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Original research
Development and assessment of the inter-rater and intra-rater reproducibility of a self-administration version of the ALSFRS-R
  1. Leonhard A Bakker1,2,
  2. Carin D Schröder2,3,
  3. Harold H G Tan1,
  4. Simone M A G Vugts1,
  5. Ruben P A van Eijk1,4,
  6. Michael A van Es1,
  7. Johanna M A Visser-Meily2,3,
  8. Leonard H van den Berg1
  1. 1 Department of Neurology, Brain Center Rudolf Magnus, University Medical Center Utrecht, Utrecht, The Netherlands
  2. 2 Center of Excellence for Rehabilitation Medicine, Brain Center Rudolf Magnus, Utrecht University, University Medical Center Utrecht, and De Hoogstraat Rehabilitation, Utrecht, The Netherlands
  3. 3 Department of Rehabilitation, Physical Therapy Science, and Sports Medicine, Brain Center Rudolf Magnus, University Medical Center Utrecht, Utrecht, The Netherlands
  4. 4 Biostatistics & Research Support, Julius Center for Health Sciences and Primary Care, University Medical Center Utrecht, Utrecht, The Netherlands
  1. Correspondence to Professor Leonard H van den Berg, Department of Neurology, Brain Centre Rudolf Magnus, University Medical Centre Utrecht, Utrecht 3584CX, The Netherlands; L.H.vandenBerg{at}umcutrecht.nl

Abstract

Objective The Amyotrophic Lateral Sclerosis Functional Rating Scale-Revised (ALSFRS-R) is widely applied to assess disease severity and progression in patients with motor neuron disease (MND). The objective of the study is to assess the inter-rater and intra-rater reproducibility, i.e., the inter-rater and intra-rater reliability and agreement, of a self-administration version of the ALSFRS-R for use in apps, online platforms, clinical care and trials.

Methods The self-administration version of the ALSFRS-R was developed based on both patient and expert feedback. To assess the inter-rater reproducibility, 59 patients with MND filled out the ALSFRS-R online and were subsequently assessed on the ALSFRS-R by three raters. To assess the intra-rater reproducibility, patients were invited on two occasions to complete the ALSFRS-R online. Reliability was assessed with intraclass correlation coefficients, agreement was assessed with Bland-Altman plots and paired samples t-tests, and internal consistency was examined with Cronbach’s coefficient alpha.

Results The self-administration version of the ALSFRS-R demonstrated excellent inter-rater and intra-rater reliability. The assessment of inter-rater agreement demonstrated small systematic differences between patients and raters and acceptable limits of agreement. The assessment of intra-rater agreement demonstrated no systematic changes between time points; limits of agreement were 4.3 points for the total score and ranged from 1.6 to 2.4 points for the domain scores. Coefficient alpha values were acceptable.

Discussion The self-administration version of the ALSFRS-R demonstrates high reproducibility and can be used in apps and online portals for both individual comparisons, facilitating the management of clinical care and group comparisons in clinical trials.

  • agreement
  • amyotrophic lateral sclerosis
  • amyotrophic lateral sclerosis functional rating scale-revised
  • inter-rater
  • intra-rater
  • reliability
  • reproducibility
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Footnotes

  • Contributors LAB, CDS and LHvdB designed the study. LAB wrote the manuscript. LAB, HHGT and SMAGV participated in the data collection. LAB performed the statistical analyses. LAB, CDS, HHGT, SMAGV, RPAVE, MAvE, JMAV-M and LHvdB contributed to the interpretation of data. LHvdB provided study supervision. All authors critically reviewed and revised the manuscript.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests LAB, CDS, HHGT, SMAGV, RPAVE and JMAV-M have nothing to disclose. MAvE received grants from the Netherlands Organization for Health Research and Development (Veni scheme), The Thierry Latran Foundation, Joint Programme – Neurodegenerative Disease Research (JPND) and the Netherlands ALS Foundation (Stichting ALS Nederland). He received travel grants from Shire (previously Baxalta) and serves on the medical ethical review board of University Medical Centre Utrecht. LHvdB reports grants from ALS Foundation Netherlands, grants from The Netherlands Organization for Health Research and Development (Vici scheme), grants from The Netherlands Organization for Health Research and Development (SOPHIA, STRENGTH, ALS-CarE project), funded through the EU JPND, personal fees from Shire (previously Baxalta), personal fees from Biogen, personal fees from Cytokinetics, other from Prinses Beatrix SpierFonds, other from Latran Foundation, outside the submitted work.

  • Patient consent for publication Not required.

  • Provenance and peer review Not commissioned; externally peer reviewed.

  • Data availability statement Data are available on reasonable request.

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