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Measuring the Functional Impact of Behavioral Inflexibility in Children with Autism Using the Behavioral Inflexibility Scale: Clinical Interview (BIS-CI)

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Title: Measuring the Functional Impact of Behavioral Inflexibility in Children with Autism Using the Behavioral Inflexibility Scale: Clinical Interview (BIS-CI)
Language: English
Authors: Bodfish, James W., Lecavalier, Luc, Harrop, Clare, Dallman, Aaron, Kalburgi, Sahana Nagabhushan, Hollway, Jill, Faldowski, Richard, Boyd, Brian A. (ORCID 0000-0002-6482-9577)
Source: Journal of Autism and Developmental Disorders. Feb 2022 52(2):782-790.
Availability: Springer. Available from: Springer Nature. One New York Plaza, Suite 4600, New York, NY 10004. Tel: 800-777-4643; Tel: 212-460-1500; Fax: 212-460-1700; e-mail: customerservice@springernature.com; Web site: https://link.springer.com/
Peer Reviewed: Y
Page Count: 9
Publication Date: 2022
Sponsoring Agency: Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD) (DHHS/NIH)
Contract Number: R01HD082127
P30HD03110
Document Type: Journal Articles
Reports - Research
Descriptors: Children, Autism, Pervasive Developmental Disorders, Child Behavior, Behavior Problems, Parents, Test Validity, Interviews, Test Reliability, Interrater Reliability
DOI: 10.1007/s10803-021-04984-z
ISSN: 0162-3257
Abstract: For individuals with autism spectrum disorder (ASD), behavioral inflexibility can affect multiple domains of functioning and family life. The objective of this study was to develop and validate a clinical interview version of the Behavioral Inflexibility Scale. Trained interviewers conducted interviews with parents of 144 children with ASD and 70 typically developing children (ages: 3-17 years). Using exploratory factor analysis, the Behavioral Inflexibility Scale-Clinical Interview (BIS-CI) was found to be unidimensional. Reliability data indicated the measure was internally consistent ([alpha] = 0.80), achieved excellent inter-rater reliability (ICC = 0.97) and test-retest reliability (ICC = 0.87). These findings demonstrate that the BIS-CI is a reliable and valid measure to determine the functional impact of behavioral inflexibility.
Abstractor: As Provided
Entry Date: 2022
Accession Number: EJ1327284
Database: ERIC
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  Value: <anid>AN0155062837;aut01feb.22;2022Feb08.05:38;v2.2.500</anid> <title id="AN0155062837-1">Measuring the Functional Impact of Behavioral Inflexibility in Children with Autism Using the Behavioral Inflexibility Scale: Clinical Interview (BIS-CI) </title> <p>For individuals with autism spectrum disorder (ASD), behavioral inflexibility can affect multiple domains of functioning and family life. The objective of this study was to develop and validate a clinical interview version of the Behavioral Inflexibility Scale. Trained interviewers conducted interviews with parents of 144 children with ASD and 70 typically developing children (ages: 3–17 years). Using exploratory factor analysis, the Behavioral Inflexibility Scale-Clinical Interview (BIS-CI) was found to be unidimensional. Reliability data indicated the measure was internally consistent (α = 0.80), achieved excellent inter-rater reliability (ICC = 0.97) and test–retest reliability (ICC = 0.87). These findings demonstrate that the BIS-CI is a reliable and valid measure to determine the functional impact of behavioral inflexibility.</p> <p>Keywords: Autism spectrum disorder; Behavioral inflexibility; Measurement; Outcomes; Repetitive behavior</p> <hd id="AN0155062837-2">Introduction</hd> <p>Behavioral inflexibility can be defined as rigid and inflexible patterns of behavior that contrast with the need to be flexible, open to change, and adaptable to the inevitable need to change and adapt in the context of the complex and often unpredictable demands of one's surroundings (Lecavalier et al., [<reflink idref="bib19" id="ref1">19</reflink>]). Inflexibility has been found to be a characteristic of a wide range of domains of functioning in ASD including cognition (e.g., cognitive flexibility or set shifting, Miller et al., [<reflink idref="bib24" id="ref2">24</reflink>]; rumination or repetitive thinking, Gotham et al., [<reflink idref="bib10" id="ref3">10</reflink>]), language (Muskett et al., [<reflink idref="bib25" id="ref4">25</reflink>]; e.g., perseverative language, echolalia), social function (Loftin et al., [<reflink idref="bib20" id="ref5">20</reflink>]), play (Honey et al., [<reflink idref="bib11" id="ref6">11</reflink>]), and eating/mealtime (Johnson et al., [<reflink idref="bib15" id="ref7">15</reflink>]). In a previous study we examined caregiver and clinician perspectives on behavioral inflexibility in ASD (Sethi et al., [<reflink idref="bib35" id="ref8">35</reflink>]) and found that caregivers and clinicians reported that some children's repetitive and inflexible behavior produced functional benefits (e.g. routines as a way to deal with unpredictability, intense interests as a way to make friends or develop vocational skills) while in other children their repetitive and inflexible behaviors produced functional deficits (e.g. movements that when interrupted lead to aggression or self-injury, rigidly held routines or overly narrow interests that lead to avoidance, restriction of experience, and stress within the larger family, school, or community context). This suggests the possibility that the discrete type or form of repetitive inflexible behavior may be less important from an intervention and treatment outcome perspective than if the behavior is associated with a rigid and inflexible style or trait that interferes with everyday activities, learning, and development.</p> <p>By definition all persons with ASD manifest repetitive and inflexible patterns of behavior; however, it is likely that there is a specific subset of individuals with ASD who manifest inflexibility in such a manner that they are associated with a clinically significant degree of functional impairment. From the perspective of treatment development and treatment outcome measurement, it is the goal of intervention studies to identify and treat this subset of cases whose repetitive and inflexible behavior is leading to functional impairment as opposed to identifying and treating all individuals who exhibit these patterns of behavior. Although instruments have been specifically developed and used to study repetitive behaviors in the context of ASD (see Honey et al., [<reflink idref="bib13" id="ref9">13</reflink>] for a review), these instruments all have at least some limitations as treatment outcome measures (see Scahill et al., [<reflink idref="bib34" id="ref10">34</reflink>]). Indeed, they were designed primarily to delineate specific topographies or measure the presence or absence of different types of repetitive behavior as opposed to measuring their functional impact on the daily lives of children with ASD and their families.