Postural Directionality and Head Tremor in Cervical Dystonia

Chen, Qiyu; Vu, Jeanne P.; Cisneros, Elizabeth; Benadof, Casey N.; Zhang, Zheng; Barbano, Richard L.; Goetz, Christopher G.; Jankovic, Joseph; Jinnah, Hyder A.; Perlmutter, Joel S.; Appelbaum, Mark I.; Stebbins, Glenn T.; Comella, Cynthia L.; Peterson, David A.

doi:10.5334/tohm.526

Cited by 11 publications

(8 citation statements)

References 11 publications

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“…With the exception of pitch (anterocollis vs. retrocollis), this directionality information is lost in clinical rating scales. Yet the directionality of pitch has been associated with likelihood of comorbid head tremor in CD 36 and in turn head tremor subtype is differentially associated with pain severity 37 . In our cohort, we found that there was no bias toward anterocollis versus retrocollis, left versus right in laterocollis (“tilt”, roll), and left versus right in torticollis (“rotation”, yaw).…”

Section: Discussionmentioning

confidence: 54%

Hold that pose: capturing cervical dystonia's head deviation severity from video

Zhang

Cisneros

Lee

et al. 2022

Ann Clin Transl Neurol

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Objective Deviated head posture is a defining characteristic of cervical dystonia (CD). Head posture severity is typically quantified with clinical rating scales such as the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS). Because clinical rating scales are inherently subjective, they are susceptible to variability that reduces their sensitivity as outcome measures. The variability could be circumvented with methods to measure CD head posture objectively. However, previously used objective methods require specialized equipment and have been limited to studies with a small number of cases. The objective of this study was to evaluate a novel software system—the Computational Motor Objective Rater (CMOR)—to quantify multi‐axis directionality and severity of head posture in CD using only conventional video camera recordings. Methods CMOR is based on computer vision and machine learning technology that captures 3D head angle from video. We used CMOR to quantify the axial patterns and severity of predominant head posture in a retrospective, cross‐sectional study of 185 patients with isolated CD recruited from 10 sites in the Dystonia Coalition. Results The predominant head posture involved more than one axis in 80.5% of patients and all three axes in 44.4%. CMOR's metrics for head posture severity correlated with severity ratings from movement disorders neurologists using both the TWSTRS‐2 and an adapted version of the Global Dystonia Rating Scale (rho = 0.59–0.68, all p <0.001). Conclusions CMOR's convergent validity with clinical rating scales and reliance upon only conventional video recordings supports its future potential for large scale multisite clinical trials.

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Section: Discussionmentioning

confidence: 54%

Hold that pose: capturing cervical dystonia's head deviation severity from video

Zhang

Cisneros

Lee

et al. 2022

Ann Clin Transl Neurol

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“…This information could be used to determine whether the arms up maneuver and sensory tricks have differential effects on HT even for individual patients. Third, although in our cross-sectional design we found that the likelihood that raising arms decreases HT severity is independent of demographic variables, HT likelihood in CD is higher for older age at onset [ 13 14 ], longer disease duration [ 14 15 ], and female gender [ 1 14 ]. Longitudinal studies could determine whether individual patients exhibit changes over time in the relationship of HT to the arms up maneuver.…”

Section: Discussionmentioning

confidence: 92%

Does Raising the Arms Modify Head Tremor Severity in Cervical Dystonia?

Cisneros

Lee

et al. 2021

Tremor and Other Hyperkinetic Movements

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Background: A defining characteristic of dystonia is its position-dependence. In cervical dystonia (CD), sensory tricks ameliorate head tremor (HT). But it remains unknown whether raising the arms alone has the same impact. Methods:We analyzed data collected from patients enrolled by the Dystonia Coalition. For 120 patients with HT, we assessed how raising their arms without touching their head changed their HT severity.Results: Forty-eight out of 120 patients exhibited changes in HT severity when raising their arms. These patients were more likely to exhibit decreases in HT severity (N = 35) than increases (N = 13, χ 2 (1, N = 48) = 10.1, p = 0.002). Demographic factors and sensory trick efficacy were not significant predictors of whether HT severity changed when raising their arms.Discussion: Raising the arms without touching the head is a posture that can reduce HT severity in some CD patients. Our results extend the concept of position-dependent motor symptoms in CD to include the position of the arms. HighlightsHead tremor (HT) is a prevalent symptom of cervical dystonia (CD) that can often be disabling. This study demonstrates that raising the arms without touching the head is a posture that can reduce HT severity in some CD patients. Our findings also identify a novel form of position-dependence in CD.

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“…There has been great enthusiasm in recent years for developing more objective outcome measures that could be used to monitor abnormal movements for dystonia 20‐22,34‐37 . Presumably these measures would not be impacted by patient impressions, and they should be quite reliable.…”

Section: Discussionmentioning

confidence: 99%

An Exploratory, Randomized, Double‐Blind Clinical Trial of Dipraglurant for Blepharospasm

Kilic‐Berkmen,

Kim,

Chen

et al. 2024

Movement Disorders

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BackgroundBlepharospasm is treated with botulinum toxin, but obtaining satisfactory results is sometimes challenging.ObjectiveThe aim is to conduct an exploratory trial of oral dipraglurant for blepharospasm.MethodsThis study was an exploratory, phase 2a, randomized, double‐blind, placebo‐controlled trial of 15 participants who were assigned to receive a placebo or dipraglurant (50 or 100 mg) and assessed over 2 days, 1 and 2 hours following dosing. Outcome measures included multiple scales rated by clinicians or participants, digital video, and a wearable sensor.ResultsDipraglurant was well tolerated, with no obvious impact on any of the measurement outcomes. Power analyses suggested fewer subjects would be required for studies using a within‐subject versus independent group design, especially for certain measures. Some outcome measures appeared more suitable than others.ConclusionAlthough dipraglurant appeared well tolerated, it did not produce a trend for clinical benefit. The results provide valuable information for planning further trials in blepharospasm. © 2024 International Parkinson and Movement Disorder Society.

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Postural Directionality and Head Tremor in Cervical Dystonia

Cited by 11 publications

References 11 publications

Hold that pose: capturing cervical dystonia's head deviation severity from video

Hold that pose: capturing cervical dystonia's head deviation severity from video

Does Raising the Arms Modify Head Tremor Severity in Cervical Dystonia?

An Exploratory, Randomized, Double‐Blind Clinical Trial of Dipraglurant for Blepharospasm

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