TY - JOUR
T1 - Head tremor in cervical dystonia
T2 - Quantifying severity with computer vision
AU - Vu, Jeanne P.
AU - Cisneros, Elizabeth
AU - Lee, Ha Yeon
AU - Le, Linh
AU - Chen, Qiyu
AU - Guo, Xiaoyan A.
AU - Rouzbehani, Ryin
AU - Jankovic, Joseph
AU - Factor, Stewart
AU - Goetz, Christopher G.
AU - Barbano, Richard L.
AU - Perlmutter, Joel S.
AU - Jinnah, Hyder A.
AU - Pirio Richardson, Sarah
AU - Stebbins, Glenn T.
AU - Elble, Rodger
AU - Comella, Cynthia L.
AU - Peterson, David A.
N1 - Funding Information:
Grants: research funding from National Institutes of Health NS075321, NS103957, NS107281, NS092865, U10NS077384, NS097437, U54NS116025, U19 NS110456, AG050263, AG-64937, NS097799, NS075527, ES029524, NS109487, R61 AT010753, (NCATS, NINDS, NIA), RO1NS118146, R01AG065214, Department of Defense (DOD W81XWH-217-1-0393), Michael J Fox Foundation, Barnes-Jewish Hospital Foundation (Elliot Stein Family Fund and Parkinson's disease research fund), American Parkinson's disease Association (APDA) Advanced Research Center at Washington University, Greater St. Louis Chapter of the APDA, Paula and Rodger Riney Fund, Jo Oertli Fund, Huntington Disease Society of America, Murphy Fund, Fixel Foundation, and CHDI.
Funding Information:
Dr. Peterson has received grant funding from the Dystonia Coalition, which is part of the Rare Diseases Clinical Research Network, an initiative funded by the Office of Rare Diseases Research at the National Center for Advancing Translational Sciences (U54 TR001456) in collaboration with the National Institute of Neurological Disorders and Stroke (U54 NS065701) at the National Institutes of Health (NIH) and the Office of the Assistant Secretary of Defense for Health Affairs, through the Peer-Reviewed Medical Research Program (W81XWH-17-1-0393). He reports no conflicts of interest.
Funding Information:
He is also co-director for the Dystonia Coalition, which has received the majority of its support through the NIH (grants NS116025, NS065701 from the National Institutes of Neurological Disorders and Stroke TR 001456 from the Office of Rare Diseases Research at the National Center for Advancing Translational Sciences).
Funding Information:
Dr. Elble is an employee of SIU HealthCare; has served as a consultant for Applied Therapeutics, BridgeBio, Cadent, Cydan, Jazz, Neurocrine Biosciences, Novartis, Osmotica, Praxis Precision Medicines, and Sage; has served on advisory boards for the International Essential Tremor Foundation and the Neuroscience Research Foundation of Kiwanis International, Illinois-Eastern Iowa District; and has received research grants from the Neuroscience Research Foundation of Kiwanis International, Illinois-Eastern Iowa District.
Funding Information:
H. A. Jinnah has active or recent grant support from the US government (National Institutes of Health), private philanthropic organizations (Cure Dystonia Now), and industry (Revance Therapeutics, Inc.). Dr. Jinnah has also served on advisory boards or as a consultant for Addex, Allergan, CoA Therapeutics, Cavion Therapeutics, EnePharmaceuticals, Ipsen, Retrophin, Revance, and Takaha Pharmaceuticals. He has received honoraria or stipends for lectures or administrative work from the International Parkinson's Disease and Movement Disorders Society. Dr. Jinnah serves on the Scientific Advisory Boards for several private foundations including the Benign Essential Blepharospasm Research Foundation, Cure Dystonia Now, the Dystonia Medical Research Foundation, the Tourette Association of America, and Tyler's Hope for a Cure. He also is principal investigator for the Dystonia Coalition, which has received the majority of its support through the NIH (grants NS116025, NS065701 from the National Institutes of Neurological Disorders and Stroke TR 001456 from the Office of Rare Diseases Research at the National Center for Advancing Translational Sciences). The Dystonia Coalition has received additional material or administrative support from industry sponsors (Allergan Inc. and Merz Pharmaceuticals) as well as private foundations (The Benign Essential Blepharospasm Foundation, Cure Dystonia Now, The Dystonia Medical Research Foundation, and The National Spasmodic Dysphonia Association).
Funding Information:
Dr. Barbano is employed by the University of Rochester ; has received grant support from the Dystonia Coalition , Revance, Acadia, PPMI, Vaccinex; has consultancy agreements with Abvie and Oscine Corporation; receives honoraria as Associate Editor of Neurology Clinical Practice; has a contract as section editor with Visual Dx, in which he also has stock options; and, does expert witness/ medical-legal work for a number of law firms.
Funding Information:
Dr. Pirio Richardson has received royalties from Springer; research support from the National Institutes of Health (P20 GM109899; U54 NS116025), Department of Defense (W81XWH-19-CTRR-CTA) and Pharma 2B.
