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Child Psychiatry & HumanDevelopment ISSN 0009-398X Child Psychiatry Hum DevDOI 10.1007/s10578-014-0496-9

Frequency and Clinical Correlates of Sleep-Related Problems Among Anxious Youthwith Autism Spectrum Disorders

Joshua M. Nadeau, Elysse B. Arnold,Amanda C. Keene, Amanda B. Collier,Adam B. Lewin, Tanya K. Murphy & EricA. Storch

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ORIGINAL ARTICLE

Frequency and Clinical Correlates of Sleep-Related ProblemsAmong Anxious Youth with Autism Spectrum Disorders

Joshua M. Nadeau • Elysse B. Arnold • Amanda C. Keene •

Amanda B. Collier • Adam B. Lewin • Tanya K. Murphy • Eric A. Storch

� Springer Science+Business Media New York 2014

Abstract Sleep-related problems (SRPs) are common

and problematic among anxious youth but have not been

investigated in anxious youth with autism spectrum disor-

der (ASD). Participants were 102 youth (ages 7–16 years)

with ASD and comorbid anxiety. Youth and their primary

caregiver were administered the Pediatric Anxiety Rating

Scale. Parents completed the Multidimensional Anxiety

Scale for Children–Parent (MASC-P) Report, Social

Responsiveness Scale, and the Child Behavior Checklist

(CBCL). A measure of SRPs was created from items from

the CBCL and MASC-P. Results suggest SRPs were rela-

tively common among youth with ASD and comorbid

anxiety. The number of SRPs endorsed directly associated

with parent ratings of social deficits, internalizing and

externalizing symptoms, and anxiety symptoms, as well as

with clinician-rated anxiety symptoms. Parent-rated inter-

nalizing symptoms predicted frequency of SRPs over and

above social deficits, externalizing symptoms, and parent-

and clinician-rated anxiety symptoms. A subset of 40 par-

ticipants who completed family-based cognitive–behavioral

therapy (CBT) experienced reduced SRPs following treat-

ment. Implications, study limitations, and recommendations

for future research are discussed.

Keywords Autism spectrum disorders � Anxiety � Sleep �Cognitive–behavioral therapy � Obsessive–compulsive

disorder

Introduction

Sleep related problems (SRPs) are relatively common

among typically developing adults [1] and children [2].

With respect to childhood presentation, SRPs can include

nightmares, difficulty falling asleep, difficulty sleeping

away from home, refusal to sleep alone, and general

nighttime fears [3, 4]. SRPs have been associated with

impairments in memory, vigilance, and affect [5, 6], and

may intensify difficulties with behavioral regulation [7].

Additionally, relatively minor amounts of sleep deprivation

result in significant performance decline within higher

cognitive functions, including verbal fluency and abstract

thinking [8]. As such, childhood SRPs have been associ-

ated with impaired functioning in academic, social, and

psychosocial domains [9], and may contribute to poor

outcomes with respect to emotional development [10–12].