</p> <p>In a previous paper, we described the multi-method iterative process we used to develop a novel parent report measure of inflexibility designed to assess the functional impact of behavioral inflexibility—the Behavior Inflexibility Scale (BIS; Lecavalier et al., [<reflink idref="bib19" id="ref11">19</reflink>]). The BIS was designed to be a parent report outcome measure. Psychometric analyses of the BIS using data from a large (n = 943) and representative sample of children with ASD indicated that the parent report BIS: (a) provides a unidimensional set of items that measure inflexibility, (b) yields an approximately normal distribution for the overall score, (c) has strong concurrent validity with an independent measure of RRBs, (d) varies independent of child age, gender, and parent reported cognitive and language ability, and (e) has excellent short-term test retest stability. For the present study, we extended this work in order to develop a structured clinical interview format of the BIS and examine the psychometric features of the interview instrument in a new sample of children with ASD. We reasoned that a clinical interview instrument could provide a means for blinding the measurement of behavioral inflexibility in treatment studies—a weakness of a parent report measure and an essential feature of a treatment outcome measure. Here we describe the approach we used to develop a structured clinical interview designed to measure the functional impact of behavioral inflexibility (the "Behavioral Inflexibility Scale—Clinical Interview") and examine its psychometric characteristics in an independent sample from the sample used to develop the parent report BIS. The aims of this study were: (a) to provide estimates of the magnitude of key psychometric features of the BIS-CI and, (b) to determine if BIS-CI scores are associated with child age, sex, or cognitive impairment, and (c) to contrast scores with those obtained by typically developing (TD) children.</p> <hd id="AN0155062837-3">Methods</hd> <p></p> <hd id="AN0155062837-4">BIS-CI—Instrument Development Procedure</hd> <p>The BIS-CI was developed by starting with the items developed for the BIS. The instrument was developed using a seven step multi-method iterative process for test development. (<reflink idref="bib1" id="ref12">1</reflink>) <emph>Development of construct</emph>: We began by developing a working definition of BI ("resistance to changing or adapting one's behavior in line with a changing situation") as the guiding concept for scale development and by identifying six potential dimensions that could help to define this construct more objectively (response to change, response to novelty, response to uncertainty, engagement in routines, tendency to be restrictive or picky, and oversensitivity to ambient events). (<reflink idref="bib2" id="ref13">2</reflink>) <emph>Expert panel review</emph>: We solicited feedback on this definition and set of dimensions from a panel of ten ASD experts. The experts' feedback was used to refine the working definition and to reduce the set of dimensions. (<reflink idref="bib3" id="ref14">3</reflink>) <emph>Caregiver and clinician focus groups</emph>: Four caregiver groups (n = 6–9 per group for a total of 30 parents) and three clinician groups (n = 5–11 per group for a total of 25 clinicians) were recruited from three different sites (University of North Carolina, Ohio State University, and Vanderbilt University). All sessions were audiotaped and transcribed verbatim. Using an iterative consensus process, a semantic code set was developed and then reduced to a subset of the codes used most consistently across independent raters which resulted in a set of themes that consistently emerged from both the caregiver and clinician focus groups. (<reflink idref="bib4" id="ref15">4</reflink>) <emph>Review of existing measures</emph>: Thirteen scales were selected and reviewed from the literature based on five constructs (repetitive behaviors, sensory behaviors, executive function/cognitive flexibility, temperament, and social-communication behaviors). Items from these scales were independently rated and then selected as relating to our BI construct. (<reflink idref="bib5" id="ref16">5</reflink>) <emph>Developing the initial item pool</emph>: An initial pool of items was derived by matching content from the measurement review with the themes derived from the focus group thematic analysis, followed by revision of items and item wording using a consensus process. (<reflink idref="bib6" id="ref17">6</reflink>) <emph>Expert Panel review to reduce to final item pool</emph>: The final item set for the BIS and for the BIS-CI was derived through independent review by the ten expert panelists. (<reflink idref="bib7" id="ref18">7</reflink>) We developed a clinical interview version of the BIS by converting the 38 BIS items content into a 25-item symptom checklist (part 1 of the BIS-CI), and adding a set of seven severity rating items designed to measure functional impairment (part 2 of the BIS-CI). Further details about these scale development steps can be found in previous publications from this project (Lecavalier et al., [<reflink idref="bib19" id="ref19">19</reflink>]; Sethi et al., [<reflink idref="bib35" id="ref20">35</reflink>]).</p> <hd id="AN0155062837-5">Description of BIS-CI</hd> <p>The BIS-CI is a structured clinical interview version of the parent report BIS. The BIS-CI consists of two parts. Part 1 is a 25 item symptom checklist, each symptom to be endorsed by the informant as either present or absent with respect to their child. The symptom checklist is derived from the items of the BIS parent rating scale and so includes the variety of forms that BI may be exhibited in children. In Part 1, caregivers are asked whether any of the described inflexible behaviors have been problematic for the child or family over the past month (e.g., the child has trouble accepting a change must occur, or gets stuck on particular activities or topics). Twenty-four of the 25 items in the symptom checklist were taken directly from the BIS, and one item allows respondents to list other inflexible behaviors not previously described. The items of the checklist are not analyzed individually; rather, they are rated in the aggregate by the interviewer across the seven clinical interview questions in part 2. Part 2 is a set of seven clinical interview questions designed to assess the functional impact of the items endorsed in the symptom checklist portion of the interview. In Part 2, caregivers are asked to consider all of the inflexible behaviors