Individual characteristics of students with autism spectrum disorders and school refusal behavior

Abstract

This study compared social, executive, emotional, and behavioral characteristics of students with autism spectrum disorder who did and did not display school refusal behavior. The participants were 62 students with autism spectrum disorder without intellectual disability aged 9–16 years attending inclusive schools. Parents first completed questionnaires assessing social and executive functioning as well as emotional and behavioral problems. They then documented their child’s school refusal behavior for a period of 20 days. Compared to students without school refusal behavior (n = 29), students with school refusal behavior (n = 33) were significantly less socially motivated; displayed more deficits in initiating tasks or activities, in generating ideas, responses, or problem-solving strategies; and displayed more withdrawn and depressive symptoms. Assessing social and executive functioning, as well as emotional problems, may help professionals provide tailored interventions for students with autism spectrum disorder and school refusal behavior, which will further be valuable in recognizing characteristics associated with school refusal behavior.

Keywords

autism spectrum disorder individual characteristics school refusal behavior

Introduction

Autism spectrum disorder (ASD) is defined as a lifelong set of pervasive neurodevelopmental disorders with an onset in childhood. Individuals with ASD are a heterogeneous group, sharing the core symptoms of deficits in social communication, social interaction, and restrictive and repetitive patterns of behavior, activities, or interests (American Psychiatric Association, 2013; Lai et al., 2014). Students with ASD, especially those with intellectual abilities within the normal range, often go to inclusive schools in Norway. Inclusive school settings are regarded as beneficial because they provide greater access to peer role models, relationships with peers, and access to the general curriculum (Dillon et al., 2016; Osborne and Reed, 2011). Despite these benefits, school refusal behavior (SRB) in students with ASD is reported to be common (Kurita, 1991; Munkhaugen et al., 2017).

The purpose of this study was to explore individual characteristics associated with SRB in students with ASD by comparing social and executive functioning and emotional and behavioral problems in students with and without SRB.

SRB

SRB is defined as child-motivated refusal to attend school and/or difficulties remaining in class for an entire day (Kearney and Albano, 2004; Kearney and Silverman, 1996). Specifically, SRB refers to students who (1) are completely absent from school or classes during the school day, (2) plead to not attend school and/or classes during the school day, (3) display physical refusal and tardiness at home to avoid school, and/or (4) display elevated distress during school, leading to pleads for future nonattendance (Kearney and Albano, 2004). This broad definition of SRB that includes both the refusal and the absenteeism as part of the behavior has been influential in the field in recent years (Ingles et al., 2015; Kearney and Silverman, 1996).

Although research studies are scarce, there are indications that SRB is a considerable problem in students with ASD and is far more common in students with ASD than in the general population, with rates reported at approximately 40%–53% in students with ASD compared to 5%–28% in the general child and adolescent population (Havik et al., 2015; Kearney, 2006; Kurita, 1991; Munkhaugen et al., 2017).

SRB is considered a heterogeneous, complex condition caused and maintained by multiple factors that may impact each other. Several individual, familial, and environmental characteristics are found to be associated with SRB in the general child and adolescent population (Heyne et al., 2001; Kearney, 2008; Kearney and Bensaheb, 2006). Individual characteristics include anxiety disorders, depression disorders, oppositional defiant disorder, somatic complaints, emotional instability, and poor relationships with peers. Familial factors associated with SRB are reported to be lack of parental support and involvement in homework and school, conflicts at home, unemployment, and poor health in the family. Factors within the school environment include being bullied or teased, teachers’ classroom management and support, and transition and change of classes (Egger et al., 2003; Havik et al., 2015; Ingles et al., 2015; Ingul et al., 2012; Ingul and Nordahl, 2013; Kearney, 2008).

There are few studies of characteristics associated with SRB in students with ASD. However, similar factors such as obsessive behavior, somatic complaints, poor family health, harsh discipline by teachers, teasing, change of classes, and curriculum were associated with SRB in students with ASD (Kurita, 1991; Munkhaugen et al., 2017). Furthermore, negative attitudes toward different subjects, irregular schooldays, and problems in the morning that led to being late for school were reported as factors associated with SRB in students with ASD (Munkhaugen et al., 2017).

Social functioning and emotional and behavioral problems are commonly examined in studies of SRB in the general child and adolescent population and thus of interest to explore in students with ASD (Egger et al., 2003; Havik et al., 2015; Ingles et al., 2015; Ingul et al., 2012; Kearney, 2008). Executive functioning is less studied in the SRB context but is of interest because deficits in these areas are often associated with adaptive behavior problems and with academic outcomes in students with ASD (Carretti et al., 2014; Gilotty et al., 2002; Hill, 2004). Problems within these four factors are reported as frequent and pervasive in students with ASD. However, whether these problems are related to SRB has not been studied (Bauminger and Kasari, 2000; Hill, 2004; Simonoff et al., 2008). Furthermore, whether SRB in ASD is associated with social/emotional problems or with cognitive deficits may have implications for intervention strategies.

