15 recherche sur le mot-clé 'Outcome measure'

Development of a Goal Attainment Scale (GAS) outcome measure for clinical interventional studies in paediatric autism / Hannah STAUNTON in Autism, 29-12 (December 2025)  

Public ISBD [article]  inAutism > 29-12 (December 2025) . - p.2975-2990 Titre : Development of a Goal Attainment Scale (GAS) outcome measure for clinical interventional studies in paediatric autism Type de document : texte imprimé Auteurs : Hannah STAUNTON, Auteur ; Tammy MCIVER, Auteur ; Julian TILLMANN, Auteur ; Susanne CLINCH, Auteur ; Vivienne HANRAHAN, Auteur ; Bethany EWENS, Auteur ; Caroline AVERIUS, Auteur ; Alexandra I. BARSDORF, Auteur ; Aurélie BARANGER, Auteur ; Elizabeth BERRY KRAVIS, Auteur ; Tony CHARMAN, Auteur ; Haraldt NEERLAND, Auteur ; Alison T. SINGER, Auteur ; Pamela VENTOLA, Auteur ; Zackary JK WILLIAMS, Auteur ; Louise BARRETT, Auteur Article en page(s) : p.2975-2990 Langues : Anglais (eng) Mots-clés : adolescents  autism  autism spectrum disorders  clinical study  GAS  Goal Attainment Scaling  outcome measure  personalised  qualitative research  school-age children Index. décimale : PER Périodiques Résumé : Existing clinical outcome assessments (COAs) have limitations in capturing treatment effects in autism for those who choose to enrol in clinical research. Some COAs include items that are not relevant or equally important to all autistic people; others are not sufficiently comprehensive. Goal Attainment Scaling (GAS), which measures individual experiences against personalised goals, has been proposed as a more individualised and change-sensitive approach to assess outcomes in clinical studies. We developed a novel GAS measure for use in paediatric autism studies based on qualitative interviews with autistic adolescents, parents and clinicians. Applying a methodologically robust approach, concept elicitation interviews (N = 40) were first conducted to understand and generate a bank of personalised goals, followed by cognitive debriefing interviews (N = 39) to provide feedback on a draft GAS manual. Twenty-five personalised goals were generated for core autistic characteristics (communication, socialisation, restrictive and repetitive behaviours), as well as associated characteristics and impacts. In debriefing interviews, adolescents and parents supported the use of personalised goals and clinicians recommended using such goals in clinical studies and practice. In conclusion, GAS measures may overcome some of the challenges with existing outcome measures and complement future outcome measurement strategies for clinical studies in paediatric autism.Lay Abstract Autistic children and adolescents and their parents support the use of personalised goals as a way of measuring the effects of new treatments for those who choose to enrol in clinical research. Health professionals also recommend using personalised goals in both their research and in treatment plans. Establishing personalised goals, a process known as Goal Attainment Scaling (GAS), means identifying goals according to the needs of the individual, as opposed to using standardised outcomes for all participants. Autism is a condition with many different characteristics and degrees of impact, and the tools commonly used to measure treatment effects, known as clinical outcome assessments (COAs), have limitations. Some COAs, for example, include outcomes that are not relevant to all autistic people; others do not include enough outcomes to represent the experience of all autistic people. GAS, which measures individual experiences against personalised goals, may be complementary to existing COAs to provide an individualised or tailor-made tool to evaluate outcomes as a result of an intervention. We developed a GAS measure for researchers/health professionals to use in studies with autistic children and adolescents. Our research was based on a total of 40 interviews with adolescents, parents and clinicians, asking open-ended questions about how children and adolescents experience autism in order to understand and create a bank of potential personalised goals. We then conducted 39 further interviews to ask them for their feedback on the first draft of the measure. In the end, we compiled a list of 25 personalised goals related