How Developmental Disorders Changed Before and After COVID-19 Pandemic

WPA Working Group on Defining and Managing Autism Spectrum Disorder: spreading knowledge for the next generations of psychiatrists.

The intersection of autism spectrum disorder and intellectual disability

Editorial: Introduction to Special Section on Evidence-Based Practices for Persons With Intellectual and Developmental Disabilities

In a large, retrospective, population-based cohort, Diana Schendel and her co-workers in Atlanta have studied the prevalence of major birth defects among children with autism spectrum disorders (ASD). The prevalence of autism in children with birth defects and the risk of autism associated with having such defects was also investigated.1 Such an approach may contribute to the understanding of the aetiological pathways in autism, especially regarding first trimester origin, resulting from genetic or exogenous interference with development. Schendel et al. emphasize the phenotypic diversity within the autism spectrum, especially the co-occurrence of intellectual impairment, which in itself is strongly associated with major birth defects. The authors use the term autism to refer to the three most common ASDs: autistic disorder, Asperger syndrome, and pervasive developmental disorder, not otherwise specified. They also separate autism into the following two subgroups: autism with no other developmental disorder and autism combined with at least one accompanying developmental disorder. The large study cohort was identified through the Metropolitan Atlanta Developmental Disabilities Surveillance Program that includes children with different developmental disabilities, such as intellectual impairment, autism, cerebral palsy, hearing loss, and vision loss, and the Metropolitan Atlanta Congenital Defects Program for the identification of major birth defects. The prevalence of birth defects in children with autism, evaluated between the ages of 3 and 10 years, was found to be 6.4%, which was about twice the rate in children without autism. The studied birth defects were mainly in the central nervous system/eye, or the genitourinary, musculosketelal, cardiovascular, head/neck, respiratory, oral, and gastrointestinal systems. Syndrome/chromosomal abnormalities constituted a separate category. The prevalence of autism increased twofold in children with birth defects, especially if the defect was combined with other developmental disorders, particularly intellectual disability. The risk that autism may be under diagnosed in children with major birth defects accompanied by medical sequelae is an important notion and a clinical reality. The reported findings of an association between central nervous system/eye defects and autism give clues to prenatal timing, which could also be exemplified by optic nerve hypoplasia and by the different eye malformations, mainly colobomas, related to the CHARGE syndrome. The authors report a prevalence of syndrome/chromosomal defects of 1% in children with autism and in 1.3% if other developmental disorders co-occurred. The rate of Down syndrome was 1% among children with autism and other developmental disorders and the rate of autism among children with Down syndrome was 1.7%. However, Schendel et al. suggest that this rate may be underestimated. The risk of under ascertainment of autism in children with Down syndrome has also been pointed out by Rasmussen et al. since it is commonly believed that children with Down Syndrome have superior social abilities.2 Schendel et al. discuss important factors that contribute to the increased association between Down syndrome and autism. Such factors were also considered in the study by Rasmussen et al.,2 who in addition reported that five out of 25 children with Down syndrome and autism had experienced infantile spasms with hypsarrythmia during infancy. The pathogenesis of hypsarrhythmia has recently been studied in children with infantile spasms using elecetroencephalograms and, simultaneously, functional magnetic resonance imaging (fMRI)3 revealing a hypsarrhythmia-specific activation of different neuronal networks in cortex and subcortical structures such as the brainstem, thalamus, and putamen. The data studied by Schendel et al. was gathered more than 10 years ago and the authors mention that genetic diagnoses may be underrepresented. The ability to identify genetic syndromes associated with malformations has increased during the past decade. Several syndromes are associated with ASDs and the microarray technique has revealed abundant copy number variations with duplications, deletions, and rearrangements of DNA which has, accordingly, contributed to the identification of new syndromes. In addition, there are also malformation syndromes associated with autism that are not indisputably genetic, as illustrated by the Möbius sequence and the oculoauriculovertebral spectrum.4 Similarly, there are acquired malformations, such as the valproic acid, thalidomide, and the foetal alcohol syndromes that are related to an ASD phenotype. New MRI techniques have given new insights into the aetiopathogenetic mechanisms in autism. Boddaert et al.5 found an unexpectedly high rate of MRI abnormalities (48%) in their large series of clinical MRI investigations in non-syndromic autism. Three predominant types of brain anomalies were found: white matter hyperintensity, temporal lobe signal abnormalities, and dilated Virchow–Robin spaces, i.e. dilatations of the perivascular space that surrounds penetrating arteries. The study by Schendel et al. elucidates the importance of different diagnostic concepts, i.e. differentiating between aetiological diagnoses and diagnoses based on impaired functions and/or behaviours. Whenever a child has been diagnosed with a developmental disorder such as autism, learning disability*, or cerebral palsy, there is a need for further considerations and relevant aetiological investigations. Thus, the paper also highlights that children with developmental disorders need a multidisciplinary approach, including collaboration between different specialists both in the assessment procedure and at follow-up.

