Digital gait outcomes for autosomal recessive spastic ataxia of Charlevoix-Saguenay (ARSACS): discriminative, convergent, and ecological validity in a multicenter study (PROSPAX)
Background: With treatment trials on the horizon, this study aimed to identify candidate digital-motor gait outcomes for autosomal recessive spastic ataxia of Charlevoix-Saguenay (ARSACS), capturable by wearable sensors with multicenter validity, and ideally also ecological validity during free walk...
Gespeichert in:
| Hauptverfasser: | , , , , , , , , , |
|---|---|
| Dokumenttyp: | Article (Journal) |
| Sprache: | Englisch |
| Veröffentlicht: |
7 June 2024
|
| In: |
Movement disorders
Year: 2024, Jahrgang: 39, Heft: 9, Pages: 1544-1555 |
| ISSN: | 1531-8257 |
| DOI: | 10.1002/mds.29876 |
| Online-Zugang: | Verlag, kostenfrei, Volltext: https://doi.org/10.1002/mds.29876 Verlag, kostenfrei, Volltext: https://onlinelibrary.wiley.com/doi/abs/10.1002/mds.29876 
|
| Verfasserangaben: | Lukas Beichert, Winfried Ilg, Christoph Kessler, Andreas Traschütz, Selina Reich, Filippo M. Santorelli, Ayşe Nazli Başak, Cynthia Gagnon, Prospax Consortium, Rebecca Schüle, and Matthis Synofzik |
MARC
| LEADER | 00000caa a2200000 c 4500 | ||
|---|---|---|---|
| 001 | 1910012742 | ||
| 003 | DE-627 | ||
| 005 | 20250716213327.0 | ||
| 007 | cr uuu---uuuuu | ||
| 008 | 241202s2024 xx |||||o 00| ||eng c | ||
| 024 | 7 | |a 10.1002/mds.29876 |2 doi | |
| 035 | |a (DE-627)1910012742 | ||
| 035 | |a (DE-599)KXP1910012742 | ||
| 035 | |a (OCoLC)1528014386 | ||
| 040 | |a DE-627 |b ger |c DE-627 |e rda | ||
| 041 | |a eng | ||
| 084 | |a 33 |2 sdnb | ||
| 100 | 1 | |a Beichert, Lukas David |d 1991- |e VerfasserIn |0 (DE-588)125045302X |0 (DE-627)1787330249 |4 aut | |
| 245 | 1 | 0 | |a Digital gait outcomes for autosomal recessive spastic ataxia of Charlevoix-Saguenay (ARSACS) |b discriminative, convergent, and ecological validity in a multicenter study (PROSPAX) |c Lukas Beichert, Winfried Ilg, Christoph Kessler, Andreas Traschütz, Selina Reich, Filippo M. Santorelli, Ayşe Nazli Başak, Cynthia Gagnon, Prospax Consortium, Rebecca Schüle, and Matthis Synofzik |
| 264 | 1 | |c 7 June 2024 | |
| 300 | |b Illustrationen | ||
| 300 | |a 12 | ||
| 336 | |a Text |b txt |2 rdacontent | ||
| 337 | |a Computermedien |b c |2 rdamedia | ||
| 338 | |a Online-Ressource |b cr |2 rdacarrier | ||
| 500 | |a Gesehen am 02.12.2024 | ||
| 520 | |a Background: With treatment trials on the horizon, this study aimed to identify candidate digital-motor gait outcomes for autosomal recessive spastic ataxia of Charlevoix-Saguenay (ARSACS), capturable by wearable sensors with multicenter validity, and ideally also ecological validity during free walking outside laboratory settings. Methods: Cross-sectional multicenter study (four centers), with gait assessments in 36 subjects (18 ARSACS patients; 18 controls) using three body-worn sensors (Opal, APDM) in laboratory settings and free walking in public spaces. Sensor gait measures were analyzed for discriminative validity from controls, and for convergent (ie, clinical