Autism spectrum disorder symptom expression in individuals with 3q29 deletion syndrome / Rebecca M. POLLAK in Molecular Autism, 13 (2022)  

Public ISBD [article]  inMolecular Autism > 13 (2022) . - 50 p. Titre : Autism spectrum disorder symptom expression in individuals with 3q29 deletion syndrome Type de document : texte imprimé Auteurs : Rebecca M. POLLAK, Auteur ; Jordan E. PINCUS, Auteur ; T. Lindsey BURRELL, Auteur ; Joseph F. CUBELLS, Auteur ; Cheryl KLAIMAN, Auteur ; Melissa M. MURPHY, Auteur ; Celine A. SAULNIER, Auteur ; Elaine F. WALKER, Auteur ; Stormi P. WHITE, Auteur ; Jennifer G. MULLE, Auteur Article en page(s) : 50 p. Langues : Anglais (eng) Mots-clés : Male  Female  Humans  Autism Spectrum Disorder/diagnosis/genetics  Syndrome  Social Skills  Surveys and Questionnaires  Phenotype  3q29 deletion  Adi-r  Ados-2  Autism  Copy number variants  Developmental delay  Genomic disorder  Psychiatric genetics  other authors report no conflicts of interest. Index. décimale : PER Périodiques Résumé : BACKGROUND: The 1.6 Mb 3q29 deletion is associated with neurodevelopmental and neuropsychiatric phenotypes, including a 19-fold increased risk for autism spectrum disorder (ASD). Previous work by our team identified elevated social disability in this population via parent-report questionnaires. However, clinical features of ASD in this population have not been explored in detail. METHODS: Thirty-one individuals with 3q29 deletion syndrome (3q29del, 61.3% male) were evaluated using two gold-standard clinical ASD evaluations: the Autism Diagnostic Observation Schedule, Second Edition (ADOS-2), and the Autism Diagnostic Interview, Revised (ADI-R). Four matched comparators for each subject were ascertained from the National Database for Autism Research. Item-level scores on the ADOS-2 and ADI-R were compared between subjects with 3q29del and matched comparators. RESULTS: Subjects with 3q29del and no ASD (3q29del-ASD) had greater evidence of social disability compared to typically developing (TD) comparison subjects across the ADOS-2. Subjects with 3q29del and ASD (3q29del + ASD) were largely indistinguishable from non-syndromic ASD (nsASD) subjects on the ADOS-2. 3q29del + ASD performed significantly better on social communication on the ADI-R than nsASD (3q29 + ASD mean=11.36; nsASD mean=15.70; p=0.01), and this was driven by reduced deficits in nonverbal communication (3q29 + ASD mean=1.73; nsASD mean=3.63; p=0.03). 3q29del + ASD reported significantly later age at the first two-word phrase compared to nsASD (3q29del + ASD mean=43.89 months; nsASD mean=37.86 months; p=0.01). However, speech delay was not related to improved nonverbal communication in 3q29del + ASD. LIMITATIONS: There were not enough TD comparators with ADI-R data in NDAR to include in the present analysis. Additionally, our relatively small sample size made it difficult to assess race and ethnicity effects. CONCLUSIONS: 3q29del is associated with significant social disability, irrespective of ASD diagnosis. 3q29del + ASD have similar levels of social disability to nsASD, while 3q29del-ASD have significantly increased social disability compared to TD individuals. However, social communication is reasonably well preserved in 3q29del + ASD relative to nsASD. It is critical that verbal ability and social disability be examined separately in this population to ensure equal access to ASD and social skills evaluations and services. En ligne : http://dx.doi.org/10.1186/s13229-022-00533-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=491 [article] Autism spectrum disorder symptom expression in individuals with 3q29 deletion syndrome [texte imprimé] / Rebecca M. POLLAK, Auteur ; Jordan E. PINCUS, Auteur ; T. Lindsey BURRELL, Auteur ; Joseph F. CUBELLS, Auteur ; Cheryl KLAIMAN, Auteur ; Melissa M. MURPHY, Auteur ; Celine A. SAULNIER, Auteur ; Elaine F. WALKER, Auteur ; Stormi P. WHITE, Auteur ; Jennifer G. MULLE, Auteur . - 50 p. Langues : Anglais (eng) in Molecular Autism > 13 (2022) . - 50 p. Mots-clés : Male  Female  Humans  Autism Spectrum Disorder/diagnosis/genetics  Syndrome  Social Skills  Surveys and Questionnaires  Phenotype  3q29 deletion  Adi-r  Ados-2  Autism  Copy number variants  Developmental delay  Genomic disorder  Psychiatric genetics  other authors report no conflicts of interest. Index. décimale : PER Périodiques Résumé : BACKGROUND: The 1.6 Mb 3q29 deletion is associated with neurodevelopmental and neuropsychiatric phenotypes, including a 19-fold increased risk for autism spectrum disorder (ASD). Previous work by our team identified elevated social disability in this population via parent-report questionnaires. However, clinical features of ASD in this population have not been explored in detail. METHODS: Thirty-one individuals with 3q29 deletion syndrome (3q29del, 61.3% male) were evaluated using two gold-standard clinical ASD evaluations: the Autism Diagnostic Observation Schedule, Second Edition (ADOS-2), and the Autism Diagnostic Interview, Revised (ADI-R). Four matched comparators for each subject were ascertained from the National Database for Autism Research. Item-level scores on the ADOS-2 and ADI-R were compared between subjects with 3q29del and matched comparators. RESULTS: Subjects with 3q29del and no ASD (3q29del-ASD) had greater evidence of social disability compared to typically developing (TD) comparison subjects across the ADOS-2. Subjects with 3q29del and ASD (3q29del + ASD) were largely indistinguishable from non-syndromic ASD (nsASD) subjects on the ADOS-2. 3q29del + ASD performed significantly better on social communication on the ADI-R than nsASD (3q29 + ASD mean=11.36; nsASD mean=15.70; p=0.01), and this was driven by reduced deficits in nonverbal communication (3q29 + ASD mean=1.73; nsASD mean=3.63; p=0.03). 3q29del + ASD reported significantly later age at the first two-word phrase compared to nsASD (3q29del + ASD mean=43.89 months; nsASD mean=37.86 months; p=0.01). However, speech delay was not related to improved nonverbal communication in 3q29del + ASD. LIMITATIONS: There were not enough TD comparators with ADI-R data in NDAR to include in the present analysis. Additionally, our relatively small sample size made it difficult to assess race and ethnicity effects. CONCLUSIONS: 3q29del is associated with significant social disability, irrespective of ASD diagnosis. 3q29del + ASD have similar levels of social disability to nsASD, while 3q29del-ASD have significantly increased social disability compared to TD individuals. However, social communication is reasonably well preserved in 3q29del + ASD relative to nsASD. It is critical that verbal ability and social disability be examined separately in this population to ensure equal access to ASD and social skills evaluations and services. En ligne : http://dx.doi.org/10.1186/s13229-022-00533-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=491

