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BACKGROUND: There is growing understanding of the potential benefits of a multi-method approach to accurately capture language skills of children on the autism spectrum. Tools such as Language ENvironment Analysis (LENA) provide an efficient means of capturing and analysing early child vocalizations (CVs) and the language learning environment. While developed to capture whole-day recordings of child language in naturalistic settings, there is potential utility in capturing, but little knowledge about, primary LENA metrics-including CVs and conversational turns (CTs)-and novel metrics, such as vocalization ratios (VRs), sampled in clinical practice settings where children are often seen. Moreover, recent research indicates that the novel VR may offer a broad indicator of children's developmental level, beyond just their language abilities, a hypothesis yet to be investigated in a large sample of children for whom the LENA was designed (i.e., pre-schoolers). AIMS: To explore the extent to which primary and novel LENA metrics collected during brief one-to-one clinical interaction was a useful indicator of developmental outcomes for children on the autism spectrum. METHODS & PROCEDURES: Participants were recruited as part of an on-going research programme evaluating early intervention outcomes (n = 99; age 14-47 months). Language samples were collected at intake (T1) using the wearable LENA Digital Language Processors during a one-to-one, play-based assessment with a clinician. Direct (Mullen Scales of Early Learning-MSEL) and parent-report (Vineland Adaptive Behavior Scales-VABS) measures of verbal and non-verbal skills were also collected at intake (T1) and again at exit (T2), approximately 12 months later. OUTCOMES & RESULTS: Few correlations were observed between child measures and CVs, a primary LENA metric. The novel VR metric was associated with concurrent direct assessment (MSEL) (and to a lesser extent parent report; VABS) measures of verbal and non-verbal skills, with moderate positive correlations found between VRs and all directly assessed subscale scores. However, VRs did not uniquely contribute to the prediction of child outcomes when baseline skills were also considered. CONCLUSIONS & IMPLICATIONS: The novel VR may provide an insight into autistic children's overall development in addition to their language ability, suggesting that even when collected in a short recording format, LENA might be a useful component of a multi-method assessment approach. WHAT THIS PAPER ADDS: What is already known on the subject To accurately capture language skills of children on the autism spectrum, multi-method approaches, including natural language sampling, are recommended. Tools such as LENA provide an efficient means of capturing and analysing naturalistic child language and the language learning environment. What this paper adds to existing knowledge This study demonstrates the potential benefits, and limitations, of using LENA to augment assessment of young children on the autism spectrum. Specifically, LENA provides a complementary, and low burden, method for capturing child language samples. What are the potential or actual clinical implications of this work? Novel metrics, such as the VR, collected during brief clinical interactions might be a useful component of a multi-method assessment approach for children on the autism spectrum.
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Transtorno do Espectro Autista , Transtorno Autístico , Transtornos do Desenvolvimento da Linguagem , Transtorno do Espectro Autista/complicações , Transtorno do Espectro Autista/diagnóstico , Transtorno Autístico/diagnóstico , Criança , Linguagem Infantil , Pré-Escolar , Humanos , Lactente , Idioma , Transtornos do Desenvolvimento da Linguagem/complicações , Transtornos do Desenvolvimento da Linguagem/diagnóstico , Transtornos do Desenvolvimento da Linguagem/terapiaRESUMO
Given concerns that bilingual exposure might confuse children with disabilities-including autism spectrum disorder (ASD)-bilingual parents may restrict exposure to one language, often the community-dominant language. We investigated a potential consequence of this decision; the possibility that non-native language use might influence parental communicative behaviors during interaction with the child. We recruited 39 parent-child dyads, each with a young child with ASD (mostly boys) and parent/carer (mostly mothers). Parents were either monolingual speakers of community-dominant English (n = 20) or bilingual with English as the second language (n = 19). We confirmed our assumption that the latter group would have significantly poorer non-native English language via standardized assessment of expressive vocabulary, and ensured children were matched on age, ASD symptoms, and developmental level. We sampled parent-child interaction-including in each of bilinguals' native and non-native languages-and coded parents' amount and complexity of speech, communicative synchrony, and imitations and expansions of their child's speech. Few differences presented across bilingual parents' native versus non-native language samples, but this group showed reduced synchrony and use of expansions compared to monolinguals. Further, bilinguals' English-language knowledge was associated with self-reported comfort using this language and with two coded interaction measures. These empirical data only partially support qualitative accounts that non-native language use may influence bilingual parents' interaction behaviors with their young children. With growing rates of ASD diagnosis and increasing cultural/linguistic diversity around the world, further dedicated clinical and experimental attention to this issue is clearly warranted.