Social functioning and SRB

Poor relationships with peers and difficulties making friends because of shyness, aggression, or withdrawal are individual characteristics reported in students with SRB (Baker and Bishop, 2015; Egger et al., 2003; Gren-Landell et al., 2015; McShane et al., 2001; Thambirajah et al., 2008). We have not identified studies reporting on characteristics within the social domain associated with SRB in students with ASD. However, studies of engagement in social interactions show that students with ASD initiate interactions with other children to a lesser extent than their peers (Kasari et al., 2011; Locke et al., 2016). Although social deficits are core symptoms in ASD, diversity within the social domain has been reported (Howlin et al., 2004; Orsmond et al., 2004; Scheeren et al., 2012).

Executive functioning and SRB

Executive functions comprise several neurocognitive components including inhibition, working memory, flexibility, emotional control, initiation, planning, organization, and self-control. These components enable the individual to disengage from the present context to effectuate future goals (Hill, 2004; Miyake et al., 2000). Studies of SRB and executive functions are scarce. The only study found reported that nearly all 14 inpatients with prolonged SRB attending a cognitive behavioral group therapy program displayed impaired executive functions (Ohmann et al., 2007). Studies of executive functions in students with ASD and SRB have not been found. However, it is consistently reported that children with ASD have executive deficits (Geurts et al., 2004; Hill, 2004; Pellicano, 2010). Furthermore, it has been shown that executive deficits are associated with adaptive behavior problems assessed in everyday settings (Gilotty et al., 2002; Pugliese et al., 2016; Rosenthal et al., 2013; Semrud-Clikeman et al., 2016).

Emotional and behavioral problems and SRB

Studies of SRB in the general child and adolescent population have reported high rates of emotional and behavioral problems (Egger et al., 2003; Kearney and Albano, 2004). The occurrence of emotional and behavioral problems in students with ASD and SRB has not been studied. However, Kurita (1991) reported that students with ASD and SRB displayed a tendency toward more obsessive behavior. Nevertheless, comorbid psychiatric conditions have been reported to be highly prevalent in students with ASD, mainly anxiety disorders, attention deficit hyperactivity disorder (ADHD), oppositional defiant disorders, and depressive disorders (Gjevik et al., 2011; Mattila et al., 2010; Simonoff et al., 2008).

Regular school attendance is important for students with ASD to benefit from academic and social advantages provided in the inclusive school. Almost half of the students with ASD and SRB displayed partial or complete absenteeism (Munkhaugen et al., 2017). Consequences of staying out of school are, in the general child and adolescence population, reported to negatively impact the students’ academic and social growth and increase the risk for chronic absenteeism and later school dropout (Balfanz and Byrnes, 2012; Fremont, 2003; Havik et al., 2015; Kearney, 2008; Sanchez, 2012; Thambirajah et al., 2008). Consequences of SRB have not been studied in students with ASD, but it may be anticipated that this behavior may have similar negative effects. The high rates of SRB in students with ASD, as well as the duration of the behavior indicated by a previous study, require a better understanding of individual characteristics associated with SRB in these students (Munkhaugen et al., 2017). For preventive purposes, knowledge about individual characteristics associated with SRB in students with ASD may be relevant for the early identification of SRB. Such knowledge may be valuable for supervision by parents and teachers. For the parents, this supervision may include proper ways to organize the morning routine and how to encourage their children to attend school. For the teachers, it may include how to make the school environment pleasant for the students. Furthermore, when SRB is present, knowledge of individual characteristics might help the clinicians tailor interventions to the student.

The main aim of this study was to explore social and executive functioning and emotional and behavioral problems associated with SRB in students with ASD without intellectual disability aged 9–16 years in inclusive schools. Measures of the four factors were compared between students with ASD and SRB and students with ASD without SRB. The research question for this study was as follows: Are executive deficits, social impairments, and emotional and behavioral problems associated with SRB in students with ASD without intellectual disability aged 9–16 years?

Methods

Study participants

The study was introduced through written material to primary and secondary inclusive schools, to child and adolescent psychiatric and pediatric outpatient clinics in the southeast region of Norway, and through advertising on the Norwegian Autism Association website.

