Skip to main content
Erschienen in: Clinical Autonomic Research 6/2022

01.10.2022 | Research Article

Responsiveness of UMSARS and other clinical measures in a longitudinal structured care clinic for multiple system atrophy

verfasst von: Elisabeth P. Golden, Morgan McCreary, Steven Vernino

Erschienen in: Clinical Autonomic Research | Ausgabe 6/2022

Einloggen, um Zugang zu erhalten

Abstract

Purpose

As understanding of multiple system atrophy (MSA) pathophysiology improves, clinical trials of disease-modifying therapies are starting. Outcome measures responsive to disease progression will be critical, but the United MSA Rating Scale (UMSARS) has limitations. The MSA multidisciplinary clinic at the University of Texas Southwestern is a longitudinal clinic with structured assessments performed at fixed time intervals. The objective of this study was to evaluate the performance of clinical measures in assessing MSA progression over time.

Methods

Data from 73 subjects with clinically diagnosed MSA were analyzed using repeated measures correlation models. Observations were made every 4 months, with up to 3 years of data included for each patient.

Results

UMSARS-I and UMSARS-II correlated positively with the MSA Quality of Life (QOL) scale. The rate of change was 3.12 points per year (ppy) for UMSARS-I and 5.55 ppy for UMSARS-II.
Some individual UMSARS questions contributed more significantly than others to overall UMSARS rate of change. Based on this finding, and using repeated measures correlations between question combinations and QOL, an optimization of UMSARS parts I and II was curated. The amended UMSARS-I included 8 of the 12 subquestions, and the amended UMSARS-II included 10 of the 14 subquestions.

