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Equating norms between the ALS Cognitive Behavioral Screen (ALS-CBS™) and the Edinburgh Cognitive and Behavioural ALS Screen (ECAS) in non-demented ALS patients

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Abstract

Background

The present study aimed at deriving equating norms to estimate scores on the Edinburgh Cognitive and Behavioural ALS Screen (ECAS) based on those on the ALS Cognitive Behavioral Screen (ALS-CBS™) in an Italian cohort of non-demented ALS patients.

Methods

ALS-CBS™ and ECAS scores of 293 ALS patients without frontotemporal dementia were retrospectively retrieved. Concurrent validity of the ALS-CBS™ towards the ECAS was tested by covarying for demographics, disease duration and severity, presence of C9orf72 hexanucleotide repeat expansion and behavioural features. A linear-smoothing equipercentile equating (LSEE) model was employed to derive ALS-CBS™-to-ECAS cross-walks. Gaps in LSEE-based estimation were managed via a linear regression-based equating approach. Equivalence between empirical and derived ECAS scores was tested via a two-one-sided test (TOST) procedure for the dependent sample.

Results

The ALS-CBS™ predicted the ECAS (β = 0.75), accounting for the vast majority of its variance (60% out of an R2 = 0.71). Consistently, a strong, one-to-one linear association between ALS-CBS™ and ECAS scores was detected (r = 0.84; R2 = 0.73). The LSEE was able to estimate conversions for the full range of the ALS-CBS™, except for raw scores equal to 1 and 6 – for whom a linear equating-based equation was derived. Empirical ECAS scores were equivalent to those derived with both methods.

Discussion

Italian practitioners and researchers have been herewith provided with valid, straightforward cross-walks to estimate the ECAS based on ALS-CBS™ scores in non-demented ALS patients. Conversions herewith provided will help avoid cross-sectional/longitudinal inconsistencies in test adoption within research, and possibly clinical, settings.

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Fig. 1

Data availability

Datasets associated with the present study are available, upon reasonable request of interested researchers to the Corresponding Author, at the following link: https://doi.org/10.5281/zenodo.7898435.

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Acknowledgements

The Authors are thankful to patients and their caregivers.

Funding

This research was funded by the Italian Ministry of Health to IRCCS Istituto Auxologico Italiano (Ricerca Corrente, project 23C302). This research was also partially supported by the Ricerca Corrente funding scheme of the Italian Ministry of Health to Istituti Clinici Scientifici Maugeri IRCCS.

Author information

Author notes

  1. Edoardo Nicolò Aiello and Federica Solca have contributed equally to this work.

  2. Nicola Ticozzi and Barbara Poletti have contributed equally to this work.

Authors and Affiliations

  1. Department of Neurology and Laboratory of Neuroscience, IRCCS Istituto Auxologico Italiano, Piazzale Brescia 20, 20149, Milan, MI, Italy

    Edoardo Nicolò Aiello, Federica Solca, Silvia Torre, Laura Carelli, Claudia Morelli, Alberto Doretti, Eleonora Colombo, Stefano Messina, Federico Verde, Vincenzo Silani, Nicola Ticozzi & Barbara Poletti

  2. PhD Program in Neuroscience, School of Medicine and Surgery, University of Milano-Bicocca, Monza, Italy

    Edoardo Nicolò Aiello

  3. Neuromuscular Omnicentre (NEMO), Fondazione Serena Onlus, Milan, Italy

    Lucia Catherine Greco, Andrea Lizio, Jacopo Casiraghi, Federica Cerri & Valeria Ada Sansone

  4. NeMO Lab, ASST Grande Ospedale Metropolitano Niguarda, Milan, Italy

    Lucia Catherine Greco

  5. Istituti Clinici Scientifici Maugeri IRCCS, Neurorehabilitation Department of Milan Institute, Milan, Italy

    Debora Pain, Alice Radici & Christian Lunetta

  6. Syneos Health, Morrisville, NC, USA

    Susan Woolley

  7. Biogen, Cambridge, MA, USA

    Jennifer Murphy

  8. School of Medicine and Surgery, University of Milano-Bicocca, Monza, Italy

    Lucio Tremolizzo & Ildebrando Appollonio

  9. Department of Pathophysiology and Transplantation, “Dino Ferrari” Center, Università degli Studi di Milano, Milan, Italy