Teachers and clinicians were asked to distribute written information and a consent form to the parents of students with ASD. Students with ASD were eligible to participate after their parents provided consent. The participants, aged 9–16 years, were attending 4th–10th grades in 59 inclusive primary and secondary schools, both in rural and urban areas. In all, 28 of the students were recruited from schools, 24 from child and adolescent psychiatric and pediatric outpatient clinics, and 10 students were recruited through advertising on the Norwegian Autism Association website.

The 62 children and adolescents represented a subsample of 78 participants in a previous study assessing the rates of SRB in children with ASD (Munkhaugen et al., 2017).

In the study sample of 62 children and adolescents with ASD, 8 were diagnosed with childhood autism, 3 with atypical autism, 45 with Asperger’s syndrome, and 6 with pervasive developmental disorder not otherwise specified according to International Classification of Diseases 10th edition (ICD-10). The mean age of the participants was 12.3 (standard deviation (SD) = 2.0 (range: 9–16)) years. The male-to-female ratio was 6:1 (53 boys, 9 girls). Intellectual ability was within the normal range (IQ > 70) and confirmed by specialists in child and adolescent psychiatric and pediatric outpatient hospital clinics. Full Scale Intelligence Quotient (FSIQ) scores were available for 51 participants; the mean was 100.8 (SD = 18.1 (range: 70–135)). Of the 62 students, 23 (38%) with ASD were diagnosed with a comorbid condition, mainly ADHD; Tic disorder; and obsessive compulsive disorder (Table 1). The diagnoses, both the ASD and the additional disorders, were assessed and confirmed by specialists in child and adolescent psychiatric and pediatric outpatient clinics using a comprehensive diagnostic process involving interviews and clinical observations for all the participants.

Table 1.

Characteristics of the 62 participants with ASD with and without SRB.

	ASDn = 29n (%)	ASD and SRBn = 33n (%)	t value (df)	Chi-square (df = 1)	p value
Proportion of males/females	26 (89.7)/3 (10.3)	27 (81.8)/6 (18.2)		0.764	0.382
Age, mean (SD)	12.1 (2.3)	12.5 (1.8)	−0.739 (60)		0.463
Total score IQ^a, mean (SD)	99.8 (19.0)	101.6 (17.6)	−0.350 (49)		0.728
Proportion students with comorbid conditions	9 (31)	14 (42.4)		0.858	0.354
Childhood autism	7 (24.1)	1 (3.0)		6.119	0.013
Atypical autism	1 (3.4)	1 (3.0)		0.009	0.926
Asperger’s syndrome	18 (62.1)	27 (81.8)		3.025	0.082
Pervasive developmental disorder not specified (PDD NOS)	3 (10.3)	4 (12.1)		0.049	0.825
Frequency of SRB	–	33 (53.2)
1–3 days displayed SRB	–	10 (30.3)
4–10 days displayed SRB	–	14 (42.4)
11–20 days displayed SRB	–	9 (27.3)
Expressions of school refusal behavior
Proportion with refusal	–	18 (54.5)
Proportion with absenteeism	–	15 (45.5)

ASD: autism spectrum disorder; SRB: school refusal behavior; SD: standard deviation; df: degree of freedom.

Values are reported as n (%) unless otherwise stated.

51 participants, n = 22 ASD, n = 29 ASD and SRB.

The sociodemographic characteristics of the participants, assessed by parents in a revised questionnaire (Taylor, 1986), showed that 42 (67.7%) participants lived with only one biological parent, that 35 (56.5%) of the mothers had a university degree, and that 33 (53.2%) families experienced an illness in another family member. In all, 33 (53.2%) of the participants lived in an urban area, and 55 (88.7%) of the families owned their own apartment or house.

In total, 33 of the students with ASD (53.2%) displayed SRB. Both duration and expression of SRB in the students with ASD were analyzed and showed that approximately 70% displayed the behavior on four or more days in the 20 days assessed. Furthermore, almost half of the students displayed SRB as partial or complete absenteeism (Table 1).

Measures

SRB questionnaire

SRB was assessed by a parent-rated questionnaire developed for the study based on Kearney and Albano’s (2004) description of SRB. A parent placed an x in one or several of the categories in the questionnaire for each of the 20 days: (0) attendance, (1) pleads for not attending school, (2) misbehavior in the morning to avoid school, (3) tardiness followed by attendance, (4) pleads to not attend classes during the day, (5) did not attend classes, or (6) did not attend school. If parents marked an x in one of the categories from (1) to (6), they were asked to write the child’s reasons in the comment field in the questionnaire. If a parent had marked more than one x within the same day, only one was counted. If one of those x’s were noted in categories (3), (5), or (6) without a written excuse for the absenteeism, it was summarized as partial/complete SRB. Partial and complete absenteeism were counted as equal in the partial/complete SRB category. The (0) attendance category served as a control variable to ensure that the questionnaire had been answered and was not summarized. SRB was defined as 1–20 days of refusal to attend school/classes expressed verbally or physically or as partial or complete absenteeism without any legal excuse written in the comment field. If the parents did not mark an x in any of the categories three times or less, it was counted as attendance, and more than three times excluded them from the study.