Although little is known about SRPs among youth with

ASD and anxiety, several studies have been conducted

among typically developing anxious youth. Findings sug-

gest that youth with anxiety disorders show dispropor-

tionately large prevalence of SRPs, with more than 50 % of

youth endorsing three or more SRPs via parent report

checklists [3, 13]. These findings are similar to those

reported among youth with obsessive–compulsive disorder

(OCD), where more than one-quarter of the sample

J. M. Nadeau (&) � E. B. Arnold � A. C. Keene �A. B. Collier � A. B. Lewin � T. K. Murphy � E. A. Storch

Department of Pediatrics, University of South Florida, 880 6th

Street South, Box 7523, St. Petersburg, FL 33701, USA

e-mail: jnadeau@health.usf.edu

J. M. Nadeau � E. A. Storch

Rogers Behavioral Health – Tampa Bay, Tampa, FL, USA

E. B. Arnold � A. B. Lewin � E. A. Storch

Department of Psychology, University of South Florida,

St. Petersburg, FL, USA

A. B. Lewin � T. K. Murphy � E. A. Storch

Department of Psychiatry and Behavioral Neurosciences,

University of South Florida, St. Petersburg, FL, USA

123

Child Psychiatry Hum Dev

DOI 10.1007/s10578-014-0496-9

Author's personal copy

endorsed five or more of eight assessed SRPs [14]. Inter-

estingly, although age and gender have not significantly

correlated with SRP endorsement, the number of SRPs

endorsed was positively associated with severity of child-

rated anxiety symptoms [3, 13, 14]. Additionally, evidence

suggests that the increase in frequency of SRPs endorsed

was associated with the specific anxiety disorder diag-

nosed; youth with social phobia and separation anxiety

disorder demonstrated higher rates of SRPs than youth with

other anxiety disorders, although SRP endorsement rates

were higher among youth with any anxiety disorder as

compared to youth without these diagnoses [13]. Of par-

ticular relevance to the current study, treatment of anxiety

symptoms resulted in concomitant reduction of endorsed

SRPs, whether treatment was pharmacological (i.e., flu-

voxamine; [3]) or psychological (i.e., cognitive–behavioral

therapy (CBT); [14]).

Recent research has investigated the nature of SRPs

among youth with psychiatric disorders, with results gen-

erally indicating that SRPs occur at increased rates among

youth with emotional and behavioral problems [14–16].

Disordered sleep has been associated with cognitive per-

formance and behavioral problems in multiple pediatric

populations, including youth with autism spectrum disorder

(ASD; see [17] for a review). The various cognitive, social,

communicative, and behavioral impairments that com-

monly occur among youth diagnosed with ASD suggest

that youth with ASD should endorse increased rates of

SRPs as compared to typically developing youth [18–20].

Findings among youth with ASD consistently suggest

increased rates of SRPs (44–83 %; [21]) as compared to

same-age typically developing peers (32 %; [22]), includ-

ing increased sleep latency, decreased duration of sleep,

and frequent nighttime wakings [8, 21]. Youth with ASD

classified as ‘‘poor sleepers’’ are rated as having more

affective problems on the Child Behavior Checklist

(CBCL), as well as more problems with reciprocal social

interaction on the Autism Diagnostic Observation Schedule

[23], as compared to youth with ASD classified as ‘‘good

sleepers’’ [8]. Increased endorsement of sleep problems, as

well as significantly greater response to medication for

sleep problems, has been reported among youth with aut-

ism and Asperger’s Disorder as compared to typically

developing youth [24]. Tudor et al. [25] found that sleep

latency among youth with ASD was a strong predictor of

communication deficits, stereotyped behaviors, and sever-

ity of core ASD symptoms. Neither cognitive nor adaptive

development was a significant predictor of SRP frequency

or severity among children with autism [22]. Within this

literature, information about the frequency of SRPs among

youth with ASD has been relatively well-represented rel-

ative to typically developing youth; however, no research

exists examining relationships of SRPs with severity of

core ASD symptoms and comorbid anxiety symptoms,

despite the high comorbidity rate of anxiety among youth

with ASD [26–28].

It is clear from existing data that youth with ASD fre-

quently experience SRPs. Similarly, existing data support

elevated rates of SRPs among typically developing youth

with anxiety disorders. However, the magnitude and nature

of relationship between ASD symptoms and comorbid

anxiety symptoms with respect to SRPs remains unclear.

The present study examines the prevalence and correlates

of SRPs in a large sample of children and adolescents with

ASD and comorbid anxiety, and was designed to answer

five research questions. First, what is the frequency of 8

SRPs (i.e., being overtired, sleeping less than other youth,

sleeping more than other youth, talking or walking while

asleep, having trouble sleeping, needing a nightlight to

sleep, co-sleeping, and having nightmares) in youth with

ASD and comorbid anxiety? We expected to find that SRPs

occurred frequently (i.e., more than 60 % of sample)

among anxious youth with ASD. Second, are SRPs related

to age and gender? We hypothesized that younger children

(ages 7–11) and females would endorse more SRPs than

older children (ages 12–16) and males, consistent with

prior findings [3]. Third, what are the associations between

SRPs, ASD symptoms, and anxiety symptoms? We

expected that SRPs would be positively related to ASD

symptoms and anxiety symptom severity. Fourth, do anx-

iety and/or internalizing symptoms predict SRPs above and

beyond ASD symptoms? We hypothesized that anxiety and

internalizing symptoms would significantly predict SRP

endorsement while controlling for ASD symptom severity.