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Transtorno do Espectro Autista/psicologia , Pesquisa Empírica , Multilinguismo , Relações Pais-Filho , Pais/psicologia , Adulto , Idoso , Transtorno do Espectro Autista/diagnóstico , Criança , Pré-Escolar , Estudos de Coortes , Feminino , Humanos , Masculino , Pessoa de Meia-IdadeRESUMO
BACKGROUND: Language and communication skills are essential aspects of child development, which are often disrupted in children with neurodevelopmental disorders. Cutting edge research in psycholinguistics suggests that multilingualism has potential to influence social, linguistic and cognitive development. Thus, multilingualism has implications for clinical assessment, diagnostic formulation, intervention and support offered to families. We present a systematic review and synthesis of the effects of multilingualism for children with neurodevelopmental disorders and discuss clinical implications. METHODS: We conducted systematic searches for studies on multilingualism in neurodevelopmental disorders. Keywords for neurodevelopmental disorders were based on Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition categories as follows; Intellectual Disabilities, Communication Disorders, Autism Spectrum Disorder (ASD), Attention-Deficit/Hyperactivity Disorder, Specific Learning Disorder, Motor Disorders, Other Neurodevelopmental Disorders. We included only studies based on empirical research and published in peer-reviewed journals. RESULTS: Fifty studies met inclusion criteria. Thirty-eight studies explored multilingualism in Communication Disorders, 10 in ASD and two in Intellectual Disability. No studies on multilingualism in Specific Learning Disorder or Motor Disorders were identified. Studies which found a disadvantage for multilingual children with neurodevelopmental disorders were rare, and there appears little reason to assume that multilingualism has negative effects on various aspects of functioning across a range of conditions. In fact, when considering only those studies which have compared a multilingual group with developmental disorders to a monolingual group with similar disorders, the findings consistently show no adverse effects on language development or other aspects of functioning. In the case of ASD, a positive effect on communication and social functioning has been observed. CONCLUSIONS: There is little evidence to support the widely held view that multilingual exposure is detrimental to the linguistic or social development of individuals with neurodevelopmental disorders. However, we also note that the available pool of studies is small and the number of methodologically high quality studies is relatively low. We discuss implications of multilingualism for clinical management of neurodevelopmental disorders, and discuss possible directions for future research.
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Multilinguismo , Transtornos do Neurodesenvolvimento , Criança , HumanosRESUMO
BACKGROUND: The PACT randomised-controlled trial evaluated a parent-mediated communication-focused treatment for children with autism, intended to reduce symptom severity as measured by a modified Autism Diagnostic Observation Schedule-Generic (ADOS-G) algorithm score. The therapy targeted parental behaviour, with no direct interaction between therapist and child. While nonsignificant group differences were found on ADOS-G score, significant group differences were found for both parent and child intermediate outcomes. This study aimed to better understand the mechanism by which the PACT treatment influenced changes in child behaviour though the targeted parent behaviour. METHODS: Mediation analysis was used to assess the direct and indirect effects of treatment via parent behaviour on child behaviour and via child behaviour on ADOS-G score. Alternative mediation was explored to study whether the treatment effect acted as hypothesised or via another plausible pathway. Mediation models typically assume no unobserved confounding between mediator and outcome and no measurement error in the mediator. We show how to better exploit the information often available within a trial to begin to address these issues, examining scope for instrumental variable and measurement error models. RESULTS: Estimates of mediation changed substantially when account was taken of the confounder effects of the baseline value of the mediator and of measurement error. Our best estimates that accounted for both suggested that the treatment effect on the ADOS-G score was very substantially mediated by parent synchrony and child initiations. CONCLUSIONS: The results highlighted the value of repeated measurement of mediators during trials. The theoretical model underlying the PACT treatment was supported. However, the substantial fall-off in treatment effect highlighted both the need for additional data and for additional target behaviours for therapy.