The Social Responsiveness Scale

The Social Responsiveness Scale (SRS) is a 65-item questionnaire that assesses the severity of social impairment and ASD symptoms in children and adolescents aged 4–18 years (Constantino and Christian, 2005). A parent who is familiar with the individual’s current behavior and developmental history completes the questionnaire by circling a number: (1) not true, (2) sometimes true, (3) often true, or (4) almost always true, in a way that best describes the child’s behavior over the past 6 months. The SRS consists of five treatment subscales, Social Awareness, Social Cognition, Social Communication, Social Motivation, and Autistic Mannerisms, and a Total Score. The T scores in the range of 60–75 indicate deficiencies in reciprocal social behavior that are clinically significant and show a mild to moderate interference in everyday social interactions. The T scores of 76 or above indicate severe interference in daily social interactions and are strongly associated with ASD. Findings from several studies have shown that SRS is useful in screening for ASD symptoms (Bolte et al., 2008; Constantino and Christian, 2005; Wigham et al., 2012).

The Behavior Inventory of Executive Function

The Behavior Inventory of Executive Function (BRIEF) assesses executive function in daily living in children and adolescents aged 5–18 years (Fallmyr and Egeland, 2011; Gioia et al., 2000). The parent inventory was utilized in this study, and a parent marked (1) never a problem, (2) sometimes a problem, or (3) often a problem for each of the 86 items describing the child’s behavior for the past 6 months. The BRIEF is commonly used in clinical practice and research and consists of eight subscales: Inhibit, Shift, Emotional Control, Initiate, Working Memory, Plan/Organize, Organization of Materials, and Monitor. The scales are transformed into a total score, the Global Executive Composite (GEC), and two different indexes: the Behavioral Regulation Index (BRI) and the Metacognition Index (MI). The BRI is composed of the scales Emotional Control, Inhibit, and Shift and the MI of the scales Initiate, Working Memory, Plan/Organize, Organization of Materials, and Monitor. The BRIEF provides gender- and age-standardized T scores for each scale. The clinical cut-off is 65, and scores above 50 indicate a borderline level of concern.

The Child Behavior Checklist

The Child Behavior Checklist (CBCL) is a parent-rated questionnaire assessing emotional and behavioral problems in children and adolescents aged 6–18 years. The CBCL is commonly used in clinical practice and research and has well-documented psychometric properties (Achenbach and Rescorla, 2001; Koot and Verhulst, 1992; Novik, 1999). The CBCL was also used in several studies to assess comorbid emotional and behavioral problems in children with ASD (Gjevik et al., 2015; Hartley et al., 2008; Hurtig et al., 2009; Kanne et al., 2009; Kuusikko et al., 2008; Skokauskas and Gallagher, 2012). The CBCL consists of 112 items that are rated on a three-point scale and are transferred into eight syndrome scales and six Diagnostic and Statistical Manual of Mental Disorders (DSM)-oriented scales. Three broadband scales (Internalizing, Externalizing, and Total Problems) were created from the syndrome scales. In this study, we wanted to explore the association between SRB and the eight syndrome scales: Anxiety/Depressed, Withdrawn/Depressed, Somatic Complaints, Social Problems, Thought Problems, Attention Problems, Rule-Breaking Behaviors, and Aggressive Behaviors. The CBCL syndrome scales were developed through factor analysis in the general child population. Gender- and age-standardized T scores for each CBCL scale were calculated. A T score of 50 represents the average score in typically developing children at the same age and gender, and each 10 points represent 1 SD. Scores in the borderline or in the clinical range represent reasons for concern: the range for the syndrome scales is 65–70, and the range for the broadband scale is 60–64.

Statistics

Continuous variables, such as scales from the SRS, the BRIEF, and the CBCL, are presented as the mean ± SD. Categorical variables, such as SRB with answers “yes” or “no,” are presented as the number of observations (percentages). Statistical comparisons between groups were assessed by the chi-square test for crosstabs or the independent samples t test as appropriate.

Analyses comparing SRB and no-SRB groups on the SRS, BRIEF, and CBCL were performed with independent samples t test. Multivariate analysis of variance (MANOVA) was performed to assess the independent contributions of the SRS, BRIEF, and CBCL to SRB. Logistic regression analysis was used to analyze the associations between the independent variables with large effect sizes and SRB. Stepwise logistic multiple regression analyses, using both a forward and backward approach, were performed to analyze the subscale scores on the BRIEF and the CBCL. The criteria for including a variable in the model for both forward and backward stepwise regression were set to p < 10. The assumptions of linearity, independence of errors, and normality of residuals were met. Multicollinearity was assessed by estimating correlations between the predictors in the logistic regression and found satisfactory to conduct regression analysis.

