Developmental associations between motor and communication outcomes in Fragile X syndrome: Variation in the context of co-occurring autism.

LAY ABSTRACT: Fragile X syndrome (FXS), the leading heritable cause of intellectual disability, has a co-occurrence rate of autism spectrum disorder (ASD) estimated at ~60%. Children with FXS experience delayed achievement and slower development of key motor abilities, which happens to an even greater extent for children with both FXS and ASD. A multitude of studies have demonstrated that motor abilities are foundational skills related to later communication outcomes in neurotypical development, as well as in the context of ASD. However, these associations remain unexamined in FXS, or FXS + ASD. In this study, we aimed to determine the associations between early motor skills and their rate of development on communication outcomes in FXS. Furthermore, we investigated whether these associations varied in the context of co-occurring FXS + ASD. Results revealed within-FXS variation in the context of co-occurring ASD between some aspects of motor development and communication outcomes, yet within-FXS consistency between others. Findings provide evidence for variability in developmental processes and outcomes in FXS in the context of co-occurring ASD and offer implications for intervention.

Expanding School-Based Motivational Interviewing Through Delivery by Paraprofessional Providers: A Preliminary Scoping Review.

The supply of school mental health (SMH) providers and services cannot meet the demand of students in-need, and this gap is expected to widen in coming years. One way to increase the reach of helpful services for youth is to grow the SMH workforce through task-shifting to paraprofessionals. Task-shifting could be especially promising in expanding Motivational Interviewing (MI) interventions, as MI can be molded to target a number of academic and behavioral outcomes important to schools. However, no review of training exclusively paraprofessional samples in MI has yet been conducted. The current paper provides a scoping review of 19 studies of training paraprofessional providers to use MI to evaluate trainee characteristics, training content and format, and outcomes. Of these 19 studies, 15 reported that paraprofessionals improved in using MI following training. Nine studies reported that task-shifting MI was positively received by clients and/or providers. Six studies examined task-shifting MI in youth-serving contexts, and four examined the practice in traditional school contexts, suggesting its potential for use in SMH. Other findings and implications, such as client behavior change and provider fidelity, are shared, along with ideas for advancing research, practice, and policy in this subfield.

Brief Report: Prevalence and Predictors of DSM-Specific and Distinct Anxiety in Cognitively Impaired Autistic Preschool Children.

Autistic individuals are twice as likely to meet criteria for anxiety than neurotypical children; yet we lack understanding of early presentations of anxiety in young autistic children, especially those with cognitive impairment. This study is the first to utilize an autism-specific anxiety diagnostic interview with 28 preschool cognitively impaired, autistic children and 18 neurotypical, age-matched controls. Results indicate that 64% of autistic children met criteria for DSM-specified or "other specified," herein referred to as "distinct," anxiety disorders; 32% met criteria for multiple anxiety disorders, with phobias occurring most often. Results indicate that anxiety is highly prevalent in cognitively-impaired, autistic preschool children, highlighting the need for developmentally-tailored assessment and treatment in early childhood.

Predictors, Parental Views, and Concordance Across Diagnostic Sources of Autism in Male Youth with Fragile X Syndrome: Clinical Best Estimate and Community Diagnoses.

Persons with fragile X syndrome (FXS) with cooccurring autism spectrum disorder (ASD) are at risk for poorer educational, medical, employment, and independent living outcomes. Thus, the identification of ASD in those with FXS is fundamental to ensuring access to appropriate supports to achieve good quality of life. Yet, optimal diagnostic methods and the exact rate of ASD comorbidity remains controversial, and description of ASD identification in the community in FXS has been limited. This study characterized ASD in a sample of 49 male youth with FXS across multiple diagnostic sources: parent-reported community diagnoses, classification derived from ADOS-2 and ADI-R thresholds, and clinical best-estimate classifications from an expert multidisciplinary team. High concordance was found between ADOS-2/ADI-R and clinical best estimate classifications, with both methods supporting ASD in ~ 75% of male youth with FXS. In contrast, 31% had a community diagnosis. Findings supported gross under-identification of ASD in male youth with FXS in community settings; 60% of those who met clinical best estimate criteria for ASD had not received a diagnosis in the community. Moreover, community diagnoses were poorly aligned with the presence of ASD symptoms as perceived by parents and professionals and, unlike clinical best estimate diagnoses, were not associated with cognitive, behavioral, or language features. Findings highlight under-identification of ASD in community settings as a significant barrier to service access for male youth with FXS. Clinical recommendations should emphasize the benefits of seeking a professional ASD evaluation for children with FXS who are noted to display key ASD symptoms.

Attention/Deficit Hyperactivity Disorder in Adolescent and Young Adult Males With Fragile X Syndrome.