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Transtorno Autístico/terapia , Comunicação , Relações Pais-Filho , Pais/psicologia , Psicoterapia de Grupo/métodos , Projetos de Pesquisa/normas , Adulto , Transtorno Autístico/diagnóstico , Pré-Escolar , Feminino , Humanos , Masculino , Método Simples-Cego , Resultado do TratamentoRESUMO
BACKGROUND: Autism is associated with impairments that have life-time consequences for diagnosed individuals and a substantial impact on families. There is growing interest in early interventions for children with autism, yet despite the substantial economic burden, there is little evidence of the cost-effectiveness of such interventions with which to support resource allocation decisions. This study reports an economic evaluation of a parent-mediated, communication-focused therapy carried out within the Pre-School Autism Communication Trial (PACT). METHODS: 152 pre-school children with autism were randomly assigned to treatment as usual (TAU) or PACT + TAU. Primary outcome was severity of autism symptoms at 13-month follow-up. Economic data included health, education and social services, childcare, parental productivity losses and informal care. RESULTS: Clinically meaningful symptom improvement was evident for 53 % of PACT + TAU versus 41 % of TAU (odds ratio 1.91, p = 0.074). Service costs were significantly higher for PACT + TAU (mean difference £4,489, p < 0.001), but the difference in societal costs was smaller and non-significant (mean difference £1,385, p = 0.788) due to lower informal care rates for PACT + TAU. CONCLUSIONS: Improvements in outcome generated by PACT come at a cost. Although this cost is lower when burden on parents is included, the cost and effectiveness results presented do not support the cost-effectiveness of PACT + TAU compared to TAU alone. TRIAL REGISTRATION: Current Controlled Trials ISRCTN58133827.
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Transtorno Autístico/economia , Transtorno Autístico/terapia , Comunicação , Psicoterapia/economia , Pré-Escolar , Análise Custo-Benefício , Intervenção Médica Precoce , Feminino , Recursos em Saúde/estatística & dados numéricos , Humanos , Masculino , Pais , Psicoterapia/métodos , Instituições Acadêmicas/economia , Seguridade Social/economiaRESUMO
Parenting is a rewarding experience but is not without its challenges. Parents of Autistic children face additional challenges, and as a result can experience lower levels of wellbeing and more mental health problems (i.e., depression, anxiety, stress). Previous studies have identified concurrent correlates of wellbeing and mental health. However, few have investigated predictors of subsequent wellbeing and mental health, or of change over time, among parents of pre-school aged autistic children. We examined child-, parent-, and family/sociodemographic factors associated with change in parents' mental health and wellbeing across three timepoints (spanning approximately one year) among 53 parents of Autistic pre-schoolers (M = 35.48, SD = 6.36 months. At each timepoint, parents reported lower wellbeing and greater mental health difficulties compared to normative data. There was no significant group-level change over time in parent outcomes. However, individual variability in short-term (~ 5 months) wellbeing and mental health change was predicted by a combination of child- and parent-related factors, while variability in medium-term (~ 10 months) change was predicted by parent factors alone. Parents' description of their child and their relationship predicted change in both wellbeing and mental health. Furthermore, participating in a parent-mediated intervention (available to a subgroup) was a significant predictor of change in wellbeing. Our findings highlight potentially modifiable factors (e.g., learning healthier coping strategies) that may positively impact both short- and medium-term change in parental outcomes.
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We tested the potential for Gazefinder eye-tracking to support early autism identification, including feasible use with infants, and preliminary concurrent validity of trial-level gaze data against clinical assessment scores. We embedded the ~ 2-min 'Scene 1S4' protocol within a comprehensive clinical assessment for 54 consecutively-referred, clinically-indicated infants (prematurity-corrected age 9-14 months). Alongside % tracking rate as a broad indicator of feasible assessment/data capture, we report infant gaze data to pre-specified regions of interest (ROI) across four trial types and associations with scores on established clinical/behavioural tools. Most infants tolerated Gazefinder eye-tracking well, returning high overall % tracking rate. As a group, infants directed more gaze towards social vs. non-social (or more vs. less socially-salient) ROIs within trials. Behavioural autism features were correlated with increased gaze towards non-social/geometry (vs. social/people) scenes. No associations were found for gaze directed to ROIs within other stimulus types. Notably, there were no associations between developmental/cognitive ability or adaptive behaviour with gaze towards any ROI. Gazefinder assessment seems highly feasible with clinically-indicated infants, and the people vs. geometry stimuli show concurrent predictive validity for behavioural autism features. Aggregating data across the ~ 2-min autism identification protocol might plausibly offer greater utility than stimulus-level analysis alone.
