Behavioral Markers of Emergent Stranger Anxiety in Infants and Toddlers with Fragile X Syndrome / Bridgette TONNSEN in Journal of Autism and Developmental Disorders, 47-11 (November 2017)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 47-11 (November 2017) . - p.3646-3658 Titre : Behavioral Markers of Emergent Stranger Anxiety in Infants and Toddlers with Fragile X Syndrome Type de document : texte imprimé Auteurs : Bridgette TONNSEN, Auteur ; Jessica F. SCHERR, Auteur ; Debra REISINGER, Auteur ; Jacqueline ROBERTS, Auteur Article en page(s) : p.3646-3658 Langues : Anglais (eng) Mots-clés : Anxiety  Autism  Behavior  Fragile X syndrome  Longitudinal  Social approach Index. décimale : PER Périodiques Résumé : Studying anxiety in neurogenetic syndromes may inform the intersection of biological and developmental risks, facilitating effective and targeted interventions. We longitudinally examined stranger fear in infants and toddlers with fragile X syndrome (FXS; n = 46) and typical controls (n = 33), as well as associations between observed stranger fear and rating scales of anxiety, withdrawal and autism features within FXS. Results indicated atypical facial fear in FXS, although facial fear did not index anxiety, autistic symptoms or social withdrawal. Instead, lower withdrawal was associated with decreased distress vocalizations across age, and higher autistic symptoms were associated with lower intensity escape behaviors. Early stranger fear in FXS reflects both typical and atypical dimensions and may help index emergence of social anxiety in this population. En ligne : http://dx.doi.org/10.1007/s10803-017-3270-6 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=325 [article] Behavioral Markers of Emergent Stranger Anxiety in Infants and Toddlers with Fragile X Syndrome [texte imprimé] / Bridgette TONNSEN, Auteur ; Jessica F. SCHERR, Auteur ; Debra REISINGER, Auteur ; Jacqueline ROBERTS, Auteur . - p.3646-3658. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 47-11 (November 2017) . - p.3646-3658 Mots-clés : Anxiety  Autism  Behavior  Fragile X syndrome  Longitudinal  Social approach Index. décimale : PER Périodiques Résumé : Studying anxiety in neurogenetic syndromes may inform the intersection of biological and developmental risks, facilitating effective and targeted interventions. We longitudinally examined stranger fear in infants and toddlers with fragile X syndrome (FXS; n = 46) and typical controls (n = 33), as well as associations between observed stranger fear and rating scales of anxiety, withdrawal and autism features within FXS. Results indicated atypical facial fear in FXS, although facial fear did not index anxiety, autistic symptoms or social withdrawal. Instead, lower withdrawal was associated with decreased distress vocalizations across age, and higher autistic symptoms were associated with lower intensity escape behaviors. Early stranger fear in FXS reflects both typical and atypical dimensions and may help index emergence of social anxiety in this population. En ligne : http://dx.doi.org/10.1007/s10803-017-3270-6 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=325

Brief Report: Intranasal Ketamine in Adolescents and Young Adults with Autism Spectrum Disorder-Initial Results of a Randomized, Controlled, Crossover, Pilot Study / Logan K. WINK in Journal of Autism and Developmental Disorders, 51-4 (April 2021)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 51-4 (April 2021) . - p.1392-1399 Titre : Brief Report: Intranasal Ketamine in Adolescents and Young Adults with Autism Spectrum Disorder-Initial Results of a Randomized, Controlled, Crossover, Pilot Study Type de document : texte imprimé Auteurs : Logan K. WINK, Auteur ; Debra L. REISINGER, Auteur ; Paul S. HORN, Auteur ; Rebecca C. SHAFFER, Auteur ; Kaela O'BRIEN, Auteur ; Lauren M. SCHMITT, Auteur ; Kelli R. DOMINICK, Auteur ; Ernest V. PEDAPATI, Auteur ; Craig ERICKSON, Auteur Article en page(s) : p.1392-1399 Langues : Anglais (eng) Mots-clés : Autism  Clinical trial  Ketamine Index. décimale : PER Périodiques Résumé : Dysregulation of glutamate neurotransmission plays a critical role in autism spectrum disorder (ASD) pathophysiology and is a primary target for core deficit research treatment trials. The mechanism of action of ketamine has striking overlap with the theory of ASD as a disorder of synaptic communication and neuronal networks. This two-dose, double-blind, placebo controlled, cross-over pilot trial of intranasal (IN) ketamine targeting