    Federico Verde, Vincenzo Silani & Nicola Ticozzi

  10. Department of Biomedical Sciences of Health, University of Milan, Milan, Italy

    Valeria Ada Sansone

Authors
  1. Edoardo Nicolò Aiello
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  2. Federica Solca
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  3. Lucia Catherine Greco
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  4. Silvia Torre
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  5. Laura Carelli
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  6. Claudia Morelli
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  7. Alberto Doretti
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  8. Eleonora Colombo
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  9. Stefano Messina
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  10. Debora Pain
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  11. Alice Radici
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  12. Andrea Lizio
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  13. Jacopo Casiraghi
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  14. Federica Cerri
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  15. Susan Woolley
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  16. Jennifer Murphy
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  17. Lucio Tremolizzo
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  18. Ildebrando Appollonio
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  19. Federico Verde
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  20. Valeria Ada Sansone
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  21. Christian Lunetta
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  22. Vincenzo Silani
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  23. Nicola Ticozzi
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  24. Barbara Poletti
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Contributions

ENA: conceptualization, analyses, drafting, revision; FS, LCG: data collection, drafting, revision; ST, LC, CM, AD, EC, SM, DP, AR, AL, JC, FC: data collection, revision; SW, JM, LT, IA, FV, VAS, CL, VS, NT: resources, revision; BP: conceptualization, drafting, revision, resources.

Corresponding author

Correspondence to Barbara Poletti.

Ethics declarations

Conflicts of interest

V. S. received compensation for consulting services and/or speaking activities from AveXis, Cytokinetics, Italfarmaco, Liquidweb S.r.l., and Novartis Pharma AG, receives or has received research support from the Italian Ministry of Health, AriSLA, and E-Rare Joint Transnational Call. He is on the Editorial Board of Amyotrophic Lateral Sclerosis and Frontotemporal Degeneration, European Neurology, American Journal of Neurodegenerative Diseases, Frontiers in Neurology. B.P. and L.C. received compensation for consulting services and/or speaking activities from Liquidweb S.r.l. B.P is Associate Editor for Frontier in Neuroscience. N. T. received compensation for consulting services from Amylyx Pharmaceuticals and Zambon Biotech SA. He is Associate Editor for Frontiers in Aging Neuroscience. C.L. received compensation for consulting services and/or speaking activities from Cytokinetics, Italfarmaco, and Mitsubishi Tanabe Pharma Europe. He is Associate Editor for Frontiers in Neurology. V.A.S. participates in Advisory Boards or teaching activities for Biogen, Roche, Avexis, PTC, Santhera, Sarepta, Dyne. S.W. receives licensing fees when the ALS Cognitive Behavioural Screen (ALS-CBS™) is used in pharmaceutical trials. J.M. is employed full time at Biogen; she does not receives compensations related to the ALS-CBS™. Remaining Authors have nothing to declare.

Ethics

participants provided informed consent and data were treated according to current regulations. This study was approved by the Ethics Committee of IRCCS Istituto Auxologico Italiano (I.D.: 2013_06_25), by the Ethics Committee of Milano Niguarda Area 3 (I.D.: 393-09062021) and by the Ethics Committee of Istituti Clinici Scientifici Maugeri IRCCS (I.D.: CE 2495-12012021).

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Aiello, E.N., Solca, F., Greco, L.C. et al. Equating norms between the ALS Cognitive Behavioral Screen (ALS-CBS™) and the Edinburgh Cognitive and Behavioural ALS Screen (ECAS) in non-demented ALS patients. J Neurol 270, 4090–4095 (2023). https://doi.org/10.1007/s00415-023-11749-4

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