Finally, what effect does CBT modified for youth with

ASD and anxiety have upon SRPs? Given the effectiveness

of CBT for anxiety in youth with ASD [20] and past

findings in typically developing youth with anxiety [29,

30], we hypothesized that a reduction in endorsed SRPs

would be found from pre- to post-treatment assessment.

Method

Participants

Participants were 102 children and adolescents with ASD

and comorbid anxiety disorders (81 males) recruited from

4 completed studies. The first study [31] examined the

efficacy of a modularized CBT protocol, Behavioral

Interventions for Anxiety in Children with Autism [32]

and contributed 44 participants (ranging in age from 7 to

11 years) to the current study. This study examined the

efficacy of 16 sessions of a modular treatment protocol

(BIACA) relative to a treatment as usual (TAU) control

condition. Although the associated publication for the

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study [31] explains the design and participant information

in great detail, use of a modular treatment approach

allowed inclusion of anxiety and non-anxiety symptoms

as treatment goals, as well as the opportunity to address

treatment issues commonly identified among youth with

ASD (i.e., comorbid conditions, low motivation). The

second study was an open trial evaluating a computer-

assisted CBT program, Camp Cope-A-Lot (CCAL; [33])

and contributed five participants (ranging in age from 7 to

11 years) to the current study. The CCAL protocol con-

sisted of six computer-guided and six therapist-guided

sessions over 12 weeks. The computer-guided sessions

focus upon helping youth with anxiety to recognize their

emotions, relax, and modify anxiety-related cognitions.

The therapist-guided sessions are designed to promote

application of learned skills through exposure tasks to

help the youth better manage thoughts, feelings and

behaviors when anxious. The third study [34] examined

use of the BIACA protocol among adolescents with high-

functioning ASD and comorbid anxiety ranging in age

from 11 to 15 years, and contributed 28 participants to the

current study. The fourth study, the manuscript for which

has been submitted for publication consideration, also

examined the use of the BIACA protocol among adoles-

cents, and contributed 25 participants (ranging in age

from 11 to 16 years) to the current study. In the last two

studies (those examining application of the BIACA pro-

tocol among adolescents), the study designs were essen-

tially replications of the first study [31] among adolescent

populations.

Participant descriptive data are provided in Table 1.

Data from the baseline assessment of each study were used

to examine cross-sectional associations among study vari-

ables for the sample as a whole, while post-treatment data

were also used to examine changes in SRP rates for a

subset of this sample (N = 40) completing the associated

course of CBT (i.e., 12 sessions of CCAL or 16 sessions of

BIACA). In any event, participants met inclusion criteria

for the presence of ASD and anxiety based on the Autism

Diagnostic Interview-Revised [35], Autism Diagnostic

Observation Schedule Module 3 [23], and the Anxiety

Disorders Interview Schedule for DSM-IV Parent and

Child versions [36], respectively. At the time of study

entry, participants ranged in age from 7 to 16 years

(M = 10.81 ± 2.3 years). The majority of youth were

Caucasian (87 %), with 7 % reporting as Hispanic, 3 % as

Asian, and 3 % as mixed. Of the sample, 59.1 % of youth

were taking one or more psychiatric medications, with

serotonin reuptake inhibitors (35.4 %), stimulants (32.3 %)

and anti-psychotics (8.2 %) representing the top three

classes of medication. Medication status was not available

for one youth.