Due to multiple comparisons, statistical analysis using Bonferroni correction was conducted within the SRS, the BRIEF, and the CBCL. Thus, the significance levels for the t tests within SRS, BRIEF, and CBCL were adjusted according to the number of subscales: SRS 0.05/5 = 0.01, BRIEF 0.05/8 = 0.006, and CBCL 0.05/8 = 0.006. The significance level for MANOVA and logistic regression analysis were set to 0.05. To analyze the effect size for SRB on the variables assessed with the SRS, the BRIEF, and the CBCL, Cohen’s d was calculated. Cohen’s d defines “small” (0.2), “medium” (0.5), and “large” (0.8) effect sizes and it might be larger than 1 (Cohen, 1988).

In the power calculations, we used different scenarios of the population parameter of Cohen’s d effect size expressed as follows

δ = \frac{μ_{1} - μ_{2}}{σ}

where δ is the population parameter of Cohen’s d, μ₁ and μ₂ are the mean of the respective populations, and σ = σ1 = σ2 is the homogeneous population SD. The estimation of the statistical power for an independent samples t test with two groups of 29 and 33 subjects, respectively, was conducted using different scenarios of the population parameter of Cohen’s d. A population parameter of Cohen’s d of 0.2, 0.5, and 0.8 corresponded to a statistical power of 12%, 49%, and 87%, respectively. Thus, our sample size was adequate to detect medium to large effect sizes with sufficient statistical power.

All analyses were performed using SPSS version 21.0 (IBM SPSS Statistics; IBM Corporation, Armonk, NY, USA), except for estimations of statistical power that was performed using Stata/SE version 14.1 (Stata Corp, College Station, TX, USA). The study protocol was approved on 11 April 2011 by the Norwegian National Committee for Research Ethics and conducted in accordance with the Declaration of Helsinki.

Results

Social functioning

The MANOVA for SRS was not statistically significant (p = 0.083). However, results from the t test showed a difference between the students with and without SRB in the SRS Social Motivation subscale (p = 0.002) (Table 2).

Table 2.

Results from the SRS, the BRIEF, and the CBCL in 62 students with ASD with and without SRB.

