Self-determination in minoritized autistic adolescents and adults.

Self-determination plays an important role in outcomes in autism and shows intersectional disparities. Yet, little is known about the role of individual differences or social drivers of health in the development of self-determination. Understanding these factors is key for developing effective supports. This mixed-methods convergent study examined self-determination in racially and ethnically minoritized autistic individuals and caregivers. Participants ages 13 to 30 ( N = 73) varying widely in language and cognitive ability and caregivers ( n =52) completed the Self-Determination Inventory. Autism traits and sense of community predicted caregiver report of self-determination, and autism traits and language predicted self-report of self-determination, consistent with DisCrit and Diversity Science. Self-Determination Inventory interviews of a subset of participants ( n = 13) and caregivers ( n = 9) were analyzed using inductive thematic analysis. Themes pointed to the role of the intersection of race and disability in shaping self-determination. Altogether, findings point to the importance of these frameworks, environmental influences, and multi-informant perspectives in characterizing self-determination. Future work should focus on the impact of environmental factors in self-determination in minoritized autistic individuals during the transition to adulthood.

Opportunities to Respond During Dyadic Caregiver-Child and Naturalistic Family Interactions Among Children With Down Syndrome: A Preliminary Investigation.

PURPOSE: Dyadic caregiver-child interactions are commonly used to examine children's language learning environments. However, children frequently interact with multiple caregivers and/or siblings if they come from homes with multiple caregivers and siblings. Thus, we examined if and how caregiver opportunities to respond (OTRs) varied when sampled across three interaction configurations. METHOD: Twelve children with Down syndrome (Mage = 40.82 months) and their biological parents participated in the current study. We collected separate mother-child and father-child dyadic interactions and one family choice interaction (i.e., both caregivers present and occasionally siblings) in families' homes. We analyzed if differences in the caregiver's OTR frequency and type-explicit and implicit-existed among dyadic and family choice configurations. RESULTS: We found that, during family choice interactions, children were exposed to fewer OTRs when combining the total number of father and mother OTRs compared to dyadic caregiver-child interactions. This effect was large for explicit OTRs (mother-dyadic vs. combined family choice: g = -1.99, confidence interval [CI] [-3.00, -1.00]; father-dyadic vs. combined family choice: g = -0.84, CI [-1.84, -0.11]). For implicit OTRs, effects were small for mother-dyadic versus combined family choice (g = -0.34, CI [-1.17, 0.48]) and negligible when comparing father-dyadic with combined family choice (g = -0.08, CI [-0.90, 0.73]). CONCLUSION: Our preliminary findings highlight the need for a more nuanced understanding of children's language learning environments to better understand how caregivers support their children's language development. SUPPLEMENTAL MATERIAL: https://doi.org/10.23641/asha.25579905.

Parental Responsivity and Child Communication During Mother-Child and Father-Child Interactions in Fragile X Syndrome.

PURPOSE: Past research shows that parentally responsive behavior toward the child positively influences language development in both neurotypical children and children with intellectual and developmental disabilities, including those with fragile X syndrome (FXS); however, most studies have focused exclusively on the mother-child relationship. The current study examined relationships between parent behavior (i.e., responsivity and behavior management) and child language performance in both mother-child and father-child interactions, as well as relationships between child characteristics and both parent behavior and child language. METHOD: Participants were 23 families of young boys with FXS between 3 and 7 years of age. Mothers and fathers independently completed questionnaires assessing child characteristics and separately engaged in 12-min play-based interactions with their child via telehealth. One parent also completed a comprehensive interview assessing child adaptive behavior. Video recordings of the parent-child interactions were transcribed and coded for parent and child behavior, and measures of parent and child language were obtained from the transcripts. RESULTS: Mothers and fathers used similar rates of responsive behaviors during parent-child interactions, and parental responsivity was positively associated with some aspects of child language performance (i.e., talkativeness and lexical diversity). Parental behavior, however, was not associated with syntactic complexity. Older children and children with higher levels of adaptive behavior had parents who used higher rates of responsive behaviors. Fathers used higher rates of behavior management strategies compared to mothers, and this type of parent behavior was not associated with child language. CONCLUSION: Overall, this study provides evidence that interventions focused on increasing parental responsiveness would be beneficial for families of children with FXS and that these interventions should be delivered early given the association between responsivity and child age. SUPPLEMENTAL MATERIAL: https://doi.org/10.23641/asha.25229939.

