopmental disorder characterized by restricted, repetitive patterns of behavior
and interests (RRBIs). With the latest update to the Diagnostic and Statistical Manual of Mental Disorders, a severity level
rating is assigned to the two core features of ASD (American Psychiatric Association in Diagnostic and statistical manual
of mental disorders 5 American Psychiatric Association Washington, D.C., 2013). Previous studies have identified factors
associated with RRBI severity; however, the relationship among RRBIs, adaptive functioning, and diagnostic severity level
remains unclear. The present study investigated whether adaptive functioning and parent-reported ASD symptoms predict
RRBI severity in young children with ASD. Additionally, a fine-grained analysis was conducted to examine the factors asso-
ciated with diagnostic severity level ratings. Several significant associations were found. Study findings and implications for
assessment and treatment of RRBIs are discussed.

Keywords Restricted behaviors · Repetitive behaviors · ASD severity level · Adaptive functioning · Autism spectrum
disorder

Individuals with autism spectrum disorder (ASD) exhibit an emphasis on the early detection and treatment of the core
impairments in social communication and social interac- symptoms of ASD (Chawarska et al. 2007). Although early
tion as well as restricted, repetitive behaviors and inter- intensive behavioral intervention (EIBI) has demonstrated
ests (RRBIs; American Psychological Association [APA] significant improvements in the areas of socialization, cog-
2013). According to the Diagnostic and Statistical Manual of nition, and language in children with ASD (Landa 2018;
Mental Disorders, Fifth Edition (DSM-5; APA 2013), there Zwaigenbaum et al. 2015), RRBIs have not been a primary
are four categories of RRBIs: (1) stereotyped or repetitive focus of early interventions (Leekam et al. 2011; Lewis and
speech, motor movements, or use of objects, (2) excessive Bodfish 1998; MacDonald et al. 2007; Odom et al. 2010).
adherence to routines, ritualized patterns of verbal or non- Given that the presence of RRBIs is a core feature of
verbal behavior, or excessive resistance to change, (3) highly ASD, they manifest early in a child’s development and can
restricted, fixated interests that are abnormal in intensity be detected at as early as 17–37 months of age (Matson et al.
or focus, (4) hyper- or hypo-reactivity to sensory input or 2009a, b, c; Ozonoff et al. 2008a, b; Rogers 2009; Yirmiya
unusual interest in sensory aspects of the environment. The and Charman 2010). However, the assessment of RRBIs can
presence of RRBIs can impair daily functioning and result in be challenging, as RRBIs are behaviorally-defined symp-
poor, long-term outcomes (Koegel and Covert 1972; Pierce toms that can take many forms (Bodfish et al. 2000; Lewis
and Courchesne 2001; Raulston and Machalicek 2018). With and Bodfish 1998), present in other conditions (e.g., intellec-
increased prevalence and awareness of ASD, there has been tual disability [ID]; Berkson et al. 1995; Matson et al. 1997),
and overlap in presentation (APA 2013). Moreover, there is
a lack of sensitive screening and assessment measures that
* Esther Hong can detect the subtle differences in RRBIs and changes in
ehong1@lsu.edu RRBIs over time (Honey et al. 2012; Raultson and Macha-
1
Department of Psychology, Louisiana State University, 236 licek 2018). In response to these assessment challenges,
Audubon Hall, Baton Rouge, LA 70803, USA researchers have developed several measurement tools for