Correction: A Survey of Community Providers on Feeding Problems in Autism Spectrum Disorder / Teresa Lindsey BURRELL in Journal of Autism and Developmental Disorders, 53-11 (November 2023)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 53-11 (November 2023) . - p.4496-4496 Titre : Correction: A Survey of Community Providers on Feeding Problems in Autism Spectrum Disorder Type de document : texte imprimé Auteurs : Teresa Lindsey BURRELL, Auteur ; William G. SHARP, Auteur ; Scott E. GILLESPIE, Auteur ; Katherine PICKARD, Auteur ; Susan BRASHER, Auteur ; Derianne BUCKLEY, Auteur ; Lawrence SCAHILL, Auteur Article en page(s) : p.4496-4496 Langues : Anglais (eng) Index. décimale : PER Périodiques En ligne : https://doi.org/10.1007/s10803-023-06055-x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=512 [article] Correction: A Survey of Community Providers on Feeding Problems in Autism Spectrum Disorder [texte imprimé] / Teresa Lindsey BURRELL, Auteur ; William G. SHARP, Auteur ; Scott E. GILLESPIE, Auteur ; Katherine PICKARD, Auteur ; Susan BRASHER, Auteur ; Derianne BUCKLEY, Auteur ; Lawrence SCAHILL, Auteur . - p.4496-4496. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 53-11 (November 2023) . - p.4496-4496 Index. décimale : PER Périodiques En ligne : https://doi.org/10.1007/s10803-023-06055-x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=512