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Transtorno do Espectro Autista , Transtorno Autístico , Lactente , Humanos , Transtorno Autístico/diagnóstico , Transtorno do Espectro Autista/psicologia , Tecnologia de Rastreamento Ocular , Estudos de ViabilidadeRESUMO
LAY ABSTRACT: Most support programmes for Autistic children are available only after they are diagnosed. Research suggests that parenting supports may be helpful for parents and their infants, when provided in the first 2 years of life - before a formal diagnosis is given, but when information suggests an infant is more likely to be Autistic. However, we do not know how acceptable these types of supports might be to the Autistic and autism communities. We asked 238 Autistic and non-autistic people - some of whom were parents, and some of whom were professionals working in research, health and education - about their perspectives on very-early supports. People generally agreed that it could be acceptable to work with parents to help them understand and support their child's specific needs and unique ways of communicating. People suggested a variety of support strategies could be acceptable, including parent education, changing the environment to meet an infant's needs, and creating opportunities for infants' to make choices and exercise control. People preferred respectful and accurate language - including the term 'support' (rather than 'intervention') and 'early-in-life' (rather than 'at-risk' of autism, or 'pre-emptive' when describing developmental stage). Continuing to work with community members will help to make sure autism support programmes are relevant and helpful.
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The effects of selectively different experience of eye contact and gaze behaviour on the early development of five sighted infants of blind parents were investigated. Infants were assessed longitudinally at 6-10, 12-15 and 24-47 months. Face scanning and gaze following were assessed using eye tracking. In addition, established measures of autistic-like behaviours and standardized tests of cognitive, motor and linguistic development, as well as observations of naturalistic parent-child interaction were collected. These data were compared with those obtained from a larger group of sighted infants of sighted parents. Infants with blind parents did not show an overall decrease in eye contact or gaze following when they observed sighted adults on video or in live interactions, nor did they show any autistic-like behaviours. However, they directed their own eye gaze somewhat less frequently towards their blind mothers and also showed improved performance in visual memory and attention at younger ages. Being reared with significantly reduced experience of eye contact and gaze behaviour does not preclude sighted infants from developing typical gaze processing and other social-communication skills. Indeed, the need to switch between different types of communication strategy may actually enhance other skills during development.
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Desenvolvimento Infantil/fisiologia , Comunicação , Relações Interpessoais , Pais , Pessoas com Deficiência Visual , Pré-Escolar , Movimentos Oculares/fisiologia , Face , Feminino , Humanos , Lactente , Estudos Longitudinais , Masculino , Relações Pais-FilhoRESUMO
Language is typically a highly lateralized function, with atypically reduced or reversed lateralization linked to language impairments. Given the diagnostic and prognostic role of impaired language for autism spectrum disorders (ASDs), this paper reviews the growing body of literature that examines patterns of lateralization in individuals with ASDs. Including research from structural and functional imaging paradigms, and behavioral evidence from investigations of handedness, the review confirms that atypical lateralization is common in people with ASDs. The evidence indicates reduced structural asymmetry in fronto-temporal language regions, attenuated functional activation in response to language and pre-linguistic stimuli, and more ambiguous (mixed) hand preferences, in individuals with ASDs. Critically, the evidence emphasizes an intimate relationship between atypical lateralization and language impairment, with more atypical asymmetries linked to more substantive language impairment. Such evidence highlights opportunities for the identification of structural and functional biomarkers of ASDs, affording the potential for earlier diagnosis and intervention implementation.
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Córtex Cerebral/patologia , Transtornos Globais do Desenvolvimento Infantil/psicologia , Lateralidade Funcional/fisiologia , Idioma , Transtornos Globais do Desenvolvimento Infantil/fisiopatologia , Humanos , Transtornos da Linguagem/fisiopatologiaRESUMO
LAY ABSTRACT: Support for autistic children early in life should help them to lead flourishing lives. However, many of the early intervention programmes for young autistic children are time-consuming and costly for families. These programmes are also often conducted in settings that are not closely matched to real life. We spoke to 23 parents (of 22 autistic children) to understand their experiences of their children's involvement in early intervention. Parents told us they were grateful for the opportunity, that they had 'hit the jackpot', and their children had 'gained so much' from the programme. They seemed to value the service because it made them feel safe and secure during an uncertain time in their children's lives. Parents told us they trusted staff, felt that they weren't 'doing it alone', and this 'took that pressure off' and helped them feel empowered. They also spoke of feelings of safety from being linked to the university research programme which offered 'accountability' and 'integrity'. Parents' comments showed a strong commitment to the early intervention model and staff - but also common feelings of abandonment and disempowerment as their child's time with the programme came to an end and they went 'back to the real world' and needed to find new supports for their children. These parents' insights should help to inform the design and delivery of community supports for preschool-aged autistic children and their families, which match the reality of their lived experiences.