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the screening and assessment of RRBIs in young children adaptive behavior composite scores were associated with
with or at risk for ASD, which include standardized, direct higher repetitive motor and sensory behaviors (Cuccaro et al.
observational methods (Lord et al. 2012) and indirect meth- 2003). Further, more severe deficits in adaptive behavior
ods such as questionnaires, rating scales, and informant- in conjunction with high frequency of RRBIs were found
based interviews (Bodfish et al. 1999; Honey et al. 2012; to be risk factors for challenging behaviors (Oliver et al.
Kim and Lord 2010; Le Couteur et al. 2003; Lewis and 2012). Consistent with the recent emphasis on increasing
Bodfish 1998). Despite existing limitations in RRBI assess- adaptive behaviors and decreasing challenging behaviors in
ment (Honey et al. 2012; Hus et al. 2007), informant-based EIBI programs for young children with ASD (Makrygianni
measures have been supported in the research, as parents and et al. 2018; Minjarez et al. 2020), further exploration of
caregivers have demonstrated the ability to reliably identify the relationship between adaptive functioning and RRBIs
ASD symptoms in their children as early as 12–18 months is warranted to optimize treatment outcomes and mitigate
of age (Gray and Tonge 2005; Reznick et al. 2007). the detrimental effects of challenging behaviors. Findings
The assessment and monitoring of RRBIs is further may have significant implications for clinical practice (e.g.,
complicated by changes in the topography, severity, and teaching adaptive skills to decrease RRBIs and challenging
frequency of RRBIs across the lifespan (Johnson et al. behaviors, identifying subtypes of ASD).
2006; Lam and Aman 2007). Indeed, the prevalence and In the most recent update to the DSM-5, a dimensional
presentation of RRBIs vary considerably across individu- assessment of ASD severity was introduced, allowing clini-
als (Bradley et al. 2016; Leekam et al. 2011) depending on cians to assign a diagnostic severity level rating to each of
several individual-specific variables such as age, gender, the two core features of ASD (APA 2013). Although the
symptom severity, cognitive functioning, language skills, DSM-5 provides some qualitative guidance for clinicians
adaptive functioning. For instance, low-level RRBIs (e.g., and researchers, there are no objective methods on how
stereotyped, repetitive motor movements and object use; severity level ratings should be determined (Mechling and
sensory behaviors; Rapp and Vollmer 2005; South et al. Tassé 2016). For instance, it is not clear if clinicians and
2005) have been typically observed in children with younger researchers assign ASD severity level ratings according to
age, greater developmental delays, and/or lower cognitive the severity of core ASD symptoms or if they make deter-
ability (Prior and Macmillan 1973; Turner 1999), whereas minations strictly based on the child’s need for support and
high-level RRBIs (e.g. perseverative interests; repetitive intervention. In the latter case, it is unclear if other areas of
questioning, obsessions, and compulsions; Boyd et al. 2012) impairment (e.g., cognition, language, challenging behav-
have been observed in children with higher cognitive and iors) contribute to the perceived level of support required
language abilities (Bishop et al. 2006; Esbensen et al. 2009; (Mazurek et al. 2018). Thus, it is possible that clinicians
Richler et al. 2010). and researchers are conceptualizing severity level ratings
Regarding ASD symptom severity, previous studies have subjectively.
indicated a strong relationship between the core symptoms Although this area of research has been understudied,
of ASD (Dworzynski et al. 2009; Kuenssberg and McKenzie researchers have recently begun to investigate factors that
2011). For instance, young children (17–37 months) with contribute to determinations of ASD severity level ratings.
higher ASD severity exhibited a greater number of RRBIs In regard to the level of functional impairment and ASD
than those lower ASD severity (Matson et al. 2009a, b, c). severity level, Weitlauf et al. (2014) reported mixed asso-
Further, participants with higher ASD severity also had ciations among skill domains (i.e., cognitive functioning,
higher severity ratings on all of the 30 RRBI items exam- adaptive functioning, ASD symptoms) and assigned ASD
ined in the study. These findings suggest that greater overall severity level ratings, which suggests that there is no uniform
ASD severity has a positive association with RRBI severity. method of assigning ASD severity level ratings based on a
Deficits in adaptive skills is not a diagnostic requirement child’s level of impairment. Nevertheless, there appears to
for ASD (APA 2013). Nevertheless, many individuals with be some consistency among parental ratings of symptom
ASD experience pervasive impairments in adaptive func- severity, clinicians’ ratings of symptom severity, and behav-
tioning (Klin et al. 1992; Volkmar et al. 2004). Although ioral observations. In a recent study, Mazurek et al. (2018)
adaptive functioning appears to covary with RRBIs, this found consistency between parent-reported RRBI severity
relationship has been overlooked in the existing literature scores (i.e., according to the Aberrant Behavior Checklist;
(Cuccaro et al. 2003) and remains unclear. In individuals Aman and Singh 1986), diagnostic observation score (i.e.,
with ID, higher rates of stereotypic behaviors have been according to the Autism Diagnostic Observation Schedule,
found to be associated with lower scores on all three adap- Second Edition; Lord et al. 2012), and clinician-assigned
tive domains (i.e., communication, daily living skills, ASD severity level ratings. However, there was no associa-
socialization; Matson et al. 1997). Similar findings have tion between parental ratings and ASD severity level rat-
been observed in individuals with ASD, such that lower ings on the social communication domain. Further, cognitive

