A Meta-Analysis of Treatment for Self-Injurious Behavior in Children and Adolescents With Intellectual and Developmental Disabilities.

Self-injurious behavior (SIB) among children and youth with developmental disabilities has not diminished in prevalence despite the availability of effective interventions, and the impact on quality of life for people and their families is devastating. The current meta-analysis reviews SIB intervention research between 2011 and 2021 using single-case experimental designs with children and youth up to 21 years old and provides a quantitative synthesis of data from high-quality studies including moderator analyses to determine effects of participant and study characteristics on intervention outcomes. Encouraging findings include a high level of effectiveness across studies in the decrease of SIB (Tau-U = -0.90) and increase of positive behavior (Tau-U = 0.73), as well as an increase in studies (relative to prior reviews) reporting intervention fidelity, generalization, maintenance, and social validity. However, our findings shed limited light on potential moderating variables in the development of interventions for children and youth who exhibit SIB. Of the potential moderators of intervention effects, only implementer (researcher/therapist vs. parent/caregiver) and setting (clinic vs. home) were significantly associated with improved outcomes. We discuss the need for more robust involvement of natural communities of implementers in SIB intervention research to better equip them to effectively and sustainably meet the needs of people they care for. We also discuss the importance of creating systems enabling broad access for children with SIB to effective interventions in service of reducing burden for people, families, and society over time.

Teaching Requesting to Individuals with Rett Syndrome Using Alternative Augmentative Communication (AAC) Through Caregiver Coaching via Telehealth.

Rett syndrome is a severe neurodevelopmental disorder that results in both motor and language skill regression with a wide range of severity in symptom presentation. Communication intervention may be particularly challenging for this population due to the decline in speech, motor skills, and motor planning difficulties that characterize the disorder (Townend et al., 2020), often resulting in the need for augmentative and alternative communication (AAC) technology. Very limited research has evaluated communication interventions for individuals with Rett syndrome and even fewer have targeted expressive communication, an important skill required for improved autonomy and quality of life (Sigafoos et al., 2009; Townend et al., 2020). The current study sought to systematically replicate the Simacek et al. (2017) mand training procedures to teach three girls with Rett Syndrome to use AAC to make requests through caregiver coaching by researchers via telehealth. Results suggest that mand training was successful in increasing AAC use for all three participants. Barriers to intervention for this population and implications of results for future research and clinical practice are discussed.

Predicting self-injurious behavior at age three among infant siblings of children with autism.

Existing research suggests that self-injurious behavior (SIB) is a relatively common interfering behavior that can occur across the lifespan of individuals with autism spectrum disorder (ASD). We previously reported that SIB or proto-injurious SIB at 12 months was related to increased risk of SIB at 24 months among a preschool sample of children with a high familial likelihood for ASD (Dimian et al., 2017). In the present study, we extend these findings, examine SIB occurrence, and associated potential risk factors at 36 months. The present sample included 149 infants with an older sibling with ASD (65.8% male) who completed assessments at ages 12, 24, and 36 months. Descriptive analyses and binary logistic regression models were utilized. SIB was more prevalent among those children who received a diagnosis of ASD. Logistic regression indicated that presence of SIB, stereotypy, hyper- and hypo- sensory responsivity, and lower intellectual functioning at age 12 months significantly predicted the occurrence of SIB at 36 months. These findings have implications for understanding developmental processes culminating in persistent SIB and may inform prevention programming.

Statewide county-level autism spectrum disorder prevalence estimates-seven U.S. states, 2018.

