Modelling disease course in amyotrophic lateral Sclerosis: Pseudo-longitudinal insights from cross-sectional health-related quality of life data

Tino Prell*, Nayana Gaur, Robert Steinbach, Otto W. Witte, Julian Grosskreutz

*Corresponding author for this work
8 Citations (Scopus)

Abstract

Background: Amyotrophic Lateral Sclerosis (ALS) is a rapidly progressive neurodegenerative disorder with limited robust disease-modifying therapies presently available. While several treatments are aimed at improving health-related quality of life (HRQoL), longitudinal data on how QoL changes across the disease course are rare. Objectives: To explore longitudinal changes in emotional well-being and HRQoL in ALS. Methods: Of the 161 subjects initially recruited, 39 received 2 subsequent follow-up assessments at 6 and 12 months after baseline. The ALS Functional Rating Scale-Revised (ALSFRS-R) was used to assess physical impairment. HRQoL was assessed using the ALS Assessment Questionnaire (ALSAQ-40). The D50 disease progression model was applied to explore longitudinal changes in HRQoL. Results: Patients were primarily in the early semi-stable and early progressive model-derived disease phases. Non-linear correlation analyses showed that the ALSAQ-40 summary index and emotional well-being subdomain behaved differently across disease phases, indicating that the response shift occurs early in disease. Both the ALSFRS-R and ALSAQ-40 significantly declined at 6-and 12-monthly follow-ups. Conclusion: ALSAQ-40 summary index and emotional well-being change comparably over both actual time and model-derived phases, indicating that the D50 model enables pseudo-longitudinal interpretations of cross-sectional data in ALS.

Original languageEnglish
Article number117
JournalHealth and Quality of Life Outcomes
Volume18
Issue number1
ISSN1477-7525
DOIs
Publication statusPublished - 01.05.2020
Externally publishedYes

Research Areas and Centers

  • Centers: Center for Neuromuscular Diseases

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