to core and associated characteristics of autistic children and adolescents, including goals related to communication, socialisation, and restricted and repetitive behaviours. We hope that these findings will help to improve the measurement of meaningful outcomes for autistic children and adolescents in future studies. En ligne : https://dx.doi.org/10.1177/13623613251349904 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=571 [article] Development of a Goal Attainment Scale (GAS) outcome measure for clinical interventional studies in paediatric autism [texte imprimé] / Hannah STAUNTON, Auteur ; Tammy MCIVER, Auteur ; Julian TILLMANN, Auteur ; Susanne CLINCH, Auteur ; Vivienne HANRAHAN, Auteur ; Bethany EWENS, Auteur ; Caroline AVERIUS, Auteur ; Alexandra I. BARSDORF, Auteur ; Aurélie BARANGER, Auteur ; Elizabeth BERRY KRAVIS, Auteur ; Tony CHARMAN, Auteur ; Haraldt NEERLAND, Auteur ; Alison T. SINGER, Auteur ; Pamela VENTOLA, Auteur ; Zackary JK WILLIAMS, Auteur ; Louise BARRETT, Auteur . - p.2975-2990. Langues : Anglais (eng) in Autism > 29-12 (December 2025) . - p.2975-2990 Mots-clés : adolescents  autism  autism spectrum disorders  clinical study  GAS  Goal Attainment Scaling  outcome measure  personalised  qualitative research  school-age children Index. décimale : PER Périodiques Résumé : Existing clinical outcome assessments (COAs) have limitations in capturing treatment effects in autism for those who choose to enrol in clinical research. Some COAs include items that are not relevant or equally important to all autistic people; others are not sufficiently comprehensive. Goal Attainment Scaling (GAS), which measures individual experiences against personalised goals, has been proposed as a more individualised and change-sensitive approach to assess outcomes in clinical studies. We developed a novel GAS measure for use in paediatric autism studies based on qualitative interviews with autistic adolescents, parents and clinicians. Applying a methodologically robust approach, concept elicitation interviews (N = 40) were first conducted to understand and generate a bank of personalised goals, followed by cognitive debriefing interviews (N = 39) to provide feedback on a draft GAS manual. Twenty-five personalised goals were generated for core autistic characteristics (communication, socialisation, restrictive and repetitive behaviours), as well as associated characteristics and impacts. In debriefing interviews, adolescents and parents supported the use of personalised goals and clinicians recommended using such goals in clinical studies and practice. In conclusion, GAS measures may overcome some of the challenges with existing outcome measures and complement future outcome measurement strategies for clinical studies in paediatric autism.Lay Abstract Autistic children and adolescents and their parents support the use of personalised goals as a way of measuring the effects of new treatments for those who choose to enrol in clinical research. Health professionals also recommend using personalised goals in both their research and in treatment plans. Establishing personalised goals, a process known as Goal Attainment Scaling (GAS), means identifying goals according to the needs of the individual, as opposed to using standardised outcomes for all participants. Autism is a condition with many different characteristics and degrees of impact, and the tools commonly used to measure treatment effects, known as clinical outcome assessments (COAs), have limitations. Some COAs, for example, include outcomes that are not relevant to all autistic people; others do not include enough outcomes to represent the experience of all autistic people. GAS, which measures individual experiences against personalised goals, may be complementary to existing COAs to provide an individualised or tailor-made tool to evaluate outcomes as a result of an intervention. We developed a GAS measure for researchers/health professionals to use in studies with autistic children and adolescents. Our research was based on a total of 40 interviews with adolescents, parents and clinicians, asking open-ended questions about how children and adolescents experience autism in order to understand and create a bank of potential personalised goals. We then conducted 39 further interviews to ask them for their feedback on the first draft of the measure. In the end, we compiled a list of 25 personalised goals related to core and associated characteristics of autistic children and adolescents, including goals related to communication, socialisation, and restricted and repetitive behaviours. We hope that these findings will help to improve the measurement of meaningful outcomes for autistic children and adolescents in future studies. En ligne : https://dx.doi.org/10.1177/13623613251349904 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=571