The first aim of this study was to evaluate the association between different subgroups of autism spectrum disorders (ASDs) (childhood autism, Asperger syndrome, and pervasive developmental disorder/pervasive developmental disorder - not otherwise specified [PDD/PDD-NOS]) and congenital anomalies. Second, we assessed the association among intellectually disabled children with ASDs in the subgroups of childhood autism and PDD/PDD-NOS. Nationwide population-based register data for children with a diagnosis of ASD (n=4449; 3548 males, 901 females) were collected during years 1987-2000 from the Finnish Hospital Discharge Register. Data on congenital anomalies were derived from the National Register of Congenital Malformations. Conditional logistic regression models were used as a statistical method. The association between ASD subgroups and congenital anomalies was stratified by the presence or absence of intellectual disability. Congenital anomalies occurred more frequently in all subgroups of ASD than in comparison participants (adjusted odds ratio [OR] for major congenital anomalies 1.8, 95% confidence interval [CI] 1.5-2.2, p<0.001). The association between congenital anomalies and childhood autism (OR 2.4, 95% CI 1.6-3.6, p<0.001) and between congenital anomalies and PDD/PDD-NOS (OR 3.7, 95% CI 2.4-5.7, p<0.001) among children with an intellectual disability was strong but remained significant also without intellectual disability (childhood autism: OR 1.7, 95% CI 1.3-2.3, p<0.001; PDD/PDD-NOS: OR 2.3, 95% CI 1.9-2.8, p<0.001). The results suggest a significant association between ASDs and congenital anomalies regardless of the ASD subgroup. The association between childhood autism and PDD/PDD-NOS and congenital anomalies is stronger among children with intellectual disability is stronger than among those without intellectual disability. These results may have relevance in examining early risk factors in autism during fetal neurodevelopment.

Advances in Our Understanding of Genetic Risk Factors for Autism Spectrum Disorders

COVID-19 has greatly affected the population, especially those in the low socio-economic group, including residents of the People's Housing Project (PPR) in Malaysia. Adolescents residing in PPR communities are among the most vulnerable groups of young people in urban areas, given their pre-existing conditions of vulnerability, face even greater challenges due to the pandemic. Understanding their mental health and coping strategies is vital to grasp how the pandemic impacts their well-being. Hence, this study aims to explore the coping strategies and barriers to help-seeking behaviour among adolescents living in the Malaysia's PPR communities, focusing on the unique mental health challenges exacerbated by the COVID-19 pandemic. Given the socio-economic vulnerabilities and the heightened mental health challenges during the pandemic, this study provides critical insights into how adolescents in PPR communities navigate psychological distress and mental health support. This qualitative study used a phenomenological research design and was conducted from January to December 2022, involving 47 adolescents aged 10 to 17 years old from 37 PPRs in the Klang Valley. Participants were recruited using the purposive sampling method as this study purposely selected adolescents with moderate, moderately severe, and severe for PHQ-9 and/or moderate and severe for GAD-7 based on the screening. Participants who agreed to participate were recruited (with consent from parents/guardians) and interviews were set at the participants' convenience. Data were collected using a semi-structured interview guide to conduct the in-depth interviews (IDI). After each IDI session, the recorded interviews were transcribed. Data from the voice recorder were stored on a password-protected computer, and participants' names were replaced with specific codes to ensure confidentiality. The researchers coded all transcripts independently. The transcripts were analysed inductively using a thematic approach to identify recurring themes. From the 37 PPRs, 194 adolescents were identified as having psychological distress based on the screening. Among them, 47 agreed to participate in the IDIs, which revealed that these adolescents utilized mainly maladaptive coping strategies, such as avoidance (cognitive distancing, externalization, and internalization), self-harm, vaping, and smoking to deal with stressors related to COVID-19. As for hindrances to help-seeking, three themes were identified such as lack of trust, perceived ineffectiveness of support, and personality. Psychological distress among adolescents was prevalent during the pandemic, and they faced hindrances in seeking help. Coping strategies have been identified to help adolescents manage their psychological distress during the pandemic. It is concerning that some had resorted to maladaptive coping mechanisms. These findings emphasized the need for targeted mental health interventions and support systems tailored to vulnerable communities. These interventions could inform policies aimed at strengthening mental health services, fostering better coping strategies and promoting help-seeking behaviours among adolescents in socio-economically challenged communities.