they identified as problematic for their child during Part 1 of the interview in their aggregate in order to answer a set of questions about the severity or functional impact of the child's inflexible behaviors. The seven severity questions in Part 2 were designed to assess the following: frequency of occurrence, interference at home, interference with social relationships, interference with activities away from home, need for family members to accommodate, child avoidance of situations, and child distress when inflexible behaviors are interrupted. Each of these was phrased as an interview question that asked parents to consider the aggregate of behaviors endorsed on the symptom checklist (e.g., for the avoidance question: "How often has your child had to avoid certain things, or going certain places because of these behaviors?"). The Part 2 severity questions are scored using a 5-point Likert severity rating scale for each item ranging from 0 to 4 (with higher scores indicating greater severity) based on the caregiver's response to the question. The BIS-CI yields one total score derived by summing the responses for the seven severity items from Part 2. The BIS-CI total score ranges from 0 (no impairment) to a maximum score of 28 (maximum impairment). On average, we have found that the BIS-CI takes approximately 10–15 min to complete by a trained rater.</p> <hd id="AN0155062837-6">Participants</hd> <p>A sample of 163 children with ASD and their caregivers was recruited for in-person assessment at three different sites (University of North Carolina, Ohio State University, and Vanderbilt University). Recruitment was designed to be nonspecific with respect to the construct of interest (behavioral inflexibility) using social media and print recruitment adds that described a study to examine "patterns of behavior in children with and without autism." This was designed to recruit a relatively diverse sample of children with ASD (e.g., a range of ages, level of cognitive/language ability and level of ASD severity), as opposed to recruiting children whose caregivers were interested in or motivated to participate in a study on repetitive behaviors or behavioral inflexibility. Inclusion criteria for the ASD sample were: (a) child with ASD between the ages of 3–17 years, (b) a clinical diagnosis of ASD confirmed by the Autism Diagnostic Observation Schedule (ADOS-2; above established cutoff for ASD on this measure), and (c) ability to travel to one of the three testing sites. Exclusion criteria for the ASD sample were limited to the presence of a comorbid genetic disorder, including Fragile X. Following diagnostic assessments, 19 cases were excluded from this study as they were not evaluated with the BIS-CI during the course of the project, leaving a final sample of 144 children with ASD for analysis. A comparison sample of 70 typically developing (TD) children was recruited at the same time and from the same sites as the ASD sample. Inclusion criteria for the TD sample were: (a) no diagnosis of a developmental condition including ASD, (b) no first degree relatives with a diagnosis of ASD, and (c) ages between 3 and 17 years. Clinical and demographic characteristics of the final ASD and TD samples are provided in Table 1. Following screening and testing, no TD children were excluded from the study leaving a final sample of 70 TD participants for analysis. We included TD children simply to contrast scores with those obtained with young people with ASD. This research was prospectively reviewed and approved by the appropriate Institutional Review Boards.</p> <p>Table 1 Demographic and clinical characteristics of participants in the ASD (n = 144) and TD (n = 70) groups</p> <p> <ephtml> <table frame="hsides" rules="groups"><thead><tr><th align="left" /><th align="left"><p>ASD</p></th><th align="left"><p>TD</p></th></tr></thead><tbody><tr><td align="left"><p>Age in years [mean (SD)]</p></td><td align="left"><p>9.4 (3.9)</p></td><td align="left"><p>8.9 (3.4)</p></td></tr><tr><td align="left" colspan="3"><p>Sex (%)</p></td></tr><tr><td align="left"><p> Males</p></td><td align="left"><p>93%</p></td><td align="left"><p>69%</p></td></tr><tr><td align="left"><p> Females</p></td><td align="left"><p>7%</p></td><td align="left"><p>31%</p></td></tr><tr><td align="left" colspan="3"><p>Caregiver race (%)</p></td></tr><tr><td align="left"><p> White</p></td><td align="left"><p>70%</p></td><td align="left"><p>77%</p></td></tr><tr><td align="left"><p> Black</p></td><td align="left"><p>17%</p></td><td align="left"><p>14%</p></td></tr><tr><td align="left"><p> Other</p></td><td align="left"><p>9%</p></td><td align="left"><p>9%</p></td></tr><tr><td align="left"><p>IQ—standard score [mean (SD)]</p></td><td align="left"><p>88.9 (23.6)</p></td><td align="left"><p>104.1 (12.9)</p></td></tr><tr><td align="left" colspan="3"><p>Caregiver education (%)</p></td></tr><tr><td align="left"><p> Some high school</p></td><td align="left"><p>1%</p></td><td align="left"><p>1%</p></td></tr><tr><td align="left"><p> High school graduate or GED</p></td><td align="left"><p>5%</p></td><td align="left"><p>4%</p></td></tr><tr><td align="left"><p> Some college</p></td><td align="left"><p>31%</p></td><td align="left"><p>18%</p></td></tr><tr><td align="left"><p> College graduate</p></td><td align="left"><p>39%</p></td><td align="left"><p>44%</p></td></tr><tr><td align="left"><p> Advanced degree</p></td><td align="left"><p>24%</p></td><td align="left"><p>33%</p></td></tr><tr><td align="left" colspan="3"><p>Household income (n, %)</p></td></tr><tr><td align="left"><p> ≤$20,000</p></td><td align="left"><p>11%</p></td><td align="left"><p>3%</p></td></tr><tr><td align="left"><p> $20,001–$40,000</p></td><td align="left"><p>13%</p></td><td align="left"><p>5%</p></td></tr><tr><td align="left"><p> $40,001–$60,000</p></td><td align="left"><p>15%</p></td><td align="left"><p>11%</p></td></tr><tr><td align="left"><p> $60,001–$90,000</p></td><td align="left"><p>22%</p></td><td align="left"><p>34%</p></td></tr><tr><td align="left"><p> $90,001+</p></td><td align="left"><p>39%</p></td><td align="left"><p>46%</p></td></tr></tbody></table> </ephtml> </p> <hd id="AN0155062837-7">Diagnostic Testing and Clinical Characterization</hd> <p></p> <hd id="AN0155062837-8">Autism Diagnostic Observation Schedule (ADOS)</hd> <p>The ADOS-2 (Lord et al., [<reflink idref="bib21" id="ref21">21</reflink>]) is a gold-standard, standardized observational measure used to diagnose ASD. It was administered or directly supervised by research-reliable staff at each site to confirm the clinical diagnosis of ASD. The following modules were administered: Module 1, n = 32 (22.2%); Module 2, n = 15 11.4%); Module 3, n = 88 (61.1%), module 4 n = 4 (6.3%).