An Exploratory Study of Pragmatic Language Use Across Contexts With the Pragmatic Rating Scale-School Age Among Autistic Boys and Boys With Fragile X Syndrome Plus Autism.

PURPOSE: Autistic boys and boys with co-occurring fragile X syndrome and autism spectrum disorder (FXS + ASD) demonstrate similar pragmatic language difficulties. The Pragmatic Rating Scale-School Age (PRS-SA) captures ecologically valid metrics of pragmatic language impairments in these populations. It is traditionally scored based on the Autism Diagnostic Observation Schedule (ADOS), which may limit the use of the PRS-SA more broadly in research and clinical contexts. METHOD: This study evaluated the feasibility of the PRS-SA based on a shorter, semistructured conversational context compared to the ADOS in school-age autistic boys (n = 16) and boys with FXS + ASD (n = 16), matched on ASD traits. Differences across ADOS and conversational contexts and associations with ASD-related social difficulties were evaluated. RESULTS: Findings revealed differences in PRS-SA scores between ADOS and conversational contexts, but only for the FXS + ASD group. Limited associations were observed between PRS-SA scores and ASD traits. CONCLUSIONS: Results from this study indicate the feasibility of using the PRS-SA in a shorter conversational context than the ADOS to assess pragmatic language among autistic boys. For boys with FXS + ASD, contextual differences warrant careful consideration in future work.

Expressive language sampling and outcome measures for treatment trials in fragile X and down syndromes: composite scores and psychometric properties.

The lack of psychometrically sound outcome measures has been a barrier to evaluating the efficacy of treatments proposed for core symptoms of intellectual disability (ID). Research on Expressive Language Sampling (ELS) procedures suggest it is a promising approach to measuring treatment efficacy. ELS entails collecting samples of a participant's talk in interactions with an examiner that are naturalistic but sufficiently structured to ensure consistency and limit examiner effects on the language produced. In this study, we extended previous research on ELS by analyzing an existing dataset to determine whether psychometrically adequate composite scores reflecting multiple dimensions of language can be derived from ELS procedures administered to 6- to 23-year-olds with fragile X syndrome (n = 80) or Down syndrome (n = 78). Data came from ELS conversation and narration procedures administered twice in a 4-week test-retest interval. We found that several composites emerged from variables indexing syntax, vocabulary, planning processes, speech articulation, and talkativeness, although there were some differences in the composites for the two syndromes. Evidence of strong test-retest reliability and construct validity of two of three composites were obtained for each syndrome. Situations in which the composite scores would be useful in evaluating treatment efficacy are outlined.

Comparison of Linguistic Error Production in Conversational Language Among Boys With Fragile X Syndrome + Autism Spectrum Disorder and Autistic Boys.

PURPOSE: Expressive language impairments are common among school-age boys with fragile X syndrome (FXS) and autistic boys. Given the high co-occurrence of autism spectrum disorder (ASD) among individuals with FXS, cross-condition comparisons can elucidate the specificity of such impairments as they relate to ASD. Language samples can provide fruitful information regarding individuals' grammatical skills in less structured formats relative to standardized measures. This study examined grammatical errors produced during a conversational language sample among 20 boys with FXS and co-occurring ASD (FXS + ASD) and 19 autistic boys matched on ASD severity. METHOD: Language samples were coded for omissions and errors at the word and utterance levels. Participants' grammatical errors were also compared to separate mental age-matched and mean length of utterance-matched boys from a reference database. RESULTS: Boys with FXS + ASD and autistic boys produced similar rates of errors across all categories. Relative to their matched comparison groups, boys with FXS + ASD and autistic boys produced significantly more omissions during conversation. CONCLUSIONS: These findings suggest that omissions may be a unique grammatical marker associated with the ASD phenotype. Further examination of omissions across diagnostic groups would aid in clarifying the specificity of omissions in the language phenotype of ASD.

The association between expressive language skills and adaptive behavior in individuals with Down syndrome.