This study characterized the rates of attention-deficit/hyperactivity disorder (ADHD) in adolescent and young adult males with fragile X syndrome (FXS) using a multi-method approach integrating a DSM-based parent interview (Children's Interview for Psychiatric Syndromes; P-ChIPS, Fristad et al., 1998) and a parent rating scale (Child Behavior Checklist; CBCL, Achenbach, 2001). Thirty-one males with FXS, aged 16-24 years, participated. Forty-two percent met DSM-5 criteria for ADHD and 35% exceeded the CBCL cut-offs. Agreement between the two classification methods was fair (κ = 0.38). Autism symptom severity and nonverbal cognitive ability did not predict ADHD diagnoses/symptoms. Results show high rates of ADHD in males with FXS during late adolescence and young adulthood, which are not accounted for by impaired nonverbal cognitive skills or autism symptom severity. DSM-based ADHD-specific scales are recommended over broadband symptom scales to improve accurate identification.

Emergence and rate of autism in fragile X syndrome across the first years of life.

Prospective longitudinal studies of idiopathic autism spectrum disorder (ASD) have provided insights into early symptoms and predictors of ASD during infancy, well before ASD can be diagnosed at age 2-3 years. However, research on the emergence of ASD in disorders with a known genetic etiology, contextualized in a developmental framework, is currently lacking. Using a biobehavioral multimethod approach, we (a) determined the rate of ASD in N = 51 preschoolers with fragile X syndrome (FXS) using a clinical best estimate (CBE) procedure with differential diagnoses of comorbid psychiatric disorders and (b) investigated trajectories of ASD symptoms and physiological arousal across infancy as predictors of ASD in preschoolers with FXS. ASD was not diagnosed if intellectual ability or psychiatric disorders better accounted for the symptoms. Our results determined that 60.7% of preschoolers with FXS met the Diagnostic and Statistical Manual of Mental Disorders (fifth edition) (DSM-5) criteria for ASD using the CBE procedure. In addition, 92% of these preschoolers presented with developmental delay and 45.4% also met criteria for psychiatric disorders, either anxiety, ADHD, or both. ASD diagnoses in preschoolers with FXS were predicted by elevated scores on traditional ASD screeners in addition to elevated autonomic arousal and avoidant eye contact from infancy.

Life Satisfaction in Adolescents with Autism Spectrum Disorder.

We provided evidence regarding the reliability and validity of measures of assets and life satisfaction (LS) for adolescents with autism spectrum disorder (ASD). We identified levels of LS within this population, compared these levels to those of typically developing adolescents, and described the relation between assets and LS. Forty-six adolescents with ASD and their caregivers completed questionnaires assessing LS and assets. Preliminary support was provided for the internal consistency reliability and validity of these measures in adolescents with ASD. Youth with ASD reported moderate to high levels of LS; these were lower than those of typically developing peers. Age moderated the relation between self-reported LS and some assets. Implications were discussed within the context of Schalock's (J Disabil Policy Stud 14:204-215, 2004) emerging disability paradigm.

Prevalence and Predictors of Anxiety Disorders in Adolescent and Adult Males with Autism Spectrum Disorder and Fragile X Syndrome.

Anxiety disorders affect ~ 15-20% of youths without neurodevelopmental disorders, with persons having autism spectrum disorder (ASD) and fragile X syndrome (FXS) at elevated risk for anxiety disorders. Few studies have compared rates and predictors of anxiety disorders in adolescents with FXS or ASD. This study directly compares rates, predictors, and medication of anxiety disorders between age-matched, male adolescents with FXS (n = 31) or ASD (n = 20). Results indicate that 51.6% of FXS and 50.0% of ASD adolescents met criteria for an anxiety disorder. Cognitive scores and ASD severity did not predict anxiety. Of those with anxiety, ~ 40% of the FXS and 20% of the ASD participants were prescribed medications for anxiety.

Autism Spectrum Disorder Symptoms in Infants with Fragile X Syndrome: A Prospective Case Series.

No studies to date have prospectively examined early autism spectrum disorder (ASD) markers in infants with fragile X syndrome (FXS), who are at elevated risk for ASD. This paper describes the developmental profiles of eight infants with FXS from 9 to 24 months of age. Four meet diagnostic criteria for ASD at 24 months of age, and four do not. Trends in these case studies suggest that early social-communicative deficits differentiate infants with and without later ASD diagnoses in ways that are similar to later-born siblings of children with ASD. Repetitive behaviors and cognitive and adaptive impairments are present in all FXS infants throughout development, suggesting that these deficits reflect the general FXS phenotype and not ASD in FXS specifically.

Life Satisfaction and Academic Performance in Early Adolescents: Evidence for Reciprocal Association.

