An innovative and collaborative method to develop a model care and service trajectory for the assessment, diagnosis, and support of children with developmental disabilities.

This paper documents an innovative research approach undertaken to co-develop an integrated assessment, diagnosis, and support service trajectory for children suspected of having a developmental disability. It employed data-driven practices and involved multiple stakeholders such as parents, professionals, managers, and researchers. It emphasized the importance of incorporating experiential knowledge adopting an integrated care and service trajectory perspective, and using an implementation science framework. The first part of this article presents the theoretical roots and the collaborative method used to co-construct the model trajectory. The second part of this article presents the results of a survey in which participating stakeholders shared their point of view on the value and impact of this approach Overall, this article provides a step-by-step operationalization of participative research in the context of public health and social services. This may help guide future initiatives to improve services for developmental disabilities in partnership with those directly concerned by these services.

A Window into the Use of Post-diagnostic Services for Autism and Parents' Perspective on the Quality of Early Childhood Services Trajectory in a Canadian Province.

The pathway for post-diagnostic services for autism tends to be complex, heterogeneous, and fraught with delays. This situation has repercussions for families' experience with care systems, the quality of early interventions, and family adjustment. To date, studies of parents' point of view on autism services have predominantly focused on the diagnostic phase of the services trajectory. The goal of the present study was to investigate the perspective of parents on autism early childhood services within a public healthcare and social services system in a Canadian province (Québec). Participants were 91 parents who completed a structured framework for the evaluation of the quality of services trajectories, the Evaluation of the services Trajectory in Autism by Parents (ETAP; Mello et al., in Research in Developmental Disabilities 133, 2023). Approximately three-quarters of families received interim services (e.g., punctual speech therapy interventions) for which they had overall neutral-to-positive appraisals but viewed as having comparably low validity and continuity. Although all children diagnosed with autism are entitled to receive early behavioral intervention (EBI) dispensed by the public health and social services system, approximately a quarter of families did not receive this service. Families who received EBI evaluated this service positively on all ETAP dimensions. The empathy (i.e., parent-provider relationships) dimension was rated highest for both interim and EBI services. Some systemic, family, and child-related factors were associated with the perceived quality of services for both interim and early childhood services. Our results emphasize the need for services for children and families as soon as possible, both during assessment and after diagnosis.

Barriers and facilitators to accessibility, continuity, validity, flexibility, and provider-family relationship along the diagnostic pathway in developmental disabilities.

BACKGROUND: Parents of children with developmental disabilities must navigate a complex network to obtain a diagnosis and interventions for their child. However, their subjective experience of this journey has yet to be analyzed through the lens of a theorical framework that could support research, organizational program evaluation, and facilitate providers' reflection on how to enhance families' diagnostic services trajectory. AIMS: This study sought to examine the diagnostic journey as experienced by 77 parents whose children were recently diagnosed with developmental disabilities (e.g., autism, intellectual disability) in the metropolitan area of Montréal, Québec (Canada). METHODS AND PROCEDURES: A mixed qualitative content analysis approach was used to describe their perspective on barriers and facilitators in reference to the five dimensions of the Evaluation of the Trajectory Autism for Parents (ETAP) model (Rivard et al., 2020): accessibility, continuity, validity, flexibility, and provider-family relationship. OUTCOMES AND RESULTS: The barriers and facilitators related to systemic factors identified by parents were consistent with the five dimensions outlined by the ETAP model. However, beyond these characteristics of the service delivery system, parents additionally identified their own, personal facilitators CONCLUSIONS AND IMPLICATIONS: This study supports the relevance of the ETAP framework to understanding the experience of families seeking a diagnosis. It also reinforces the potential contributions of this model to organize extant and future research as well as structure program evaluation and improvements.

Competency drivers to support implementation of early intensive behavioral intervention in large-scale community-based services: Perspectives of caseworkers and organization representatives.