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4646 Journal of Autism and Developmental Disorders (2021) 51:4644–4654

functioning and age were found to influence ASD severity C, EarlySteps provides services to infants and toddlers
level ratings on both the social communication and RRBI under the age of 36 months, who have or are at risk for
domains (Mazurek et al. 2018), which indicates that other having a developmental delay. Children enrolled in Ear-
areas factors conflated ASD severity level ratings. lySteps who were found to be “at risk” for ASD accord-
Although research on the factors associated with RRBIs ing to an ASD screener (i.e., Baby and Infant Screen for
have expanded considerably over the years, the relationship Children with aUtism Traits, Part 1; Matson et al. 2007)
among these factors and RRBIs still remains unclear. In par- were referred to Louisiana State University’s Psychologi-
ticular, the existing literature has not sufficiently examined cal Services Center for a formal assessment of develop-
associations between adaptive functioning and RRBIs (Cuc- mental functioning.
caro et al. 2003) in infants and toddlers with ASD. There- To be included in the study, participants had to meet the
fore, the current study aimed to answer several questions. following criteria: (a) were 37 months or under at the time
First, are any demographic factors (i.e., age, gender) and of assessment, (b) have a DSM-5 diagnosis of ASD, (c)
adaptive skill domains (i.e., socialization, communication, were administered the BISCUIT- Part 1, (d) were admin-
daily living skills, motor skills) predictive of RRBI severity? istered the VABS-3, and (e) were administered the CARS2.
Second, do parent-reported ratings of socialization deficits, These criteria were applied to a pool of 317 children (age
communication deficits, and RRBI severity predict diagnos- ranging from 19 to 37 months) in a pre-existing database
tic ASD severity level ratings? Lastly, a fine-grained analysis comprised of data from diagnostic evaluations. Cases with
was conducted to determine which factors (i.e., Baby and missing relevant data and/or errors (n = 226) were removed
Infant Screen for Children with aUtIsm Traits- Part 1 items, from the sample, which resulted in a final sample size of
Childhood Autism Rating Scale, Second Edition total score 91 participants. The age of participants ranged from 19 to
and subscale scores, Vineland Adaptive Behavior Scales, 37 months (M = 30.08, SD = 4.31). The study participants
Third Edition composite score and subdomain scores) are were 84.6% male (n = 77) and 15.4% female (n = 14). Of
associated with diagnostic ASD severity level ratings. Impli- the total sample, 20.9% were African American, 64.8%
cations of the present study’s findings are expected to poten- were White, 4.4% were Hispanic, and 9.9% were identified
tially identify the predictive relationships among RRBIs and as another ethnicity. Participant characteristics of the study
other related factors and to guide individualized intervention sample are reported in Table 1. Participants were assigned
plans for the treatment of RRBIs. to one of three groups based on their diagnostic ASD
severity level rating per the DSM-5 criteria: ASD-Level 1,
ASD-Level 2, and ASD-Level 3. The ASD diagnoses were
Methods given by a licensed clinical psychologist based on results
from formal assessment measures, parent interview, and
Participants direct observation of the child in the clinic. There were no
significant group differences in age, gender, and ethnicity.
Participants in the current sample were recruited through
EarlySteps, Louisiana’s statewide early intervention pro-
gram. Under the Individuals with Disabilities Act, Part

Table 1  Demographic Total (N = 91) ASD-Level 1 ASD-Level 2 ASD-Level


information of the total sample (n = 6, 6.6%) (n = 30, 33%) 3 (n = 55,
and by group 60.4%)

Gender [N (%)]
Male 77 (84.6%) 6 (100%) 24 (80%) 47 (85.45%)
Female 14 (15.4%) 0 (0%) 6 (20%) 8 (14.55%)
Age in months
M (SD) 30.08 (4.31) 29.17 (2.99) 29.80 (5.23) 30.33 (3.91)
Range 19–37 24–33 19–37 20–37
Ethnicity [N (%)]
African American 19 (20.9%) 0 (0%) 5 (16.67%) 14 (25.45%)
White 59 (64.8%) 6 (100%) 19 (63.33%) 34 (61.82%)
Hispanic 4 (4.4%) 0 (0%) 1 (3.33%) 3 (5.45%)
Other 9 (9.9%) 0 (0%) 5 (16.67%) 4 (7.27%)