Examining adaptations necessary to support the implementation of a parent-mediated intervention for children with autism spectrum disorder and moderate feeding problems / Katherine PICKARD in Autism, 27-8 (November 2023)  

Public ISBD [article]  inAutism > 27-8 (November 2023) . - p.2434-2445 Titre : Examining adaptations necessary to support the implementation of a parent-mediated intervention for children with autism spectrum disorder and moderate feeding problems Type de document : texte imprimé Auteurs : Katherine PICKARD, Auteur ; T. Lindsey BURRELL, Auteur ; Susan BRASHER, Auteur ; Derianne BUCKLEY, Auteur ; Scott E. GILLESPIE, Auteur ; William SHARP, Auteur ; Lawrence SCAHILL, Auteur Article en page(s) : p.2434-2445 Mots-clés : autism spectrum disorders  implementation  nutrition/feeding  parent-mediated intervention  qualitative research Index. décimale : PER Périodiques Résumé : Moderate feeding problems affect many children with autism spectrum disorder and often co-occur with disruptive mealtime behaviors. Although parent-mediated interventions have shown promise to support feeding problems, research has primarily occurred within randomized controlled trials when delivered by highly trained clinicians in specialty clinics. Thus, the alignment of these approaches within community settings is unclear. To address this limitation, this study explored adaptations to a parent-mediated intervention, Managing Eating Aversions and Limited Variety (i.e. MEAL Plan), to enhance its adoption, implementation, and sustainment within community settings, including factors that may support or hinder its translation. Participants were 14 multidisciplinary providers who attended one of the three workgroups that included semi-structured focus groups regarding the fit of MEAL Plan in their practice setting. Thematic analysis was used to determine emergent themes, which fell into the domains of content, contextual considerations, and training needs. Specific themes included the appropriateness MEAL Plan for autistic and non-autistic children, adaptations to MEAL Plan delivery, billing considerations, agency-level support, and the format of ongoing training and consultation. By proactively responding to these factors, it may be possible to enhance the implementation and sustainability of parent-mediated interventions, including MEAL Plan, within community practices that support autistic children.Lay abstractModerate feeding problems and disruptive mealtime behaviors are common in children with autism spectrum disorder. Although parent-mediated interventions are able to support feeding problems in autistic children, most research has occurred within specialty clinics when delivered by highly trained clinicians. Thus, the fit of these interventions within community settings is not clear. To address this limitation, this study explored adaptations to a parent-mediated intervention, Managing Eating Aversions and Limited Variety (i.e. MEAL Plan), to improve its fit and use within community settings. Participants were 14 multidisciplinary providers who attended one of the three intensive workgroups that included focus groups about the fit of MEAL Plan in their practice setting. Qualitative analysis was used to determine the main themes that came up within the focus groups. Specific themes included the appropriateness MEAL Plan for autistic and non-autistic children, how providers might adapt their delivery of MEAL Plan, billing and insurance considerations, administrator support for MEAL Plan, and the content and format of ongoing training and consultation. By proactively considering and responding to these factors, it may be possible to enhance MEAL Plan so that it is better able to be delivered and sustained within community practices that support autistic children. En ligne : http://dx.doi.org/10.1177/13623613231166181 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=513 [article] Examining adaptations necessary to support the implementation of a parent-mediated intervention for children with autism spectrum disorder and moderate feeding problems [texte imprimé] / Katherine PICKARD, Auteur ; T. Lindsey BURRELL, Auteur ; Susan BRASHER, Auteur ; Derianne BUCKLEY, Auteur ; Scott E. GILLESPIE, Auteur ; William SHARP, Auteur ; Lawrence SCAHILL, Auteur . - p.2434-2445. in Autism > 27-8 (November 2023) . - p.2434-2445 Mots-clés : autism spectrum disorders  implementation  nutrition/feeding  parent-mediated intervention  qualitative research Index. décimale : PER Périodiques Résumé : Moderate feeding problems affect many children with autism spectrum disorder and often co-occur with disruptive mealtime behaviors. Although parent-mediated interventions have shown promise to support feeding problems, research has primarily occurred within randomized controlled trials when delivered by highly trained clinicians in specialty clinics. Thus, the alignment of these approaches within community settings is unclear. To address this limitation, this study explored adaptations to a parent-mediated intervention, Managing Eating Aversions and Limited Variety (i.e. MEAL Plan), to enhance its adoption, implementation, and sustainment within community settings, including factors that may support or hinder its translation. Participants were 14 multidisciplinary providers who attended one of the three workgroups that included semi-structured focus groups regarding the fit of MEAL Plan in their practice setting. Thematic analysis was used to determine emergent themes, which fell into the domains of content, contextual considerations, and training needs. Specific themes included the appropriateness MEAL Plan for autistic and non-autistic children, adaptations to MEAL Plan delivery, billing considerations, agency-level support, and the format of ongoing training and consultation. By proactively responding to these factors, it may be possible to enhance the implementation and sustainability of parent-mediated interventions, including MEAL Plan, within community practices that support autistic children.Lay abstractModerate feeding problems and disruptive mealtime behaviors are common in children with autism spectrum disorder. Although parent-mediated interventions are able to support feeding problems in autistic children, most research has occurred within specialty clinics when delivered by highly trained clinicians. Thus, the fit of these interventions within community settings is not clear. To address this limitation, this study explored adaptations to a parent-mediated intervention, Managing Eating Aversions and Limited Variety (i.e. MEAL Plan), to improve its fit and use within community settings. Participants were 14 multidisciplinary providers who attended one of the three intensive workgroups that included focus groups about the fit of MEAL Plan in their practice setting. Qualitative analysis was used to determine the main themes that came up within the focus groups. Specific themes included the appropriateness MEAL Plan for autistic and non-autistic children, how providers might adapt their delivery of MEAL Plan, billing and insurance considerations, administrator support for MEAL Plan, and the content and format of ongoing training and consultation. By proactively considering and responding to these factors, it may be possible to enhance MEAL Plan so that it is better able to be delivered and sustained within community practices that support autistic children. En ligne : http://dx.doi.org/10.1177/13623613231166181 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=513