PURPOSE: Autism spectrum disorder (ASD) prevalence information is necessary for identifying community needs such as addressing disparities in identification and services. METHODS: Seven Autism and Developmental Disabilities Monitoring (ADDM) Network sites participated in a pilot project to link statewide health and education data to generate statewide and county-level prevalence estimates for a broader age range for their states for the first time. RESULTS: Statewide prevalence of ASD for ages 3-21 years in 2018 ranged from 1.5% in Tennessee and Wisconsin to 2.3% in Arizona. The median county-level prevalence of ASD was 1.4% of residents ages 3-21 years. More boys than girls had ASD at all sites, and prevalence was lower among non-Hispanic Black, Hispanic, Asian/Pacific Islander, and American Indian/Alaska Native residents compared to non-Hispanic White residents at most sites. ASD prevalence estimates for children aged 8 years were similar to 2018 ADDM Network estimates that used record review to provide more in-depth information, but showed greater variation for children aged 4 years. CONCLUSIONS: Linkage of statewide data sets provides less detailed but actionable local information when more resource-intensive methods are not possible.

Restricted and repetitive behavior in children with autism during the first three years of life: A systematic review.

Restricted and repetitive behavior (RRB) is a core diagnostic feature of autism spectrum disorder (ASD). Previous research shows that RRB is prevalent early in life and observed in neurotypical development as well. Less is known, however, about early RRB patterns, developmental trajectories, and the relation to outcomes for autistic children. The purpose of this systematic review was to synthesize findings from studies examining RRB in autistic children from birth through age 3. A detailed protocol was designed a priori based on PRISMA guidelines for systematic reviews. From the published literature, 41 peer reviewed journal articles were identified and included in this review. Our synthesis of the literature suggests that differences in RRB are evident prior to age 2 in children with or who go onto be diagnosed with autism. These differences were evident for both frequency and intensity of RRB across multiple topographies. There were mixed results regarding functional outcomes associated with early RRB, such as cognitive and adaptive behavior, though relations appeared to become stronger as children aged beyond toddlerhood. Notably, level of RRB appears unrelated to autism severity in young autistic children. A wide range of RRB have been reported to be elevated in autistic children during the first years of life, including repetitive motor behaviors, atypical sensory behaviors, insistence on sameness (IS), and self-injurious behaviors (SIBs). In contrast to studies of older children, RRB in very young autistic children do not appear to be associated with functional outcomes but may be valuable to include in early screening efforts. Systematic review registration: https://osf.io/huzf3, unique identifier: doi: 10.17605/OSF.IO/HUZF3.

A systematic review of risk for the development and persistence of self-injurious behavior in intellectual and developmental disabilities.

Self-injurious behavior (SIB) by individuals with intellectual and developmental disabilities including autism (I/DD) is among the most clinically disturbing, socially costly, and scientifically challenging behavior disorders. Forty years of clinical research has produced a knowledge base supporting idiographic behavioral assessment and treatment approaches. Despite the treatment progress, from a public health and population perspective, we argue it is less clear that we have reduced the disorder's burden. The developmental course of the disorder is mostly unknown and empirically informed population-level models of risk are absent. In this review, we systematically examined the published scientific literature specific to risk for SIB in the I/DD population. We reviewed study methodology in detail intentionally informed by an epidemiological perspective with a set of questions intended to test the quality of the inferences about risk. Results are discussed in terms of conceptual, methodological, and translational issues with respect to what needs to be done to create credible and useful clinical models for SIB risk in the I/DD population.

Current Trends in Telehealth Applications to Deliver Social Communication Interventions for Young Children with or at Risk for Autism Spectrum Disorder.

PURPOSE OF REVIEW: Early, intensive, and high-quality interventions can often improve social communication outcomes for children with autism spectrum disorder (ASD). Many children experience barriers to intervention, resulting in significant delays to intervention onset or missed opportunities for intervention altogether. With constant advances in technology, the field is experiencing a rapid increase in investigation of telehealth applications to intervention delivery. This article highlights the current trends in social communication intervention via telehealth used in early intervention practices for children with ASD over the past 5 years, including a brief review of studies (from 2014 to January 2020) and our team's experiences in this area. RECENT FINDINGS: Based on our experience and the 22 studies we identified in this area, we describe the current trends in telehealth applications used and how interventions were delivered. We also provide recommendations, limitations, and future directions on this topic. SUMMARY: Telehealth offers innovative intervention delivery options by increasing intervention access, overcoming barriers such as geography and costs of service delivery for young children with ASD.