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Measures that the child is sometimes able to perform the task, and
a “2” indicates that the child is usually able to perform
Baby and Infant Screen for Children with aUtIsm Traits‑Part the task. Some items are rated as “yes” or “no”. The items
1 (BISCUIT‑Part 1; Matson et al. 2007) are scored to yield an overall adaptive behavior composite
(ABC) score and four subdomain scores: Communication
The BISCUIT-Part 1 is the diagnostic component of the (COMM), Daily Living Skills (DLS), Socialization (SOC),
BISCUIT, a three-part, informant-based assessment battery Motor Skills (MOT). The ABC, COMM, DLS, SOC, and
designed to detect symptoms of ASD in infants and toddlers MOT scores will be used in the present study as a measure
aged 17 to 37 months. The BISCUIT-Part 1 is comprised of a child’s adaptive functioning.
of 62 items that are scored on a 3-point Likert scale. The
parent/caregiver of the child is instructed to rate each item Childhood Autism Rating Scale, Second (CARS2; Schopler
in comparison to other same-aged children as: “0”—not et al. 2010)
different; no impairment, “1”—somewhat different; mild
impairment, or “2”—very different, severe impairment. The CARS2 is an instrument used to assist in the diagno-
The total BISCUIT-Part 1 score is calculated by adding each sis of ASD. The CARS2 was designed to identify children
item score. A total score between 0 and 16 is categorized in 2 years and older with mild to severe symptoms of ASD. The
the “No ASD/Atypical Development” range, a total score CARS2 has been found to differentiate among children with
between 17 and 38 is categorized in the “Possible ASD” ASD and children with other developmental disabilities. The
range, and a total score between 39 and 124 is categorized CARS2 measures functioning in 15 categories: Relating to
under “Probable ASD” range. Thus, children who receive a People; Imitation; Emotional Response; Body Use; Object
cut-off score of 17 or higher are considered at risk for ASD Use; Adaptation to Change; Visual Response; Listening
and should receive further assessment. Response; Taste, Smell, and Touch Response and Use; Fear
The BISCUIT-Part 1 has been found to have strong psy- or Nervousness; Verbal Communication; Nonverbal Com-
chometric properties, with an internal reliability of 0.97 munication; Activity Level; Level and Consistency of Intel-
(Matson et al. 2009a, b, c) and high validity (i.e., sensitiv- lectual Response; General Impressions.
ity of 93.4, specificity of 86.6, overall correct classification The clinician rates the items based on direct observation,
rate of 88.8) against the Modified Checklist for Autism in parent or caregiver report, and/or other sources of informa-
Toddlers (Matson et al. 2009a, b, c). An exploratory fac- tion (e.g., medical records, teacher reports). Each item is
tor analysis of the BISCUIT-1 yielded three distinct factors: scored on 4-point scale: a score of “1” indicates no impair-
Socialization/Nonverbal Communication (S/NVC), Repeti- ment/normal development, “2” indicates mildly abnormal
tive Behavior/Restricted Interests (RRBI), and Communi- behavior, “3” indicates moderately abnormal behavior,
cation (Matson et al. 2010). Internal consistency of each and “4” indicates severely abnormal behavior. The item
factor was high, with a Cronbach’s alpha of 0.93 in Factor 1, scores are summed to produce a total score (i.e., severity
Cronbach’s alpha of 0.90 in Factor 2, and Cronbach’s alpha rating), which ranges from 15 to 60. The total score is then
of 0.87 in Factor 3. The S/NVC subscale is comprised of used to assign a severity group: a total score between 15
24 items, the RRBI subscale is comprised of 23 items, and and 29 indicates “Minimal-to-No Symptoms of ASD”, a
the Communication subscale is comprised of 7 items, with total score between 30 and 36 indicates “Mild-to-Moderate
item-total correlations ranging from 0.320 to 0.702 (Matson Symptoms of ASD”, and a total score of 37 and higher indi-
et al. 2010). cates “Severe Symptoms of ASD”. Reliability and validity
evidence for the CARS2 is unavailable; however, the original
Vineland Adaptive Behavior Scales, Third Edition (VABS‑3; CARS (Schopler et al. 1986), has demonstrated high internal
Sparrow et al. 2016) consistency (reliability coefficient alpha of 0.94) and validity
(r = 0.84; Schopler et al. 1980).
The VABS-3 is an assessment tool designed to aid in the
assessment of intellectual and developmental disabilities. Procedure
There are three forms of the VABS-3, including the Inter-
view Form, Parent/Caregiver Form, and Teacher Form. The The Louisiana State University Institutional Review Board
Interview Form is administered by the examiner using a and the Office for Citizens with Developmental Disabili-
semi-structured interview method, and the examiner rates ties (OCDD) of the State of Louisiana approved the study
each item based on the parent/caregiver’s responses. Items prior to data collection. The BISCUIT, Vineland-3, and
are rated on a 3-point Likert scale, according to the child’s CARS2 were administered by graduate student clinicians as
ability to complete a task. A rating of “0” indicates that part of the formal assessment of developmental function-
the child is never able to perform the task, a “1” indicates ing, which was comprised of a parent/caregiver interview,