</p> <hd id="AN0155062837-9">Stanford-Binet IQ</hd> <p>All participants were administered the abbreviated Stanford-Binet Intelligence Test, Fifth Edition (Roid, [<reflink idref="bib30" id="ref22">30</reflink>]) to determine verbal, nonverbal, and general IQ (Table 1). The full-scale IQ scores and abbreviated IQ scores are highly correlated in the ASD population (Twomey et al., [<reflink idref="bib39" id="ref23">39</reflink>]).</p> <hd id="AN0155062837-10">Parent-Reported Demographic and Clinical Characteristics</hd> <p>Caregivers reported information on child race, parental education, and household income.</p> <hd id="AN0155062837-11">Behavior Inflexibility Scale (BIS)</hd> <p>Caregivers of the participants were asked to complete the BIS (Lecavalier et al., [<reflink idref="bib19" id="ref24">19</reflink>]), a 38-item caregiver completed scale. Items are rated on a 6-point rating scale going from "Not at all a problem" to "Very severe or Extreme problem." Raters assessed behaviors over the past month. BIS total score was used to examine convergent validity with the BIS-CI.</p> <hd id="AN0155062837-12">Repetitive Behavior Scale—Revised (RBS-R)</hd> <p>Caregivers of the participants were asked to complete the RBS-R (Bodfish et al. [<reflink idref="bib3" id="ref25">3</reflink>]), a 43-item caregiver report measuring a variety of repetitive behaviors. Items are rated on a four-point Likert-scale and distributed along six subscales: stereotyped behavior, self-injurious behavior, compulsive behavior, ritualistic behavior, sameness behavior, and restricted behavior. The RBSR subscale and total scores were used to examine convergent validity with the BIS-CI.</p> <hd id="AN0155062837-13">Social Responsiveness Scale (SRS)</hd> <p>Caregivers of participants with ASD completed the SRS (Constantino and Gruber, [<reflink idref="bib6" id="ref26">6</reflink>]) to measure autistic traits. The SRS is a quantitative parent-completed questionnaire that can be used to measure the severity of deficits in social-communication skills. Items are distributed across both social-communication subscales (social awareness, social cognition, social motivation) and a repetitive behavior subscale (restricted repetitive behavior and interests). The social-communication subscale T scores were used to examine divergent validity with the BIS-CI.</p> <hd id="AN0155062837-14">Social Communication Questionnaire (SCQ)</hd> <p>The SCQ (Rutter et al., [<reflink idref="bib32" id="ref27">32</reflink>]) consists of 40 items arranged onto three subscales: social interaction, communication, and stereotyped behaviors. The SCQ total score was used to examine divergent validity with the BIS-CI.</p> <hd id="AN0155062837-15">Data Collection Procedure</hd> <p></p> <hd id="AN0155062837-16">BIS-CI Rater Training</hd> <p>Raters at all sites were trained using a standardized training approach. Raters read a standard administration manual and then participated together in a cross-site training meeting to review administration procedures for the BIS-CI to help insure consistent application of the measure across sites. Following these training steps, all raters separately rated a set of three previously recorded BIS-CI administrations. Each rater's scores were compared to a "gold standard" rater and discrepancies in scores from the standard were discussed as a group to review and clarify relevant scoring guidelines. Raters then independently administered and scored three additional BIS-CIs at their respective sites and each administration was audio recorded. These audio-recorded BIS-CI administrations were independently scored by a trained rater at another site. The raters at each site were required to reach at least 80% overall inter-rater reliability and item scores within 1 point on all items in order to proceed with BIS-CI data collection at their site.</p> <hd id="AN0155062837-17">BIS-CI Data Collection</hd> <p>BIS-CIs were completed in person at each of the three data collection sites by raters using a standardized structured interview format. Parents of the participants served as informants for the BIS-CI. Informants were asked to base their responses on their child's behavior over the past month during the interview.</p> <hd id="AN0155062837-18">BIS-CI Inter-Rater Reliability</hd> <p>31 participants (22% of sample) were randomly selected from all three sites for examination of inter-rater reliability of the BIS-CI. IRR was conducted based on the scores from the original interviewer and two independent coders who rated the same selected BIS-CI recording.</p> <hd id="AN0155062837-19">BIS-CI Test–Retest</hd> <p>30 participants (21% of sample) were evaluated using the BIS-CI both at study entry and approximately 2 weeks later via phone to reduce the need for parents to have to return to the clinical site for the completion of one measure.</p> <hd id="AN0155062837-20">Analytical Plan</hd> <p>We used exploratory factor analysis (EFA) to assess the structure of the BIS-CI's seven severity items in the ASD group and to obtain indices of model fit. We chose EFA over confirmatory factor analysis (CFA) for two reasons: (a) this is the initial psychometric study of the BIS-CI and EFA is warranted at this stage, and (b) there are no existing empirical studies on the factor structure of the BI construct in the literature and thus we had no a priori hypothesis about the factor structure of the BIS-CI with which to guide a CFA. Model fit was evaluated using root mean squared error (RMSE), comparative fit index (CFI), Tucker Lewis index (TLI), and standardized root mean squared residual (SRMR). Guidelines to evaluate whether a given model provided a reasonable approximation to the data included: RMSEA and SRMR less than 0.1, and CFI and TLI greater than 0.9 (Browne & Cudeck, [<reflink idref="bib4" id="ref28">4</reflink>]). Factor analysis models were estimated employing mean and variance adjusted weighted least squares (WLSMV) estimators with robust standard errors (DiStefano & Morgan, [<reflink idref="bib8" id="ref29">8</reflink>]; Muthén [<reflink idref="bib26" id="ref30">26</reflink>]; Muthén & Muthén, [<reflink idref="bib28" id="ref31">28</reflink>]; Wirth & Edwards, [<reflink idref="bib42" id="ref32">42</reflink>]). All factor analyses were based on the polychoric correlations between the items. All analyses were conducted using the SAS (SAS Institute Inc. [<reflink idref="bib33" id="ref33">33</reflink>]), MPlus (Muthén & Muthén, [<reflink idref="bib28" id="ref34">28</reflink>]), and FACTOR (Ferrando & Lorenzo-Seva, [<reflink idref="bib9" id="ref35">9</reflink>]; Lorenzo-Seva & Ferrando, [<reflink idref="bib22" id="ref36">22</reflink>]) software packages to verify accuracy. We report results from MPlus below. All factor analysis models were identified by setting latent factor means to 0 and latent factor variances to 1, such that all item factor