Delay to Early Intensive Behavioral Intervention and Educational Outcomes for a Medicaid-Enrolled Cohort of Children with Autism.

Increased prevalence of autism spectrum disorder (ASD) has underscored the need for early intervention services. Early Intensive Behavioral Intervention (EIBI) is among the most common evidence-based approaches, however, stakeholders report significant waitlists. The effects of these delays to intervention are unknown. The purpose of this study was to evaluate the effects of delay to EIBI for preschool aged children with ASD on later educational outcomes. Medicaid records from Minnesota (2008-2010) were used to evaluate a cohort diagnosed with ASD and their later educational outcomes from 2010 to 2014 (n = 667) using generalized estimating equations. Approximately 70% of children experienced a delay to EIBI and children that experienced less delay and started EIBI at a younger age had better educational outcomes.

Preliminary Evidence That Resting State Heart Rate Variability Predicts Reactivity to Tactile Stimuli in Rett Syndrome.

Patients with Rett syndrome may manifest altered pain perception/experience and are vulnerable to conditions associated with chronic pain. Pain response is difficult to measure, however, because of severe communicative impairment. There is also documented autonomic dysfunction, including decreased heart rate variability. Given the relation between pain and the autonomic nervous system, we tested the feasibility of using resting heart rate variability to predict nonverbal pain/discomfort behavior during a standardized modified quantitative sensory test in Rett syndrome. All stimulus applications resulted in increased behavioral reactivity compared to baseline, with repeated von Frey significantly greater than all other stimuli. Resting heart rate variability predicted behavioral reactivity to repeated von Frey. These preliminary findings provide feasibility evidence for an integrated autonomic-sensory measurement approach and are consistent at a construct level with preclinical evidence in Rett syndrome. Further work is needed to determine how heart rate variability changes during stimulus application.

A longitudinal study of parent-reported sensory responsiveness in toddlers at-risk for autism.

BACKGROUND: Atypical sensory responsivity and sensory interests are now included in the DSM 5 diagnostic criteria for autism spectrum disorder (ASD) under the broad domain of restricted and repetitive behavior (RRB). However, relatively little is known about the emergence of sensory-related features and their relation to conventionally defined RRB in the first years of life. METHODS: Prospective, longitudinal parent-report data using the Sensory Experiences Questionnaire (SEQ) were collected for 331 high-risk toddlers (74 of whom met diagnostic criteria for ASD at age 2) and 135 low-risk controls. Longitudinal profiles for SEQ scores were compared between groups across ages 12-24 months. Associations between SEQ measures and measures of RRB subtypes (based on the Repetitive Behavior Scale, Revised) were also examined. RESULTS: Longitudinal profiles for all SEQ scores significantly differed between groups. SEQ scores were elevated for the ASD group from age 12 months, with differences becoming more pronounced across the 12-24 month interval. At both 12 and 24 months, most measures derived from the SEQ were significantly associated with all subtypes of RRB. CONCLUSIONS: These findings suggest that differences in sensory responsivity may be evident in high-risk infants later diagnosed with ASD in early toddlerhood, and that the magnitude of these differences increases over the second year of life. The high degree of association between SEQ scores and RRB supports the conceptual alignment of these features but also raises questions as to explanatory mechanisms.

Agreement Between Structured Descriptive Assessments and Functional Analyses Conducted Over a Telehealth System.

This study examined whether experimental functional analyses (FAs) conducted by parents at home with coaching via telehealth would produce differentiated results, and compared these results to the functions identified from structured descriptive assessments (SDAs) also conducted by parents at home via telehealth. Four boys between the ages of 4- and 8-years old with intellectual and developmental disabilities and their parents participated. All assessments were conducted in the children's homes with their parents serving as intervention agents and with coaching from remote behavior therapists using videoconferencing technology. Parent-implemented FAs produced differentiated results for all 4 children in the study. Overall, analyzing antecedent-behavior (A-B) and behavior-consequence (B-C) relations from the SDA videos identified only half of the functions identified by the FAs. For children whose SDA results were differentiated, analyzing A-B relations correctly identified 4 of 5 functions. Analyzing B-C relations correctly identified 5 of 6 functions identified by the experimental FA, but overidentified attention for all children. Implications for conducting functional analyses and interpreting structured descriptive assessment via telehealth are discussed.