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administration of standardized measures, and direct observa- Table 2  Correlations between Variables ρ
tion of the child. All evaluations were conducted by gradu- RRBI severity and potential
independent variables for the Age − 0.119
ate student clinicians and supervised by a licensed clinical stepwise regression model
psychologist. Prior to the start of the evaluation, informed Gender − 0.055
consent to participate in research was obtained from the VABS-3- MOT − 0.423***
parent or caregiver of the child receiving the evaluation. VABS-3- SOC − 0.615***
Relevant data from the evaluation (e.g., gender, age, race, VABS-3- COM − 0.307**
assessment measure scores, clinical diagnosis) were inputted VABS-3- DLS − 0.356***
into a pre-existing database for ongoing research purposes. N = 89. *p ≤ 0.05, **p < 0.01,
Personal identifiers (e.g., name, date of birth) were removed ***p ≤ 0.001
from the database.

as measured by the VABS-3, improved the prediction of


Statistical Analyses RRBI severity scores, as measured by the BISCUIT-Part
1 RRBI subscale score. The average scores for the VABS-3
All statistical analyses were performed using SPSS 26.0. subdomains and RRBI subscale were as follows: VABS-3-
First, a stepwise multiple regression was conducted to deter- MOT (M = 82.64, SD = 12.94), VABS-3- SOC (M = 66.99,
mine the predictive influence of several independent vari- SD = 13.64), VABS-3- COMM (M = 55.27, SD = 20.38),
ables on RRBI severity. Predictor variables included MOT, VABS-3- DLS (M = 70.81, SD = 15.40), RRBI subscale
SOC, COMM, and DLS subscale scores from the VABS-3, score (M = 20.52, SD = 9.11).
and the dependent variable was the BISCUIT-Part 1 RRBI There was independence of observations, as assessed
subscale score. An ordinal regression was conducted to by a Durbin-Watson statistic of 2.194. According to Field
determine which BISCUIT-Part 1 subscales (i.e., S/NVC, (2013), the Durbin-Watson statistic can range from 0 and
RRBI, Communication) predicted diagnostic ASD sever- 4, with a value of approximately 2 indicating independ-
ity level rating. The BISCUIT-Part 1 subscale scores were ence of residuals. There was linearity between the depend-
the independent (predictor) variables and diagnostic ASD ent variable and each of the independent variables, as
severity level rating was the dependent variable. Finally, a assessed using a plot of studentized residuals against the
series of Spearman’s rank-order correlations were conducted predicted values and partial regression plots. There was
to assess the strength of the relationship between several homoscedasticity, as assed by visual inspection of a plot
factors (i.e., BISCUIT-Part 1 items, CARS2 total score, of studentized residuals versus unstandardized predicted
CARS2 severity group, CARS2 subscale scores, VABS-3 values. Multicollinearity was assessed using the tolerance
ABC, VABS-3 subdomain scores) and diagnostic severity and variance inflation factors (VIF) values. There was no
level rating. evidence of multicollinearity, as assessed by tolerance val-
ues of greater than 0.1 and VIF values of less than 10 (Hair
et al. 2014). There were no outliers, such that there were
no studentized deleted residuals greater than ± 3 standard
Results deviations, no leverage values greater than 0.2, and no
values for Cook’s distance above 1. The assumption of
RRBIs and Adaptive Functioning normality was met, as assessed by a Normal Q-Q Plot of
the studentized residuals.
To identify influential predictors for the stepwise multi- In Model 1, RRBI severity was significantly predicted
ple regression model, Spearman’s rank correlations were by the SOC subdomain score alone, R 2 = 0.40, F (1,
first conducted to examine the strength of the association 87) = 58.69, p = 0.00. The addition of the MOT subdo-
between the six potential predictor variables and the depend- main score (Model 2) also led to a statistically signifi-
ent variable. Four variables (i.e., MOT, SOC, COMM, and cant increase in variance, ΔR2 = 0.005, F (2, 86) = 29.63,
DLS of the VABS-3) had significant negative correlations p = 0.00. In Model 3, the addition of DLS also led to a
with RRBI severity. Two variables (i.e., age, gender) were statistically significant increase in variance, ΔR2 = 0.008,
not significantly associated with RRBI severity. See Table 2 F (3, 85) = 20.22, p = 0.00. Finally, the addition of the
for Spearman’s correlation coefficients. Therefore, MOT, COMM subdomain score (Model 4) led to a statisti-
SOC, COMM, AND DLS were included in the regression cally significant change, ΔR 2 = 0.026, F (4, 84) = 16.68,
model and age and gender were excluded from the model. p = 0.00. See Table 3 for the stepwise multiple regres-
A stepwise multiple regression was run to deter- sion model prediction of RRBI severity using VABS-3
mine if the addition of MOT, SOC, COMM, and DLS, subdomains.

