% IMPORTANT: The following is UTF-8 encoded.  This means that in the presence
% of non-ASCII characters, it will not work with BibTeX 0.99 or older.
% Instead, you should use an up-to-date BibTeX implementation like “bibtex8” or
% “biber”.

@ARTICLE{Regensburger:164659,
      author       = {Regensburger, Martin and Spatz, Imke Tabea and
                      Ollenschläger, Malte and Martindale, Christine F and
                      Lindeburg, Philipp and Kohl, Zacharias and Eskofier, Björn
                      and Klucken, Jochen and Schüle, Rebecca and Klebe, Stephan
                      and Winkler, Jürgen and Gaßner, Heiko},
      title        = {{I}nertial {G}ait {S}ensors to {M}easure {M}obility and
                      {F}unctioning in {H}ereditary {S}pastic {P}araplegia: {A}
                      {C}ross-{S}ectional {M}ulticenter {C}linical {S}tudy},
      journal      = {Neurology},
      volume       = {99},
      number       = {10},
      issn         = {0028-3878},
      address      = {[S.l.]},
      publisher    = {Ovid},
      reportid     = {DZNE-2022-01189},
      pages        = {e1079 - e1089},
      year         = {2022},
      abstract     = {Hereditary spastic paraplegia (HSP) causes progressive
                      spasticity and weakness of the lower limbs. As neurological
                      examination and the clinical Spastic Paraplegia Rating Scale
                      (SPRS) are subject to potential patient- and
                      clinician-dependent bias, instrumented gait analysis bears
                      the potential to objectively quantify impaired gait. The aim
                      of the present study was to investigate gait cyclicity
                      parameters by application of a mobile gait analysis system
                      in a cross sectional cohort of HSP patients and a
                      longitudinal fast progressing subcohort.Methods: Using
                      wearable sensors attached to the shoes, HSP patients and
                      controls performed a 4x10 meters walking test during regular
                      visits in three outpatient centers. Patients were also rated
                      according to the SPRS and in a subset, questionnaires on
                      quality of life and fear of falling were obtained. An
                      unsupervised segmentation algorithm was employed to extract
                      stride parameters and respective coefficients of
                      variation.Results: Mobile gait analysis was performed in a
                      total of 112 ambulatory HSP patients and 112 age and gender
                      matched controls. While swing time was unchanged compared to
                      controls, there were significant increases in the duration
                      of the total stride phase and the duration of the stance
                      phase, both regarding absolute values and coefficients of
                      variation values. While stride parameters did not correlate
                      to age, weight or height of the patients, there were
                      significant associations of absolute stride parameters to
                      single SPRS items reflecting impaired mobility (|r| > 0.50),
                      to patients' quality of life (|r| > 0.44), and notably to
                      disease duration (|r| > 0.27). Sensor-derived coefficients
                      of variation, on the other hand, were associated with
                      patient-reported fear of falling (|r| > 0.41) and cognitive
                      impairment (|r| > 0.40). In a small 1-year follow-up
                      analysis of patients with complicated HSP and fast
                      progression, absolute values of mobile gait parameters had
                      significantly worsened compared to baseline.Discussion: The
                      presented wearable sensor system provides parameters of
                      stride characteristics which appear clinically valid to
                      reflect gait impairment in HSP. Due to the feasibility with
                      regard to time, space and costs, the present study forms the
                      basis for larger scale longitudinal and interventional
                      studies in HSP.},
      keywords     = {Gait Analysis (Other) / Wearables (Other) / [163] Gait
                      disorders/ataxia (Other) / [174] Spastic paraplegia (Other)},
      cin          = {AG Maetzler},
      ddc          = {610},
      cid          = {I:(DE-2719)5000024},
      pnm          = {353 - Clinical and Health Care Research (POF4-353)},
      pid          = {G:(DE-HGF)POF4-353},
      typ          = {PUB:(DE-HGF)16},
      pubmed       = {pmid:35667840},
      doi          = {10.1212/WNL.0000000000200819},
      url          = {https://pub.dzne.de/record/164659},
}