Promoting Conditional Use of Communication Skills for Learners With Complex Communication Needs: A Tutorial.

Purpose: Conditional use of communication skills refers to the ability of a learner to appropriately generalize and discriminate when, where, and how to communicate based on constant variation and shifts in environmental cues. Method: We describe discrimination and generalization challenges encountered by learners with complex communication needs and ways in which these challenges are fostered through traditional communication intervention programming. We address arrangements in instruction that maximize the probability of learners acquiring the conditional use of new vocabulary and the modest instructional technology implemented when planning for generalization. Results: We propose establishing well-discriminated and generalized use of new vocabulary items through the application of a general case instruction framework to communication intervention programming. Conclusion: We provide intervention methodology, including intervention steps for general case instruction, a plethora of functional examples, and graphic displays to assess and intervene to promote conditional use of communication skills for learners with complex communication needs.

Potential Risk Factors for the Development of Self-Injurious Behavior among Infants at Risk for Autism Spectrum Disorder.

Prevalence of self-injurious behavior (SIB) is as high as 50% among children with autism spectrum disorder (ASD). Identification of risk factors for the development of SIB is critical to early intervention and prevention. However, there is little empirical research utilizing a prospective design to identify early risk factors for SIB. The purpose of this study was to evaluate behavioral characteristics predicting SIB at age 2 years among 235 infants at high familial risk for ASD. Logistic regression results indicated that presence of SIB or proto-SIB and lower developmental functioning at age 12 months significantly predicted SIB at 24 months. A pattern of persistent SIB over this period was associated with a diagnosis of autism and poorer cognitive and adaptive outcomes.

Communication Intervention for Young Children with Severe Neurodevelopmental Disabilities Via Telehealth.

Young children with neurodevelopmental disorders such as autism spectrum disorders (ASD) and Rett syndrome often experience severe communication impairments. This study examined the efficacy of parent-implemented communication assessment and intervention with remote coaching via telehealth on the acquisition of early communication skills of three young children with ASD (2) and Rett syndrome (1). Efficacy of the intervention was evaluated using single-case experimental designs. First, functional assessment was used to identify idiosyncratic/potentially communicative responses and contexts for each child. Next, parents implemented functional communication training (FCT). All of the children acquired the targeted communication responses. The findings support the efficacy of telehealth as a service delivery model to coach parents on intervention strategies for their children's early communication skills.

A Direct Comparison of Self-Injurious and Stereotyped Motor Behavior Between Preschool-Aged Children With and Without Developmental Delays.

OBJECTIVE: To compare the prevalence of self-injurious behavior (SIB) and stereotyped motor behavior (STY) of preschool-aged children with developmental delays (DD group) and their peers without developmental delays (TD group) using a standardized caregiver report scale. METHODS: The Repetitive Behavior Scale-Revised was completed by caregivers of children with developmental delays and their peers without developmental delays. Frequency of occurrence and severity ratings for SIB and STY were compared between groups. RESULTS: SIB and STY were reported more often and at a greater level of severity in the DD group. Older chronological age was associated with more severe STY in the DD group but not the TD group. Gender was not related to STY or SIB for either group. CONCLUSIONS: Differences in STY and SIB were evident between preschoolers with and without DD. Findings are discussed from developmental and behavioral psychology perspectives regarding the expression of repetitive behavior in developmentally at-risk pediatric populations.

Infrared Thermal Analysis and Individual Differences in Skin Temperature Asymmetry in Rett Syndrome.

