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Background <p>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.</p> Methods <p>The Scorecard development process included four phases: (1) video source material study, (2) identification of initial scoring criteria, (3) scorecard drafts, and (4)&#xa0;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.</p> Results <p>In the communication, ADLs, and external direction domains, scorecard criteria reached 80 to 100% agreement among caregivers (<i>n</i> = 8) and clinicians (<i>n</i> = 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 (<i>n</i> = 8) with a total of 347 scoring criteria and levels across 8 tasks.</p> Conclusions <p>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.</p>

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Development of the Angelman syndrome video assessment: quantifying meaningful change

  • 期刊论文

  • 出版模式：

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  • 发表日期：

    2025年11月3日

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  • Kriszha A. Sheehy,
  • Mindy G. Leffler,
  • Rebecca J. Woods,
  • Robert Komorowski,
  • Rebecca Crean,
  • Christina K. Zigler,
  • Jessica Duis,
  • Olivia Boorom,
  • Nancy Brady,
  • Lauren DeValk,
  • Nicole Harris,
  • Amber Sapp,
  • Caroline Woeber,
  • Anjali Sadhwani,
  • Wen-Hann Tan,
  • Kara Arps,
  • Lynne M. Bird,
  • Betsy Black,
  • Amanda Guevara,
  • Curtis Hanson,
  • Jennifer Jones,
  • Rebecca Leonard,
  • Jessica Lopes,
  • Kristen K. Madison,
  • Candace McClellan,
  • Michelle Ozick,
  • Kelsey Pauley,
  • Maria Fragala-Pinkham,
  • Sandra Rivero,
  • Claudia Senesac,
  • Courtney Swafford,
  • Marie Thomas,
  • Rhonda Thomas,
  • Carol Washburn,
  • Anne Wheeler

摘要

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.