Procedure

Study procedures were approved by the local Institutional

Review Board. In the case of each parent study, written

parental consent and child assent were obtained. After

obtaining consent/assent, families completed a 2 day in-

person screening with the 2 days occurring no more than

7 days apart. Anxiety-specific diagnostic assessments were

administered on the first visit, and autism-focused diag-

nostic assessments occurred at the second visit. Those

youth who qualified returned to complete a baseline

assessment, including all clinician- and parent-report

measures. For those participants completing CBT, post-

treatment assessments were completed within 1 week after

the final CBT session.

Measures

Establishment of ASD diagnosis and diagnostic differen-

tiation was accomplished via the use of combined diag-

nostic information from the ADI-R and ADOS-Module 3,

which have demonstrated good diagnostic utility when

used in combination [37]. Administration of the ADI-R and

ADOS were completed by a certified and experienced

doctoral-level evaluator. Independent evaluators, who were

Table 1 Characteristics of study participants

Age (years)

Range 7–16 years

Mean (SD) 10.81

(2.31)

Gender [n (%)]

Male 81 (79.4)

Female 21 (20.6)

Ethnicity [n (%)]

White/Caucasian 89 (87.3)

Hispanic 7 (6.9)

Asian 3 (2.9)

Multi-racial 3 (2.9)

Autism spectrum disorder [n (%)]

Asperger’s disorder (AS) 42 (41.2)

Autistic disorder (AD) 30 (29.4)

Pervasive developmental disorder, not otherwise

specified (PDD-NOS)

30 (29.4)

Primary anxiety disorder [n (%)]

Social phobia (SoP) 42 (35.6)

Generalized anxiety disorder (GAD) 31 (26.3)

Obsessive–compulsive disorder (OCD) 13 (11.0)

Separation anxiety disorder (SAD) 12 (10.2)

Specific phobia (SP) 2 (1.7)

Anxiety disorder, not otherwise specified (ANX-NOS) 10 (8.5)

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blind to participants’ intervention condition and not

involved in treatment, administered all clinician-rated

measures. Prior to the onset of all studies, evaluators

completed instructional training, observed multiple

administrations of measures, and completed measure

administrations under supervision.

Pediatric Anxiety Rating Scale (PARS)

The PARS [38] is a clinician-rated measure of the presence

and severity of anxiety symptoms in youth. Total scores

represent the severity and frequency of anxiety symptoms

as well as associated distress, avoidance, and interference

during the previous week. For the current study, severity of

physical symptoms of anxiety was assessed using an item

from the PARS as rated by the parent, youth, and clinician.

The PARS has acceptable reliability and validity [39] and

has been shown to be acceptable for use among youth with

ASD [40]. Internal consistency ratings for the PARS in the

sample were .53 at baseline, and .80 at post-treatment.

Multidimensional Anxiety Scale for Children–Parent

Version (MASC-P)

The MASC-P [41] is a 39-item parent-report measure of

anxiety symptoms. Items are rated on a four-point Likert

scale (0: never true about my child, 1: rarely true about my

child, 2: sometimes true about my child, 3: often true about

my child) with higher scores corresponding to greater

anxiety. The MASC-P has demonstrated strong psycho-

metric properties [42]. Internal consistency for the MASC-

P in this sample was .87.

Social Responsiveness Scale (SRS)

The SRS [43] is a 65-item scale measuring severity of

autism spectrum symptoms (social awareness, autistic

preoccupations) with excellent psychometrics [44]. Internal

consistency for the SRS in this sample was .81.

Sleep Composite Measure

Similar to previous studies [3, 13, 14, 45], we formed a

composite measure of SRPs by combining six relevant

items from the CBCL (i.e., sleep walks, has nightmares,

overtired, sleeps less than most children, sleeps more than

most children, and trouble sleeping) and two items from

the MASC-P (i.e., keeps the light on at night, sleeps next to

someone from my family). In contrast to utilizing child-

reported anxiety via the MASC [42], the current study used

parent proxy report via the MASC-P. Given that the items

from the MASC-P differ in response format form those of

the CBCL, responses were recoded. However, to provide a

more conservative estimate of sleep problem severity than

in prior studies, response recoding was such that more

positive endorsement of a SRP (i.e., rating of ‘‘2’’ on the

CBCL, rating of ‘‘2’’ or ‘‘3’’ on the MASC-P) was recoded

as ‘‘1,’’ while ‘‘1’’ ratings on both instruments were reco-

ded as ‘‘0.’’ Note that ‘‘0’’ ratings were not recoded. These

eight items were standardized and then summed to create a

composite SRP score.