The primary goal of this study was to determine whether expressive language skills contribute to adaptive behavior (e.g., socialization and daily living skills) in children, adolescents, and young adults with Down syndrome (DS) whilst controlling for age and nonverbal cognitive ability. Expressive language was assessed using the psychometrically validated Expressive Language Sampling (ELS) conversation and narration procedures. The language produced was transcribed and analyzed to yield measures of expressive vocabulary, syntax, and intelligibility. Socialization and daily living skills of participants with DS were measured with the Vineland Adaptive Behavior Scales, 2nd edition (VABS-2) parent/caregiver rating form. Our results show that the three ELS measures were significantly correlated with multiple measures from the VABS-2 when controlling for age. Several correlations remained significant even when nonverbal cognitive ability was included as a control variable. Our results suggest that expressive language skills contribute to adaptive behavior in children, adolescents, and young adults with DS regardless of age and some of these associations are not explained solely by overall cognitive delays. Further studies including longitudinal data are needed to extend our results.

Associations Among Sex, Cognitive Ability, and Autism Symptoms in Individuals with Down Syndrome.

This study explores sex-differences in (a) rates and profiles of autism symptoms as well as in (b) the contribution of intellectual quotient (IQ) to autism symptom presentation in Down syndrome (DS). Participants were 40 males and 38 females with DS, aged 6 to 23 years. Autism symptoms were rated through the Autism Diagnostic Observation Schedule-Second Edition (ADOS-2). Results show no sex differences in the ADOS-2 Calibrated Severity Scores (CSS). However, only females with DS who are classified as DS-Only have higher scores on verbal IQ than those classified as DS + autism. Furthermore, associations between IQ and all CSSs are found for females, but not for males. Findings suggest that verbal cognition may play differential roles for females and males with DS.

Analysis of a Repetitive Language Coding System: Comparisons between Fragile X Syndrome, Autism, and Down Syndrome.

Expressive language sampling (ELS) is a frequently used tool for language analysis, as it can be used across widely ranging cognitive and language abilities. ELS can also evaluate pragmatic language, including excessive self-repetition, which is challenging to assess with traditional standardized assessments. This study explored how a well-established ELS protocol can assess three types of linguistic self-repetition in three neurodevelopmental disabilities: fragile X syndrome (FXS), autism spectrum disorder (ASD), and Down syndrome (DS). We examined its ability to differentiate between these disorders, the relationships between repetitive language and other participant characteristics, and initial construct validity. We found that the groups with FXS and ASD differed significantly on each of the three repetitive language measure, and that the group with DS differed from either ASD or FXS on two. Cognitive ability was significantly related to phrase repetition in the group with ASD. When the groups were combined, there was evidence of convergent and divergent validity. This study extends previous research on ELS and supports its use as a means to characterize pragmatic language. It also provides information about the relationships between repetitive language and other phenotypic characteristics.

Mental Health Challenges, Parenting Stress, and Features of the Couple Relationship in Parents of Children With Fragile X Syndrome.

Background: Individuals with fragile X syndrome (FXS) have significant delays in cognition and language, as well as anxiety, symptoms of autism spectrum disorder, and challenging behaviors such as hyperactivity and aggression. Biological mothers of children with FXS, who are themselves FMR1 premutation or full mutation carriers, are at elevated risk for mental health challenges in addition to experiencing stress associated with parenting a child with significant disabilities. However, little is known about fathers in these families, including the ways in which parental well-being influences the mother-father relationship and the impact of child characteristics on paternal and couple functioning. Method: The current study examined features of, and relationships between, parental well-being, couple well-being, and child functioning in 23 families of young boys with FXS. Mothers and fathers independently completed multiple questionnaires about their individual well-being, couple functioning, and child behavior. One parent per family also completed an interview about the child's adaptive skills. Results: Results suggest that both mothers and fathers in these families experience clinically significant levels of mental health challenges and elevated rates of parenting stress relative to the general population. Findings also indicate that the couples' relationship may be a source of strength that potentially buffers against some of the daily stressors faced by these families. Additionally, parents who reported less parenting stress had higher couples satisfaction and dyadic coping. Finally, parents of children with less severe challenging behaviors exhibited fewer mental health challenges, less parenting stress, and higher levels of both couples satisfaction and dyadic coping. Parents of children with higher levels of adaptive behavior also reported less parenting stress and higher couples satisfaction. Conclusion: Overall, this study provides evidence that families of children with FXS need access to services that not only target improvements in the child's functioning, but also ameliorate parental stress. Family-based services that include both mothers and fathers would lead to better outcomes for all family members.