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Transtorno do Espectro Autista , Transtorno Autístico , Pré-Escolar , Criança , Humanos , Pais , Pessoal de Saúde , EmoçõesRESUMO
Many autistic children require support to meet their learning needs. Given the heterogeneity within the autism spectrum it is plausible that different types of support might be better suited to different children. However, knowledge on what interventions work best for which children is limited. We examined the outcomes of autistic preschool-aged children receiving one of two community early intervention approaches. Our main objective was to understand which baseline child characteristics might be associated with the degree of individual response to intervention-whether prognostically (i.e., irrespective of intervention received) or predictively (i.e., specifically in the context of one or other EI approach). Participants comprised two matched groups of preschool-aged autistic children receiving either Group-based Early Start Denver Model (G-ESDM; n = 42) delivered in a 1:3-4 staff:child ratio or an Early Intensive Behavioural Intervention (EIBI; n = 40) delivered in combination of 1:1 and 1:2 staff:child ratio. Over an approximate one-year follow-up period, children in both groups made significant gains in Developmental Quotient (DQ) scores, and trend-level gains in adaptive behaviour composite scores. Higher attention to a playful adult measured via an eye-tracking task was prognostically indicative of better verbal DQ and adaptive behaviour outcomes for the cohort overall. Moderation analyses indicated a single predictive effect-of pre-program sustained attention for subsequent NVDQ outcomes specific to those children receiving G-ESDM. These findings suggest that fine-grained measures of learning skills offer promise towards the selection and tailoring of intervention approaches to meet individual children's learning needs.
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PURPOSE: While research indicates that both the core features of autism and associated developmental skills influence adaptive behaviour outcomes, results to date suggest greater influence of the latter than the former, and little attention has been given to how the interaction of both together might impact functional disability. Seeking to expand understanding of associations between young children's core social autism features, developmental skills, and functional ability/disability, we specifically tested whether early developmental skills might have a moderating effect on the association between early social features and subsequent functional disability. METHODS: Data from 162 preschool children were available for this study. These included time-1 measures of social autism features (ADOS-Social Affect score) and developmental skills (MSEL-Developmental Quotient; DQ), and a measure of functional ability/disability (VABS-Adaptive Behaviour Composite; ABC) available at follow-up 1-year later (time-2). RESULTS: Time-1 ADOS-SA and MSEL-DQ scores were concurrently associated with one another, and both prospectively associated with time-2 VABS-ABC scores. Examination of partial correlations (i.e., controlling for MSEL-DQ) demonstrated that the association of time-1 ADOS-SA and time-2 VABS-ABC was accounted for by shared variance with DQ. Formal moderation analysis returned a non-significant overall interaction term, but showed a lower-bound region of significance whereby the association of time-1 ADOS-SA with time-2 VABS-ABC was significant for children with baseline DQ ≤ 48.33. CONCLUSION: Our results add to a body of empirical evidence consistent with an understanding of the needs of and resources available to autistic people through a 'cognitive compensation' lens.
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PURPOSE: Autistic children are reported to display higher levels of externalizing and internalizing behaviors than neurotypical children, and their parents report more stress than parents of neurotypical children. It is unclear whether child behavior difficulties contribute to increased parenting stress, whether parenting stress contributes to child behavior difficulties, or whether the relationship may be bidirectional. METHODS: We investigated prospective bidirectional associations between parenting stress and child externalizing and internalizing behaviors when autistic children were aged on average 3.5, 4.5, and 5.5 years. Data collected at these three timepoints were examined across two panels: Time 1 to Time 2 (n = 38 parent-child dyads) and Time 2 to Time 3 (n = 27 dyads). RESULTS: Across Time 1 to Time 2, early parenting stress was significantly associated with later child externalizing behavior, and cross-lagged panel analysis supported a uni- rather than a bidirectional association between these factors. There was some evidence of a bidirectional association between parenting stress and child internalizing behavior, though this was non-significant when the strong stability of child internalizing behavior was statistically controlled. In contrast, across Time 2 to Time 3, there were no significant prospective associations found between variables, highlighting the importance of considering the impact of parenting stress early in the course of childhood autism. CONCLUSION: Our results add to research indicating that support targeting parent characteristics, especially parenting stress, could ameliorate subsequent outcomes for both parents and children.