Anxiety Disorders Interview Schedule for DSM-IV-Child

and Parent Versions (ADIS-C/P)

The ADIS-C/P [36] are clinician-administered, semi-

structured interviews that assess parent and child

endorsement of DSM-IV-TR Axis I disorders. Diagnostic

presence and severity are established using a CSR on a

scale of 0–8, with a score of 4 or more indicating diag-

nostic presence. The ADIS-C/P demonstrates strong reli-

ability and validity among typically developing youth [40]

and treatment sensitivity among youth with ASD [31, 46].

Child Behavior Checklist (CBCL)

The CBCL [47] is a 113-item parent report form used to

assess a wide range of child internalizing and externalizing

symptoms over the past 6 months. Items are rated on a

three-point scale (0: never true, 1: sometimes true, 2: often

or always true). The CBCL has eight individual subscales,

Internalizing and Externalizing Scales, and a Total Score

that is derived by summing the Internalizing and Exter-

nalizing Scales. Numerous studies have supported the

psychometric properties of the updated version of the

CBCL [48, 49]. Internal consistency ratings for the CBCL

in this sample were .82 for the internalizing subscale, and

.92 for the externalizing subscale.

Data Analysis

Missing data were determined to be at random using pro-

cedures provided by Schlomer et al. [50]. As listwise

deletion occurred in variables of interest in less than 5 % of

response opportunities [51], hotdeck multiple imputation

was completed, ensuring that none of the missing data

constituted sleep composite items. Data were analyzed

using SPSS 21.0. Descriptive data and zero-order correla-

tion coefficients were calculated to determine the preva-

lence of SRPs in the current sample, as well as to identify

the relationships between SRPs and other study variables of

interest (e.g., PARS scores, CBCL Internalizing and

Externalizing subscale scores, MASC-P Total Score, SRS

scores). Sleep items were removed from all CBCL scales

and MASC-P scores before calculating internalizing,

externalizing, and anxiety scores to remove overlapping

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items from analyses. To examine possible differences in

overall and individual SRPs due to age and gender, paired

sample t tests and Chi square tests were used. The rela-

tionships between SRPs, SRS scores, parent-rated inter-

nalizing and externalizing symptoms, and parent- and

clinician-rated anxiety symptoms were examined via

backward regression. The dependent variable for the

regression model was overall SRPs. Finally, to examine

treatment effects, data representing a subset of participants

who completed CBT were used. From these data, t tests

were used to test for significant changes in total number of

SRPs endorsed, as well as in individual SRPs endorsed,

from pre- to post-treatment. Given the exploratory nature of

this study, no statistical correction was used to minimize the

chance of obfuscating potentially important relationships.

Results

Associations Between Clinical Characteristics

and SRPs

Table 2 presents descriptive statistics for and correlations

among administered measures. As shown, the total number

of SRPs showed strong positive relationships with parent-

reported ASD-related social deficits, parent-rated internal-

izing symptoms, and child- and clinician-rated anxiety

symptoms. A moderate positive association was observed

between total number of SRPs and parent-rated external-

izing symptoms. Endorsement rates for overall number of

SRPs, as well as for each of the eight SRPs examined, are

presented in Table 4. Less than one-quarter (24.8 %) of the

current sample did not have a SRP, with more than

one-third (35.6 %) of the sample reporting three or more

SRPs. The most commonly reported problems included

needing a night light (54.5 %), and feeling unable to sleep

alone (43.6 %).

Age and Gender Effects

Gender

The difference between the mean number of SRPs for

males and females was not statistically significant

[t(101) = 1.54, p [ .05]. Analysis of individual SRPs

revealed that nightmares [t(101) = 2.60, p = .011]

occurred with greater frequency in female than male

participants.