Speech-language pathologists' practices in augmentative and alternative communication during early intervention.

This survey study examined augmentative and alternative communication (AAC) practices reported by early intervention speech-language pathologists (SLPs) across the United States (N = 376). The study examined (a) types of AAC that SLPs reported using (i.e., sign language, photographs, pictures, symbols, talking switches, and iPad apps or dedicated speech-generating devices); (b) SLPs' perspectives on the influence of child spoken language ability on AAC recommendations; (c) factors that influenced AAC decision-making within early intervention; and (d) perceived barriers associated with AAC implementation. SLPs reported that they were significantly more likely to introduce all types of AAC to children without spoken language abilities compared to children in later stages of language development. On average, they were most likely to report using or recommending sign language and photographs, and least likely to report using or recommending talking switches or speech-generating devices. Of the options provided, child expressive and receptive language abilities were rated as the most important factors to consider when determining AAC use, followed by cognitive ability, diagnosis, and chronological age. SLPs identified caregiver buy-in and carryover across providers as the most significant barriers to AAC implementation. Recommendations for future research and current AAC practices within early intervention are discussed.

Assessing the Quantity and Quality of Language Used by Mothers and Fathers of Children with Down Syndrome During Shared Book Reading.

Young children with Down syndrome (DS) have language delays beginning early in life. Book reading with parents provides a context for capitalizing on language learning opportunities. This study evaluated the quantity and quality of language input among mothers and fathers of young children with DS during book reading interactions and investigated associations with child language. Findings revealed that mothers were more talkative and used more descriptive language, whereas fathers spent more time reading the book text. Moreover, maternal and paternal input were correlated with different measures of child language, suggesting that mothers and fathers may use divergent approaches to support language development.

Verbal inhibition declines among older women with high FMR1 premutation expansions: A prospective study.

The FMR1 premutation has been associated with difficulties in executive functioning, including verbal inhibition. However, little is known about the longitudinal profiles of verbal inhibition among FMR1 premutation carriers, particularly in women, and how individual factors such as aging and CGG repeat length may contribute to changes in verbal inhibition over time. The present study examined verbal inhibition performance (i.e., inhibition errors) on the Hayling Sentence Completion Task in a cohort of 92 women with the FMR1 premutation across two timepoints approximately three years apart. We examined the effects of age, CGG repeat length, and their interactions on verbal inhibition over time. We also evaluated whether response latency affected verbal inhibition errors. We found no significant change in verbal inhibition in the full cohort during the three-year study period. However, a subset of FMR1 premutation carriers, namely older participants with higher CGG repeats, evidenced greater declines in verbal inhibition over time. Longer response latencies did not compensate for verbal inhibition errors. The findings suggest that a subset of women with the FMR1 premutation may be at earlier, increased risk for changes in executive functioning, which if confirmed, should be considered as part of the clinical profile associated with the premutation.

Brief Report: Linguistic Mazes and Perseverations in School-Age Boys with Fragile X Syndrome and Autism Spectrum Disorder and Relationships with Maternal Maze Use.

Autism spectrum disorder (ASD) and fragile X syndrome (FXS) are neurodevelopmental disorders with overlapping pragmatic language impairments. Prior work suggests pragmatic language differences may run in families. This study examined specific pragmatic difficulties (i.e., linguistic mazes and perseverations) in boys (9-18 years) with idiopathic ASD (n = 26) and FXS+ASD (n = 29), and relationships with maternal maze use. Language samples were obtained separately for boys and mothers. Nonparametric analyses suggested that boys largely did not differ in their rates of mazes, but that boys with FXS+ASD exhibited more perseverations. Mazes were correlated between fragile X dyads. Maternal mazes were correlated with child perseverations among idiopathic ASD dyads. These findings have implications for the etiological significance of ASD-related language phenotypes.