Exploration of Treatment Response in Parent Training for Children with Autism Spectrum Disorder and Moderate Food Selectivity / T. Lindsey BURRELL in Journal of Autism and Developmental Disorders, 53-1 (January 2023)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 53-1 (January 2023) . - p.229-235 Titre : Exploration of Treatment Response in Parent Training for Children with Autism Spectrum Disorder and Moderate Food Selectivity Type de document : texte imprimé Auteurs : T. Lindsey BURRELL, Auteur ; Lawrence SCAHILL, Auteur ; Nadratu NUHU, Auteur ; Scott E. GILLESPIE, Auteur ; William SHARP, Auteur Article en page(s) : p.229-235 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : Managing Eating Aversions and Limited Variety (MEAL) Plan is a structured parent-mediated intervention for children with autism spectrum disorder and moderate food selectivity. Our previously reported group-based clinical trial revealed a positive treatment response rate of 47.3%. Although encouraging, this response rate raises questions about factors that may affect treatment outcomes. Here, we examine the impact of child and parent characteristics and feeding behaviors on treatment response. Higher maternal education and higher child communication abilities at baseline were associated with positive treatment response. Improvement in sitting at the table and reductions in disruptive mealtime behavior promoted treatment success. Results also suggest that individually delivered MEAL Plan may offer more flexibility than group-based intervention for some parents. En ligne : https://doi.org/10.1007/s10803-021-05406-w Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=493 [article] Exploration of Treatment Response in Parent Training for Children with Autism Spectrum Disorder and Moderate Food Selectivity [texte imprimé] / T. Lindsey BURRELL, Auteur ; Lawrence SCAHILL, Auteur ; Nadratu NUHU, Auteur ; Scott E. GILLESPIE, Auteur ; William SHARP, Auteur . - p.229-235. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 53-1 (January 2023) . - p.229-235 Index. décimale : PER Périodiques Résumé : Managing Eating Aversions and Limited Variety (MEAL) Plan is a structured parent-mediated intervention for children with autism spectrum disorder and moderate food selectivity. Our previously reported group-based clinical trial revealed a positive treatment response rate of 47.3%. Although encouraging, this response rate raises questions about factors that may affect treatment outcomes. Here, we examine the impact of child and parent characteristics and feeding behaviors on treatment response. Higher maternal education and higher child communication abilities at baseline were associated with positive treatment response. Improvement in sitting at the table and reductions in disruptive mealtime behavior promoted treatment success. Results also suggest that individually delivered MEAL Plan may offer more flexibility than group-based intervention for some parents. En ligne : https://doi.org/10.1007/s10803-021-05406-w Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=493