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The COVID-19 pandemic has impacted mental health at a population level. Families of children with health vulnerabilities have been disproportionately affected by pandemic-related policies and service disruptions as they substantially rely on the health and social care system. We elicited the impact of the COVID-19 pandemic on children with health and disability-related vulnerabilities, their families, and their health care providers (HCPs). Children with diverse health vulnerabilities (cardiac transplantation, respiratory conditions, sickle cell disease, autism spectrum disorder, mental health issues, and nearing the end of life due to a range of underlying causes), as well as their parents and HCPs, participated in semi-structured interviews. Data were analyzed using qualitative content analysis in determining themes related to impact and recommendations for practice improvement. A total of 262 participants (30 children, 76 parents, 156 HCPs) were interviewed. Children described loneliness and isolation; parents described feeling burnt out; and HCPs described strain and a sense of moral distress. Themes reflected mental health impacts on children, families, and HCPs, with insufficient resources to support mental health; organizational and policy influences that shaped service delivery; and recommendations to enhance service delivery. Children with health vulnerabilities, their families and HCPs incurred profound mental health impacts due to pandemic-imposed public health restrictions and care shifts. Recommendations include the development and application of targeted pandemic information and mental health supports. These findings amplify the need for capacity building, including proactive strategies and mitigative planning in the event of a future pandemic.

Home movies of infants later diagnosed as having early childhood psychoses were compared with movies of normal control infants. A scale of attachment indicators measured mother-infant dyadic avoidance and reciprocity in the first 6 months of the infant's life. Three judges blind to diagnosis saw a general trend to dampened interaction for prepsychotic infants and their mothers. Prepsychotic and normal infants were not significantly different in touching and eye gaze behaviors, whereas mothers of prepsychotic infants exhibited less adequate eye gaze and touching than mothers of normal infants.

Autism spectrum disorders (ASD) represent a phenotypically heterogeneous group of patients that strongly intertwine with other neurodevelopmental disorders (NDDs), with genetics playing a significant role in their etiology. Whole exome sequencing (WES) has become predominant in molecular diagnostics for ASD by considerably increasing the diagnostic yield. However, the proportion of undiagnosed patients still remains high due to complex clinical presentation, reduced penetrance, and lack of segregation analysis or clinical information. Thus, reverse phenotyping, where we first identified a possible genetic cause and then determine its clinical relevance, has been shown to be a more efficient approach. WES was performed on 147 Slovenian pediatric patients with suspected ASD. Data analysis was focused on identifying ultrarare or “single event” variants in ASD-associated genes and further expanded to NDD-associated genes. Protein function and gene prioritization were performed on detected clinically relevant variants to determine their role in ASD etiology and phenotype. Reverse phenotyping revealed a pathogenic or likely pathogenic variant in ASD-associated genes in 20.4% of patients, with subsequent segregation analysis indicating that 14 were de novo variants and 1 was presumed compound heterozygous. The diagnostic yield was further increased by 2.7% by the analysis of ultrarare or “single event” variants in all NDD-associated genes. Protein function analysis established that genes in which variants of unknown significance (VUS) were detected were predominantly the cause of intellectual disability (ID), and in most cases, features of ASD as well. Using such an approach, variants in rarely described ASD-associated genes, such as SIN3B, NR4A2, and GRIA1, were detected. By expanding the analysis to include functionally similar NDD genes, variants in KCNK9, GNE, and other genes were identified. These would probably have been missed by classic genotype–phenotype analysis. Our study thus demonstrates that in patients with ASD, analysis of ultrarare or “single event” variants obtained using WES with the inclusion of functionally similar genes and reverse phenotyping obtained a higher diagnostic yield despite limited clinical data. The present study also demonstrates that most of the causative genes in our cohort were involved in the syndromic form of ASD and confirms their comorbidity with other developmental disorders.