	ASD without SRBn = 29Mean (SD)	ASDwith SRBn = 33Mean (SD)	Mean difference (MD)	t value (df)	95% CI	p value	Cohen’s d
SRS
Social Communication	72.3 (13.4)	77.4 (11.9)	−5.1	−1.6 (59)	−11.6 to 1.4	0.12	0.4
Social Awareness	63.7 (9.1)	65.6 (13.0)	−1.8	−0.6 (59)	−7.7 to 4.0	0.53	0.2
Social Cognition	73.0 (13.3)	79.2 (13.3)	−6.2	−1.8 (59)	−13.0 to 3.9	0.07	0.5
Social Motivation	73.0 (11.4)	83.7 (13.7)	−10.6	−3.3 (59)	−17.1 to −4.2	0.002	0.8
Autistic Mannerisms	79.2 (14.2)	85.9 (13.9)	−6.7	−1.9 (59)	−13.3 to 0.4	0.06	0.5
Total Score	76.3 (12.1)	83.5 (11.9)	−7.2	−2.3 (59)	−13.3 to −1.0	0.02	0.6
BRIEF
Shift	69.5 (11.1)	77.0 (11.7)	−7.5	−2.6 (60)	−13.3 to −1.6	0.01	0.7
Inhibit	61.2 (11.8)	60.7 (12.3)	0.6	0.2 (60)	−5.570 to 6.7	0.85	−0.1
Initiate	56.0 (10.4)	67.9 (10.8)	−11.9	−4.4 (60)	−17.3 to −6.5	<0.001	1.1
Emotional Control	60.7 (11.8)	67.4 (11.6)	−6.6	−2.2 (60)	−12.6 to 0.7	0.03	0.6
Working Memory	60.2 (10.7)	66.1 (13.2)	−5.9	−1.9 (60)	−12.1 to 0.3	0.06	0.5
Plan/Organize	58.0 (9.2)	67.8 (11.5)	−9.9	−3.7 (60)	−15.2 to −4.5	<0.001	0.9
Organization of Materials	50.0 (10.8)	54.5 (12.0)	−4.5	−1.5 (60)	−10.3 to 1.3	0.13	0.4
Monitor	62.2 (10.6)	66.0 (10.4)	−3.8	−1.4 (60)	−9.1 to 1.6	0.16	0.4
BRI (Shift, Inhibit, Emotional Control)	65.3 (11.4)	70.3 (11.5)	−5.1	−1.7 (60)	−10.9 to 0.8	0.09	0.4
MI (Initiate, Working Memory, Plan/Organize, Organization of Materials, Monitor)	58.4 (9.2)	67.2 (11.2)	−8.8	−3.3 (60)	−14.0 to −3.5	0.001	0.9
Total score GEC	62.0 (9.6)	69.6 (10.6)	−7.7	−3.0 (60)	−12.8 to −2.5	0.004	0.7
CBCL
Anxiety/Depressed	61.3 (8.1)	70.2 (12.8)	−9.0	−3.2 (58)	−12.7 to −2.1	0.002	0.8
Withdrawn/Depressed	59.7 (7.1)	69.7 (10.0)	−10.0	−4.4 (58)	−14.3 to −5.0	<0.001	1.2
Somatic Complaints	57.5 (7.7)	64.6 (10.7)	−7.0	−2.2 (58)	−10.8 to −0.6	0.005	0.8
Social Problems	64.9 9.3)	68.3 (8.9)	−3.4	−1.4 (58)	−7.4 to 2.2	0.15	0.4
Thought Problems	62.9 (8.6)	69.4 (8.8)	−6.5	−2.9 (58)	−11.0 to −1.9	0.005	0.7
Attention Problems	60.3 (8.2)	65.6 (10.7)	−5.2	−2.1 (58)	−9.1 to 1.1	0.04	0.6
Rule-breaking Behaviors	56.0 (5.8)	59.4 (7.6)	−3.4	−2.0 (58)	−6.4 to 0.9	0.06	0.5
Aggressive Behaviors	60.2 (10.4)	63.9 (9.2)	−3.7	−1.5 (58)	−8.0 to 2.3	0.15	0.4
Internalized	61.0 (8.9)	72.9 (12.3)	−12.0	−4.3 (58)	−16.5 to −4.7	<0.001	1.1
Externalized	56.4 (12.3)	62.7 (9.4)	−6.2	−2.2 (58)	−11.4 to 0.1	0.03	0.6
Total problem score	62.6 (8.7)	71.0 (10.2)	−8.4	−3.4 (58)	−12.6 to −2.5	0.001	0.9

SRS: Social Responsiveness Scale; BRIEF: Behavior Inventory of Executive Function; CBCL: Child Behavior Checklist; ASD: autism spectrum disorder; SRB: school refusal behavior; SD: standard deviation; df: degree of freedom; CI: confidence interval; BRI: Behavioral Regulation Index; MI: Metacognition Index; GEC: Global Executive Composite.

Executive function

The MANOVA for BRIEF was statistically significant (p = 0.002). Results from the t test showed that the BRIEF GEC scores were higher in the students with ASD displaying SRB compared to those without SRB (p = 0.004). Furthermore, two of the eight subscales from the BRIEF showed differences between students displaying SRB and those who did not: Initiate (p < 0.001) and Plan/Organize (p < 0.001) (Table 2).

Emotional and behavioral problems

The MANOVA for CBCL was statistically significant (p = 0.004). Results from the t test showed that the CBCL Total Problems scores were higher in students with SRB compared to those without SRB (p = 0.001). Furthermore, differences between students with and without SRB were found in four of the eight CBCL subscales: Anxiety/Depressed (p = 0.002), Withdrawn/Depressed (p < 0.001), Somatic Complaints (p = 0.005), and Thought Problems (p = 0.005) (Table 2).

Logistic regression of executive functioning and emotional problems

The subscale Initiate (BRIEF) (p < 0.001) and the Withdrawn/Depressed subscale (CBCL) (p = 0.001) remained significant after performing the logistic multiple regression analyses (Table 3).

Table 3.

Logistic regression analysis of associations between SRB in students with ASD and subscale variables from the BRIEF and the CBCL.

	Simple logistic regression			Multiple logistic regression
	OR	95% CI	p value	OR	95% CI	p value
BRIEF
Shift	1.06	1.01–1.11	0.017	–	–	–
Initiate	1.11	1.05–1.17	<0.001	1.11	1.05–1.17	<0.001
Plan/Organize	1.10	1.03–1.15	0.002	–	–	–
CBCL
Anxiety/Withdrawn	1.10	1.02–1.14	0.005	–	–	–
Depressed/Withdrawn	1.14	1.05–1.23	0.001	1.14	1.06–1.23	0.001
Somatic Complaints	1.10	1.02–1.20	0.009	–	–	–

BRIEF: Behavior Inventory of Executive Function; CBCL: Child Behavior Checklist; ASD: autism spectrum disorder; SRB: school refusal behavior; SD: standard deviation; df: degree of freedom; CI: confidence interval; OR: odds ratio.