Age

The difference for the mean number of SRPs between age

groups was not statistically significant [t(101) = .65,

p [ .05]. On individual types of SRP, sleeping less

[t(101) = 2.22, p = .028] and co-sleeping [t(101) = 2.34,

p = .021] were found with greater frequency in younger

versus older youth.

Predictive Utility of ASD and Comorbid Anxiety

Symptoms

Results of the backward regression model are summarized

in Table 3. In step one, the overall model was a significant

predictor of SRP endorsement, accounting for 51 % of the

observed variance (p \ .01); however, investigation of

individual contributions indicated no significant predictors

within the model. Removal of parent-rated externalizing

symptoms in step 2 maintained significant prediction of

Table 2 Correlations, internal consistency, and descriptive statistics for study variables

Variable SRPs ASD symptoms Parent-rated anxiety Parent-rated Clinician-rated anxiety

Internalizing Externalizing

Total SRPs – .456** .446** .590** .271** .453**

Overtired .561** .270* .262* .499** .072 .414**

Sleep less .499** .397** .266* .323** .247* .252*

Sleep more .279** .229 .128 .413** .090 .234*

Sleep walk .404** .207 .050 .285** .140 .206

Sleep trouble .703** .277* .318** .298** .102 .470**

Nightlight .614** .206 .320** .248* .134 .111

Co-sleep .548** -.030 .241* .261** .261** .205

Nightmares .610** .429** .254* .319** .118 .240*

Mean (SD) 2.01 (1.74) 166.78 (18.00) 59.99 (13.66) 22.11 (9.44) 15.93 (11.12) 23.68 (3.41)

Reliability (a) .57 .81 .87 .83 .92 –

* p \ .05; ** p \ .01; *** p \ .001

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SRP endorsement by the model overall (51 %, p \ .01),

although none of the individual variables significantly

predicted variance in SRP endorsement. Clinician-rated

anxiety symptoms were removed in step 3 of the regres-

sion, yielding a non-significant reduction in overall model

predictive utility (50 %, p \ .01); however, no individual

significant predictors of SRP endorsement were revealed.

Step 4 of the regression involved removal of parent-rated

anxiety symptoms. Note that, although overall model pre-

dictive utility exhibited a non-significant reduction (49 %,

p \ .001), parent-rated internalizing symptoms were a

significant individual predictor of SRP endorsement

(p \ .01). Finally, social deficits were removed in step 5 of

the regression, with the result that the parent-rated inter-

nalizing symptoms exhibited significant predictive utility

(46 %, p \ .001) of SRP endorsement.

Treatment Effects

A subsample of 40 youth who completed a full course of

family-based CBT (16 sessions for the 3 studies utilizing

the BIACA protocol; 12 sessions for the CCAL protocol)

was examined. Endorsement rates of SRPs for all partici-

pants at baseline and post-treatment are provided in

Table 4. The youth completing CBT displayed a statisti-

cally significant reduction in overall number of reported

SRPs from baseline to post-treatment [t(39) = 2.30,

p = .027, d = .74]. In terms of specific symptom reduc-

tions, nightmares [t(39) = 2.36, p = .023], overtired

[t(39) = 2.78, p = .008], and sleeps more [t(39) = 2.22,

p = .032] were all significantly reduced in the current

sample (see Table 4). The remaining SRPs, ‘‘talks or walks

in sleep’’, ‘‘sleeps less than other kids’’, ‘‘has trouble

sleeping’’, ‘‘sleeps next to someone in family’’, and ‘‘needs

nightlight to sleep’’ showed no significant reductions in

endorsement rate from baseline to post-treatment among

the treatment-completing subset.