To assess to what extent child neurologists (CNs) and developmental-behavioral pediatricians (DBPs) order diagnostic tests that are not recommended/indicated and/or fail to order tests that are recommended/indicated when evaluating children with an autism spectrum disorder (ASD). CNs and DBPs in the United States were asked which laboratory tests they would "routinely order" for a preschool child with ASD and IQ = 58 (ASD + Intellectual Disability (ID)), and a preschool child with ASD and IQ = 85 (ASD-ID). Chi-square tests were performed to identify differences (CNs vs DBPs) in laboratory testing. The sample consisted of 267 respondents (127 CN's; 140 DBPs). When evaluating ASD + ID or ASD - ID, inappropriate tests (≥1) were ordered by 76.8% and 76.4% of MDs, respectively. There was no significant difference between specialties in compliance with evaluation guidelines for ASD + ID (CN = 20.5% vs DBP = 16.4%; χ = 0.73). No significant differences were noted (DBP vs CN) regarding the percent ordering inappropriate tests for either clinical case or within each specialty when comparing testing for ASD + ID versus ASD - ID. Relative to DBPs, CNs were more likely to order EEGs and MRIs when evaluating children with ASD + ID or ASD - ID. 10% and 40% of respondents did not order any recommended genetic tests when evaluating ASD + ID and ASD - ID, respectively. When evaluating children with ASD, many CNs and DBPs fail to order tests that should be routinely performed and often order tests that are not routinely indicated yet are neither benign nor inexpensive. Recommended molecular genetic tests are often not ordered. Clinical guidelines must be updated and better promulgated.

Background: Several studies have reported contradictory results regarding the benefits of music interventions in children and adolescents with neurodevelopmental disorders (NDDs), including autism spectrum disorder (ASD).Methods: We performed a systematic review according to the PRISMA guidelines. We searched the Cochrane, PubMed and Medline databases from January 1970 to September 2020 to review all empirical findings, except case reports, measuring the effect of music therapy on youths with ASD, intellectual disability (ID), communication disorder (CD), developmental coordination disorder (DCD), specific learning disorder, and attention/deficit hyperactivity disorder (ADHD).Results: Thirty-nine studies (N = 1,774 participants) were included in this review (ASD: n = 22; ID: n = 7; CD and dyslexia: n = 5; DCD: n = 0; ADHD: n = 5 studies). Two main music therapies were used: educational music therapy and improvisational music therapy. A positive effect of educational music therapy on patients with ASD was reported in most controlled studies (6/7), particularly in terms of speech production. A positive effect of improvisational music therapy was reported in most controlled studies (6/8), particularly in terms of social functioning. The subgroup of patients with both ASD and ID had a higher response rate. Data are lacking for children with other NDDs, although preliminary evidence appears encouraging for educational music therapy in children with dyslexia.Discussion: Improvisational music therapy in children with NDDs appears relevant for individuals with both ASD and ID. More research should be encouraged to explore whether oral and written language skills may improve after educational music therapy, as preliminary data are encouraging.

BackgroundAustralian rural and regional communities are marked by geographic isolation and increasingly frequent and severe natural disasters such as drought, bushfires and floods. These circumstances strain the mental health of their inhabitants and jeopardise the healthy mental and emotional development of their adolescent populations. Professional mental health care in these communities is often inconsistent and un-coordinated. While substantial research has examined the barriers of young people’s mental health and help-seeking behaviours in these communities, there is a lack of research exploring what adolescents in rural and regional areas view as facilitators to their mental health and to seeking help when it is needed. This study aims to establish an in-depth understanding of those young people’s experiences and needs regarding mental health, what facilitates their help-seeking, and what kind of mental health education and support they want and find useful.MethodWe conducted a qualitative study in 11 drought-affected rural and regional communities of New South Wales, Australia. Seventeen semi-structured (14 group; 3 individual) interviews were held with 42 year 9 and 10 high school students, 14 high school staff, and 2 parents, exploring participants’ experiences of how geographical isolation and natural disasters impacted their mental health. We further examined participants’ understandings and needs regarding locally available mental health support resources and their views and experiences regarding mental illness, stigma and help-seeking.ResultsThematic analysis highlighted that, through the lens of participants, young people’s mental health and help-seeking needs would best be enabled by a well-coordinated multi-pronged community approach consisting of mental health education and support services that are locally available, free of charge, engaging, and empowering. Participants also highlighted the need to integrate young people’s existing mental health supporters such as teachers, parents and school counselling services into such a community approach, recognising their strengths, limitations and own education and support needs.ConclusionsWe propose a three-dimensional Engagement, Empowerment, Integration model to strengthen young people’s mental health development which comprises: 1) maximising young people’s emotional investment (engagement); 2) developing young people’s mental health self-management skills (empowerment); and, 3) integrating mental health education and support programs into existing community and school structures and resources (integration).