PURPOSE: We evaluated the feasibility of using a portable infrared thermal camera to quantify the degree of thermal dysregulation (cold hands/feet) and test for naturally occurring within-patient skin temperature asymmetry in Rett syndrome. PROCEDURES: Infrared thermal images were acquired passively from 15 patients (mean age = 13.7 years, range 4-47) with clinical diagnoses of Rett. Images were acquired using a FLIR T400 infrared thermal camera (still images recorded at 5 Hz, resolution of 320 × 240 pixels, thermal sensitivity = 0.05 °C; capture session lasted approximately 3 minutes). The infrared thermal camera was orthogonal to the body part (hands, feet) and positioned approximately 1 meter from the skin's surface. RESULTS: There were large intraindividual left/right differences in temperature. Seven (47%) and eight (53%) patients had statistically significant (P <0.05) left/right asymmetries between hands (mean difference = 0.87 °C, standard deviation = 1.21) and feet (mean difference = 1.73 °C, standard deviation = 3.03), respectively. Coders were reliable (intraclass correlations 0.97-0.99) on temperatures and selection of anatomical regions of interest. CONCLUSIONS: The degree of thermal asymmetry may reflect prolonged activity of the sympathetic nervous system and individual differences in sympathetic regulation. As clinical trials emerge and endpoints are considered, portable infrared thermal camera may provide one noninvasive means of evaluating changes in sympathetic regulation.

A Case-controlled Investigation of Pain Experience and Sensory Function in Neuronal Ceroid Lipofuscinosis.

OBJECTIVES: This case-control study explored pain experience and expression among individuals with neuronal ceroid lipofuscinosis (NCL) through parental report, tactile sensory testing, and infrared thermography (IRT). MATERIALS AND METHODS: Participants with NCL (n=8; M=14.8 y) and their unaffected siblings (n=8; M=23.5 y) were characterized in terms of pain response to a brief tactile sensory test (light touch, Von Frey monofilament). During sensory testing, behavioral expression was measured using the Battens Observational Pain Scale and infrared thermography (IRT) was used to quantify changes in skin/eye temperature. RESULTS: Children with NCL experienced pain frequently and from multiple sources that negatively impacted their lives. Children with NCL were reactive to the sensory testing as indexed by significant increased IRT temperature change (P<0.001). Across combined sensory conditions, individuals with NCL were significantly more reactive (Battens Observational Pain Scale total score) to sensory testing compared with siblings (P<0.05). Similarly, IRT difference scores between sensory conditions revealed a significant increase in temperature for individuals with NCL compared with siblings (P<0.001). DISCUSSION: Ongoing reported pain was a problem for the children with NCL in this sample. Increased pain expression during the repeated Von Frey filament suggests that the pathophysiology of the ongoing pain may be centrally mediated.

Functional communication training in rett syndrome: a preliminary study.

Rett syndrome (RTT) is associated with a range of serious neurodevelopmental consequences including severe communicative impairments. Currently, no evidence-based communication interventions exist for the population ( Sigafoos et al., 2009 ). The purpose of the current study was to examine the effectiveness of functional assessment (FA) and functional communication training (FCT) methods for teaching 3 individuals (ages 15-47 years) with classic RTT novel communicative behaviors. Using single-case experimental designs, functional reinforcers were identified (FA) and each participant quickly learned to activate a voice-output switch to obtain a reinforcer (FCT). These results suggest that individuals with classic RTT can learn novel communicative responses, which has important implications for future intervention research.

Effects of Positive and Negative Reinforcement in a Concurrent Operants Arrangement on Compliance and Problem Behavior / Efectos del Reforzamiento Positivo y Negativo en un Arreglo de Operantes Concurrentes sobre la Obediencia y la Conducta Problemática