Implementing evidence-based practices in "real-world" settings poses significant challenges. Organizations involved must address this issue by providing supportive infrastructures. Among the elements to consider are competency drivers, which refer to the selection, training, and supervision of caseworkers. The purpose of this study was to describe the perspectives of caseworkers and representatives on competency drivers that organizations put in place to support the implementation of early intensive behavioral intervention (EIBI) in large-scale community-based services. The sample consisted of 109 caseworkers and 23 organization representatives who completed questionnaires. Results demonstrated that respondents consider clinical support and training as key elements in EIBI implementation. However, despite recognizing these factors, respondents reported considerable variability in practices. It appears necessary to better define and plan the implementation of these competency drivers with a view to improving EIBI implementation.

Changes in Autistic Symptoms and Adaptive Functioning of Children Receiving Early Behavioral Intervention in a Community Setting: A Latent Growth Curve Analysis.

Despite showing effects in well-controlled studies, the extent to which early intensive behavioral intervention (EBI) produces positive changes in community-based settings remains uncertain. Thus, our study examined changes in autistic symptoms and adaptive functioning in 233 children with autism receiving EBI in a community setting. The results revealed nonlinear changes in adaptive functioning characterized by significant improvements during the intervention and a small linear decrease in autistic symptoms from baseline to follow-up. The intensity of intervention, initial age, IQ and autistic symptoms were associated either with progress during the intervention or maintenance during the follow-up. The next step to extend this line of research involves collecting detailed data about intervention strategies and implementation fidelity to produce concrete recommendations for practitioners.

Cluster Analysis of Clinical Features of Children Suspected to Have Neurodevelopmental Disorders.

Early identification of neurodevelopmental disabilities (NDDs) is critical to a good prognosis. Several factors such as overlapping diagnoses can complicate this process and thus delay access to services. This study sought to identify meaningful clinical profiles, beyond diagnostic labels, in 194 children with NDDs referred to an assessment clinic. Cluster analyses were applied to eight selected behavioral and cognitive variables. Results suggested a cluster structure in which three homogenous groups differed significantly from one another: children who presented either (1) heterogeneous diagnoses and ambiguous profiles, (2) a clinical profile closely aligned to a classic presentation of ASD, and (3) emotional and behavioral challenges. These distinct profiles may have implications for assessment and clinical practices.

Assessing the quality of care and service trajectories in autism from families' perspective: Early intervention and interim services.

BACKGROUND: Following the Evaluation of the Autism Trajectory for Parents - Diagnostic Services (ETAP-1), the ETAP-2 instrument was created to assess the quality of the post-diagnostic phase of the care and service trajectory of families of children with autism. The instrument, based on an integrated care perspective, was developed with the input of multiple stakeholders (parents, service providers, researchers). AIMS: This study sought to evaluate the factor structure, reliability, and convergent and discriminant validity of ETAP-2. METHODS AND PROCEDURES: Parents (N = 197) of children recently diagnosed with autism (M = 5.1 years) were recruited from an assessment center and organizations providing early behavioral intervention and other supports for autism in the province of Québec, Canada. They completed the ETAP-2 questionnaire along with measures of satisfaction and family quality of life. OUTCOMES AND RESULTS: The instrument presented a five-construct structure generally consistent with previously identified dimensions of quality, except for three items previously associated with the continuity of the service trajectory. ETAP-2 had excellent internal consistency and demonstrated convergent and discriminant validity with other measures. CONCLUSIONS AND IMPLICATIONS: ETAP-2 is a brief parent-report measure with good psychometric properties. It can assist in gathering information on families' perception and experiences with early intervention and other post-diagnostic, interim services.

Developmental and behavioral groupings can predict changes in adaptive behavior over time in young children with neurodevelopmental disorders.