The COVID-19 pandemic has led to a devastating loss of life, staggering economic losses, and untold psychological distress for people all over the world. People with intellectual and developmental disabilities (IDD) have been impacted as well and, in many ways, they are particularly vulnerable to the pernicious effects of the pandemic. Preliminary data reported in the popular press, for example, suggest that people with IDD who contracted COVID-19 have died at more than twice the rate of that of the general population. In addition, most of the services and supports required to optimize inclusion of people with IDD in their communities were severely disrupted during the pandemic. Diagnostic and developmental evaluations to determine eligibility for services, in-home behavioral services, special education and ancillary school-based therapies, and supported employment were limited or halted completely. Indeed, the vulnerability of our systems of services and supports for people with IDD to disruption by natural and human-made disasters was made abundantly clear during the past few months.The pandemic, however, has also provided glimpses into how we could redesign services and supports for people with IDD in ways that could make them more robust and less susceptible to disruption. At the heart of this redesign is technology. At the UC Davis MIND Institute and at other academic health systems around the country, clinic visits with psychologists, psychiatrists, developmental-behavioral pediatricians, genetic counselors, and other professionals were quickly shifted to video visits through video teleconferencing technology platforms, such as Skype© and Zoom©. K-12 schools shifted to online classrooms and curricula, including for students receiving special education services. Families even took educational and fun trips to museums and zoos through virtual reality technology.I believe that a lesson from the pandemic is that technology-delivered services should be more fully developed so as to allow us to continue to support people with IDD when the next disaster makes face-to-face contact difficult. Moreover, I would encourage the widespread use of technology-delivered services as a complement to face-to-face services even during non-disaster "normal" periods of life. Technology offers the promise of making professional expertise accessible to more people, removing barriers that create health disparities based on race, ethnicity, geography, and economic circumstances, and all while providing services in cost-effective ways.At the same time, not every service will lend itself to technology-based delivery, or at least not without considerable adaptation. Anecdotal reports from many parents suggest that online classrooms for students with special needs, without consideration of how to support engagement and reduce challenging behaviors, have not been effective. So, rather than just assuming that every service can be delivered through technology, we must do the hard work of adapting the services and the technologies in an iterative process designed to provide evidence of efficacy, to develop procedures for individualization, and to show that the technology-delivered services can be brought to scale and be cost effective. And finally, there must be evidence that the technology-delivered services reduce rather than reinforce or exacerbate disparities in access that are common according to race, ethnicity, income, etc. If technology just makes getting care more convenient for those citizens who already have access to the best care and does not reach those citizens who are typically marginalized, we have failed as researchers and professionals.In the remainder of this address, I will summarize some of the research conducted by my lab group to develop a distance-delivered, parent-implemented language intervention (PILI). I will also briefly touch on a few other examples of technology-delivered services by other researchers to provide a sense of the scope of work ongoing in the field.There is considerable evidence from longitudinal correlational studies that the ways in which caregivers interact with, and talk to, children shape language development in typically developing children (Hart & Risley, 1995). Importantly, there is also evidence that the same types of parent input and interaction that optimize typical development are facilitative of language in children with developmental challenges, including those with autism spectrum disorder (ASD) or an intellectual disability (ID) associated with conditions, such as Down syndrome and fragile X syndrome (e.g., Haebig et al., 2013; Warren et al., 2010). The unique learning and behavioral challenges associated with IDD, however, make it difficult for parents and other adult caregivers to engage in these language-facilitating behaviors – behaviors toward which the adults would be naturally inclined. In no way is this to suggest that parents are at fault for their children's language problems. Instead, it is the case that the unique characteristics of the child with IDD, which slow language learning in and of themselves, also "create" an environment that is less conducive to learning than it could be. The goal of PILI is to help parents learn ways to engage their children in adaptive interactions in which the use of language-facilitating strategies is again possible. In other words, we train parents to serve as "therapists" and deliver a naturalistic intervention to their children.The PILI training is largely delivered to parents individually in their homes through video teleconferencing and other digital technologies. Once trained, parents can then engage in supportive, "therapeutic" interactions with their children at home or in other settings that they experience together in daily life. There are many potential