Toward better outcome measurement for insomnia in children with autism spectrum disorder / Luc LECAVALIER ; Michael C. EDWARDS ; Megan L. WENZELL ; Leah M. BARTO ; Arielle MULLIGAN ; Auscia T. WILLIAMS ; Opal Y. OUSLEY ; Cynthia B. SINHA ; Christopher A. TAYLOR ; Soo Youn KIM ; Laura M. JOHNSON ; Scott E. GILLESPIE ; Cynthia JOHNSON in Autism, 28-12 (December 2024)  

Public ISBD [article]  inAutism > 28-12 (December 2024) . - p.3131-3142 Titre : Toward better outcome measurement for insomnia in children with autism spectrum disorder Type de document : texte imprimé Auteurs : Luc LECAVALIER, Auteur ; Michael C. EDWARDS, Auteur ; Megan L. WENZELL, Auteur ; Leah M. BARTO, Auteur ; Arielle MULLIGAN, Auteur ; Auscia T. WILLIAMS, Auteur ; Opal Y. OUSLEY, Auteur ; Cynthia B. SINHA, Auteur ; Christopher A. TAYLOR, Auteur ; Soo Youn KIM, Auteur ; Laura M. JOHNSON, Auteur ; Scott E. GILLESPIE, Auteur ; Cynthia JOHNSON, Auteur Article en page(s) : p.3131-3142 Langues : Anglais (eng) Mots-clés : autism  children  insomnia  outcome measure Index. décimale : PER Périodiques Résumé : This report presents a new parent-rated outcome measure of insomnia for children with autism spectrum disorder. Parents of 1185 children with autism spectrum disorder (aged 3-12; 80.3% male) completed the first draft of the measure online. Factor and item response theory analyses reduced the set of 40 items to the final 21-item Pediatric Insomnia Autism Rating Scale. In this online sample, Pediatric Insomnia Autism Rating Scale mean was 21.8+15.0 (range 0-63; coefficient alpha = 0.94). Item response theory results indicated excellent reliability across a range of scores. Next, we conducted a telehealth assessment of 134 children with autism spectrum disorder (aged 3-12; 73% male). In this clinical sample, Pediatric Insomnia Autism Rating Scale mean was 28.9+14.62 (range 0-61; coefficient alpha = 0.93). Pearson correlations of Pediatric Insomnia Autism Rating Scale with the PROMIS Sleep Disturbance and the modified Children s Sleep Habits Questionnaire was 0.74 for each. This was significantly higher than correlations of parent-rated disruptive behavior, hyperactivity, repetitive behavior, and anxiety measures (range 0.29-0.59)-supporting convergent and divergent validity. Parents of 63 children completed the Pediatric Insomnia Autism Rating Scale three times over 4 weeks. Intraclass correlation was excellent (0.88) over three time points. Pediatric Insomnia Autism Rating Scale is a valid dimensional measure of insomnia in autistic children that produces reliable scores. Lay Abstract Insomnia, trouble falling asleep or staying asleep, is common in autistic children. In a previous report, we described the results of focus groups with parents of autistic children toward the development of the Pediatric Autism Insomnia Rating Scale. In this article, we report on the steps taken to complete the Pediatric Autism Insomnia Rating Scale. With help from the Simons Foundation registry, we collected information from parents on 1185 children with autism spectrum disorder to test the new measure. These results were evaluated using standard statistical methods such as factor analysis. To confirm the validity of the new measure, we enrolled a separate sample of 134 autistic children for a detailed assessment by video conference. This step showed that the Pediatric Autism Insomnia Rating Scale is clearly measuring symptoms of insomnia in children with autism spectrum disorder and not related problems such as hyperactivity, repetitive behavior, or anxiety. We also showed that the total score on the Pediatric Autism Insomnia Rating Scale is stable when repeated over a brief period of time. This