core social impairment included individuals with ASD (N = 21) between 14 and 29 years. Participants were randomized to received two doses of IN ketamine (30 and 50 mg) and two doses of matching placebo. No significant impact was noted on the Aberrant Behavior Checklist Social Withdraw subscale. The IN ketamine was well tolerated, with only transient mild adverse effects. En ligne : http://dx.doi.org/10.1007/s10803-020-04542-z Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=445 [article] Brief Report: Intranasal Ketamine in Adolescents and Young Adults with Autism Spectrum Disorder-Initial Results of a Randomized, Controlled, Crossover, Pilot Study [texte imprimé] / Logan K. WINK, Auteur ; Debra L. REISINGER, Auteur ; Paul S. HORN, Auteur ; Rebecca C. SHAFFER, Auteur ; Kaela O'BRIEN, Auteur ; Lauren M. SCHMITT, Auteur ; Kelli R. DOMINICK, Auteur ; Ernest V. PEDAPATI, Auteur ; Craig ERICKSON, Auteur . - p.1392-1399. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 51-4 (April 2021) . - p.1392-1399 Mots-clés : Autism  Clinical trial  Ketamine Index. décimale : PER Périodiques Résumé : Dysregulation of glutamate neurotransmission plays a critical role in autism spectrum disorder (ASD) pathophysiology and is a primary target for core deficit research treatment trials. The mechanism of action of ketamine has striking overlap with the theory of ASD as a disorder of synaptic communication and neuronal networks. This two-dose, double-blind, placebo controlled, cross-over pilot trial of intranasal (IN) ketamine targeting core social impairment included individuals with ASD (N = 21) between 14 and 29 years. Participants were randomized to received two doses of IN ketamine (30 and 50 mg) and two doses of matching placebo. No significant impact was noted on the Aberrant Behavior Checklist Social Withdraw subscale. The IN ketamine was well tolerated, with only transient mild adverse effects. En ligne : http://dx.doi.org/10.1007/s10803-020-04542-z Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=445

Health Related Quality of Life in Autistic Youth and Their Families / Rebecca C. SHAFFER ; Lauren M. SCHMITT ; Marika C. COFFMAN ; Paul S. HORN ; Debra L. REISINGER ; Craig ERICKSON in Journal of Autism and Developmental Disorders, 55-6 (June 2025)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 55-6 (June 2025) . - p.1982-1987 Titre : Health Related Quality of Life in Autistic Youth and Their Families Type de document : texte imprimé Auteurs : Rebecca C. SHAFFER, Auteur ; Lauren M. SCHMITT, Auteur ; Marika C. COFFMAN, Auteur ; Paul S. HORN, Auteur ; Debra L. REISINGER, Auteur ; Craig ERICKSON, Auteur Article en page(s) : p.1982-1987 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : The construct Quality of Life (QoL) involves a range of factors related to one?s well-being. Individuals on the autism spectrum have been previously reported to have lower QoL. The purpose of the present study is to examine QoL in autistic individuals and their families and to evaluate associations between QoL and measures of functioning using the PedsQL 4.0. En ligne : https://doi.org/10.1007/s10803-023-06054-y Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=556 [article] Health Related Quality of Life in Autistic Youth and Their Families [texte imprimé] / Rebecca C. SHAFFER, Auteur ; Lauren M. SCHMITT, Auteur ; Marika C. COFFMAN, Auteur ; Paul S. HORN, Auteur ; Debra L. REISINGER, Auteur ; Craig ERICKSON, Auteur . - p.1982-1987. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 55-6 (June 2025) . - p.1982-1987 Index. décimale : PER Périodiques Résumé : The construct Quality of Life (QoL) involves a range of factors related to one?s well-being. Individuals on the autism spectrum have been previously reported to have lower QoL. The purpose of the present study is to examine QoL in autistic individuals and their families and to evaluate associations between QoL and measures of functioning using the PedsQL 4.0. En ligne : https://doi.org/10.1007/s10803-023-06054-y Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=556