The heterogeneity within, and the overlap between, diagnostic categories for neurodevelopmental disorders (NDDs) remain poorly understood. Developmental trajectories may diverge among children with the same diagnosis, who may also respond very differently to treatment. In a previous study, we used statistical clustering methods in a sample of 194 preschoolers who were referred for NDD assessment. We identified three distinct subgroups based on multiple developmental and behavioral variables. The present study aimed to identify: (1) early developmental markers at the surveillance and screening period that are predictive of subgroup membership at the diagnostic period (i.e., around age 5), (2) associations between subgroups and the evolution of adaptive behavior over the course of two years, and (3) predictors of adaptive behavior change. Subgroup membership was the only significant predictor of adaptive behavior change over time, which suggests that a clustering method based on developmental and behavioral profiles may be useful in treatment planning.

The Diagnostic Period for Autism: Risk and Protective Factors for Family Quality of Life in Early Childhood.

During the diagnostic evaluation period for autism or intellectual disability (ID), families of young children are at risk for poor adjustment. The present study aimed to document family quality of life (FQOL), along with associated risk and protective factors, during this critical step of families' services trajectory. FQOL was measured in a large sample of families of children recently diagnosed with a neurodevelopmental disorder and examined in relation to the type of services received, children's clinical profile, and family variables. FQOL was related to types of services, children's challenging behavior, parenting stress, and several aspects of family composition and status. These findings highlight a need for mental health support for parents, coaching interventions for challenging behaviors, and family-centered supports.

Group intervention programs and their impact on well-being and quality-of-life for adults living with a rare or orphan disease - realist review of literature.

PURPOSE: Adults living with a rare or orphan diseases (ROD) experience common psychosocial difficulties that are often poorly addressed in usual care. This realist literature review aims to inform the development, evaluation and implementation of evidence based group therapy programs adapted to shared needs of patients living with various ROD. METHOD: The review is based on an analysis of Context-Mechanism-Outcome configurations. It included 21 primary studies published between 2010 and April 2022 and used a PRISMA process for study selection and inclusion. RESULTS: Our results show that group psychosocial interventions can help reduce perception of symptoms and psychological impacts of disease, improve social functioning and support and quality of life in patients. CONCLUSION: Group therapy programs seem promising for ROD-patients and should be considered within comprehensive treatment and support plans. However, more comprehensive studies of group therapies in context should aim to identify core active components of these interventions with ROD-patients. Implications for Rehabilitation:Rare or Orphan Diseases are varied, difficult to diagnose and have a major impact on all aspects of the patients' lives (physical, emotional, psychological, social, professional).Psychosocial support is a key but underdeveloped component to support the recovery trajectory for these patients.In this review of group interventions, we identified a few promising practices adaptable to patients living with Rare or Orphan Diseases (Acceptance and commitment therapy, cognitive behavioural therapies, psychoeducational programs).Patients who received psychosocial group interventions are likely to experience improvement in their quality of life.

Brief Report: Machine Learning for Estimating Prognosis of Children with Autism Receiving Early Behavioral Intervention-A Proof of Concept.

Although early behavioral intervention is considered as empirically-supported for children with autism, estimating treatment prognosis is a challenge for practitioners. One potential solution is to use machine learning to guide the prediction of the response to intervention. Thus, our study compared five machine algorithms in estimating treatment prognosis on two outcomes (i.e., adaptive functioning and autistic symptoms) in children with autism receiving early behavioral intervention in a community setting. Each machine learning algorithm produced better predictions than random sampling on both outcomes. Those results indicate that machine learning is a promising approach to estimating prognosis in children with autism, but studies comparing these predictions with those produced by qualified practitioners remain necessary.

Parents' satisfaction with a Canadian pilot clinic to reduce waiting lists for the assessment and diagnosis of autism spectrum disorder and intellectual disability in young children.