is important because a measure that is not stable over a brief period of time would not be suitable as an outcome measure. In summary, the Pediatric Autism Insomnia Rating Scale is a brief and valid measure of insomnia in children with autism spectrum disorder that provides reliable scores. En ligne : https://dx.doi.org/10.1177/13623613241255814 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=543 [article] Toward better outcome measurement for insomnia in children with autism spectrum disorder [texte imprimé] / Luc LECAVALIER, Auteur ; Michael C. EDWARDS, Auteur ; Megan L. WENZELL, Auteur ; Leah M. BARTO, Auteur ; Arielle MULLIGAN, Auteur ; Auscia T. WILLIAMS, Auteur ; Opal Y. OUSLEY, Auteur ; Cynthia B. SINHA, Auteur ; Christopher A. TAYLOR, Auteur ; Soo Youn KIM, Auteur ; Laura M. JOHNSON, Auteur ; Scott E. GILLESPIE, Auteur ; Cynthia JOHNSON, Auteur . - p.3131-3142. Langues : Anglais (eng) in Autism > 28-12 (December 2024) . - p.3131-3142 Mots-clés : autism  children  insomnia  outcome measure Index. décimale : PER Périodiques Résumé : This report presents a new parent-rated outcome measure of insomnia for children with autism spectrum disorder. Parents of 1185 children with autism spectrum disorder (aged 3-12; 80.3% male) completed the first draft of the measure online. Factor and item response theory analyses reduced the set of 40 items to the final 21-item Pediatric Insomnia Autism Rating Scale. In this online sample, Pediatric Insomnia Autism Rating Scale mean was 21.8+15.0 (range 0-63; coefficient alpha = 0.94). Item response theory results indicated excellent reliability across a range of scores. Next, we conducted a telehealth assessment of 134 children with autism spectrum disorder (aged 3-12; 73% male). In this clinical sample, Pediatric Insomnia Autism Rating Scale mean was 28.9+14.62 (range 0-61; coefficient alpha = 0.93). Pearson correlations of Pediatric Insomnia Autism Rating Scale with the PROMIS Sleep Disturbance and the modified Children s Sleep Habits Questionnaire was 0.74 for each. This was significantly higher than correlations of parent-rated disruptive behavior, hyperactivity, repetitive behavior, and anxiety measures (range 0.29-0.59)-supporting convergent and divergent validity. Parents of 63 children completed the Pediatric Insomnia Autism Rating Scale three times over 4 weeks. Intraclass correlation was excellent (0.88) over three time points. Pediatric Insomnia Autism Rating Scale is a valid dimensional measure of insomnia in autistic children that produces reliable scores. Lay Abstract Insomnia, trouble falling asleep or staying asleep, is common in autistic children. In a previous report, we described the results of focus groups with parents of autistic children toward the development of the Pediatric Autism Insomnia Rating Scale. In this article, we report on the steps taken to complete the Pediatric Autism Insomnia Rating Scale. With help from the Simons Foundation registry, we collected information from parents on 1185 children with autism spectrum disorder to test the new measure. These results were evaluated using standard statistical methods such as factor analysis. To confirm the validity of the new measure, we enrolled a separate sample of 134 autistic children for a detailed assessment by video conference. This step showed that the Pediatric Autism Insomnia Rating Scale is clearly measuring symptoms of insomnia in children with autism spectrum disorder and not related problems such as hyperactivity, repetitive behavior, or anxiety. We also showed that the total score on the Pediatric Autism Insomnia Rating Scale is stable when repeated over a brief period of time. This is important because a measure that is not stable over a brief period of time would not be suitable as an outcome measure. In summary, the Pediatric Autism Insomnia Rating Scale is a brief and valid measure of insomnia in children with autism spectrum disorder that provides reliable scores. En ligne : https://dx.doi.org/10.1177/13623613241255814 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=543