Parent-Reported Outcome Measures for Individuals with Fragile X Syndrome: Clinically Meaningful Change Thresholds / Meredith A. NELSON in Journal of Autism and Developmental Disorders, 56-4 (April 2026)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 56-4 (April 2026) . - p.1658-1670 Titre : Parent-Reported Outcome Measures for Individuals with Fragile X Syndrome: Clinically Meaningful Change Thresholds Type de document : texte imprimé Auteurs : Meredith A. NELSON, Auteur ; Lauren M. SCHMITT, Auteur ; Paul S. HORN, Auteur ; Elizabeth BERRY-KRAVIS, Auteur ; David HESSL, Auteur ; Rebecca C. SHAFFER, Auteur ; Randall CARPENTER, Auteur ; Dejan B. BUDIMIROVIC, Auteur ; Paul WANG, Auteur ; Debra L. REISINGER, Auteur ; Karen WALTON-BOWEN, Auteur ; Craig A. ERICKSON, Auteur Article en page(s) : p.1658-1670 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : Estimating meaningful change thresholds (MCT) on clinical outcome assessments is an important consideration when evaluating treatments. In fragile X syndrome (FXS) research, there has been no consensus on how to define MCT’s on several commonly used outcome measures. The purpose of the current study was to determine clinically relevant MCT’s of caregiver-rated assessments using data from a phase 3 clinical trials of arbaclofen (Berry-Kravis et al., 2017). Data were collected as a part of previous phase 3, double-blind, placebo-controlled studies of arbaclofen in individuals with FXS (Berry-Kravis et al., 2017). The two studies enrolled age groups of 5–11-years (n = 159) and 12–50-years (n = 119). The current study examines meaningful within-patient change thresholds from baseline to treatment week 8 across several measures: ABC-CFXS; PSI; Vineland-II; and a Visual Analog Scale (VAS) of Anxiety and Disruptive Behaviors. MCT’s were established by using anchor-based methods, using the CGI-S and CGI-I as anchors. Examining the results of the anchor-based analyses and visual CDF plots, MCT’s were observed for the pediatric study for the ABC-CFXS subscales (with a range depending on use of CGI-S or CGI-I as anchor): Irritability: 11.1–14.8 points; Hyperactivity: 6.7–8.9 points; and Socially Unresponsive/Lethargic: 6.6–8.1 points; as well both VAS subscales: Anxiety: 28.3–36.2 mm; and Disruptive Behavior: 22.4–27.4 mm. Such thresholds were not observed for the Vineland-II and PSI subscales. Our analysis of MCT’s helps set the stage for interpreting clinical trial results in FXS. This may include use of relevant subscales of the ABC-CFXS and VAS as primary outcomes using the MCT’s for response definition. This work may help define future study inclusion criteria and enable future interpretation of treatment outcome results in the field. En ligne : https://doi.org/10.1007/s10803-024-06634-6 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=583 [article] Parent-Reported Outcome Measures for Individuals with Fragile X Syndrome: Clinically Meaningful Change Thresholds [texte imprimé] / Meredith A. NELSON, Auteur ; Lauren M. SCHMITT, Auteur ; Paul S. HORN, Auteur ; Elizabeth BERRY-KRAVIS, Auteur ; David HESSL, Auteur ; Rebecca C. SHAFFER, Auteur ; Randall CARPENTER, Auteur ; Dejan B. BUDIMIROVIC, Auteur ; Paul WANG, Auteur ; Debra L. REISINGER, Auteur ; Karen WALTON-BOWEN, Auteur ; Craig A. ERICKSON, Auteur . - p.1658-1670. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 56-4 (April 2026) . - p.1658-1670 Index. décimale : PER Périodiques Résumé : Estimating meaningful change thresholds (MCT) on clinical outcome assessments is an important consideration when evaluating treatments. In fragile X syndrome (FXS) research, there has been no consensus on how to define MCT’s on several commonly used outcome measures. The purpose of the current study was to determine clinically relevant MCT’s of caregiver-rated assessments using data from a phase 3 clinical trials of arbaclofen (Berry-Kravis et al., 2017). Data were collected as a part of previous phase 3, double-blind, placebo-controlled studies of arbaclofen in individuals with FXS (Berry-Kravis et al., 2017). The two studies enrolled age groups of 5–11-years (n = 159) and 12–50-years (n = 119). The current study examines meaningful within-patient change thresholds from baseline to treatment week 8 across several measures: ABC-CFXS; PSI; Vineland-II; and a Visual Analog Scale (VAS) of Anxiety and Disruptive Behaviors. MCT’s were established by using anchor-based methods, using the CGI-S and CGI-I as anchors. Examining the results of the anchor-based analyses and visual CDF plots, MCT’s were observed for the pediatric study for the ABC-CFXS subscales (with a range depending on use of CGI-S or CGI-I as anchor): Irritability: 11.1–14.8 points; Hyperactivity: 6.7–8.9 points; and Socially Unresponsive/Lethargic: 6.6–8.1 points; as well both VAS subscales: Anxiety: 28.3–36.2 mm; and Disruptive Behavior: 22.4–27.4 mm. Such thresholds were not observed for the Vineland-II and PSI subscales. Our analysis of MCT’s helps set the stage for interpreting clinical trial results in FXS. This may include use of relevant subscales of the ABC-CFXS and VAS as primary outcomes using the MCT’s for response definition. This work may help define future study inclusion criteria and enable future interpretation of treatment outcome results in the field. En ligne : https://doi.org/10.1007/s10803-024-06634-6 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=583