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LAY ABSTRACT: What is already known about the topic?The delivery of evidence-based interventions is an important part of the clinical pathway for many autistic children and their families. However, parents, practitioners, and policymakers face challenges making evidence informed decisions, due to the wide variety of interventions available and the large, and often inconsistent, body of evidence regarding their effectiveness.What this paper adds?This is a comprehensive umbrella review, also known as a 'review of reviews', which examined the range of interventions available, the evidence for their effectiveness, and whether effects were influenced by factors relating to individual children (e.g. chronological age, core autism characteristics, and related skills) or the ways interventions were delivered (by whom and in what setting, format, mode, and amount). There was evidence for positive therapeutic effects for some, but not all, interventions. No single intervention had a positive effect for all child and family outcomes of interest. The influence of child and delivery characteristics on effects was unclear.Implications for practice, research, and policyThe findings provide parents, practitioners, and policymakers with a synthesis of the research evidence to inform decision-making and highlight the importance of individualised approaches in the absence of clear and consistent evidence. The findings also highlight the need to improve consistency and completeness in reporting of research studies, so that the same questions may be answered more comprehensively in the future.
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Transtorno do Espectro Autista , Transtorno Autístico , Criança , Humanos , Transtorno Autístico/terapia , Transtorno do Espectro Autista/terapia , PaisRESUMO
Both the amount and responsiveness of adult language input contribute to the language development of autistic and non-autistic children. From parent-child interaction footage, we measured the amount of adult language input, overall parent responsiveness, and six discrete parent responsive behaviours (imitations, expansions, open-ended questions, yes/no questions, comments and acknowledgements) to explore which types of responsiveness predicted autistic preschoolers' language five months later, after controlling for adult language input. We found expansions and particularly imitations to be more important for later language than overall responsiveness. This study emphasises the need to capture what exactly about parent language input influences child language acquisition, and adds to the evidence that imitating and expanding early language might be particularly beneficial for autistic preschoolers.
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Transtorno do Espectro Autista , Transtorno Autístico , Adulto , Criança , Humanos , Linguagem Infantil , Transtorno Autístico/diagnóstico , Comportamento Imitativo , PaisRESUMO
While theory supports bidirectional effects between caregiver sensitivity and language use, and infant language acquisition-both caregiver-to-infant and also infant-to-caregiver effects-empirical research has chiefly explored the former unidirectional path. In the context of infants showing early signs of autism, we investigated prospective bidirectional associations with 6-min free-play interaction samples collected for 103 caregivers and their infants (mean age 12-months; and followed up 6-months later). We anticipated that measures of caregiver sensitivity/language input and infant language would show within-domain temporal stability/continuity, but also that there would be predictive associations from earlier caregiver input to subsequent child language, and vice versa. Caregiver sensitive responsiveness (from the Manchester Assessment of Caregiver-Infant interaction [MACI]) predicted subsequent infant word tokens (i.e., amount of language, coded following the Systematic Analysis of Language Transcripts [SALT]). Further, earlier infant Mean Length of Utterance (MLU; reflecting language complexity, also derived from SALT coding) predicted later caregiver MLU, even when controlling for variability in infant ages and clear within-domain temporal stability/continuity in key measures (i.e., caregiver sensitive responsiveness and infant word tokens; and infant and caregiver MLU). These data add empirical support to theorization on how caregiver input can be both supportive of, and potentially influenced by, infant capacities, when infants have social-communication differences and/or communication/language delays suggestive of possible emerging autism.