Functional analysis (Iwata, Dorsey, Slifer, Bauman, & Richman, 1982/1994) is a robust approach to identifying function-based interventions for problem behavior, including self-injury, aggression, and destruction. Such interventions, however, may be difficult for untrained caregivers to implement with fidelity in natural environments. Further research is needed to identify simple antecedent strategies for promoting appropriate behavior among children with significant problem behavior. The purpose of the current study was to utilize a concurrent schedules arrangement to identify conditions under which two children with autism spectrum disorder (ASD) and developmental delays who engaged in problem behaviors would choose to complete academic tasks to earn access to preferred items. In both cases, problem behaviors were shown to be sensitive to reinforcement in the forms of escape from task demands and access to preferred items. A concurrent operant arrangement in which the participants could choose to complete work tasks to earn access to preferred activities, or to take a break without demands or preferred items, was implemented. The schedule requirements in the demand component were systematically increased across opportunities, while the amount and type of reinforcement was kept constant. The results show, at the lowest levels of task demands, both participants allocated more opportunities to the work option. At higher levels, however, both participants allocated a majority of their choices to the break option. Despite the absence of preferred items in the break component, no instances of problem behavior were observed following selection of the break option. This indicates that this type of analysis could be used to identify conditions for compliance among individuals who engage in escape- or multiply-maintained problem behaviors, without the need to provoke or reinforce problem behavior. Limitations of the current study and recommendations for future research are discussed.

El análisis funcional (Iwata, Dorsey, Slifer, Bauman, & Richman, 1982/1994) es una aproximación robusta para identificar intervenciones basadas en la función para la conducta problemática, incluyendo la autolesión, agresión y destrucción. Tales intervenciones, no obstante, pueden ser difíciles de implementar fidedignamente en escenarios naturales por cuidadores no entrenados. Se requiere más investigación para identificar estrategias antecedentes simples para promover la conducta apropiada entre niños con problemas significativos de conducta. El propósito del presente estudio fue utilizar un arreglo de programas concurrentes para identificar las condiciones bajo las cuales dos niños con trastorno del espectro autista (ASD, por sus siglas en inglés) y retraso en el desarrollo que emitían conducta problemática escogerían completar tareas académicas para ganar acceso a ítems preferidos. En ambos casos, las conductas problemáticas fueron sensibles al reforzamiento consistente en escape de las demandas de la tarea y en acceso a ítems preferidos. Se implementó un arreglo de programas concurrentes en el que los participantes podían escoger entre completar una tarea académica para ganar acceso a ítems preferidos o tomar un descanso sin demandas y sin ítems preferidos. Los requisitos del programa en el componente de demanda fueron incrementados sistemáticamente a través de las oportunidades de elección, mientras que el tipo y cantidad de reforzamiento se mantuvo constante. Los resultados mostraron que en el nivel más bajo de demandas, ambos participantes prefirieron la opción de trabajo. A niveles de demanda más altos, no obstante, ambos participantes eligieron la opción de tomar un descanso. A pesar de la ausencia de ítems preferidos en el componente de descanso, no se observaron instancias de conducta problemática después de esta opción. Esto indicó que este tipo de análisis puede ser usado para identificar condiciones que conducen a la obediencia entre individuos cuya conducta problemática se mantiene por escape o bien por múltiples reforzadores, sin la necesidad de provocar o reforzar la conducta problemática. Se discuten las limitaciones del presente estudio y se ofrecen recomendaciones para futura investigación.

Communication modality sampling for a toddler with Angelman syndrome.

PURPOSE: Vocal, gestural, and graphic communication modes were implemented concurrently with a toddler with Angelman syndrome to identify the most efficiently learned communication mode to emphasize in an initial augmentative communication system. METHOD: Symbols representing preferred objects were introduced in vocal, gestural, and graphic communication modes using an alternating treatment single-subject experimental design. Conventionally accepted prompting strategies were used to teach symbols in each communication mode. Because the learner did not vocally imitate, vocal mode intervention focused on increasing vocal frequency as an initial step. RESULTS: When graphic and gestural mode performances were compared, the learner most accurately produced requests in graphic mode (percentage of nonoverlapping data = 96). Given the lack of success in prompting vocal productions, a comparison between vocal and the other two communication modes was not made. CONCLUSION: A growing body of evidence suggests that concurrent modality sampling is a promising low-inference, data-driven procedure that can be used to inform selection of a communication mode(s) for initial emphasis with young children. Concurrent modality sampling can guide clinical decisions regarding the allocation of treatment resources to promote success in building an initial communicative repertoire.