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ASD Severity Level Rating

53.88*** (5.15)
− 0.49*** (0.08)
− 0.08 (0.07)
− 0.01 (0.075)
0.11* (0.06)
A logistic regression was conducted to determine if BIS-
CUIT-Part 1 subscale scores (i.e., S/NVC, RRBI, Commu-

B (SE)
nication) based on parent ratings predicted diagnostic ASD
severity level ratings. There was no evidence of multicol-
linearity, as assessed by tolerance values of greater than 0.1

16.68***
and VIF values of less than 10. The assumption of propor-
tional odds was met, as assessed by a full likelihood ratio test
F

comparing the fit of the proportional odds location model to


Model 4

0.44*

a model with varying location parameters. Increased RRBI


R2

severity did not significantly predict diagnostic severity level


ratings, b = 0.06, χ2(1) = 3.47, p = 0.06. Increased severity
51.24*** (5.06)
− 0.43*** (0.07)
− 0.09 (0.07)
0.07 (0.07)
of SOC did not significantly predict diagnostic severity level
rating, b = -0.03, χ2(1) = 1.336, p = 0.25. Finally, increased
severity of COMM did not significantly predict diagnostic
B (SE)

severity level rating, b = 0.08, χ2(1) = 0.58, p = 0.45.


Finally, a series of Spearman’s rank-order correlations
were conducted to examine relationships between several
20.22***

variables and diagnostic ASD severity level. Given the large


number of comparisons in this analysis, a Bonferroni adjust-
F

ment for multiple comparisons was made, with an adjusted


Model 3

0.42**

alpha level of p ≤ 0.001. Table 4 displays the Spearman’s


R2

correlation coefficients for each variable examined. On the


BISCUIT- Part 1, no items or subscales were found to be sig-
51.80*** (5.04)
− 0.39*** (0.07)
− 0.06 (0.07)

nificantly correlated with diagnostic severity level. Regard-


ing the CARS2, the Body Use (ρ(91) = 0.341, p = 0.001) and
General Impressions (ρ(91) = 0.393, p = 0.000) subscales
B (SE)

were significantly positively correlated with diagnostic


severity level. The CARS2 severity group (ρ(91) = 0.359,
p = 0.000) was also significantly positively correlated with
29.63***

diagnostic severity level rating. On the VABS-3, COMM


(ρ(87) = − 0.328, p = 0.001), MOT (ρ(87) = − 0.336,
Table 3  Stepwise multiple regression for variables predicting RRBI severity

p = 0.001), and ABC (ρ(87) = − 0.362, p = 0.000) were


Model 2

0.41**

significantly negatively correlated with diagnostic severity


level. No other variables were significantly correlated with
R2

ASD severity level.


48.92*** (3.78)
− 0.42*** (0.06)
RRBI Severity (RRBI Subscale Score)

Discussion
B (SE)

The present study examined several factors associated with


N = 89. *p ≤ 0.05, **p < 0.01, ***p ≤ 0.001

both RRBI severity and diagnostic severity level ratings


58.69***

among infants and toddlers with ASD. Consistent with pre-


vious studies (Cuccaro et al. 2003; Matson et al. 1997), the
F

current study found that lower adaptive functioning scores


Model 1

were significantly associated with higher RRBI severity.


0.40
R2

Of the four adaptive subdomains examined in this study,


the Socialization subdomain had the strongest correlation
VABS-3 COMM

with RRBI severity (ρ = − 0.615), followed by Motor Skills


VABS-3 MOT
VABS-3 SOC

VABS-3 DLS

(ρ = − 0.423), Daily Living Skills (ρ = − 0.356), and Com-


Constant
Variable

munication (ρ = − 0.307). This study is among the first to


examine the predictive influence of adaptive functioning on

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Table 4  Correlations between Variable Rho (ρ) Variable Rho (ρ)