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Transtorno do Espectro Autista , Transtorno Autístico , Criança , Humanos , Lactente , Cuidadores , Estudos Prospectivos , Idioma , Desenvolvimento da LinguagemRESUMO
Our previous cross-sectional investigation (Chetcuti et al., 2020) showed that infants with autism traits could be divided into distinct subgroups based on temperament. This longitudinal study builds on this existing work by exploring the continuity of temperament subgroup classifications and their associations with behavioral/clinical phenotypic features from infancy to toddlerhood. 103 infants (68% male) showing early signs of autism were referred to the study by community healthcare professionals and seen for assessments when aged around 12-months (Time 1), 18-months (Time 2), and 24-months (Time 3). Latent profile analysis revealed inhibited/low positive, active/negative reactive, and sociable/well-regulated subgroups at each timepoint, and a unique reactive/regulated subgroup at Time 3. Cross-tabulations indicated a significant likelihood of children having a recurrent subgroup classification from one timepoint to the next, and no apparent patterns to the movement of children who did change from one subgroup to another over time. Temperament subgroups were associated with concurrent child social-emotional functioning and autism traits, but unrelated to child age, sex, or developmental level. These findings suggest that temperament subgroup classifications might represent a reliable and very early indicator of autism characteristics and social-emotional functioning among infants/toddlers with autism traits.
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Transtorno do Espectro Autista , Transtorno Autístico , Humanos , Masculino , Lactente , Idoso , Feminino , Temperamento , Transtorno Autístico/diagnóstico , Estudos Longitudinais , Estudos TransversaisRESUMO
Importance: The growing global prevalence of autism spectrum disorder (ASD) is associated with increasing costs for support services. Ascertaining the effects of a successful preemptive intervention for infants showing early behavioral signs of autism on human services budgets is highly policy relevant. Objective: To estimate the net cost impact of the iBASIS-Video Interaction to Promote Positive Parenting (iBASIS-VIPP) intervention on the Australian government. Design, Setting, and Participants: Infants (aged 12 months) showing early behavioral indicators of autism were recruited through community settings into the multicenter Australian iBASIS-VIPP randomized clinical trial (RCT), a 5- to 6-month preemptive parent-mediated intervention, between June 9, 2016, and March 30, 2018, and were followed up for 18 months to age 3 years. This economic evaluation, including cost analysis (intervention and cost consequences) and cost-effectiveness analyses of iBASIS-VIPP compared with usual care (treatment as usual [TAU]), modeled outcomes observed at age 3 through to 12 years (13th birthday) and was conducted from April 1, 2021, to January 30, 2023. Data analysis was conducted from July 1, 2021, to January 29, 2023. Exposures: iBASIS-VIPP intervention. Main Outcomes and Measures: To project the diagnostic trajectory and associated disability support costs drawing on the Australian National Disability Insurance Scheme (NDIS), the main outcome was the differential treatment cost of iBASIS-VIPP plus TAU vs TAU and disability-related government costs modeled to age 12 years, using a clinical diagnosis of ASD and developmental delay (with autism traits) at 3 years. Costs were calculated in Australian dollars and converted to US dollars. Economic performance was measured through the following: (1) differential net present value (NPV) cost (iBASIS-VIPP less TAU), (2) investment return (dollars saved for each dollar invested, taking a third-party payer perspective), (3) break-even age when treatment cost was offset by downstream cost savings, and (4) cost-effectiveness in terms of the differential treatment cost per differential ASD diagnosis at age 3 years. Alternate values of key parameters were modeled in 1-way and probabilistic sensitivity analysis, the latter identifying the likelihood of an NPV cost savings. Results: Of the 103 infants enrolled in the iBASIS-VIPP RCT, 70 (68.0%) were boys. Follow-up data at age 3 years were available for 89 children who received TAU (44 [49.4%]) or iBASIS-VIPP (45 [50.6%]) and were included in this analysis. The estimated mean differential treatment cost was A $5131 (US $3607) per child for iBASIS-VIPP less TAU. The best estimate of NPV cost savings was A $10â¯695 (US $7519) per child (discounted at 3% per annum). For each dollar invested in treatment, a savings of A $3.08 (US $3.08) was estimated; the break-even cost occurred at age 5.3 years (approximately 4 years after intervention delivery). The mean differential treatment cost per lower incident case of ASD was A $37â¯181 (US $26â¯138). We estimated that there was an 88.9% chance that iBASIS-VIPP would deliver a cost savings for the NDIS, the dominant third-party payer. Conclusions and Relevance: The results of this study suggest that iBASIS-VIPP represents a likely good-value societal investment for supporting neurodivergent children. The estimated net cost savings were considered conservative, as they covered only third-party payer costs incurred by the NDIS and outcomes were modeled to just age 12 years. These findings further suggest that preemptive interventions may be a feasible, effective, and efficient new clinical pathway for ASD, reducing disability and the costs of support services. Long-term follow-up of children receiving preemptive intervention is needed to confirm the modeled results.