loadings and residual covariances are estimable under the MPlus "delta" parameterization (Muthén & Muthén [<reflink idref="bib28" id="ref37">28</reflink>]; Muthén & Asparounov, [<reflink idref="bib27" id="ref38">27</reflink>]). Internal consistency of the BIS-CI was measured with Cronbach's alpha, temporal stability of the BIS-CI total score was estimated with intraclass correlations with a mixed effect for absolute agreement under a generalizability theory framework. Validity was assessed using Pearson's correlations by examining the associations between the BIS-CI total score and: (a) BIS total score, RBS-R subscale and total scores (convergent validity), and (b) SRS social-communication subscale scores and SCQ total score (divergent validity). Associations of the BIS-CI total score with demographic (age, sex) and clinical (IQ) variables were assessed with Pearson's correlations. Discriminant validity was assessed by comparing the mean BIS-CI total scores for the ASD and TD groups using a t-test. Given that the ASD and the non ASD groups differed in both sex distribution and parental income level, we also examined the differences between the ASD and the TD groups on BIS-CI Total Score in a multiple regression model that includes these covariates [model: BIS-CI Total Score as dependent variable and Group (ASD, non-ASD), Sex, and Parent Income as independent variables].</p> <hd id="AN0155062837-21">Results</hd> <p>EFA on the inter-item polychoric correlation matrix converged on a one-factor solution, with one dominant eigenvalue that accounted for 49.7% of the total item variance and a magnitude more than four times larger than the second eigenvalue. Models with additional factors did not converge. Fit indices for the single factor solution were: RMSEA = 0.09 (90% CI 0.05–0.14), CFI = 0.96, TLI = 0.94, SRMR = 0.06. In addition to the scree plot (Cattell, [<reflink idref="bib5" id="ref39">5</reflink>]), the Hull method, item and global unidimensional congruence indices, item and global explained common variance indices, and item and global residual absolute loadings indices all suggested a unidimensional solution (Ferrando & Lorenzo-Seva, [<reflink idref="bib9" id="ref40">9</reflink>]; Lorenzo-Seva et al., [<reflink idref="bib23" id="ref41">23</reflink>]). Table 2 shows summary items and factor loadings for the ASD sample. All loadings were statistically significant at p ≤ 0.0001.</p> <p>Table 2 BIS-CI items and factor loadings from EFA model</p> <p> <ephtml> <table frame="hsides" rules="groups"><thead><tr><th align="left"><p>BIS-CI item</p></th><th align="left"><p>Estimated loading</p></th><th align="left"><p>SE</p></th></tr></thead><tbody><tr><td align="left"><p>1. Frequency of occurrence</p></td><td char="." align="char"><p>0.586</p></td><td char="." align="char"><p>0.07</p></td></tr><tr><td align="left"><p>2. Interference with activities at home</p></td><td char="." align="char"><p>0.685</p></td><td char="." align="char"><p>0.05</p></td></tr><tr><td align="left"><p>3. Interference with social relationships</p></td><td char="." align="char"><p>0.740</p></td><td char="." align="char"><p>0.05</p></td></tr><tr><td align="left"><p>4. Interference with activities out of the home</p></td><td char="." align="char"><p>0.576</p></td><td char="." align="char"><p>0.07</p></td></tr><tr><td align="left"><p>5. Accommodation</p></td><td char="." align="char"><p>0.732</p></td><td char="." align="char"><p>0.05</p></td></tr><tr><td align="left"><p>6. Avoidance</p></td><td char="." align="char"><p>0.502</p></td><td char="." align="char"><p>0.07</p></td></tr><tr><td align="left"><p>7. Child distress</p></td><td char="." align="char"><p>0.694</p></td><td char="." align="char"><p>0.05</p></td></tr></tbody></table> </ephtml> </p> <p>Cronbach's alpha is a measure of the degree of homogeneity of responses and provides a lower-bound estimate to the measure's reliability for a specific population. The Cronbach's alpha for the BIS-CI scale was 0.80. Test–retest reliability of the BIS-CI for a subset of 30 ASD participants was ICC = 0.87 (p < 0.001), suggesting high temporal stability over a short time frame. Inter-rater reliability of the BIS-CI total score for a subset of 30 ASD participants was ICC = 0.97 (range = 0.95–0.98), suggesting high reliability.</p> <p>Table 3 shows the relationships between the BIS-CI score and demographic and clinical variables derived from other instruments for the ASD group (Table 3). Correlations of the BIS-CI scores with another measure of behavioral inflexibility (BIS parent report), measures of RRBs (RBSR Total and subscales), and measures of social–communication impairment (SRS Total score and subscales) were all statistically significant (all p's < 0.0001). Correlation of the BIS-CI scores and social-communication impairment as measured by the SCQ was not significant. No significant correlations were found between the BIS-CI and sex or IQ. As the age rage in our ASD sample was broad, we examined the association between BIS-CI Total Score and age in the ASD group. Child age did not significantly predict BIS-CI scores [b = − 0.21, t(<reflink idref="bib225" id="ref42">225</reflink>) = − 1.79, p > 0.05] and only explained a small proportion of variance in BIS scores [R<sups>2</sups> = 0.02, F(<reflink idref="bib1" id="ref43">1</reflink>, 138) = 3.19, p > 0.05; 95% CI − 0/43 to 0.02].</p> <p>Table 3 Correlations of BIS-CI total score with demographic and clinical variables in the ASD group (n = 144)</p> <p> <ephtml> <table frame="hsides" rules="groups"><tbody><tr><td align="left"><p>BIS parent report</p></td><td char="0." align="char"><p>0.65**</p></td></tr><tr><td align="left"><p>RBS-R total</p></td><td char="0." align="char"><p>0.57**</p></td></tr><tr><td align="left"><p>RBS-R stereotypy</p></td><td char="0." align="char"><p>0.39**</p></td></tr><tr><td align="left"><p>RBS-R self-injury</p></td><td char="0." align="char"><p>0.32*</p></td></tr><tr><td align="left"><p>RBS-R compulsive</p></td><td char="0." align="char"><p>0.48**</p></td></tr><tr><td align="left"><p>RBS-R ritualistic</p></td><td char="0." align="char"><p>0.46**</p></td></tr><tr><td align="left"><p>RBS-R sameness</p></td><td char="0." align="char"><p>0.53**</p></td></tr><tr><td align="left"><p>RBS-R restricted</p></td><td char="0." align="char"><p>0.41**</p></td></tr><tr><td align="left"><p>SRS total</p></td><td char="0." align="char"><p>0.47**</p></td></tr><tr><td align="left"><p>SRS—social communication</p></td><td char="0." align="char"><p>0.42**</p></td></tr><tr><td align="left"><p>SRS—social cognition</p></td><td char="0." align="char"><p>0.41**</p></td></tr><tr><td align="left"><p>SCQ—total</p></td><td char="0." align="char"><p>0.25</p></td></tr><tr><td align="left"><p>SBIQ</p></td><td char="0." align="char"><p>0.01</p></td></tr><tr><td