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@ARTICLE{Thierfelder:163303,
      author       = {Thierfelder, Annika and Seemann, Jens and John, Natalie and
                      Harmuth, Florian and Giese, Martin and Schüle, Rebecca and
                      Schöls, Ludger and Timmann, Dagmar and Synofzik, Matthis
                      and Ilg, Winfried},
      title        = {{R}eal-{L}ife {T}urning {M}ovements {C}apture {S}ubtle
                      {L}ongitudinal and {P}reataxic {C}hanges in {C}erebellar
                      {A}taxia.},
      journal      = {Movement disorders},
      volume       = {37},
      number       = {5},
      issn         = {1531-8257},
      address      = {New York, NY},
      publisher    = {Wiley},
      reportid     = {DZNE-2022-00083},
      pages        = {1047-1058},
      year         = {2022},
      note         = {(CC BY-NC-ND 4.0)},
      abstract     = {Clinical and regulatory acceptance of upcoming molecular
                      treatments in degenerative ataxias might greatly benefit
                      from ecologically valid endpoints that capture change in
                      ataxia severity in patients' real life.This longitudinal
                      study aimed to unravel quantitative motor biomarkers in
                      degenerative ataxias in real-life turning movements that are
                      sensitive for changes both longitudinally and at the
                      preataxic stage.Combined cross-sectional (n = 30) and
                      longitudinal (n = 14, 1-year interval) observational study
                      in degenerative cerebellar disease (including eight
                      preataxic mutation carriers) compared to 23 healthy
                      controls. Turning movements were assessed by three body-worn
                      inertial sensors in three conditions: (1) instructed
                      laboratory assessment, (2) supervised free walking, and (3)
                      unsupervised real-life movements.Measures that quantified
                      dynamic balance during turning-lateral velocity change (LVC)
                      and outward acceleration-but not general turning measures
                      such as speed, allowed differentiating ataxic against
                      healthy subjects in real life (effect size δ = 0.68), with
                      LVC also differentiating preataxic against healthy subjects
                      (δ = 0.53). LVC was highly correlated with clinical ataxia
                      severity (scale for the assessment and rating of ataxia
                      [SARA] score, effect size ρ = 0.79) and patient reported
                      balance confidence (activity-specific balance confidence
                      scale [ABC] score, ρ = 0.66). Moreover, LVC in real
                      life-but not general turning measures or the SARA
                      score-allowed detecting significant longitudinal change in
                      1-year follow-up with high effect size (rprb =
                      0.66).Measures of turning allow capturing specific changes
                      of dynamic balance in degenerative ataxia in real life, with
                      high sensitivity to longitudinal differences in ataxia
                      severity and to the preataxic stage. They thus present
                      promising ecologically valid motor biomarkers, even in the
                      highly treatment-relevant early stages of degenerative
                      cerebellar disease. © 2022 The Authors. Movement Disorders
                      published by Wiley Periodicals LLC on behalf of
                      International Parkinson and Movement Disorder Society.},
      keywords     = {Ataxia / Biomarkers / Cerebellar Ataxia / Cross-Sectional
                      Studies / Humans / Longitudinal Studies / Spinocerebellar
                      Ataxias: genetics / cerebellar ataxia (Other) / motor
                      biomarker (Other) / real-life walking (Other) / turning
                      (Other) / wearable sensors (Other)},
      cin          = {AG Maetzler / AG Gasser 1},
      ddc          = {610},
      cid          = {I:(DE-2719)5000024 / I:(DE-2719)1210000},
      pnm          = {353 - Clinical and Health Care Research (POF4-353)},
      pid          = {G:(DE-HGF)POF4-353},
      typ          = {PUB:(DE-HGF)16},
      pubmed       = {pmid:35067979},
      doi          = {10.1002/mds.28930},
      url          = {https://pub.dzne.de/record/163303},
}