Quantifying Caregiver Change Across Early Autism Interventions Using the Measure of NDBI Strategy Implementation: Caregiver Change (MONSI-CC) / Bethany A. VIBERT in Journal of Autism and Developmental Disorders, 50-4 (April 2020)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 50-4 (April 2020) . - p.1364-1379 Titre : Quantifying Caregiver Change Across Early Autism Interventions Using the Measure of NDBI Strategy Implementation: Caregiver Change (MONSI-CC) Type de document : texte imprimé Auteurs : Bethany A. VIBERT, Auteur ; Sarah DUFEK, Auteur ; Claire B. KLEIN, Auteur ; Yeo Bi CHOI, Auteur ; Jamie WINTER, Auteur ; Catherine LORD, Auteur ; So Hyun KIM, Auteur Article en page(s) : p.1364-1379 Langues : Anglais (eng) Mots-clés : Autism spectrum disorder  Early intervention  Ndbi  Outcome measure  Parent-mediated intervention Index. décimale : PER Périodiques Résumé : This study aimed to provide initial validity and reliability of the Measure of NDBI Strategy Implementation-Caregiver Change (MONSI-CC), a novel measure that captures changes in caregivers' implementation of NDBI strategies during early intervention. The MONSI-CC was applied to 119 observations of 43 caregiver-child dyads of preschoolers with autism spectrum disorders (ASD). The MONSI-CC showed high inter-rater and test-retest reliability and captured significant improvements in caregivers' implementation of NDBI strategies. Significant associations between improvements in caregiver NDBI implementation and improvements in the child's ASD symptoms also emerged. Our work shows promising evidence for the utility of the MONSI-CC to evaluate implementation of NDBI strategies by caregivers as a mediating and moderating factor for treatment effects on children with ASD. En ligne : http://dx.doi.org/10.1007/s10803-019-04342-0 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=421 [article] Quantifying Caregiver Change Across Early Autism Interventions Using the Measure of NDBI Strategy Implementation: Caregiver Change (MONSI-CC) [texte imprimé] / Bethany A. VIBERT, Auteur ; Sarah DUFEK, Auteur ; Claire B. KLEIN, Auteur ; Yeo Bi CHOI, Auteur ; Jamie WINTER, Auteur ; Catherine LORD, Auteur ; So Hyun KIM, Auteur . - p.1364-1379. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 50-4 (April 2020) . - p.1364-1379 Mots-clés : Autism spectrum disorder  Early intervention  Ndbi  Outcome measure  Parent-mediated intervention Index. décimale : PER Périodiques Résumé : This study aimed to provide initial validity and reliability of the Measure of NDBI Strategy Implementation-Caregiver Change (MONSI-CC), a novel measure that captures changes in caregivers' implementation of NDBI strategies during early intervention. The MONSI-CC was applied to 119 observations of 43 caregiver-child dyads of preschoolers with autism spectrum disorders (ASD). The MONSI-CC showed high inter-rater and test-retest reliability and captured significant improvements in caregivers' implementation of NDBI strategies. Significant associations between improvements in caregiver NDBI implementation and improvements in the child's ASD symptoms also emerged. Our work shows promising evidence for the utility of the MONSI-CC to evaluate implementation of NDBI strategies by caregivers as a mediating and moderating factor for treatment effects on children with ASD. En ligne : http://dx.doi.org/10.1007/s10803-019-04342-0 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=421

Development of the Angelman syndrome video assessment: quantifying meaningful change / Kriszha A. SHEEHY in Journal of Neurodevelopmental Disorders, 17 (2025)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 17 (2025) Titre : Development of the Angelman syndrome video assessment: quantifying meaningful change Type de document : texte imprimé Auteurs : Kriszha A. SHEEHY, Auteur ; Mindy G. LEFFLER, Auteur ; Rebecca J. WOODS, Auteur ; Robert KOMOROWSKI, Auteur ; Rebecca CREAN, Auteur ; Christina K. ZIGLER, Auteur ; Jessica DUIS, Auteur ; Olivia BOOROM, Auteur ; Nancy BRADY, Auteur ; Lauren DEVALK, Auteur ; Nicole HARRIS, Auteur ; Amber SAPP, Auteur ; Caroline WOEBER, Auteur ; Anjali SADHWANI, Auteur ; Wen-Hann TAN, Auteur ; ASVA DELPHI PANELISTS, Auteur Langues : Anglais (eng) Mots-clés : Humans  Angelman Syndrome/diagnosis/physiopathology  Activities of Daily Living  Video Recording  Male  Female  Delphi Technique  Child  Caregivers  Communication  Adult  Adolescent  Activities of daily living  Angelman syndrome  Meaningful