align="left"><p>Child age</p></td><td char="." align="char"><p>− 0.18</p></td></tr><tr><td align="left"><p>Child sex</p></td><td char="." align="char"><p>− 0.03</p></td></tr></tbody></table> </ephtml> </p> <p>*p ≤.001; **p ≤.0001</p> <p>The distribution of BIS-CI total scores in the ASD group was normally distributed. Figure 1 shows the distribution of BIS-CI total scores for the ASD sample only. Mean BIS-CI total score for the ASD group (mean = 17.01, SD = 5.39) was significantly different [t(<reflink idref="bib209" id="ref44">209</reflink>) = 16.71, p < 0.0001] from the score for the TD group (mean = 4.45, SD = 4.57). As the groups (ASD, TD) differed in both child sex distribution and in parent income levels, we examined group differences taking these covariates into account. This regression model explained 61.78% of the total variance with a significant Group term (p < 0.0001; 95% CI 11.08–13.84) and nonsignificant effects of either Sex (p = 0.49; 95% CI − 2.06 to 1.00) or Parent Income (p = 0.095; 95% CI − 0.95 to 0.08). The parameter estimate for the Group effect is 12.46, which suggests that those in the ASD group are predicted to be 12.46 units higher on the BIS-CI Total Score than those in the non-ASD group holding the additional covariates constant.</p> <p>Graph: Fig. 1 Distribution of BIS-CI sum scores</p> <hd id="AN0155062837-22">Discussion</hd> <p>We developed a structured clinical interview version of the parent report Behavioral Inflexibility Scale as a dimensional measure of the functional impact of behavioral inflexibility in children with ASD. Psychometric evaluation of the data derived from the interview instrument in a sample of 144 children with ASD indicated: (<reflink idref="bib1" id="ref45">1</reflink>) that the instrument provides a unidimensional measure of inflexibility, (<reflink idref="bib2" id="ref46">2</reflink>) that the instrument yields an approximately normal distribution for the overall score in a large sample of children with ASD, (<reflink idref="bib3" id="ref47">3</reflink>) that the instrument demonstrated acceptable levels of internal consistency, and excellent levels of test–retest stability and inter-rater reliability in this ASD sample, (<reflink idref="bib4" id="ref48">4</reflink>) that the instrument has strong convergent validity with parent report measures of behavioral inflexibility and of RRBs, and (<reflink idref="bib5" id="ref49">5</reflink>) that scores on the instrument appear to vary independently of child age, sex, and cognitive ability in this sample. Feasibility (low user burden), unidimensionality (results in one overall severity score), and excellent stability are critical features of treatment outcome measures, and so these findings suggest that the BIS-CI is a reasonable candidate as treatment outcome for children with ASD. High temporal stability is particularly important in measuring change as it indicates that changes in scores are likely not due to error variance (Kraemer, [<reflink idref="bib17" id="ref50">17</reflink>]).</p> <p>A key aspect of a behavioral inflexibility model of functional impact associated with RRBs is that inflexibility is associated with all types of discrete repetitive behaviors that occur in persons with ASD as opposed to being linked in a more limited way to a specific type or form of repetitive behavior. Several previous phenomenological studies of RRBs in ASD have shown that there is a variety of discrete types of RRBs associated with ASD including "lower order" forms such as stereotyped movements or stereotyped use of objects and "higher order" forms such as rituals, routines, insistence on sameness, and circumscribed interests (Bodfish et al., [<reflink idref="bib3" id="ref51">3</reflink>]; Honey et al., [<reflink idref="bib12" id="ref52">12</reflink>], [<reflink idref="bib13" id="ref53">13</reflink>]; Wolff et al., [<reflink idref="bib43" id="ref54">43</reflink>]). For example, a consistently replicated finding in this area is that factor analyses of data from various repetitive behavior measures in large samples of children with ASD yields evidence for multiple factors in this domain (Bishop et al., [<reflink idref="bib2" id="ref55">2</reflink>]; Cuccaro et al., [<reflink idref="bib7" id="ref56">7</reflink>]; Hus et al., [<reflink idref="bib14" id="ref57">14</reflink>]; Lam et al., [<reflink idref="bib18" id="ref58">18</reflink>]; Szatmari et al., [<reflink idref="bib36" id="ref59">36</reflink>]). In the present study we found that inflexibility as measured by the BIS-CI was significantly correlated with each subscale of the RBSR (stereotyped movements, repetitive self-injury, compulsive behaviors, ritualistic behaviors, sameness behaviors, and restricted interests), as opposed to being correlated with just one or a few RRB subscales. However, the correlations were in the moderate range (0.32–0.57), which importantly suggests that the BIS-CI and RBS-R are not redundant measures. Still this finding supports the hypothesis that inflexibility can be associated with the full variety of discrete types of RRB in ASD. This also suggests that an intervention model that focuses on identifying the subset of persons with ASD whose RRBs are producing functional impairments is valid, as opposed to approaches based on identifying and treating individuals with RRBs more broadly. A strength of an instrument such as the BIS-CI is that it can be applied within an intervention model that focuses on identifying the subset of RRB cases associated with functional impact.</p> <p>Our finding that a variety of types of RRBs can be associated with inflexibility is consistent with both clinical experience and findings from previous studies that have examined the clinical phenomenology of RRBs in ASD. For example, stereotyped movements like hand-flapping or body rocking are often benign and therefore not specifically targeted for intervention. However, in a subset of cases, these behaviors are associated with a significant degree of inflexibility as interruption of these actions can lead to emotional outbursts and more severe behavior challenges (e.g. aggression, self-injury) that do become the target of treatment (Richards et al., [<reflink idref="bib29" id="ref60">29</reflink>]; Rojahn et al., [<reflink idref="bib31" id="ref61">31</reflink>]) Likewise, "higher-order" RRBs, such as insistence on sameness or circumscribed interests have been reported to be adaptive or a source of resilience (e.g. routines as a way to deal with unpredictability, intense interests as a way to make friends or develop vocational skills). Yet, in some circumstances the opposite is true and rigidly held routines or overly narrow interests are associated with a significant degree of inflexibility that can lead to avoidance, restriction of experience, and stress within the larger family, school, or community context (Gotham et al., [<reflink idref="bib10" id="ref62">10</reflink>]; Turner-Brown et al., [<reflink idref="bib38" id="ref63">38</reflink>]; Uljarević et al., [<reflink idref="bib40" id="ref64">40</reflink>]). As these clinical scenarios show, there is a subset of individuals with ASD whose RRBs are associated with a clinically significant degree of functional impairment.