Symptoms of Autism Spectrum Disorder in Individuals with Down Syndrome.

There is a growing body of evidence to suggest that individuals with Down syndrome (DS) are diagnosed with autism spectrum disorders (ASD) at a higher rate than individuals in the general population. Nonetheless, little is known regarding the unique presentation of ASD symptoms in DS. The current study aims to explore the prevalence and profiles of ASD symptoms in a sample of individuals with DS (n = 83), aged between 6 and 23 years. Analysis of this sample (MAge = 15.13) revealed that approximately 37% of the sample met the classification cut-off for ASD using the Autism Diagnostic Observation Schedule 2 (ADOS-2) Calibrated Severity Score (CSS), an indicator of the participants' severity of ASD-related symptoms. Item-level analyses revealed that multiple items on Module 2 and Module 3 of the ADOS-2, mostly in the Social Affect (SA) subdomain, differentiated the children with DS who did not meet ASD classification (DS-only) from those who did (DS + ASD). Lastly, comparisons of individuals with DS-only and those with DS + ASD differed significantly on the syntactic complexity of their expressive language. These findings shed light on the unique presentation of ASD symptoms in a sample of individuals with DS and suggest that expressive language abilities may play a pivotal role in the presentation of ASD symptoms in DS.

Maternal Use of Decontextualized and Contextualized Talk: An In-Depth Investigation of Early Parent-Child Interactions in Down Syndrome.

Purpose The goal of this study was to characterize and quantify maternal use of decontextualized and contextualized input during mother-child interactions including young children with Down syndrome (DS). Method Participants included 22 mother-child dyads with DS (M age = 42.8 months) and 22 mother-child dyads with typical development (M age = 44.0 months). Parent-child language samples were collected during free-play, book reading, and snack time, and coded for maternal decontextualized (i.e., pretend, explanatory, and narrative talk) and contextualized input (i.e., descriptions, conversation, praise, questions, and directives). Results Mothers of children with DS used a larger proportion of pretend talk compared to other types of decontextualized input and also used a larger proportion of questions, conversation, and descriptions compared to other types of contextualized language. Mothers of children with DS generally used a smaller proportion of decontextualized input compared to mothers of children with typical development, with the exception of pretend talk. Maternal decontextualized input was not related to children's age or language ability in DS. Conclusions Findings shed new light on the early language environments of children with DS, providing important insight into the ways that mothers of children with DS are incorporating decontextualized and contextualized talk into early mother-child conversations. Additional implications and future directions are discussed.

Parent Command Use and Child Compliance During Parent-Child Interactions Including Children With Down Syndrome.

Purpose This study investigated maternal and paternal command use and child compliance in children with Down syndrome during mother-child and father-child free-play interactions. We also examined child compliance to direct versus indirect commands, and the relationships between maternal command use, paternal command use, child compliance, and child speech and language abilities. Method Fifteen families including a mother, father, and child with Down syndrome participated. Mother-child and father-child dyads participated in free-play interactions. We coded overall parent command use, command type (direct or indirect), and child compliance with the commands. Child language abilities were measured using a standardized assessment as well as during free-play. Results Mothers and fathers of children with Down syndrome used a similar number of commands, and children complied similarly across interactions. Children were more likely to comply with direct versus indirect commands. Parent command use was related to child language abilities in different ways for mothers and fathers. Child compliance was not related to receptive or expressive language abilities. Conclusions Mothers and fathers of children with Down syndrome use commands in similar ways but may base their command use on different child factors. Using direct commands with children with Down syndrome may be beneficial when increasing compliance during early interactions.

Spoken language outcome measures for treatment studies in Down syndrome: feasibility, practice effects, test-retest reliability, and construct validity of variables generated from expressive language sampling.