Discussion

In this study, we explored individual characteristics in a sample of students with ASD and SRB. The students were aged 9–16 years without intellectual disability and were going to inclusive schools. To explore characteristics in these students, our study focused on four factors: social and executive functioning and emotional and behavioral problems. Problems within these four factors are known to be common in students with ASD but not previous examined related to SRB in this population. However, except executive functioning, these factors are commonly examined in studies of SRB in the general child and adolescent population (Egger et al., 2003; Havik et al., 2015; Ingul and Nordahl, 2013; Kearney, 2008a). Executive deficits are reported to negatively impact daily living skills and academic outcomes in students with ASD and are thus of interest in our study (Carretti et al., 2014; Gilotty et al., 2002; Hill, 2004a).

The analyses of the subscales in each of the three assessments tools, the SRS, the BRIEF, and the CBCL, showed differences in social and executive functioning and emotional problems between students with ASD and SRB and students with ASD without SRB. Our results suggest that the students with ASD and SRB were characterized by lower social motivation, more impaired abilities to initiate activities or tasks and to generate ideas, responses, or problem-solving strategies and showed more symptoms of withdrawal and depression. The findings will be discussed in turn as they are related to previous studies of these characteristics in students with ASD and to SRB in the general child and adolescent population.

Social functioning in the students with ASD

Although all the students with ASD displayed high rates of overall impaired social functioning, the students with SRB were more socially impaired than those without SRB. Furthermore, low social motivation showed the strongest association with SRB. We found no difference between students with and without SRB in social communication and awareness of self or others’ nonverbal social behavior.

Previous studies have reported that students with ASD attending inclusive schools are mostly socially unengaged in the schoolyard and more in the periphery of the social networks than their classmates (Kasari et al., 2011; Macintosh and Dissanayake, 2006). However, the students were not found to be more exposed to active social rejection than their typically developing peers (Kasari et al., 2011). Whether students with ASD tend to distance themselves, or whether they are overlooked as potential playmates by the other students, was not identifiable in these studies (Kasari et al., 2011; Macintosh and Dissanayake, 2006). Our findings revealed that students with ASD and SRB showed low motivation to engage in social settings, rather than impaired social communication skills and ability to recognize socially relevant cues. Whether impaired social motivation reported in the students with SRB represents temperamental and personality traits or social withdrawal due to past negative social experiences, and how social motivation is related to SRB, needs to be further explored. Since MANOVA for SRS was not statistically significant, results should be interpreted with some caution due to multiple comparisons.

Executive functioning in students with ASD

Overall, students with SRB displayed higher rates of executive deficits (GEC) than students without SRB. Thus, consistent with the study by Ohmann et al. (2007) reporting executive deficits in other clinical populations of adolescents with prolonged SRB, our findings suggest that SRB in students with ASD is associated with neurocognitive components. Furthermore, several of the BRIEF subscales differentiate between the students with ASD and SRB and those without SRB. Impaired ability to initiate showed the strongest association with SRB, followed by planning/organizing and shifting. However, students with SRB did not differ from those without SRB in their capacities to inhibit and monitor behavior, in the organization of material or in working memory. The findings in our study show similarities with findings in studies of quality of life and adaptive behavior problems in students with ASD. Lack of initiative and impaired shifting, planning and organizing, and working memory abilities were strongly associated with problems in overall adaptive, social, and school functioning (De Vries and Geurts, 2015; Gilotty et al., 2002; Leung et al., 2016; Rosenthal et al., 2013). Other than deficits in working memory, these elements were remarkably similar to those that differentiated between the students with and without SRB. We may speculate that the initiation deficits shown in students with SRB may impede the students’ ability to ask for information and support from teachers and peers necessary for them to start tasks and social activities. Furthermore, the impaired initiation might be interpreted from teachers and peers as a lack of interest and not as a need for support. At home, it is possible that this deficit impacts the morning routine such that the students will be late for school. To refuse to go school when it is too late was reported as a reason for displaying SRB from parents in the comments field of the questionnaire. However, the relation between executive dysfunction and SRB needs to be further explored both by reports from students, parents, and teachers and by clinicians in standardized neuropsychological assessments.