Funding Information:
This research was conducted under the auspices of the Dystonia Coalition, which is part of the Rare Diseases Clinical Research Network, an initiative funded by the Office of Rare Diseases Research at the National Center for Advancing Translational Sciences (U54 TR001456) in collaboration with the National Institute of Neurological Disorders and Stroke (U54 NS065701 and U54 NS116025) at the National Institute of Health (NIH). This work was also supported by the Office of the Assistant Secretary of Defense for Health Affairs, through the Peer-Reviewed Medical Research Program under Awards W81XWH-17-1-0393 and W81XWH-19-1-0146. Opinions, interpretations, conclusions, and recommendations are those of the authors and are not necessarily endorsed by the Department of Defense.Dr. Barbano is employed by the University of Rochester; has received grant support from the Dystonia Coalition, Revance, Acadia, PPMI, Vaccinex; has consultancy agreements with Abvie and Oscine Corporation; receives honoraria as Associate Editor of Neurology Clinical Practice; has a contract as section editor with Visual Dx, in which he also has stock options; and, does expert witness/ medical-legal work for a number of law firms.
Funding Information:
Grants: Medtronics, Boston Scientific, Sun Pharmaceuticals Advanced Research Company, Biohaven, Impax, Lilly, US World Meds, Sunovion Therapeutics, Neurocrine, Vaccinex, Voyager, Jazz Pharmaceuticals, CHDI Foundation, Michael J. Fox Foundation, NIH(U10 NS077366), Parkinson Foundation.
Funding Information:
Dr. Comella serves on the editorial board of Clinical Neuropharmacology and Sleep Medicine. She receives compensation/honoraria for services as a consultant or an advisory committee member: Acorda Therapeutics, Allergan, Inc.; Lundbeck Ltd.; Merz Pharmaceuticals; Acadia Pharmaceuticals; Ipsen Pharmaceuticals, Jazz Pharmaceuticals, Neurocrine Biosciences Inc., Revance Therapeutic, Sunovion., AEON Biopharma. She receives royalties from Cambridge, Wolters Kluwer. She receives research support from the Parkinson's Disease Foundation.
Publisher Copyright:
© 2022 Elsevier B.V.
PY - 2022/3/15
Y1 - 2022/3/15
N2 - Background: Head tremor (HT) is a common feature of cervical dystonia (CD), usually quantified by subjective observation. Technological developments offer alternatives for measuring HT severity that are objective and amenable to automation. Objectives: Our objectives were to develop CMOR (Computational Motor Objective Rater; a computer vision-based software system) to quantify oscillatory and directional aspects of HT from video recordings during a clinical examination and to test its convergent validity with clinical rating scales. Methods: For 93 participants with isolated CD and HT enrolled by the Dystonia Coalition, we analyzed video recordings from an examination segment in which participants were instructed to let their head drift to its most comfortable dystonic position. We evaluated peak power, frequency, and directional dominance, and used Spearman's correlation to measure the agreement between CMOR and clinical ratings. Results: Power averaged 0.90 (SD 1.80) deg2/Hz, and peak frequency 1.95 (SD 0.94) Hz. The dominant HT axis was pitch (antero/retrocollis) for 50%, roll (laterocollis) for 6%, and yaw (torticollis) for 44% of participants. One-sided t-tests showed substantial contributions from the secondary (t = 18.17, p < 0.0001) and tertiary (t = 12.89, p < 0.0001) HT axes. CMOR's HT severity measure positively correlated with the HT item on the Toronto Western Spasmodic Torticollis Rating Scale-2 (Spearman's rho = 0.54, p < 0.001). Conclusions: We demonstrate a new objective method to measure HT severity that requires only conventional video recordings, quantifies the complexities of HT in CD, and exhibits convergent validity with clinical severity ratings.
AB - Background: Head tremor (HT) is a common feature of cervical dystonia (CD), usually quantified by subjective observation. Technological developments offer alternatives for measuring HT severity that are objective and amenable to automation. Objectives: Our objectives were to develop CMOR (Computational Motor Objective Rater; a computer vision-based software system) to quantify oscillatory and directional aspects of HT from video recordings during a clinical examination and to test its convergent validity with clinical rating scales. Methods: For 93 participants with isolated CD and HT enrolled by the Dystonia Coalition, we analyzed video recordings from an examination segment in which participants were instructed to let their head drift to its most comfortable dystonic position. We evaluated peak power, frequency, and directional dominance, and used Spearman's correlation to measure the agreement between CMOR and clinical ratings. Results: Power averaged 0.90 (SD 1.80) deg2/Hz, and peak frequency 1.95 (SD 0.94) Hz. The dominant HT axis was pitch (antero/retrocollis) for 50%, roll (laterocollis) for 6%, and yaw (torticollis) for 44% of participants. One-sided t-tests showed substantial contributions from the secondary (t = 18.17, p < 0.0001) and tertiary (t = 12.89, p < 0.0001) HT axes. CMOR's HT severity measure positively correlated with the HT item on the Toronto Western Spasmodic Torticollis Rating Scale-2 (Spearman's rho = 0.54, p < 0.001). Conclusions: We demonstrate a new objective method to measure HT severity that requires only conventional video recordings, quantifies the complexities of HT in CD, and exhibits convergent validity with clinical severity ratings.
KW - Computer vision
KW - Head tremor
KW - Severity rating
KW - TWSTRS
KW - Video
UR - http://www.scopus.com/inward/record.url?scp=85123737528&partnerID=8YFLogxK
U2 - 10.1016/j.jns.2022.120154
DO - 10.1016/j.jns.2022.120154
M3 - Article
C2 - 35101766
AN - SCOPUS:85123737528
SN - 0022-510X
VL - 434
JO - Journal of the Neurological Sciences
JF - Journal of the Neurological Sciences
M1 - 120154
ER -