advantages of this distance-delivered approach.The goal of PILI is to teach parents to be more "verbally responsive" and thereby more supportive of their child's language learning. A verbally responsive style of interaction entails:Verbal responsivity provides the optimal interactive context for language learning throughout development, although the implementation will differ depending of the age and developmental capacity of the child. Verbally responsive interactions between a parent and preschooler with IDD will look different in many respects from a verbally responsive interaction between a parent and an adolescent with IDD.We have developed and tested different telehealth-delivered PILIs for people with fragile X syndrome (FXS), which is the leading inherited cause of ID and the leading single-gene cause of autism spectrum disorder (ASD). The different versions were adapted according to the ages and developmental levels of the target group. At the heart of all the interventions, however, was the goal of improving outcomes for people with FXS by teaching and supporting increased verbal responsivity of parents through technology.We have developed and tested the efficacy of a distance-delivered PILI for 2- to 5-year-olds with FXS (McDuffie, Oakes et al., 2016; Oakes et al., 2015). The intervention was delivered in the context of dyadic play with objects. The participants were six boys with FXS and their biological mothers. The boys ranged in age from 27 to 43 months at the start of their participation in the intervention. All had IQs in the range of ID, and the language levels ranged from no productive language to the production of an occasional multiword utterance.The primary focus of the intervention was on increasing child communication and secondarily on improving spoken language. Parents were taught three strategies. In follow-in commenting, the parent was to describe an object, activity, or event that was the focus of the child's attention. In interpreting/expanding, the parent was to respond to a child utterance with a fuller, more adult-like version and to a child nonverbal behavior with an utterance that ascribed an intention to that behavior. In indirect prompting, the parent was to create a need for the child to communicate by offering a choice or delaying a desired activity or object. The parent was also taught strategies to reduce child challenging behaviors.Parents were taught these strategies over a period of 17 weeks. The dyads visited the clinic once per month. In these clinic visits, the clinician presented the rationale for the strategies to be learned, along with video examples. This didactic session was followed by real-time coaching by the clinician as the parent played with the child. In addition to the clinic visits, there were weekly real-time dyadic play coaching sessions delivered into the home by the clinician through video teleconferencing. Parents were given a laptop computer equipped with a webcam for these in-home sessions. There were also weekly distance observation sessions for the purpose of taking data on the dyad's progress with no coaching or feedback provided. Finally, pre- and post-treatment assessments of play interactions without coaching were conducted in the clinic. Throughout the intervention, the clinician encouraged the parent to use the targeted strategies in naturally occurring interactions throughout the day.We used a multiple baseline design in which the length of the baselines varied and the initiation of the intervention was staggered across participants so that we could be more certain that changes in parent and child behavior were due to participation in the intervention. The primary data for evaluating the efficacy of the intervention came from the observation sessions. In terms of parental behavior, the frequency of use of the targeted strategies increased during the intervention relative to baseline. In fact, there was minimal overlap for most parents in terms of their frequency of use of the strategies between baseline and treatment, although there was variability across parents in the magnitude of change. In terms of child behavior, the frequency of communication acts was greater in treatment than in baseline. This was true in general both for prompted and spontaneous acts of child communication. Increased communicativeness is important because its leads to longer and more satisfying interactions for parent and child and creates opportunities for parents to provide the child with information about the language being learned, thereby creating a positive developmental cascade.Post-treatment surveys and interviews indicated that parents found the intervention to be helpful and felt that their interactions with their children were more positive and fulfilling as a result. These latter findings are important because positive parental attitudes are likely to be a prerequisite for the continued use of the targeted strategies after participation with the clinicians and the study has ended.We have developed and tested the efficacy of a parent-implemented language intervention for preadolescents and adolescents with FXS in three studies, one a series of three single-case analyses (McDuffie, Machalicek et al., 2016), the second a small-scale randomized controlled trial (RCT; McDuffie et al., 2018), and the third an RCT of a medication with all participants also receiving distance-delivered PILI (Thurman et al., 2020). Across the studies, the participants with FXS ranged from 10 to 17 years of age, most were male, and all had IQs in the range of ID. The language skills of the people with FXS ranged from largely single-word and only occasional multiword speech to frequent use of multiword utterances, with all participants well below age-level expectations as regards language.Parents were taught to