Emotional and behavioral problems in students with ASD

Students with ASD and SRB displayed overall more internalizing problems than the students without SRB. Withdrawn and depressed symptoms showed the strongest association with SRB, followed by anxiety and depressed symptoms and somatic complaints. Students with and without SRB did not differ on subscales of social, aggressive, or rule-breaking behaviors. Thus, our findings are mainly consistent with studies of SRB in the general child and adolescent population, which reported that anxiety, depression, and somatic complaints were the most prevalent conditions associated with SRB (Egger et al., 2003; Havik et al., 2015; Ingles et al., 2015; Ingul and Nordahl, 2013). Furthermore, studies of students with SRB have shown that low expectations in coping with stressful situations in school and negative automatic thoughts were associated with the condition (Heyne et al., 1998; Maric et al., 2012). Similar findings were revealed in our study; parents commented in the SRB questionnaire that negative thoughts of relationships with peers and teachers and the school subjects, especially physical education, were frequently reasons for SRB in the students with ASD.

Why high rates of withdrawal and depression are related to SRB in students with ASD in mainstream schools needs to be further explored (e.g. by including more information concerning the school environment, other life events and genetics). However, we may speculate that a sense of not coping in school possibly leads to withdrawal and depression associated with SRB.

The use of three assessment tools, the SRS, the BRIEF, and the CBCL, to compare characteristics of students with ASD with and without SRB was valuable. Findings from the various tools point in the same direction showing that the behaviors characterizing the students with SRB are lower social motivation, more impaired initiating ability, and a higher level of withdrawn and depressed behavior than students without SRB. However, although the tools assessed different domains, some questions covered similar aspects across the different questionnaires and may measure similar impairments. For instance, Gioia et al. (2000) underscored that highly elevated scores on the BRIEF Initiate subscale might reflect depression. Indeed, the relation between the characteristics assessed in the students and SRB has not been established. We may speculate that the students displayed SRB as a consequence of unmet needs for support and education and that they are especially vulnerable in coping with experiences of failure in school. However, whether the characteristics revealed in the students with ASD lead to SRB or if SRB exacerbates the deficits in the students is not clear, and the relation may be bidirectional.

Clinical implications

Our findings may increase awareness of individual characteristics in students with ASD and SRB and provide information for teachers and professionals assessing and treating SRB in these students. Because impaired social and executive functioning and emotional problems are associated with ASD, these traits may be easily overlooked as problems associated with SRB. Characteristics associated with SRB in students with ASD show similarities compared to reports of SRB in students in the general child and adolescent population. Thus, we may suggest including our results in assessment and treatment procedures utilized in general could be considered for students with ASD. However, longitudinal studies are needed to identify more specific risk factors for SRB in ASD students. Knowledge from such studies would be relevant for interventions specifically targeting SRB.

For preventive reasons, it may be valuable for teachers to know that social impairment, executive deficits, and emotional problems are associated with SRB in students with ASD. This knowledge may help teachers provide adequate and sufficient support and education needed for the individual student to cope with the inclusive school environment.

Specifically, we suggest that the students with low social motivation may need support in seeking and finding rewarding social relationships through joint interests in addition to social skills programs. Furthermore, as a supplement to psychoeducation, individual support, and training, the use of electronic devices may ease the problems associated with the students’ executive deficits in school and at home.

Strengths and limitations

The strength of this study was including the three assessment tools that did not contain questions concerning SRB and were thus appropriate for the purpose. However, there were limitations that need to be noted. Parenting a child with SRB could be stressful, and this experience may have biased the findings in the students with SRB. Furthermore, the small sample size of students limits the generalizability of the findings. The study design does not allow us to determine the causal direction of the associations between SRB and the characteristics found in the students.

Conclusion

The results from our study showed that students with ASD and SRB differ from students without SRB within the three domains measured by the SRS, the BRIEF, and the CBCL. Furthermore, the study revealed characteristics in students with SRB that might be used to recognize early signs and to construct and implement treatment interventions for SRB. However, it is important to note that this study yielded group-level findings and that interventions must be tailored to individual qualities of students and not to group characteristics.

Our study is a unique but small contribution to the exploration of a complex phenomenon prevalent in students with ASD going to inclusive schools. As this study is the first of its kind, there is a need for replication studies that address methodological and research design limitations. Further studies are needed that are both long-term follow-up studies and studies assessing individual characteristics by self-report, teacher report, and clinician assessment in lab settings. Whether characteristics of individuals with SRB differ between the genders should be explored in future studies. Studies focusing on contextual variables in the school environment, such as bullying, special educational competence, and the number of students in the schools and classes, are also needed to better understand the complexity of SRB.

Footnotes

Acknowledgements

The authors gratefully acknowledge the children and their parents and the clinicians in the South-East Health Region for their participation.

Funding

The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This study was supported by Oslo University Hospital, Regional Resource Center for Autism, ADHD, Tourette’s Syndrome, and Narcolepsy.

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