and patient relevance) validity by correlations with SPRSmobility (primary outcome) and Scale for the Assessment and Rating of Ataxia (SARA), Spastic Paraplegia Rating Scale (SPRS), and activities of daily living subscore of the Friedreich Ataxia Rating Scale (FARS-ADL) (exploratory outcomes). Results: Of 30 hypothesis-based digital gait measures, 14 measures discriminated ARSACS patients from controls with large effect sizes (|Cliff's δ| > 0.8) in laboratory settings, with strongest discrimination by measures of spatiotemporal variability Lateral Step Deviation (δ = 0.98), SPcmp (δ = 0.94), and Swing CV (δ = 0.93). Large correlations with the SPRSmobility were observed for Swing CV (Spearman's ρ = 0.84), Speed (ρ = −0.63), and Harmonic Ratio V (ρ = −0.62). During supervised free walking in a public space, 11/30 gait measures discriminated ARSACS from controls with large effect sizes. Large correlations with SPRSmobility were here observed for Swing CV (ρ = 0.78) and Speed (ρ = −0.69), without reductions in effect sizes compared with laboratory settings. Conclusions: We identified a promising set of digital-motor candidate gait outcomes for ARSACS, applicable in multicenter settings, correlating with patient-relevant health aspects, and with high validity also outside laboratory settings, thus simulating real-life walking with higher ecological validity. | ||
| 650 | 4 | |a ARSACS | |
| 650 | 4 | |a digital gait outcomes | |
| 650 | 4 | |a outcome measures | |
| 650 | 4 | |a spastic ataxia | |
| 650 | 4 | |a wearable sensors | |
| 700 | 1 | |a Ilg, Winfried |e VerfasserIn |4 aut | |
| 700 | 1 | |a Kessler, Christoph |e VerfasserIn |4 aut | |
| 700 | 1 | |a Traschütz, Andreas |e VerfasserIn |4 aut | |
| 700 | 1 | |a Reich, Selina |e VerfasserIn |4 aut | |
| 700 | 1 | |a Santorelli, Filippo M. |e VerfasserIn |4 aut | |
| 700 | 1 | |a Başak, Ayşe Nazli |e VerfasserIn |4 aut | |
| 700 | 1 | |a Gagnon, Cynthia |e VerfasserIn |4 aut | |
| 700 | 1 | |a Schüle-Freyer, Rebecca |d 1975- |e VerfasserIn |0 (DE-588)137544472 |0 (DE-627)695848283 |0 (DE-576)30411118X |4 aut | |
| 700 | 1 | |a Synofzik, Matthis |e VerfasserIn |4 aut | |
| 773 | 0 | 8 | |i Enthalten in |t Movement disorders |d New York, NY : Wiley, 1986 |g 39(2024), 9 vom: Sept., Seite 1544-1555 |h Online-Ressource |w (DE-627)326179658 |w (DE-600)2041249-6 |w (DE-576)094427003 |x 1531-8257 |7 nnas |a Digital gait outcomes for autosomal recessive spastic ataxia of Charlevoix-Saguenay (ARSACS) discriminative, convergent, and ecological validity in a multicenter study (PROSPAX) |
| 773 | 1 | 8 | |g volume:39 |g year:2024 |g number:9 |g month:09 |g pages:1544-1555 |g extent:12 |a Digital gait outcomes for autosomal recessive spastic ataxia of Charlevoix-Saguenay (ARSACS) discriminative, convergent, and ecological validity in a multicenter study (PROSPAX) |
| 856 | 4 | 0 | |u https://doi.org/10.1002/mds.29876 |x Verlag |x Resolving-System |z kostenfrei |3 Volltext |
| 856 | 4 | 0 | |u https://onlinelibrary.wiley.com/doi/abs/10.1002/mds.29876 |x Verlag |z kostenfrei |3 Volltext |
| 951 | |a AR | ||