</p> <p>In addition to being significantly correlated with the subscales of the RBSR, we also found that BIS-CI scores in this sample of children with ASD were significantly associated with social-communication impairments as measured by the SRS (although not the SCQ). This finding suggests that inflexibility as a form of functional impairment is not limited to RRBs in ASD but may also contribute to other symptom domains in ASD. For example, children with ASD may be inflexible in their social interactions (e.g., scripting interactions, or dominating the topic of conversation), play (e.g., perseverative patterns of play), and even activities of daily living such as mealtimes (e.g., picky eaters), dressing (e.g., wearing only certain clothes), and transition or travel (e.g., insisting on certain routes or ways or transitioning). Indeed, considering these broader aspects of inflexibility in the context of ASD begins to blur the line between behaviors counted as RRBs and behaviors counted as social-communication impairments (e.g., is perseverative play with a peer a ritual or evidence of impaired social reciprocity?). Importantly, this broader role that inflexibility may play in the development of ASD was implicit in the earliest formulations of autism as Kanner ([<reflink idref="bib16" id="ref65">16</reflink>]), Asperger ([<reflink idref="bib1" id="ref66">1</reflink>]), and Wing ([<reflink idref="bib41" id="ref67">41</reflink>]) all specifically referred to inflexibility as being a defining feature of the condition in their seminal works on autism.</p> <p>We found that the BIS-CI was capable of distinguishing inflexibility in the context of typical development from inflexibility in ASD. The degree of separation between the ASD and TD groups in the present study is striking—the mean BIS-CI total score of the ASD group was over three SDs greater than the mean score of the TD group. This suggests that inflexibility in children with ASD, at least as it is measured by the BIS-CI, is not simply a greater degree of inflexibility observed in the general population but may be qualitatively different especially in terms of its impact on daily functioning and family life. This is remarkable given that inflexibility is a common dimension of temperament in typically developing children (Thomas & Chess, [<reflink idref="bib37" id="ref68">37</reflink>]). This provides further support for the idea that the BIS-CI is useful for identifying and quantifying true functional impairment and therefore may be a clinically significant target for intervention. At this point however, we do not know if behavioral inflexibility as measured by the BIS-CI is a shared feature of other neuropsychiatric conditions such as ADHD, depression, OCD, or anxiety disorders. Given that persons with ASD are at increased risk for developing these comorbid conditions, future research is warranted that examines the relation of inflexibility to comorbidities within samples of ASD, and also examines the similarities and differences in behavioral inflexibility between ASD and other psychiatric groups.</p> <p>The findings of this study must be considered in light of its methodological limitations. The large and diverse sample drawn from multiple sites is a strength in terms of the potential for the findings from this sample to generalize to ASD more broadly. On the other hand, a larger sample size would have allowed for additional analyses. For instance, it may very well be the case that development moderates scores on the BIS-CI and future studies should examine behavioral inflexibility in more narrowly defined age groups. Unlike parent report measures or objective measures, the reliability of a clinical interview such as the BIS-CI relies more heavily on the training process and on interviewer experience with the instrument. This can make interview measures more susceptible to factors like rater bias, rater drift, and local differences in how the interview is actually conducted and scored. Our inter-rater and test–retest results suggest that such effects while likely present were not large at least in this sample. These potential sources of error inherent in structured clinical interviews are important to consider, however, and this makes independent replication of the psychometric characteristics of the BIS-CI with a larger and more representative sample imperative and an important step to be accomplished next. We designed the BIS-CI training process to be simple and the structured interview itself to be brief (approximately 10–15 min) in an effort to mitigate some of these sources of error. We are making both the BIS-CI and the training manual freely and publicly available through the project website in hopes that this will support replication studies. Another weakness in our approach to examining convergent and divergent validity is that all measures used were parent-report measures taken at the same time point and thus shared measurement variance is contributing to our pattern of cross-item correlations to an unknown degree. To a small extent, the pattern of differential correlations found within our dataset diminishes this concern as parents were apparently able to discriminate different aspects of their child's behavior using the instruments included; although, future studies need to examine the convergent and divergent validity of the BIS-CI using a multi-method approach (e.g., parent-report, clinical interview, direct observation).</p> <p>In conclusion, the findings of this study suggest that the BIS-CI is a reliable, stable, valid and unidimensional measure of functional impairment associated with RRBs in children with ASD. It has minimal burden in terms of data collection time and as it is a structured clinical interview it is capable of being blinded in intervention studies. If these characteristics are replicated in an independent sample, then the BIS-CI would be a reasonable candidate as an outcome measure in ASD.</p> <hd id="AN0155062837-23">Acknowledgments</hd> <p>Research reported in this publication was supported by the Eunice Kennedy Shriver National Institute of Child Health and Human Development of the National Institutes of Health under Award Number R01HD082127 and P30-HD03110. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health. We also would like to acknowledge and sincerely thank all of the children and youth with ASD and their families who participated in this study.