clinical outcome  Neurodevelopmental  Outcome measure  Patient outcome assessment  Patient-focused drug development  Video assessment  during the ASVA Pilot study, approved by the central Institutional Review Board  IntegReview (Austin, TX) and the ASVA source material study (NCT05637697),  approved by Advarra IRB (Pro00057202). All caregivers were legal guardians who  provided written consent for themselves and subjects and received compensation  for their participation. All participating Delphi panelists signed written  agreements to provide consultant services for the purposes of Delphi panel, and  they were provided with an honorarium for their time. Since panelists provided  expert opinion through consultant services, this was not considered human  subjects research. Consent for publication: Not applicable. Competing interests:  The authors declare no competing interests. Index. décimale : PER Périodiques Résumé : BACKGROUND: The Angelman Syndrome Video Assessment (ASVA) is a clinician-reported outcome measure that was developed to assess the functional ability of individuals with Angelman Syndrome (AS) in a familiar environment. Through standardized tasks and associated scorecards, clinicians assess four meaningful domains of functioning: communication, activities of daily living (ADLs, which include fine motor skills), gross motor, and external direction (i.e., the ability to follow directions) via scorecards with pre-established criteria. The aim of this project was to develop and refine the scorecards using a rigorous process in partnership with caregivers, clinicians, and researchers in the AS community. METHODS: The Scorecard development process included four phases: (1) video source material study, (2) identification of initial scoring criteria, (3) scorecard drafts, and (4) two (Caregiver and Clinician panel and PT panel) two-round modified Delphi processes to reach consensus. All phases were conducted remotely except for Round 2 of the Caregiver and Clinician Delphi Panel, which was conducted in person. Votes were held for each scoring criterion and consensus was defined as ≥ 70% agreement. RESULTS: In the communication, ADLs, and external direction domains, scorecard criteria reached 80 to 100% agreement among caregivers (n = 8) and clinicians (n = 2), resulting in a total of 218 scoring criteria and levels across 10 tasks. In the gross motor domain, scorecard criteria reached 100% agreement among physical therapists (n = 8) with a total of 347 scoring criteria and levels across 8 tasks. CONCLUSIONS: The ASVA was developed with insights from the AS community, including caregivers of individuals with AS, clinicians, and researchers. The ASVA is a novel, disease-specific, clinician-reported outcome measure that uses standardized video capture and scorecards that were developed through a rigorous process, resulting in well-developed criteria to quantify meaningful changes of function in individuals with AS in communication, ADLs, gross motor function, and external direction. En ligne : https://dx.doi.org/10.1186/s11689-025-09655-z Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=576 [article] Development of the Angelman syndrome video assessment: quantifying meaningful change [texte imprimé] / Kriszha A. SHEEHY, Auteur ; Mindy G. LEFFLER, Auteur ; Rebecca J. WOODS, Auteur ; Robert KOMOROWSKI, Auteur ; Rebecca CREAN, Auteur ; Christina K. ZIGLER, Auteur ; Jessica DUIS, Auteur ; Olivia BOOROM, Auteur ; Nancy BRADY, Auteur ; Lauren DEVALK, Auteur ; Nicole HARRIS, Auteur ; Amber SAPP, Auteur ; Caroline WOEBER, Auteur ; Anjali SADHWANI, Auteur ; Wen-Hann TAN, Auteur ; ASVA DELPHI PANELISTS, Auteur. