Individual changes in preclinical spinocerebellar ataxia identified via increased motor complexity

Ilg, Winfried; Fleszar, Zofia; Schatton, Cornelia; Hengel, Holger; Harmuth, Florian; Bauer, Péter; Timmann, Dagmar; Giese, Martin A.; Schöls, Lüdger; Synofzik, Matthis

doi:10.1002/mds.26835

Cited by 53 publications

(72 citation statements)

References 40 publications

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“…We have shown that precise spatiotemporal calibration in combination with a two-sided setup of sensors provides better statistical agreement with a gold standard motion capturing system than previously reported systems [1,2]. The excellent agreement with the VICON system suggests that, in addition to differences between patients and healthy controls, even subtle changes in gait, such as in preclinical cerebellar ataxia [36], might be detectable. Furthermore, the low-costs make multi-center studies involving clinical centers without dedicated motion laboratories feasible, allowing for distributed investigation of rare movement disorders.…”

Section: Discussionmentioning

confidence: 78%

Validation of enhanced kinect sensor based motion capturing for gait assessment

et al. 2017

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Optical motion capturing systems are expensive and require substantial dedicated space to be set up. On the other hand, they provide unsurpassed accuracy and reliability. In many situations however flexibility is required and the motion capturing system can only temporarily be placed. The Microsoft Kinect v2 sensor is comparatively cheap and with respect to gait analysis promising results have been published. We here present a motion capturing system that is easy to set up, flexible with respect to the sensor locations and delivers high accuracy in gait parameters comparable to a gold standard motion capturing system (VICON). Further, we demonstrate that sensor setups which track the person only from one-side are less accurate and should be replaced by two-sided setups. With respect to commonly analyzed gait parameters, especially step width, our system shows higher agreement with the VICON system than previous reports.

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Section: Discussionmentioning

confidence: 78%

Validation of enhanced kinect sensor based motion capturing for gait assessment

et al. 2017

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“…The definition of the prodromal stage in SCA2 comes from follow‐up and cross‐sectional studies carried out during the past 2 decades in the Cuban and European population of SCA2 preclinical individuals . These studies have included clinical somatic and autonomic assessments of the nervous system, semiquantitative rating of ataxia using clinical scales, neuropsychological studies, and imaging investigations.…”

Section: Criteria Defining the Prodromal Stage In Sca2 Preclinical Camentioning

confidence: 99%

“…Electrophysiological and quantitative neurological tests have become helpful approaches to characterize the SCA2 prodromal stage, providing several functional biomarkers with high diagnostic and predictive value and close association with the mutation size (Table ).…”

Section: Functional Criteriamentioning

confidence: 99%

Prodromal spinocerebellar ataxia type 2: Prospects for early interventions and ethical challenges

2017

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The characterization of prodromal stages in neurodegenerative disorders is becoming increasingly important because of the need for early neuroprotective therapies. Research during the past 3 decades in spinocerebellar ataxia type 2 has revealed a large body of evidence suggesting that many disease features precede the manifest cerebellar syndrome, which delineates the prodromal stage of this disorder. This stage is defined by clinical, imaging, and functional criteria, which are supported by early molecular events demonstrated in animal models. Knowledge regarding prodromal spinocerebellar ataxia type 2 provides insight into the mechanisms underlying neurodegeneration from the early stages, which enables the design of promising disease-modifying clinical trials through the identification of the optimum moment to begin the therapies, the appropriate selection of individuals, and the identification of sensitive outcome measures. The management of patients in prodromal spinocerebellar ataxia type 2 may raise ethical dilemmas related to predictive diagnosis and early interventions, which impose new challenges to clinical and therapeutic research. © 2017 International Parkinson and Movement Disorder Society.

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“…It has been shown in laboratory-based assessments that measures of spatial and temporal movement variability allow distinctively to capture and characterize the specificities of ataxic gait [3][4][5][6][7][8][9][10] . Moreover, they allow to quantify disease severity even at preclinical stages of DCA 11,12 and to capture treatment-induced improvements in a fine-grained fashion [13][14][15] , thus suggesting a high potential as both progression and treatment response parameters in the upcoming treatment trials [16][17][18] . Recently, first studies showed that such measures of spatio-temporal variability characterizing ataxic gait can also be captured using wearable inertial sensors in clinical assessments 19,20 .…”

Section: Discussionmentioning

confidence: 99%

“…Variability measures were calculated using the coefficient of variation CV=σ/μ, normalizing the standard deviation with the mean value 34 . On this basis, stride length CV (StrideL CV ) and stride time CV (StrideT CV ) were determined, which have shown to be sensitive to ataxia severity in constrained lab-based walking 3,5,7,11,12 .…”

Section: Discussionmentioning

confidence: 99%