TY - JOUR
T1 - Modelling disease course in amyotrophic lateral Sclerosis
T2 - Pseudo-longitudinal insights from cross-sectional health-related quality of life data
AU - Prell, Tino
AU - Gaur, Nayana
AU - Steinbach, Robert
AU - Witte, Otto W.
AU - Grosskreutz, Julian
N1 - Publisher Copyright:
© 2020 The Author(s).
PY - 2020/5/1
Y1 - 2020/5/1
N2 - 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.
AB - 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.
UR - http://www.scopus.com/inward/record.url?scp=85084475839&partnerID=8YFLogxK
U2 - 10.1186/s12955-020-01372-6
DO - 10.1186/s12955-020-01372-6
M3 - Journal articles
C2 - 32357946
AN - SCOPUS:85084475839
SN - 1477-7525
VL - 18
JO - Health and Quality of Life Outcomes
JF - Health and Quality of Life Outcomes
IS - 1
M1 - 117
ER -