BACKGROUND: A large body of evidence suggest that parents of young children with autism spectrum disorder or intellectual disability experience low levels of satisfaction with the diagnostic evaluation process. This study sought to document parents' satisfaction with the services of a pilot clinic implemented in Québec, Canada. METHOD: Two-hundred fifty-nine (259) parents were recruited following their child's diagnosis. A mixed methods approach was used to investigate parents' satisfaction globally and with specific aspects of the assessment process. RESULTS: Parents expressed overall high satisfaction with the assessment process. Parental satisfaction with specific aspects of the assessment process was negatively related to paternal stress, fathers' unemployment and household income and positively related to maternal stress. CONCLUSIONS: This pilot clinic could meet parents' needs at this crucial moment in their care and services trajectory. The factors associated with satisfaction in the present study may inform future improvements to its services.

Validation of the multidimensional attitudes toward preschool inclusive education scale in the bilingual context of Québec.

BACKGROUND: The Multidimensional Attitudes Toward Preschool Inclusive Education Scale (MATPIES; Lohmann et al., 2016) seeks to assess preschool educators' attitudes toward inclusive education. It has been used in wide range of settings and with varied populations, but there has been a lack of systematicity in translation, adaptation, and validation procedures associated with it. For instance, its use in French or in a French-English bilingual context such as Québec (Canada) has yet to be validated. AIMS: The present study documented the translation and validation process for the MATPIES in bilingual early childhood education settings. METHODS AND PROCEDURES: The MATPIES was completed by 211 French- and English-speaking early childhood educators and administrators in the province of Québec, Canada. OUTCOMES AND RESULTS: Confirmatory factor analyses indicated that the original factor structure for the MATPIES was not replicated within the bilingual Québec sample.Exploratory analyses suggested a four-factor structure encompassing 15 items, which had good internal consistency (α = 0.87). CONCLUSIONS AND IMPLICATIONS: The factor structure of the MATPIES may vary across populations. This study underscores the importance of evaluating instruments in contexts that differ from those in which they were originally constructed to ensure the validity of results.

Symptom Severity, Internalized and Externalized Behavioral and Emotional Problems: Links with Parenting Stress in Mothers of Children Recently Diagnosed with Autism.

The present study examined which aspects of the child's behavior and clinical profile accounted for three dimensions of parenting stress: related to parenting roles, to interactions with the child, and to the child. Measures of adaptive behavior, intellectual functioning, autism symptom severity, and challenging behavior and emotional difficulties were examined as predictors of parenting stress in 157 mothers of children recently diagnosed with autism. Children's emotional problems and aggressive behavior were most predictive of parenting distress, whereas autism symptoms along with emotional problems and aggressive behavior, respectively, were linked to stress pertaining to interactions and to the child. These findings underscore the need for comprehensive and complementary interventions that focus on children's behavior and symptoms but also on parent adjustment.

The diagnostic trajectory in autism and intellectual disability in Quebec: pathways and parents' perspective.

BACKGROUND: This paper aimed to describe the diagnostic service trajectory of families of children with autism or intellectual disability in the province of Québec and identify predictors of parents' perceptions of its quality. METHODS: The Evaluation of the services Trajectory in Autism by Parents instrument was completed by 259 parents at an assessment clinic. Children's clinical records were also examined. RESULTS: On average 26 months elapsed between their first concerns and their child's diagnosis, a period during which few (25%) received support. Parents' evaluations were generally positive but were lower for the accessibility of the pre-assessment phase and the flexibility of the assessment process. Longer delays and a greater number of professionals consulted were associated with lower quality ratings. Some language-, immigration status-, and income-related differences in families' appraisals were noted. CONCLUSION: The diagnostic trajectory for neurodevelopmental disorders within public services in Québec presents some efficiency and accessibility challenges. Possible improvements are proposed to facilitate screening and to support families throughout this phase of their trajectory.

Using Prevent Teach Reinforce for Young Children to Manage Challenging Behaviors in Public Specialized Early Intervention Services for Autism.