Student subjective well-being remains a relatively neglected topic despite its intimate link to positive school outcomes. As academic achievement is a widely used yardstick of student success and school accountability, school-based mental health research and practice have focused primarily on the assessment and treatment of learning and behavioral problems. This short-term longitudinal study sought to establish the role of student subjective well-being, specifically, global life satisfaction (LS), in academic achievement. Based on the engine model of well-being (Jayawickreme, Forgeard, & Seligman, 2012), the study focused on LS as a process variable and academic performance as an outcome variable and vice versa. Using two waves (five months apart) of data, the study examined the reciprocal relations between LS and academic achievement, and how the relations may be shaped by positive and negative affective experiences in school, in a sample of 821 middle school students. Results revealed positive reciprocal causal relations between students' LS and grades, even when demographic covariates, school-based positive and negative affect, and baseline values of the criterion variables were controlled. This study provides empirical support that LS does not undermine academic achievement (or vice versa), but rather it is synergistic with better school grades. Furthermore, the relations between students' LS and grades were not moderated by negative or positive affective experiences in school. These findings suggest that student LS should occupy a more prominent niche in the school agenda.

Life satisfaction and maladaptive behaviors in early adolescents.

This study explored the directionality of the relations between global life satisfaction (LS) and internalizing and externalizing behaviors using a sample of regular education students who were initially enrolled in Grade 7 (n = 470). Self-report measures of internalizing and externalizing behaviors and LS were administered on 2 occasions, 6 months apart, to students from a Southeastern U.S. middle school. Short-term longitudinal analyses revealed that neither externalizing behaviors nor internalizing behaviors at Time 1 predicted LS at Time 2. However, LS at Time 1 predicted externalizing behaviors at Time 2. LS at Time 1 also predicted internalizing behaviors at Time 2, but the results were moderated by student gender. At higher levels of LS, boys reported lower levels of internalizing behaviors at Time 2. The overall results suggested that lower levels of LS are an antecedent of increased maladaptive behaviors among early adolescents. Alternatively, higher levels of LS may be a protective factor against subsequent externalizing behaviors among boys and girls and internalizing behaviors among boys. Furthermore, the results provide further support for the discriminant validity of positive and negative measures of mental health and suggest that LS measures may provide useful information for comprehensive adolescent health screening and monitoring systems.

Mechanisms of change in adolescent life satisfaction: a longitudinal analysis.

This study explored the psychosocial mechanisms of change associated with differences in levels and linear change of adolescents' global life satisfaction across a 2-year time period. Based on a theoretical model proposed by Evans (1994), this study tested the relations between selected personality (i.e., extraversion and neuroticism) and environmental (stressful life events) variables and global life satisfaction when mediated by internalizing and externalizing problems. The results suggested support for internalizing problems as a mediator of the relationship of personality and environmental variables with life satisfaction. Pathways mediated by internalizing problems significantly predicted levels and linear change of life satisfaction across a 2-year time span. Furthermore, pathways mediated by externalizing problems significantly predicted the level but not the linear change of life satisfaction. Thus, behavior problems and their antecedents appear to relate significantly to adolescents' perceptions of their quality of life. Implications for adolescent mental health promotion were discussed.

Measuring school-related subjective well-being in adolescents.

The tripartite model of subjective well-being (SWB) incorporates 3 components: frequent positive emotions, infrequent negative emotions, and an overall positive evaluation of life circumstances (Diener, Suh, Lucas, & Smith, 1999). In light of the large amount of time that youth spend in school, this study investigated a tripartite model of school-related SWB among adolescents, based on 3 measures of SWB appropriate for adolescents. The measures included a measure of school satisfaction (SS) and measures of positive and negative emotions experienced specifically during school hours. Confirmatory factor analysis was conducted to analyze the factorial validity of 3- and 4-factor models of school-related SWB in a sample of 921 adolescents. Results indicated that a 4-factor model comprised of positive emotions, negative emotions, fear-related negative emotions, and SS best described the structure of school-related SWB in the current sample. Results also revealed a comparable factor structure for male and female students. The study points to the possible benefits of a contextualized approach to SWB that takes into account the specific environments in which adolescents live.

A dual-factor model of mental health: toward a more comprehensive understanding of youth functioning.

Traditional mental health models focus on psychological problems and distress; accordingly, health is viewed as the absence of illness or disability. In contrast, a dual-factor model of mental health incorporates both indicators of positive subjective well-being (SWB) and measures of psychopathological symptoms to comprehensively determine an individual's psychological adjustment. This study used such a dual-factor model to measure the mental health status of young adolescents. A total of 764 middle school students were classified into one of four distinct groups based on having high or low psychopathology and high or low SWB. Furthermore, group differences in student engagement, academic achievement, and environmental support for learning were investigated. Results demonstrated the existence of a traditionally neglected group of adolescents (low SWB and low psychopathology) who are nonetheless at risk for academic and behavior problems in school and who performed no better than the most troubled group of adolescents. Overall, both the presence of positive well-being and the absence of symptoms were necessary for ensuring the most advantageous school performance. These results highlight the importance of incorporating positive indicators of well-being along with traditional negative factors in more fully understanding relationships between individuals' mental health and educational outcomes.