Discussion

This study investigated the incidence and correlates of

SRPs among youth with ASD and comorbid anxiety dis-

orders. While SRPs have been examined separately in

youth with ASD and in typically developing youth with

anxiety, few data have been reported on SRPs in youth with

ASD and comorbid anxiety. Consistent with studies of

anxious typically developing youth [3, 14] and youth with

ASD [8, 21], 75 % of participants in this study endorsed at

least 1 SRP, with 35.6 % of participants endorsing 3 or

more SRPs. Parents most frequently reported problems

with needing a night light and co-sleeping, which is con-

sistent with findings that youth with autism demonstrate

higher frequency of endorsed co-sleeping versus typically

Table 3 Backward regression model for overall sleep-related

problems

Variables R2 R2 change F B (std. error) b

First step .51 – 4.35**

SRS .020 (.02) .20

CBCLext -.009 (.3) -.06

CBCLint .087 (.05) .43

PARS .055 (.11) .10

MASC .015 (.03) .12

Second step .51 .00 5.64**

SRS .019 (.02) .18

CBCLint .085 (.05) .42

PARS .046 (.10) .08

MASC .017 (.03) .13

Third step .50 -.01 7.72**

SRS .020 (.02) .20

CBCLint .088 (.05) .43

MASC .019 (.03) .15

Fourth step .49 -.01 11.60***

SRS .025 (.02) .24

CBCLint .103 (.04) .51*

Fifth step .46 -.03 21.61***

CBCLint .138 (.03) .68***

* p \ .05; ** p \ .01; *** p \ .001

Table 4 Mean scores and percentages for sleep-related problems for

entire sample, and for treatment-completing subset before and after

cognitive–behavioral therapy

Sleep related

problem

Overall

sample (%)

Treatment-completing

subset

Pre-

treatment

(%)

Post-

treatment

(%)

Nightmares 17.8 34.8 12.8*

Overtired 20.8 60.0 17.9**

Sleeps less than other

kids

15.8 15.4 15.4

Sleeps more than other

kids

8.9 13.0 5.1*

Talks or walks in sleep 6.9 8.7 7.7

Has trouble sleeping 32.7 29.2 23.1

Sleeps next to

someone in family

43.6 47.8 38.5

Needs nightlight to

sleep

54.5 48.7 40.0

Total mean SRPs (SD) 2.01 (1.74) 2.75 (1.81) 1.69* (1.59)

* p \ .05; ** p \ .01; *** p \ .001

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developing youth and youth with other disabilities (e.g.,

Prader–Willi Syndrome and Down Syndrome; [52]).

Consistent with findings among typically developing

anxious youth [3, 14, 15], parent-reported internalizing

symptoms, and parent- and clinician rated anxiety shared

significant positive associations with total SRPs. Parent-

rated internalizing symptoms directly predicted SRPs,

consistent with previous research [17]. The association

between SRPs and symptoms of ASD and anxiety may

suggest a complex relationship. Symptoms related to anx-

iety as well as ASD may affect sleep, which in turn reduces

coping, tolerance and behavioral and affective regulation,

which further exacerbates ASD and anxiety presentations

[15, 16, 53]. These findings indicate that symptoms asso-

ciated with anxiety and ASD may impact children’s sleep

in multiple ways. First, anxiety symptoms and ASD

behavioral characteristics may significantly impact the

bedtime routine. For example, if anxiety is centered around

bedtime or its characteristics (i.e., fear of the dark, of

separating from parents, or of experiencing nightmares), or

if behavioral characteristics commonly observed among

youth with ASD are present (i.e., rigidity with respect to

rituals and/or schedules, difficulties with transitions, diffi-

culties identifying social cues related to bedtime), then

youth with anxiety and ASD may seek and receive

increased support and attention from their parents at bed-

time. This reliance on parents, in addition to significantly

extending an anxiogenic routine (going to sleep), may also

hinder self-regulation in mood and sleep. Second, the

physiological symptoms of anxiety (e.g., heart palpitations,

stomachaches, sweating, recurrent use of the bathroom), as

well as evidence suggesting biological abnormalities

associated with ASD (i.e., melatonin synthesis and circa-

dian rhythm; [53]) could contribute to or exacerbate com-

mon difficulties with achieving and maintaining sleep (i.e.,

insomnia, delayed sleep onset, and irregular sleep patterns).