This proof-of-concept study assessed the feasibility of implementing Prevent-Teach-Reinforce-for-Young-Children (PTR-YC) program to address challenging behaviors in children with autism within the context of public, specialized early intensive behavioral intervention (EIBI) services offered in community settings. Following a 2-day training and with brief weekly supervision meetings, children's EIBI educators acted as facilitators in 35 families' home environments. Small and moderate effect sizes were observed for children's behavioral outcomes and parenting stress. Recruitment and retention rates, implementation fidelity, as well as treatment acceptability from the perspective of participating parents and the organization attested to the feasibility and relevance of implementing and evaluating this program on a larger scale as part of a multi-center randomized controlled trial.

Parent coaching intervention program based on the Early Start Denver Model for children with autism spectrum disorder: Feasibility and acceptability study.

A parent coaching intervention program was launched in a Quebec community-based organization for autism spectrum disorder. This initiative was intended to support families following the child's diagnosis, as they awaited public early intensive behavioral intervention services. The parent-mediated intervention program, based on the Early Start Denver Model, included a 3 h parent group training, an initial assessment followed by the development of an individualized intervention plan, 8 weeks of live coaching, a final assessment to evaluate children's progress, and a feedback meeting with families. The present study used a mixed-method design to assess the feasibility and acceptability of the program. Ten families (ten mothers and three fathers) and four coaches participated in the present study. Parents and coaches perceived and achieved high levels of implementation overall (91 %) and there was a moderate improvement of parents' teaching skills. All participating families and coaches were satisfied or highly satisfied with the program and found it acceptable.

Development of a questionnaire to assess the quality of service trajectories in autism spectrum disorder from families' perspective.

BACKGROUND: ETAP-1 was created to evaluate the quality of services trajectory from families' perspective. The items of ETAP-1 were developed from previous studies on integrated care, existing quality assessments, and consultations with families and experts in evaluation and in autism spectrum disorder (ASD). METHOD: The questionnaire was completed by 200 parents of children aged 5 and under who were recently diagnosed with ASD or intellectual disability. Of these, 183 received diagnostic evaluation through a clinic specialized in ASD; the other 17 underwent diagnostic evaluation in hospital settings. RESULTS: Factor analysis supported the a priori dimensions of quality and distinctions between experiences before and during diagnostic evaluation. The instrument had high internal consistency, convergent and discriminant validity with other measures and was sensitive to differences in service delivery models. DISCUSSION: ETAP-1 is useful in organizing information on families' experiences throughout their services trajectories and according to a dynamic perspective.

Quality of Life in Families of Young Children With Autism Spectrum Disorder.

The present study investigated family quality of life (FQOL) as experienced by 493 mothers and fathers (295 families) of young children with autism spectrum disorder (ASD) prior to receiving early intervention services in the province of Québec. These families were most satisfied with their physical and material well-being and least satisfied with their family interactions. Children's level of functioning across various standardized and subjective measures were positively associated with parents' FQOL. In both parents, family characteristics associated with financial and personal resources were also linked to FQOL. The child's age and the number of children were associated with mothers' FQOL. Overall, these findings provide a portrait of ASD-related stressors and resources relevant to both parents during the early childhood period.

Follow-Up of Children With Autism Spectrum Disorder 1 Year After Early Behavioral Intervention.

The effectiveness of early intensive behavioral intervention (EIBI) for children with autism spectrum disorders (ASDs) has been demonstrated by many studies and meta-analyses. Although it is considered an exemplary practice in several countries, few studies have investigated the maintenance of gains made in EIBI over time. Thirty-two children were assessed at posttreatment and 1-year follow-up after they attended a low-to-moderate-intensity (10 to 20 hr per week) EIBI program delivered by a public rehabilitation center. Between baseline and posttreatment, children showed significant gains in most areas of intellectual functioning and a significant decrease of autism symptom severity, but no change in adaptive behavior. Gains in intellectual functioning were maintained over a 1-year period after treatment termination, but autism symptom severity had increased to approximately pretreatment levels during that interval. Considerable individual variability was noted in the evolution of outcomes.