Provider and Caregiver Satisfaction with Telehealth Evaluation of Autism Spectrum Disorder in Young Children During the COVID-19 Pandemic / Debra L. REISINGER in Journal of Autism and Developmental Disorders, 52-12 (December 2022)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 52-12 (December 2022) . - p.5099-5113 Titre : Provider and Caregiver Satisfaction with Telehealth Evaluation of Autism Spectrum Disorder in Young Children During the COVID-19 Pandemic Type de document : texte imprimé Auteurs : Debra L. REISINGER, Auteur ; Elesia N. HINES, Auteur ; Christine M. RACHES, Auteur ; Qing TANG, Auteur ; Cristina JAMES, Auteur ; Rebecca MCNALLY KEEHN, Auteur Année de publication : 2022 Article en page(s) : p.5099-5113 Langues : Anglais (eng) Mots-clés : Child  Humans  Child, Preschool  Infant  covid-19  Autism Spectrum Disorder/diagnosis/epidemiology/therapy  Pandemics  Caregivers  SARS-CoV-2  Personal Satisfaction  Telemedicine  Autism spectrum disorder  Diagnostic evaluation  Satisfaction  Telehealth  Young children Index. décimale : PER Périodiques Résumé : The present study examines provider and caregiver satisfaction with telehealth evaluation of autism spectrum disorder (ASD) in young children during the coronavirus SARS-CoV-2 (COVID-19) pandemic. A telehealth model of ASD evaluation was implemented with 308 children ages 14 to 78 months between May 2020 to June 2021. Data were gathered from electronic health records, autism-specific telehealth diagnostic tools, and post-evaluation surveys. Overall, the majority of providers and caregivers were satisfied with telehealth ASD evaluation. Multiple variables were associated with ratings of satisfaction, differing by providers and caregivers. Findings have important implications for the feasibility and acceptability of telehealth ASD evaluations, in addition to key factors to consider in optimizing and sustaining telehealth evaluations beyond the COVID-19 pandemic. En ligne : http://dx.doi.org/10.1007/s10803-022-05576-1 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=489 [article] Provider and Caregiver Satisfaction with Telehealth Evaluation of Autism Spectrum Disorder in Young Children During the COVID-19 Pandemic [texte imprimé] / Debra L. REISINGER, Auteur ; Elesia N. HINES, Auteur ; Christine M. RACHES, Auteur ; Qing TANG, Auteur ; Cristina JAMES, Auteur ; Rebecca MCNALLY KEEHN, Auteur . - 2022 . - p.5099-5113. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 52-12 (December 2022) . - p.5099-5113 Mots-clés : Child  Humans  Child, Preschool  Infant  covid-19  Autism Spectrum Disorder/diagnosis/epidemiology/therapy  Pandemics  Caregivers  SARS-CoV-2  Personal Satisfaction  Telemedicine  Autism spectrum disorder  Diagnostic evaluation  Satisfaction  Telehealth  Young children Index. décimale : PER Périodiques Résumé : The present study examines provider and caregiver satisfaction with telehealth evaluation of autism spectrum disorder (ASD) in young children during the coronavirus SARS-CoV-2 (COVID-19) pandemic. A telehealth model of ASD evaluation was implemented with 308 children ages 14 to 78 months between May 2020 to June 2021. Data were gathered from electronic health records, autism-specific telehealth diagnostic tools, and post-evaluation surveys. Overall, the majority of providers and caregivers were satisfied with telehealth ASD evaluation. Multiple variables were associated with ratings of satisfaction, differing by providers and caregivers. Findings have important implications for the feasibility and acceptability of telehealth ASD evaluations, in addition to key factors to consider in optimizing and sustaining telehealth evaluations beyond the COVID-19 pandemic. En ligne : http://dx.doi.org/10.1007/s10803-022-05576-1 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=489

Regulating Together: Emotion Dysregulation Group Treatment for ASD Youth and Their Caregivers / Rebecca C. SHAFFER in Journal of Autism and Developmental Disorders, 53-5 (May 2023)  

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