Feasibility of Group Parent Training for Children with Autism Spectrum Disorder and Disruptive Behavior: A Demonstration Pilot / T. Lindsey BURRELL in Journal of Autism and Developmental Disorders, 50-11 (November 2020)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 50-11 (November 2020) . - p.3883-3894 Titre : Feasibility of Group Parent Training for Children with Autism Spectrum Disorder and Disruptive Behavior: A Demonstration Pilot Type de document : texte imprimé Auteurs : T. Lindsey BURRELL, Auteur ; Valentina POSTORINO, Auteur ; Lawrence SCAHILL, Auteur ; Hannah M. REA, Auteur ; Scott E. GILLESPIE, Auteur ; Andrea Nichole EVANS, Auteur ; Karen E. BEARSS, Auteur Article en page(s) : p.3883-3894 Langues : Anglais (eng) Mots-clés : Autism spectrum disorder  Disruptive behaviors  Feasibility  Group intervention  Parent training Index. décimale : PER Périodiques Résumé : Delivery of interventions in a group format is a potential solution to limited access to specialized services for children with autism spectrum disorder (ASD). We conducted an open feasibility trial of group-based RUBI parent training in 18 children (mean age 6.12 ± 1.95 years) with ASD and disruptive behaviors. Parents participated in one of five groups (3 to 4 parents per group). Eighty-three percent of participants completed the 24-week trial. Session attendance was moderate (74.2%). All parents indicated that they would recommend the treatment. Therapists demonstrated 98.8% fidelity to the manual. Eleven of 18 (64.7%) participants were rated as much/very much improved by an independent evaluator at Week 24. Preliminary efficacy findings justify further study. En ligne : http://dx.doi.org/10.1007/s10803-020-04427-1 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=432 [article] Feasibility of Group Parent Training for Children with Autism Spectrum Disorder and Disruptive Behavior: A Demonstration Pilot [texte imprimé] / T. Lindsey BURRELL, Auteur ; Valentina POSTORINO, Auteur ; Lawrence SCAHILL, Auteur ; Hannah M. REA, Auteur ; Scott E. GILLESPIE, Auteur ; Andrea Nichole EVANS, Auteur ; Karen E. BEARSS, Auteur . - p.3883-3894. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 50-11 (November 2020) . - p.3883-3894 Mots-clés : Autism spectrum disorder  Disruptive behaviors  Feasibility  Group intervention  Parent training Index. décimale : PER Périodiques Résumé : Delivery of interventions in a group format is a potential solution to limited access to specialized services for children with autism spectrum disorder (ASD). We conducted an open feasibility trial of group-based RUBI parent training in 18 children (mean age 6.12 ± 1.95 years) with ASD and disruptive behaviors. Parents participated in one of five groups (3 to 4 parents per group). Eighty-three percent of participants completed the 24-week trial. Session attendance was moderate (74.2%). All parents indicated that they would recommend the treatment. Therapists demonstrated 98.8% fidelity to the manual. Eleven of 18 (64.7%) participants were rated as much/very much improved by an independent evaluator at Week 24. Preliminary efficacy findings justify further study. En ligne : http://dx.doi.org/10.1007/s10803-020-04427-1 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=432

Feasibility of Parent Training via Telehealth for Children with Autism Spectrum Disorder and Disruptive Behavior: A Demonstration Pilot / Karen E. BEARSS in Journal of Autism and Developmental Disorders, 48-4 (April 2018)  

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The Autism MEAL Plan: A parent-training curriculum to manage eating aversions and low intake among children with autism / William G. SHARP in Autism, 18-6 (August 2014)  

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Visual-Motor Integration Deficits in 3q29 Deletion Syndrome / T. Lindsey BURRELL ; Joseph F. CUBELLS ; Cheryl KLAIMAN ; Melissa M. MURPHY ; Celine A. SAULNIER ; Elaine F. WALKER ; Stormi Pulver WHITE ; Jennifer G. MULLE in Journal of Autism and Developmental Disorders, 54-8 (August 2024)  

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