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 17 (2025) Mots-clés : Humans  Angelman Syndrome/diagnosis/physiopathology  Activities of Daily Living  Video Recording  Male  Female  Delphi Technique  Child  Caregivers  Communication  Adult  Adolescent  Activities of daily living  Angelman syndrome  Meaningful clinical outcome  Neurodevelopmental  Outcome measure  Patient outcome assessment  Patient-focused drug development  Video assessment  during the ASVA Pilot study, approved by the central Institutional Review Board  IntegReview (Austin, TX) and the ASVA source material study (NCT05637697),  approved by Advarra IRB (Pro00057202). All caregivers were legal guardians who  provided written consent for themselves and subjects and received compensation  for their participation. All participating Delphi panelists signed written  agreements to provide consultant services for the purposes of Delphi panel, and  they were provided with an honorarium for their time. Since panelists provided  expert opinion through consultant services, this was not considered human  subjects research. Consent for publication: Not applicable. Competing interests:  The authors declare no competing interests. Index. décimale : PER Périodiques Résumé : BACKGROUND: The Angelman Syndrome Video Assessment (ASVA) is a clinician-reported outcome measure that was developed to assess the functional ability of individuals with Angelman Syndrome (AS) in a familiar environment. Through standardized tasks and associated scorecards, clinicians assess four meaningful domains of functioning: communication, activities of daily living (ADLs, which include fine motor skills), gross motor, and external direction (i.e., the ability to follow directions) via scorecards with pre-established criteria. The aim of this project was to develop and refine the scorecards using a rigorous process in partnership with caregivers, clinicians, and researchers in the AS community. METHODS: The Scorecard development process included four phases: (1) video source material study, (2) identification of initial scoring criteria, (3) scorecard drafts, and (4) two (Caregiver and Clinician panel and PT panel) two-round modified Delphi processes to reach consensus. All phases were conducted remotely except for Round 2 of the Caregiver and Clinician Delphi Panel, which was conducted in person. Votes were held for each scoring criterion and consensus was defined as ≥ 70% agreement. RESULTS: In the communication, ADLs, and external direction domains, scorecard criteria reached 80 to 100% agreement among caregivers (n = 8) and clinicians (n = 2), resulting in a total of 218 scoring criteria and levels across 10 tasks. In the gross motor domain, scorecard criteria reached 100% agreement among physical therapists (n = 8) with a total of 347 scoring criteria and levels across 8 tasks. CONCLUSIONS: The ASVA was developed with insights from the AS community, including caregivers of individuals with AS, clinicians, and researchers. The ASVA is a novel, disease-specific, clinician-reported outcome measure that uses standardized video capture and scorecards that were developed through a rigorous process, resulting in well-developed criteria to quantify meaningful changes of function in individuals with AS in communication, ADLs, gross motor function, and external direction. En ligne : https://dx.doi.org/10.1186/s11689-025-09655-z Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=576

Measuring parent strategy use in early intervention: Reliability and validity of the Naturalistic Developmental Behavioral Intervention Fidelity Rating Scale across strategy types / Bailey J. SONE in Autism, 25-7 (October 2021)  

Public ISBD [article]  inAutism > 25-7 (October 2021) . - p.2101-2111 Titre : Measuring parent strategy use in early intervention: Reliability and validity of the Naturalistic Developmental Behavioral Intervention Fidelity Rating Scale across strategy types Type de document : texte imprimé Auteurs : Bailey J. SONE, Auteur ; Aaron J. KAAT, Auteur ; Megan Y. ROBERTS, Auteur Article en page(s) : p.2101-2111 Langues : Anglais (eng) Mots-clés : Autism Spectrum Disorder/therapy  Child  Communication  Early Intervention, Educational  Humans  Parents  Reproducibility of Results  autism spectrum disorders  communication and language  fidelity  observational methods  outcome measure  parent-implemented intervention Index. décimale : PER Périodiques Résumé : Children with autism spectrum disorder benefit from early intervention to improve social communication, and parent-implemented interventions are a feasible and family-centered way to increase the amount of treatment they receive. For these treatments to be effective, it is important for the parent to implement the strategies as intended. However, measurement of parent strategy use is inconsistent across studies of parent-implemented interventions. This