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Introduction <p>The Friedreich Ataxia Rating Scale–Activities of Daily Living (FARS-ADL) is a validated and highly utilized measure for evaluating patients with Friedreich Ataxia. While construct validity of FARS-ADL has been shown for spinocerebellar ataxia (SCA), content validity has not been established.</p> Methods <p>Individuals with SCA1 or SCA3 (<i>n</i>&#xa0;=&#xa0;7) and healthcare professionals (HCPs) with SCA expertise (<i>n</i>&#xa0;=&#xa0;8) participated in qualitative interviews evaluating the relevance, clarity, and clinical meaningfulness of FARS-ADL for assessment of individuals with SCA. Interviews were recorded, transcribed, coded, and analyzed by ATLAS.Ti v22 software.</p> Results <p>FARS-ADL concepts most frequently reported by individuals with SCA were difficulty walking (<i>n</i>&#xa0;=&#xa0;7/7), falls (<i>n</i>&#xa0;=&#xa0;6/7), speech difficulties (<i>n</i>&#xa0;=&#xa0;4/7), and swallowing (<i>n</i>&#xa0;=&#xa0;3/7). Individuals with SCA reported that all FARS-ADL items were relevant; Gait and Walking (<i>n</i>&#xa0;=&#xa0;7/7), Bladder Function (<i>n</i>&#xa0;=&#xa0;6/7), and Falling (<i>n</i>&#xa0;=&#xa0;6/7) were considered extremely relevant. All HCPs (<i>n</i>&#xa0;=&#xa0;8/8) reported that most FARS-ADL items were relevant to individuals with SCA; Quality of Sitting Position was considered least relevant. HCPs reported meaningful change as 1–2 point score change in individual FARS-ADL items (<i>n</i>&#xa0;=&#xa0;7/7), 1–3 point change in total score (<i>n</i>&#xa0;=&#xa0;6/6), and stability on any item and/or total score over&#xa0;≥&#xa0;1&#xa0;year, depending on SCA subtype (<i>n</i>&#xa0;=&#xa0;5/8). Cognitive debriefing supported clarity and comprehension of FARS-ADL. Suggested improvements included refining response options for Dressing, Falling, Walking, and Bladder Function items.</p> Conclusion <p>The findings confirm the content validity of most FARS-ADL items for use in individuals with mild-to-moderate SCA1 and SCA3, and offer suggested improvements for response options.</p>

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Content Validity of the Friedreich Ataxia Rating Scale in Patients with Spinocerebellar Ataxia

  • 期刊论文

  • 出版模式：

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  • 发表日期：

    2025年1月15日

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  • Michele Potashman,
  • Katja Rudell,
  • Naomi Suminski,
  • Rinchen Doma,
  • Maggie Heinrich,
  • Linda Abetz-Webb,
  • Melissa Wolfe Beiner,
  • Vlad Coric,
  • Liana S. Rosenthal,
  • Sheng-Han Kuo,
  • Theresa Zesiewicz,
  • Terry D. Fife,
  • Bart van de Warrenburg,
  • Giovanni Ristori,
  • Matthis Synofzik,
  • Sub Subramony,
  • Susan Perlman,
  • Jeremy D. Schmahmann,
  • Gil L’Italien

摘要

Introduction

The Friedreich Ataxia Rating Scale–Activities of Daily Living (FARS-ADL) is a validated and highly utilized measure for evaluating patients with Friedreich Ataxia. While construct validity of FARS-ADL has been shown for spinocerebellar ataxia (SCA), content validity has not been established.

Methods

Individuals with SCA1 or SCA3 (n = 7) and healthcare professionals (HCPs) with SCA expertise (n = 8) participated in qualitative interviews evaluating the relevance, clarity, and clinical meaningfulness of FARS-ADL for assessment of individuals with SCA. Interviews were recorded, transcribed, coded, and analyzed by ATLAS.Ti v22 software.

Results

FARS-ADL concepts most frequently reported by individuals with SCA were difficulty walking (n = 7/7), falls (n = 6/7), speech difficulties (n = 4/7), and swallowing (n = 3/7). Individuals with SCA reported that all FARS-ADL items were relevant; Gait and Walking (n = 7/7), Bladder Function (n = 6/7), and Falling (n = 6/7) were considered extremely relevant. All HCPs (n = 8/8) reported that most FARS-ADL items were relevant to individuals with SCA; Quality of Sitting Position was considered least relevant. HCPs reported meaningful change as 1–2 point score change in individual FARS-ADL items (n = 7/7), 1–3 point change in total score (n = 6/6), and stability on any item and/or total score over ≥ 1 year, depending on SCA subtype (n = 5/8). Cognitive debriefing supported clarity and comprehension of FARS-ADL. Suggested improvements included refining response options for Dressing, Falling, Walking, and Bladder Function items.

Conclusion

The findings confirm the content validity of most FARS-ADL items for use in individuals with mild-to-moderate SCA1 and SCA3, and offer suggested improvements for response options.