BISCUIT-Part 1, CARS2, and
VABS-3 variables and ASD BISCUIT- Part 1 BISCUIT- Part 1
severity
Item 1 − 0.071 Item 46 − 0.020
Item 2 − 0.038 Item 47 0.110
Item 3 − 0.068 Item 48 − 0.040
Item 4 0.072 Item 49 0.071
Item 5 0.000 Item 50 0.179
Item 6 0.206 Item 51 0.089
Item 7 − 0.005 Item 52 − 0.020
Item 8 − 0.106 Item 53 0.199
Item 9 0.104 Item 54 0.104
Item 10 − 0.030 Item 55 0.032
Item 11 0.073 Item 56 0.057
Item 12 0.102 Item 57 0.234
Item 13 0.117 Item 58 0.175
Item 14 − 0.111 Item 59 0.031
Item 15 − 0.060 Item 60 0.129
Item 16 − 0.071 Item 61 0.016
Item 17 − 0.094 Item 62 0.005
Item 18 − 0.049 S/NVC score 0.011
Item 19 − 0.071 RRBI score 0.122
Item 20 − 0.098 Communication score − 0.004
Item 21 0.034 Total Score 0.070
Item 22 0.149 CARS-2
Item 23 − 0.040 Relating to people 0.077
Item 24 − 0.065 Imitation 0.306
Item 25 0.114 Emotional response 0.150
Item 26 0.278 Body use 0.341*
Item 27 0.012 Object use 0.302
Item 28 − 0.037 Adaptation to change − 0.040
Item 29 − 0.086 Visual response 0.184
Item 30 0.091 Listening response − 0.019
Item 31 0.059 Taste, smell, and touch response and use 0.255
Item 32 0.121 Fear or nervousness 0.208
Item 33 0.035 Verbal communication 0.236
Item 34 0.039 Nonverbal communication 0.235
Item 35 − 0.076 Activity level 0.081
Item 36 0.097 Level and consistency of intellectual response − 0.049
Item 37 0.074 General impressions 0.393*
Item 38 0.083 Total score 0.313
Item 39 − 0.050 Severity group 0.359*
Item 40 0.012 VABS-3
Item 41 0.109 COMM − 0.328*
Item 42 0.136 DLS − 0.280
Item 43 − 0.079 SOC − 0.008
Item 44 0.078 MOT − 0.336*
Item 45 0.075 ABC − 0.362*

N = 93. *p ≤ 0.001

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Journal of Autism and Developmental Disorders (2021) 51:4644–4654 4651