Neither gender nor age was associated with SRPs. This

stands in contrast to Alfano et al. [15], where increased

SRPs were endorsed among adolescents as compared to

children in a sample of typically developing youth. The

high rate of SRPs in this sample suggests that any demo-

graphic-related effects may have been outweighed by

characteristics of diagnostic presentation; specifically,

ASD and comorbid anxiety, both of which have been

consistently associated with elevated rates of impaired

sleep [3, 17, 21, 53].

Sleep related problems were reduced following CBT,

which has been shown in youth with ASD [24] and without

[3]. A relatively straightforward explanation of this may lie

in the hypothesized relationships between ASD and anxiety

symptoms, and SRPs. Beyond the findings that modular-

ized CBT adapted to meet the needs of youth with ASD

and anxiety has been efficacious in reducing symptoms of

anxiety [20], treatment response has also been associated

with reductions in ASD-related social and behavioral

symptom severity and related impairment [31, 54]. It is

possible that reduction of anxiety and/or ASD symptoms,

which are hypothesized to act as maintaining variables for

SRPs, would reduce characteristics generating or exacer-

bating sleep impairments. Beyond attempts at replication

of current findings, future research among this population

should investigate whether CBT-related improvement in

sleep quality subsequently manifests as decreased social

and behavioral issues.

The results of the current study should be considered in

light of several limitations. First, these analyses were post

hoc and subject to multiple hypothesis testing. Second,

internal consistency for the PARS and SRPs were relatively

low, which could impact the conclusions. Although psy-

chometrically sound, the PARS has demonstrated fair

internal consistency in typically developing youth and in

those with ASD [38, 40], which has been suggested to

relate to the associated but not redundant item content.

Third, use of an open trial prevented the incorporation of a

control group with which to compare outcome data.

Finally, the limited number of female participants restricts

the generalizability of the results to all youth with ASD.

Despite these limitations, this study is the first to

examine SRPs in anxious youth with ASD. These findings

indicate that SRPs frequently occur in this population.

Furthermore, ASD symptoms and anxiety uniquely con-

tributed to the presence and frequency of SRPs, suggesting

that anxiety compounds SRPs over and above what may be

expected in youth with ASD. SRPs were reduced following

CBT. Collectively, these data emphasize the importance of

screening for SRPs and evaluating their impact upon

comorbid symptoms in youth with ASD, especially those

who are anxious. Considering the efficacy of CBT with

respect to SRP endorsement in the current study, future

research should examine the possible adaptation of com-

mon sleep interventions for anxious youth with ASD,

whether through behavioral interventions (e.g., increased

sleep hygiene, faded bedtime with response cost), phar-

macotherapy, or a combination of modalities. Particularly

given the evidence of reciprocal relationships SRPs share

with anxiety symptoms and ASD symptoms, there is a

compelling rationale to determine whether easily imple-

mented and effective sleep interventions have the potential

to reduce or minimize impairment associated with anxiety

and ASD symptoms in youth.

Summary

This study reports initial data on the frequency and clinical

correlates of sleep-related problems among 102 anxious

Child Psychiatry Hum Dev

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youth with ASD. Frequency of SRPs endorsed was ele-

vated as compared to previous findings among neurotypical

anxious youth and youth with ASD alone. Strong direct

associations were observed between frequency of SRPs

endorsed and parent-rated internalizing and externalizing

symptoms, as well as with parent- and clinician-rated

anxiety symptoms. Further, parent-rated internalizing

symptoms predicted SRP frequency above and beyond

social deficits commonly associated with ASD, parent-

rated externalizing symptoms, and parent- and clinician-

rated anxiety symptoms. Finally, the subset of participants

completing family-based CBT displayed a significant

reduction in frequency of SRPs following treatment.

Although study limitations should be considered, these data

provide information regarding the frequency and clinical

correlates of SRPs among anxious youth with ASD.

Acknowledgments This work was supported in part by grants from

the All Children’s Hospital Research Foundation, the University of

South Florida Internal Grants Program, and the National Institutes of

Child Health and Human Development to Dr. Storch

(5R34HD065274-02). The views of this manuscript do not necessarily

reflect those of the National Institutes of Health.

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