study evaluates the quality of the NDBI-Fi, an efficient measure, compared to a more time-consuming measure that is known to be precise. Videos of parents playing with their children were used to compare these two measurement methods. Results demonstrated that the NDBI-Fi was of good quality: scorers had high levels of agreement, the NDBI-Fi was similar to the more precise measure in rating parents after intervention, it detected changes from before to after intervention, and it detected differences when parents learned different types of intervention strategies. The NDBI-Fi was not as precise as the other measure across all strategies before parents learned intervention. Taken together, the findings of this study support the use of the NDBI-Fi as a high-quality outcome measure. En ligne : http://dx.doi.org/10.1177/13623613211015003 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=451 [article] Measuring parent strategy use in early intervention: Reliability and validity of the Naturalistic Developmental Behavioral Intervention Fidelity Rating Scale across strategy types [texte imprimé] / Bailey J. SONE, Auteur ; Aaron J. KAAT, Auteur ; Megan Y. ROBERTS, Auteur . - p.2101-2111. Langues : Anglais (eng) in Autism > 25-7 (October 2021) . - p.2101-2111 Mots-clés : Autism Spectrum Disorder/therapy  Child  Communication  Early Intervention, Educational  Humans  Parents  Reproducibility of Results  autism spectrum disorders  communication and language  fidelity  observational methods  outcome measure  parent-implemented intervention Index. décimale : PER Périodiques Résumé : Children with autism spectrum disorder benefit from early intervention to improve social communication, and parent-implemented interventions are a feasible and family-centered way to increase the amount of treatment they receive. For these treatments to be effective, it is important for the parent to implement the strategies as intended. However, measurement of parent strategy use is inconsistent across studies of parent-implemented interventions. This study evaluates the quality of the NDBI-Fi, an efficient measure, compared to a more time-consuming measure that is known to be precise. Videos of parents playing with their children were used to compare these two measurement methods. Results demonstrated that the NDBI-Fi was of good quality: scorers had high levels of agreement, the NDBI-Fi was similar to the more precise measure in rating parents after intervention, it detected changes from before to after intervention, and it detected differences when parents learned different types of intervention strategies. The NDBI-Fi was not as precise as the other measure across all strategies before parents learned intervention. Taken together, the findings of this study support the use of the NDBI-Fi as a high-quality outcome measure. En ligne : http://dx.doi.org/10.1177/13623613211015003 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=451

Measuring parent strategy use in early intervention: Reliability and validity of the Naturalistic Developmental Behavioral Intervention Fidelity Rating Scale across strategy types / Bailey J. SONE in Autism, 26-7 (October 2022)  

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Measuring treatment response in children with autism spectrum disorder: Applications of the Brief Observation of Social Communication Change to the Autism Diagnostic Observation Schedule / So Hyun KIM in Autism, 23-5 (July 2019)  

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Social communication in fragile X syndrome: pilot examination of the Brief Observation of Social Communication Change (BOSCC) / Rebecca SHAFFER in Journal of Neurodevelopmental Disorders, 14 (2022)  

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Associations between genotype, phenotype and behaviours measured by the Rett syndrome behaviour questionnaire in Rett syndrome / Jenny DOWNS in Journal of Neurodevelopmental Disorders, 16 (2024)  

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Brief report: Game day: A novel method of assessing change in social competence in students with autism spectrum disorder (ASD) / Stephen BENT in Research in Autism Spectrum Disorders, 84 (June 2021)  

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