BACKGROUND: The purpose of this study was to evaluate expressive language sampling (ELS) as a procedure for generating spoken language outcome measures for treatment research in Down syndrome (DS). We addressed (a) feasibility, (b) practice effects across two short-term administrations, (c) test-retest reliability across two short-term administrations, (d) convergent and discriminant construct validity, and (e) considered comparisons across the conversation and narration contexts. METHOD: Participants were 107 individuals with DS between 6 and 23 years of age who presented with intellectual disability (IQ < 70). The utility of ELS procedures designed to collect samples of spoken language in conversation and narration were evaluated separately. Variables of talkativeness, vocabulary, syntax, utterance planning, and articulation quality, derived from transcripts segmented into C-units (i.e., an independent clause and its modifiers), were considered. A 4-week interval was used to assess practice effects and test-retest reliability. Standardized direct assessments and informant report measures were collected to evaluate construct validity of the ELS variables. RESULTS: Low rates of noncompliance were observed; youth who were under 12 years of age, had phrase-level speech or less, and had a 4-year-old developmental level or less were at particular risk for experiencing difficulty completing the ELS procedures. Minimal practice effects and strong test-retest reliability across the 4-week test-retest interval was observed. The vocabulary, syntax, and speech intelligibility variables demonstrated strong convergent and discriminant validity. Although significant correlations were found between the variables derived from both the conversation and narration contexts, some differences were noted. CONCLUSION: The ELS procedures considered were feasible and yielded variables with adequate psychometric properties for most individuals with DS between 6 and 23 years old. That said, studies of outcome measures appropriate for individuals with DS with more limited spoken language skills are needed. Context differences were observed in ELS variables suggest that comprehensive evaluation of expressive language is likely best obtained when utilizing both contexts.

"Are We Done Yet?" Question-Asking in Boys With Fragile X Syndrome and Idiopathic Autism Spectrum Disorder.

Purpose Question-asking serves as a tool to learn new information and is important in both academic and social settings. Boys with idiopathic autism spectrum disorder (ASD) and boys with fragile X syndrome and comorbid ASD (FXS + ASD) have similar social communication deficits, which may have downstream effects on their question-asking ability. This study examined question-asking in school-age boys with idiopathic ASD and FXS + ASD, including the role of ASD severity, expressive grammatical complexity (measured by mean length of utterance [MLU]), and IQ. Method Twenty-five boys with FXS + ASD and 21 boys with idiopathic ASD (ages 9-16 years) were included in this study. Autism Diagnostic Observation Schedule assessments were transcribed and coded for the frequency, function, and appropriateness of spontaneous questions asked. We examined group differences in these aspects of question-asking and relationships between question-asking and ASD severity, MLU, and IQ within each group. Results Boys with FXS + ASD asked more questions than boys with idiopathic ASD, although boys with idiopathic ASD asked a higher proportion of appropriate questions. Boys with idiopathic ASD also asked the examiner more personal questions than the boys with FXS + ASD. ASD severity and MLU were related to the proportion of clarification questions in FXS + ASD, and ASD severity was also related to the proportion of personal questions in this group. For the boys with idiopathic ASD, ASD severity was related to the total number of questions asked. Conclusions Our findings highlight similarities and differences between boys with FXS + ASD and idiopathic ASD in their spontaneous question production and indicate that ASD severity and grammatical language are differentially important for question-asking. This study has implications for targeted treatment in question-asking skills for boys with FXS + ASD and ASD.

Maternal Input and Child Language Comprehension During Book Reading in Children With Down Syndrome.

Purpose Communication interactions between parents and children during shared book reading impact a child's development of both language and literacy skills. This study examined maternal language input and child expressive communication during a shared book reading activity in children with Down syndrome (DS) and children with typical development (TD). Additionally, children's receptive language was examined to understand the relationship between maternal language input and child receptive language ability. Method Participants included 22 children with DS and 22 children with TD between 22 and 63 months of age and their mothers. Each mother-child dyad participated in a 7-min naturalistic shared book reading activity. Results Compared to mothers of children with TD, mothers of children with DS used significantly more utterances with less grammatical complexity, but a similar range of vocabulary diversity. Mothers of children with DS used more questions, descriptions, gestures, and labels, whereas mothers of children with TD used nearly half of their utterances to read directly from books. Children with DS communicated at a similar frequency compared to their peers with TD; however, they produced significantly fewer spoken words. Conclusions This study reveals important differences between early shared book reading interactions and provides implications for future research targeting parent-coached intervention strategies that may enhance children's learning during shared book reading by providing access to expressive language and print instruction.