RRBI severity. As expected, the addition of Socialization functioning, lower scores on the COMM, MOT, and ABC
scores into the regression model led to a significant increase were significantly associated with higher level of diagnostic
in variance for RRBI severity (R2 = 0.40). The addition of severity level ratings; however, the DLS and SOC scores
Motor Skills scores to the model led to a significant increase were not.
in variance (ΔR2 = 0.005). Significant changes with the addi- Although there is limited research on factors associ-
tion of the Daily Living Skills scores (ΔR2 = 0.008) and ated with diagnostic severity level ratings, the present
Communication scores (ΔR2 = 0.026) were found. Overall, results from the series of correlations are consistent with
the full model, including all four adaptive subdomains, was what has been reported in the existing literature. Signifi-
found to significantly predict RRBI severity in young chil- cant relationships between clinician-assigned ASD severity
dren with ASD, with 44% of the variance in RRBI severity level ratings and motor-related categories (i.e., Body Use
explained by adaptive functioning skills. of CARS2, MOT of VABS-3) suggest that motor RRBIs and
In order to address the gap in the literature regarding motor delays are associated with ASD severity level ratings.
how DSM-5 ASD severity level ratings are determined, Indeed, retrospective studies have found that children who
parent- and clinician-rated measures of behavior were ana- were later diagnosed with ASD were reported to have early
lyzed to identify which variables predict and are associated motor delays during infancy and toddlerhood (Ozonoff et al.
with ASD severity level ratings. First, the three subscales of 2008a, b). Therefore, motor delays may be an early indicator
ASD symptoms according to the BISCUIT-Part 1 (i.e., S/ of risk for ASD and RRBIs (Bhat et al. 2012). Further, pre-
NVC, RRBI, Communication) did not significantly predict vious studies have found strong relationships between pres-
ASD severity level group membership (i.e., ASD-Level 1, ence of RRBIs and ASD severity level (Dworzynski et al.
ASD-Level 2, ASD-Level 3). This is not consistent with a 2009; Kuenssberg and McKenzie 2011; Matson et al. 2009a,
previous study that reported significant correlations between b, c). It may be that severity of motor RRBIs and delayed
parent-report ratings of RRBI severity and ASD severity motor functioning are significantly impairing and therefore,
level ratings (Mazurek et al. 2018). Given the young age may warrant more support.
of the current study’s participants (i.e., 19–37 months), it Social skills, as measured by the BISCUIT-Part 1,
may be that parents are not yet sensitive to and/or concerned CARS2, and VABS-3 were not significantly associated with
about the social and communication delays that are asso- ASD severity level ratings, which is consistent with Mazurek
ciated with ASD. Additionally, parents may not perceive et al. (2018)’s findings that there was no association between
restricted, repetitive behaviors as impairing or atypical. parent and clinician ratings of social communication and
Indeed, the existing literature indicates that parents typi- ASD severity rating. This finding is surprising given that
cally report symptoms that are not characteristic of ASD as impairment in socialization is a hallmark of the ASD pheno-
first concerns of their children’s development (Kozlowski type. This discrepancy may be explained by methodological
et al. 2011; Matheis et al. 2016). Nevertheless, parents and limitations. For instance, different studies may be using dif-
caregivers have been found to reliably identify ASD symp- ferent measures and constructs of socialization, which likely
toms in children as young as 12–18 months of age (Gray explains the mixed findings. Though no studies, to date,
and Tonge 2005; Reznick et al. 2007). Therefore, it may be have directly investigated the relationship between adap-
that clinicians are determining ASD severity level ratings tive functioning and ASD severity level ratings, the present
according to the severity of the core symptoms of ASD as findings support the idea that other areas of functioning are
well as other confounding factors. conflating diagnostic severity level ratings (Mazurek et al.
To evaluate other factors associated with diagnostic 2018). Indeed, clinicians may be assigning a more severe
severity level ratings, a series of Spearman’s correlations level of ASD to young children who have pervasive skill
were conducted. First, the individual BISCUIT-Part 1 items, deficits, as those children require more substantial support
subscale scores, and total score were examined. Of these and intervention.
variables, no statistically significant correlations were found. The current study has several limitations that should be
When clinician-rated measures of ASD symptoms were considered. First, two of the measures used in the study (i.e.,
evaluated, two significant positive correlations between the BISCUIT-Part 1, VABS-3) relied on parent report. Though
CARS2 subscale and ASD severity level were found: Body parents have been found to be reliable reporters of their chil-
Use (ρ = 0.341) and General Impressions (ρ = 0.393). The dren’s behavior (Gray and Tonge 2005; Reznick et al. 2007),
CARS2 Severity Group (ρ = 0.359) was also significantly there are several parental factors (e.g., cultural background,
positively correlated with diagnostic severity level ratings. level of education, stress level, coping skills, social support)
Of these significant relationships, the Body Use subscale that may influence parents’ perceptions of appropriate social,
can be categorized under the RRBI domain, and the General communication, and adaptive skills. Thus, researchers may
Impressions subscale and Severity Group can be categorized consider investigating factors related to RRBIs and diagnos-
as overall measures of ASD severity. Regarding adaptive tic severity level ratings while controlling for parent-specific

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4652 Journal of Autism and Developmental Disorders (2021) 51:4644–4654

factors. Second, RRBI severity was measured according to needs support and determine the level(s) of support for spe-
the BISCUIT-Part 1 RRBI subscale, which includes vari- cific skill domains. Additionally, RRBI and ASD symptom
ous topographies of RRBIs. A more fine-grained analysis severity should be studied across the lifespan to evaluate the
of evaluating the factors associated with specific topogra- developmental trajectory of RRBIs, changes in diagnostic
phies of RRBIs is warranted. Further, given that the sample ASD severity level ratings, differential responses to treat-
included children referred from a statewide early interven- ment, and treatment outcomes in young children and ado-
tion program, the participants included in the present study lescents with ASD.
were quite young (i.e., 19–37 months old). Previous studies
have found that the topography and severity of RRBIs vary
by age and developmental functioning (Prior and Macmillan Author contribution Esther Hong designed and executed the study,
analyzed the data, and wrote the manuscript. Johnny L. Matson assisted
1973; Turner 1999). Therefore, the topography and severity with the methods and data analysis and edited the final manuscript.
of RRBIs reported in this study may be limited to infants and
toddlers with ASD. Future research should investigate the Compliance with Ethical Standards
factors associated with RRBIs across the lifespan.
Though no assumptions for statistical analyses were vio- Conflict of interest Mrs. Deann Matson, Dr. Johnny L. Matson’s
lated, there was an unequal distribution of participants when spouse, is the sole owner of the Baby and Infant Screen for Children
grouped by ASD severity level, with 6 participants diag- with aUtIsm Traits-Part 1 and sells the scale. Esther Hong declares that
she has no conflict of interest.
nosed with ASD-Level 1, 24 participants diagnosed with
ASD-Level 2, and 47 participants diagnosed with ASD-
Level 3. Future studies should investigate factors associated
with ASD severity level ratings using more equal distribu- References
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