use verbally responsive language strategies; however, because of the ages of the youth with FXS, the intervention was embedded in the context of shared story telling using wordless picture books rather than play with objects. The book functioned to provide a shared conversational topic for the dyad. In contrast to the study with young children, the focus was on building language skills. The intervention was delivered entirely into the family home through video teleconferencing, with no clinic-based instructional component. The intervention began with two parent education sessions that described the rationale for the targeted strategies and video examples. The intervention then continued for 12 weeks with four types of activities each week.A different book was used each week so that the youth with FXS was not simply learning how to tell a specific story but was instead gaining more generalized language skills. The books were digitized and presented on an iPad.Parents were taught three strategies. The first was to use open-ended wh-questions, such as, "What is the boy doing?" and "How is the boy feeling?" These questions tend to elicit a verbal response and thus, draw the youth with FXS into the conversation and allow him/her to practice new language skills. The second strategy was to use expansions, which entail the parent providing a fuller, more mature version of the youth's utterance and thereby an opportunity to learn new words and syntactic forms. The third strategy was the use of fill-in-the-blank statements in which the parent started a sentence but through a rising intonation and pause conveyed the expectation that the youth with FXS was to complete the sentence. Fill-in-the blank allowed the youth with FXS to participate in the conversation in way that increased the chances of communicative success.The results across the three studies were generally consistent in supporting the efficacy of the intervention. In assessing treatment-induced changes, we examined story-telling interaction with previously unseen books with the parent at home and in the clinic, as well with an examiner in the clinic. In terms of parent behavior, participation in the treatment led to increases in the use of all three targeted strategies. In terms of the behavior of the youths with FXS, participation in the treatment led to sizable increases in a measure of expressive vocabulary (i.e., the number of different words used). The intervention also led to an increase in the length of the dyadic story-telling sessions both in time and number of story-related utterances. It was also found that changes in youth vocabulary were correlated with changes in parental behavior; that is, the more progress the parents made learning the strategies, the more progress the youth with FXS made. Finally, the same pattern of findings was observed for measures derived from parent-youth interactions at home and in the clinic and some non-targeted language skills (e.g., inferential language) also improved (Nelson et al., 2018). These data suggest that the intervention promoted growth in both mother and child behavior that was not specific to a single story or setting but more general to the dyadic shared story-telling context.At the same time, however, there were limitations in the efficacy of the intervention. First, no consistent differences related to treatment were found in the expressive syntax of the youth with FXS. This suggests the possibility that more direct and explicit instruction may be needed to improve syntax in this population. Second, the gains observed for the youth with FXS in vocabulary in their interactions with their mothers did not generalize to their interactions with the examiner in the clinic context. We believe that this lack of generalizability to the examiner-led interaction reflects the fact that unlike the scaffolding that parents were providing through their use of verbally responsive strategies, the examiner by design minimized his/her participation and provided minimal scaffolding. Thus, it may be that the vocabulary skills of the youth with FXS were still forming and not sufficiently developed so as to be used without considerable supportIn summary, a training program for parents delivered into the home through telehealth technology can lead to improvements in communication and language for people with ID across a wide age range. At the same time, however, not all aspects of language seem to benefit from the intervention in its current form and generalization of gains may be limited to highly scaffolded contexts. We plan to address these limitations in future research. We also are exploring whether the coaching can be reduced and supplemented with self-guided online modules to make the intervention more cost effective.Telehealth technology is arguably the most widely used form of technology to deliver behavioral and mental health services to people with IDD. These uses extend beyond treatment delivery of the sort I have described above to in-home assessments and evaluations. No doubt there will be an explosion of examples of other technologies and uses post-pandemic. Here are just a few examples of technologies being tested now.As we move out of the pandemic and achieve some return to "normal" life, it is imperative that we not forget the lessons we have learned over the past several months. We desperately need a more robust and equitable system of services and supports for people with IDD. Evidence-based, technology-driven approaches to delivery of services will be an important part of the solution. However, additional resources and stronger government policies that lead to more and better coordinated systems are also needed. I know that AAIDD will help lead the way forward to improve choice and quality of life for people with IDD and their families.