| 992 | |a 20241202 | ||
| 993 | |a Article | ||
| 994 | |a 2024 | ||
| 998 | |g 137544472 |a Schüle-Freyer, Rebecca |m 137544472:Schüle-Freyer, Rebecca |d 910000 |d 911100 |e 910000PS137544472 |e 911100PS137544472 |k 0/910000/ |k 1/910000/911100/ |p 9 | ||
| 999 | |a KXP-PPN1910012742 |e 4629481389 | ||
| BIB | |a Y | ||
| SER | |a journal | ||
| JSO | |a {"origin":[{"dateIssuedKey":"2024","dateIssuedDisp":"7 June 2024"}],"relHost":[{"part":{"text":"39(2024), 9 vom: Sept., Seite 1544-1555","issue":"9","extent":"12","pages":"1544-1555","year":"2024","volume":"39"},"language":["eng"],"note":["Gesehen am 13.03.2014"],"pubHistory":["1.1986 -"],"disp":"Digital gait outcomes for autosomal recessive spastic ataxia of Charlevoix-Saguenay (ARSACS) discriminative, convergent, and ecological validity in a multicenter study (PROSPAX)Movement disorders","type":{"media":"Online-Ressource","bibl":"periodical"},"corporate":[{"role":"isb","display":"Movement Disorder Society"}],"recId":"326179658","id":{"eki":["326179658"],"issn":["1531-8257"],"doi":["10.1002/(ISSN)1531-8257"],"zdb":["2041249-6"]},"physDesc":[{"extent":"Online-Ressource"}],"title":[{"title":"Movement disorders","subtitle":"official journal of the Movement Disorder Society","title_sort":"Movement disorders"}],"origin":[{"dateIssuedKey":"1986","publisherPlace":"New York, NY","publisher":"Wiley","dateIssuedDisp":"1986-"}]}],"physDesc":[{"noteIll":"Illustrationen","extent":"12 S."}],"id":{"eki":["1910012742"],"doi":["10.1002/mds.29876"]},"type":{"media":"Online-Ressource","bibl":"article-journal"},"title":[{"subtitle":"discriminative, convergent, and ecological validity in a multicenter study (PROSPAX)","title":"Digital gait outcomes for autosomal recessive spastic ataxia of Charlevoix-Saguenay (ARSACS)","title_sort":"Digital gait outcomes for autosomal recessive spastic ataxia of Charlevoix-Saguenay (ARSACS)"}],"note":["Gesehen am 02.12.2024"],"recId":"1910012742","person":[{"family":"Beichert","role":"aut","display":"Beichert, Lukas David","given":"Lukas David"},{"display":"Ilg, Winfried","given":"Winfried","family":"Ilg","role":"aut"},{"role":"aut","family":"Kessler","given":"Christoph","display":"Kessler, Christoph"},{"family":"Traschütz","role":"aut","given":"Andreas","display":"Traschütz, Andreas"},{"role":"aut","family":"Reich","display":"Reich, Selina","given":"Selina"},{"display":"Santorelli, Filippo M.","given":"Filippo M.","role":"aut","family":"Santorelli"},{"role":"aut","family":"Başak","given":"Ayşe Nazli","display":"Başak, Ayşe Nazli"},{"given":"Cynthia","display":"Gagnon, Cynthia","role":"aut","family":"Gagnon"},{"role":"aut","family":"Schüle-Freyer","given":"Rebecca","display":"Schüle-Freyer, Rebecca"},{"display":"Synofzik, Matthis","given":"Matthis","family":"Synofzik","role":"aut"}],"name":{"displayForm":["Lukas Beichert, Winfried Ilg, Christoph Kessler, Andreas Traschütz, Selina Reich, Filippo M. Santorelli, Ayşe Nazli Başak, Cynthia Gagnon, Prospax Consortium, Rebecca Schüle, and Matthis Synofzik"]},"language":["eng"]} | ||
| SRT | |a BEICHERTLUDIGITALGAI7202 | ||