Conclusions

Data from a longitudinal MSA clinic allows better characterization of the performance of UMSARS as a clinical outcome measure. A curated set of UMSARS questions appears more responsive to change and accounts for correlation with QOL, and could be the starting point for an improved MSA outcome measure.
Literatur
1.
Zurück zum Zitat Wenning GK, Tison F, Seppi K, Sampaio C, Diem A, Yekhlef F, Ghorayeb I, Ory F, Galitzky M, Scaravilli T, Bozi M, Colosimo C, Gilman S, Shults CW, Quinn NP, Rascol O, Poewe W (2004) Development and validation of the unified multiple system atrophy rating scale (UMSARS). Mov Disord 19(12):1391–1402. https://doi.org/10.1002/mds.20255CrossRefPubMed Wenning GK, Tison F, Seppi K, Sampaio C, Diem A, Yekhlef F, Ghorayeb I, Ory F, Galitzky M, Scaravilli T, Bozi M, Colosimo C, Gilman S, Shults CW, Quinn NP, Rascol O, Poewe W (2004) Development and validation of the unified multiple system atrophy rating scale (UMSARS). Mov Disord 19(12):1391–1402. https://​doi.​org/​10.​1002/​mds.​20255CrossRefPubMed
2.
Zurück zum Zitat Palma JA, Vernetti PM, Perez MA, Krismer F, Seppi K, Fanciulli A, Singer W, Low P, Biaggioni I, Norcliffe-Kaufmann L, Pellecchia MT, Martí MJ, Kim HJ, Merello M, Stankovic I, Poewe W, Betensky R, Wenning G, Kaufmann H (2021) Limitations of the unified multiple system atrophy rating scale as outcome measure for clinical trials and a roadmap for improvement. Clin Auton Res 31(2):157–164. https://doi.org/10.1007/s10286-021-00782-wCrossRefPubMedPubMedCentral Palma JA, Vernetti PM, Perez MA, Krismer F, Seppi K, Fanciulli A, Singer W, Low P, Biaggioni I, Norcliffe-Kaufmann L, Pellecchia MT, Martí MJ, Kim HJ, Merello M, Stankovic I, Poewe W, Betensky R, Wenning G, Kaufmann H (2021) Limitations of the unified multiple system atrophy rating scale as outcome measure for clinical trials and a roadmap for improvement. Clin Auton Res 31(2):157–164. https://​doi.​org/​10.​1007/​s10286-021-00782-wCrossRefPubMedPubMedCentral
4.
Zurück zum Zitat Wenning GK, Geser F, Krismer F, Seppi K, Duerr S, Boesch S, Kollensperger M, Goebel G, Pfeiffer KP, Barone P, Pellecchia MT, Quinn NP, Koukouni V, Fowler CJ, Schrag A, Mathias CJ, Giladi N, Gurevich T, Dupont E, Ostergaard K, Nilsson CF, Widner H, Oertel W, Eggert KM, Albanese A, del Sorbo F, Tolosa E, Cardozo A, Deuschl G, Hellriegel H, Klockgether T, Dodel R, Sampaio C, Coelho M, Djaldetti R, Melamed E, Gasser T, Kamm C, Meco G, Colosimo C, Rascol O, Meissner WG, Tison F, Poewe W (2013) The natural history of multiple system atrophy: a prospective European cohort study. Lancet Neurol 12(3):264–274. https://doi.org/10.1016/s1474-4422(12)70327-7CrossRefPubMedPubMedCentral Wenning GK, Geser F, Krismer F, Seppi K, Duerr S, Boesch S, Kollensperger M, Goebel G, Pfeiffer KP, Barone P, Pellecchia MT, Quinn NP, Koukouni V, Fowler CJ, Schrag A, Mathias CJ, Giladi N, Gurevich T, Dupont E, Ostergaard K, Nilsson CF, Widner H, Oertel W, Eggert KM, Albanese A, del Sorbo F, Tolosa E, Cardozo A, Deuschl G, Hellriegel H, Klockgether T, Dodel R, Sampaio C, Coelho M, Djaldetti R, Melamed E, Gasser T, Kamm C, Meco G, Colosimo C, Rascol O, Meissner WG, Tison F, Poewe W (2013) The natural history of multiple system atrophy: a prospective European cohort study. Lancet Neurol 12(3):264–274. https://​doi.​org/​10.​1016/​s1474-4422(12)70327-7CrossRefPubMedPubMedCentral
6.
Zurück zum Zitat Donadio V, Incensi A, Rizzo G, De Micco R, Tessitore A, Devigili G, Del Sorbo F, Bonvegna S, Infante R, Magnani M, Zenesini C, Vignatelli L, Cilia R, Eleopra R, Tedeschi G, Liguori R (2020) Skin biopsy may help to distinguish multiple system atrophy-parkinsonism from Parkinson’s disease with orthostatic hypotension. Mov Disord 35(9):1649–1657. https://doi.org/10.1002/mds.28126CrossRefPubMed Donadio V, Incensi A, Rizzo G, De Micco R, Tessitore A, Devigili G, Del Sorbo F, Bonvegna S, Infante R, Magnani M, Zenesini C, Vignatelli L, Cilia R, Eleopra R, Tedeschi G, Liguori R (2020) Skin biopsy may help to distinguish multiple system atrophy-parkinsonism from Parkinson’s disease with orthostatic hypotension. Mov Disord 35(9):1649–1657. https://​doi.​org/​10.​1002/​mds.​28126CrossRefPubMed
10.
Zurück zum Zitat Wenning GK, Stankovic I, Vignatelli L, Fanciulli A, Calandra-Buonaura G, Seppi K, Palma JA, Meissner WG, Krismer F, Berg D, Cortelli P, Freeman R, Halliday G, Höglinger G, Lang A, Ling H, Litvan I, Low P, Miki Y, Panicker J, Pellecchia MT, Quinn N, Sakakibara R, Stamelou M, Tolosa E, Tsuji S, Warner T, Poewe W, Kaufmann H (2022) The movement disorder society criteria for the diagnosis of multiple system atrophy. Mov Disord 37(6):1131–1148. https://doi.org/10.1002/mds.29005CrossRefPubMedPubMedCentral Wenning GK, Stankovic I, Vignatelli L, Fanciulli A, Calandra-Buonaura G, Seppi K, Palma JA, Meissner WG, Krismer F, Berg D, Cortelli P, Freeman R, Halliday G, Höglinger G, Lang A, Ling H, Litvan I, Low P, Miki Y, Panicker J, Pellecchia MT, Quinn N, Sakakibara R, Stamelou M, Tolosa E, Tsuji S, Warner T, Poewe W, Kaufmann H (2022) The movement disorder society criteria for the diagnosis of multiple system atrophy. Mov Disord 37(6):1131–1148. https://​doi.​org/​10.​1002/​mds.​29005CrossRefPubMedPubMedCentral
14.
Zurück zum Zitat R: A language and environment for statistical computing (2021) In: R Core Team, R Foundation for Statistical Computing, Vienna, Austria R: A language and environment for statistical computing (2021) In: R Core Team, R Foundation for Statistical Computing, Vienna, Austria
16.
Zurück zum Zitat Bürkner P-C (2018) Advanced Bayesian multilevel modeling with the R package brms. R J 10(1):395–411CrossRef Bürkner P-C (2018) Advanced Bayesian multilevel modeling with the R package brms. R J 10(1):395–411CrossRef
17.
Zurück zum Zitat Lüdecke D, Ben-Shachar MS, Patil I, Waggoner P, Makowski D (2021) Performance: an R package for assessment, comparison and testing of statistical models. J Open Sour Softw 6(60):3139CrossRef Lüdecke D, Ben-Shachar MS, Patil I, Waggoner P, Makowski D (2021) Performance: an R package for assessment, comparison and testing of statistical models. J Open Sour Softw 6(60):3139CrossRef
21.
Zurück zum Zitat Meissner WG, Foubert-Samier A, Dupouy S, Gerdelat-Mas A, Debs R, Marquant F, De Cock VC, Rascol O, Tison F, Pavy-Le Traon A (2012) Assessment of quality of life with the multiple system atrophy health-related quality of life scale. Mov Disord 27(12):1574–1577. https://doi.org/10.1002/mds.25174CrossRefPubMed Meissner WG, Foubert-Samier A, Dupouy S, Gerdelat-Mas A, Debs R, Marquant F, De Cock VC, Rascol O, Tison F, Pavy-Le Traon A (2012) Assessment of quality of life with the multiple system atrophy health-related quality of life scale. Mov Disord 27(12):1574–1577. https://​doi.​org/​10.​1002/​mds.​25174CrossRefPubMed
Metadaten
Titel
Responsiveness of UMSARS and other clinical measures in a longitudinal structured care clinic for multiple system atrophy
verfasst von
Elisabeth P. Golden
Morgan McCreary
Steven Vernino
Publikationsdatum
01.10.2022
Verlag
Springer Berlin Heidelberg
Erschienen in
Clinical Autonomic Research / Ausgabe 6/2022
Print ISSN: 0959-9851
Elektronische ISSN: 1619-1560
DOI
https://doi.org/10.1007/s10286-022-00898-7

Weitere Artikel der Ausgabe 6/2022

Clinical Autonomic Research 6/2022 Zur Ausgabe