</p> <hd id="AN0155062837-24">Author Contributions</hd> <p>Significant contributions to study conceptualization and design were made by Brian Boyd, James Bodfish and Luc Lecavalier. The first draft of the manuscript was written by James Bodfish, Brian Boyd, Luc Lecavalier and Clare Harrop, with all other authors providing edits and feedback. Data collection and material preparation for the manuscript were made by Clare Harrop, Aaron Dallman, Sahana Nagabhushan Kalburgi, and Jill Hollway. Data analysis was performed by Richard Faldowski.</p> <hd id="AN0155062837-25">Funding</hd> <p>The funding was supported by Eunice Kennedy Shriver National Institute of Child Health and Human Development (Grant Nos.: R01HD082127 and P30-HD03110).</p> <hd id="AN0155062837-26">Publisher's Note</hd> <p>Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.</p> <ref id="AN0155062837-27"> <title> References </title> <blist> <bibl id="bib1" idref="ref12" type="bt">1</bibl> <bibtext> Asperger, H. (1944). Die "autistischenPsychopathen"im Kindesalter. 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  Label: Title
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  Data: Measuring the Functional Impact of Behavioral Inflexibility in Children with Autism Using the Behavioral Inflexibility Scale: Clinical Interview (BIS-CI)
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  Label: Language
  Group: Lang
  Data: English
– Name: Author
  Label: Authors
  Group: Au
  Data: <searchLink fieldCode="AR" term="%22Bodfish%2C+James+W%2E%22">Bodfish, James W.</searchLink><br /><searchLink fieldCode="AR" term="%22Lecavalier%2C+Luc%22">Lecavalier, Luc</searchLink><br /><searchLink fieldCode="AR" term="%22Harrop%2C+Clare%22">Harrop, Clare</searchLink><br /><searchLink fieldCode="AR" term="%22Dallman%2C+Aaron%22">Dallman, Aaron</searchLink><br /><searchLink fieldCode="AR" term="%22Kalburgi%2C+Sahana+Nagabhushan%22">Kalburgi, Sahana Nagabhushan</searchLink><br /><searchLink fieldCode="AR" term="%22Hollway%2C+Jill%22">Hollway, Jill</searchLink><br /><searchLink fieldCode="AR" term="%22Faldowski%2C+Richard%22">Faldowski, Richard</searchLink><br /><searchLink fieldCode="AR" term="%22Boyd%2C+Brian+A%2E%22">Boyd, Brian A.</searchLink> (ORCID <externalLink term="http://orcid.org/0000-0002-6482-9577">0000-0002-6482-9577</externalLink>)
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  Data: <searchLink fieldCode="SO" term="%22Journal+of+Autism+and+Developmental+Disorders%22"><i>Journal of Autism and Developmental Disorders</i></searchLink>. Feb 2022 52(2):782-790.
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  Data: Springer. Available from: Springer Nature. One New York Plaza, Suite 4600, New York, NY 10004. Tel: 800-777-4643; Tel: 212-460-1500; Fax: 212-460-1700; e-mail: customerservice@springernature.com; Web site: https://link.springer.com/
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  Label: Peer Reviewed
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  Data: Y
– Name: Pages
  Label: Page Count
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  Data: 9
– Name: DatePubCY
  Label: Publication Date
  Group: Date
  Data: 2022
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  Label: Sponsoring Agency
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  Data: Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD) (DHHS/NIH)
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  Label: Contract Number
  Group: NumCntrct
  Data: R01HD082127<br />P30HD03110
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  Label: Document Type
  Group: TypDoc
  Data: Journal Articles<br />Reports - Research
– Name: Subject
  Label: Descriptors
  Group: Su
  Data: <searchLink fieldCode="DE" term="%22Children%22">Children</searchLink><br /><searchLink fieldCode="DE" term="%22Autism%22">Autism</searchLink><br /><searchLink fieldCode="DE" term="%22Pervasive+Developmental+Disorders%22">Pervasive Developmental Disorders</searchLink><br /><searchLink fieldCode="DE" term="%22Child+Behavior%22">Child Behavior</searchLink><br /><searchLink fieldCode="DE" term="%22Behavior+Problems%22">Behavior Problems</searchLink><br /><searchLink fieldCode="DE" term="%22Parents%22">Parents</searchLink><br /><searchLink fieldCode="DE" term="%22Test+Validity%22">Test Validity</searchLink><br /><searchLink fieldCode="DE" term="%22Interviews%22">Interviews</searchLink><br /><searchLink fieldCode="DE" term="%22Test+Reliability%22">Test Reliability</searchLink><br /><searchLink fieldCode="DE" term="%22Interrater+Reliability%22">Interrater Reliability</searchLink>
– Name: DOI
  Label: DOI
  Group: ID
  Data: 10.1007/s10803-021-04984-z
– Name: ISSN
  Label: ISSN
  Group: ISSN
  Data: 0162-3257
– Name: Abstract
  Label: Abstract
  Group: Ab
  Data: For individuals with autism spectrum disorder (ASD), behavioral inflexibility can affect multiple domains of functioning and family life. The objective of this study was to develop and validate a clinical interview version of the Behavioral Inflexibility Scale. Trained interviewers conducted interviews with parents of 144 children with ASD and 70 typically developing children (ages: 3-17 years). Using exploratory factor analysis, the Behavioral Inflexibility Scale-Clinical Interview (BIS-CI) was found to be unidimensional. Reliability data indicated the measure was internally consistent ([alpha] = 0.80), achieved excellent inter-rater reliability (ICC = 0.97) and test-retest reliability (ICC = 0.87). These findings demonstrate that the BIS-CI is a reliable and valid measure to determine the functional impact of behavioral inflexibility.
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  Group: Date
  Data: 2022
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  Label: Accession Number
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  Data: EJ1327284
PLink https://search.ebscohost.com/login.aspx?direct=true&site=eds-live&db=eric&AN=EJ1327284
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        Value: 10.1007/s10803-021-04984-z
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      – Text: English
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        PageCount: 9
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      – SubjectFull: Children
        Type: general
      – SubjectFull: Autism
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      – SubjectFull: Pervasive Developmental Disorders
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      – SubjectFull: Child Behavior
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      – TitleFull: Measuring the Functional Impact of Behavioral Inflexibility in Children with Autism Using the Behavioral Inflexibility Scale: Clinical Interview (BIS-CI)
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