Elizabeth Cisneros1, Glenn T Stebbins2, Qiyu Chen3, Jeanne P Vu4, Casey N Benadof4, Zheng Zhang4, Richard L Barbano5, Susan H Fox6, Christopher G Goetz7, Joseph Jankovic8, Hyder A Jinnah9, Joel S Perlmutter10, Charles H Adler11, Stewart A Factor12, Stephen G Reich13, Ramon Rodriguez14, Lawrence L Severt15, Natividad P Stover16, Brian D Berman17, Cynthia L Comella18, David A Peterson19. 1. Institute for Neural Computation, University of California, San Diego, 9500 Gilman Dr, La Jolla, CA 92093, United States of America. Electronic address: e1cisner@ucsd.edu. 2. Department of Neurological Sciences, Rush University Medical Center, 1620 W Harrison St, Chicago, IL 60612, United States of America. Electronic address: glenn_stebbins@rush.edu. 3. Institute for Neural Computation, University of California, San Diego, 9500 Gilman Dr, La Jolla, CA 92093, United States of America. Electronic address: qic050@ucsd.edu. 4. Institute for Neural Computation, University of California, San Diego, 9500 Gilman Dr, La Jolla, CA 92093, United States of America. 5. Department of Neurology, University of Rochester, 500 Joseph C. Wilson Blvd, Rochester, NY 14627, United States of America. Electronic address: richard_barbano@urmc.rochester.edu. 6. Movement Disorder Clinic, Toronto Western Hospital, 399 Bathurst Street, Toronto, ON M5T 2S8, Canada; Medical Sciences Building, 1 King's College Cir, Toronto, ON M5S 1A8, Canada. Electronic address: susan.fox@uhnresearch.ca. 7. Department of Neurological Sciences, Rush University Medical Center, 1620 W Harrison St, Chicago, IL 60612, United States of America. Electronic address: christopher_goetz@rush.edu. 8. Department of Neurology, Baylor College of Medicine, 1 Baylor Plaza, Houston, TX 77030, United States of America. Electronic address: josephj@bcm.edu. 9. Departments of Neurology and Human Genetics, Emory University, 1365 Clifton Rd building b suite 2200, Atlanta, GA 30322, United States of America. Electronic address: hjinnah@emory.edu. 10. Department of Neurology, Washington University School of Medicine, 660 S Euclid Ave, St. Louis, MO 63110, United States of America; Departments of Radiology, Neuroscience, Physical Therapy, and Occupational Therapy, Washington University School of Medicine, 660 S Euclid Ave, St. Louis, MO 63110, United States of America. Electronic address: perlmutterjoel@wustl.edu. 11. Department of Neurology, Mayo Clinic College of Medicine, 200 1st St SW, Rochester, MN 55905, United States of America. Electronic address: cadler@mayo.edu. 12. Department of Neurology, Emory University School of Medicine, 201 Dowman Dr, Atlanta, GA 30322, United States of America. Electronic address: sfactor@emory.edu. 13. Department of Neurology, University of Maryland Medical Centre, 22 S Greene St, Baltimore, MD 21201, United States of America. Electronic address: sreich@som.umaryland.edu. 14. UF Department of Neurology, 1149 Newell Dr, Gainesville, FL 32611, United States of America. Electronic address: ramrod@ufl.edu. 15. Department of Neurology, Beth Israel Medical Center, 529 W 42nd St # 6K, New York, NY 10036, United States of America. 16. Department of Neurology, The University of Alabama, Tuscaloosa, AL 35487, United States of America. Electronic address: npstover@uab.edu. 17. Department of Neurology, Virginia Commonwealth University, 1101 East Marshall Street, PO Box 980599, Richmond, VA 23298-0599, United States of America. Electronic address: Brian.Berman@vcuhealth.org. 18. Department of Neurological Sciences, Rush University Medical Center, 1620 W Harrison St, Chicago, IL 60612, United States of America. Electronic address: cynthia_comella@rush.edu. 19. Institute for Neural Computation, University of California, San Diego, 9500 Gilman Dr, La Jolla, CA 92093, United States of America; CNL-S, Salk Institute for Biological Studies, 10010 N Torrey Pines Rd, La Jolla, CA 92037, United States of America. Electronic address: dap@salk.edu.
Abstract
OBJECTIVES: To investigate hypothesized sources of error when quantifying the effect of the sensory trick in cervical dystonia (CD) with the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS-2), test strategies to mitigate them, and provide guidance for future research on the sensory trick. METHODS: Previous analyses suggested the sensory trick (or "alleviating maneuver", AM) item be removed from the TWSTRS-2 because of its poor clinimetric properties. We hypothesized three sources of clinimetric weakness for rating the AM: 1) whether patients were given sufficient time to demonstrate their AM; 2) whether patients' CD was sufficiently severe for detecting AM efficacy; and 3) whether raters were inadvertently rating the item in reverse of scale instructions. We tested these hypotheses with video recordings and TWSTRS-2 ratings by one "site rater" and a panel of five "video raters" for each of 185 Dystonia Coalition patients with isolated CD. RESULTS: Of 185 patients, 23 (12%) were not permitted sufficient testing time to exhibit an AM, 23 (12%) had baseline CD too mild to allow confident rating of AM effect, and 1 site- and 1 video-rater each rated the AM item with a reverse scoring convention. When these confounds were eliminated in step-wise fashion, the item's clinimetric properties improved. CONCLUSIONS: The AM's efficacy can contribute to measuring CD motor severity by addressing identified sources of error during its assessment and rating. Given the AM's sensitive diagnostic and potential pathophysiologic significance, we also provide guidance on modifications to how AMs can be assessed in future CD research.
OBJECTIVES: To investigate hypothesized sources of error when quantifying the effect of the sensory trick in cervical dystonia (CD) with the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS-2), test strategies to mitigate them, and provide guidance for future research on the sensory trick. METHODS: Previous analyses suggested the sensory trick (or "alleviating maneuver", AM) item be removed from the TWSTRS-2 because of its poor clinimetric properties. We hypothesized three sources of clinimetric weakness for rating the AM: 1) whether patients were given sufficient time to demonstrate their AM; 2) whether patients' CD was sufficiently severe for detecting AM efficacy; and 3) whether raters were inadvertently rating the item in reverse of scale instructions. We tested these hypotheses with video recordings and TWSTRS-2 ratings by one "site rater" and a panel of five "video raters" for each of 185 Dystonia Coalition patients with isolated CD. RESULTS: Of 185 patients, 23 (12%) were not permitted sufficient testing time to exhibit an AM, 23 (12%) had baseline CD too mild to allow confident rating of AM effect, and 1 site- and 1 video-rater each rated the AM item with a reverse scoring convention. When these confounds were eliminated in step-wise fashion, the item's clinimetric properties improved. CONCLUSIONS: The AM's efficacy can contribute to measuring CD motor severity by addressing identified sources of error during its assessment and rating. Given the AM's sensitive diagnostic and potential pathophysiologic significance, we also provide guidance on modifications to how AMs can be assessed in future CD research.
Authors: M F Lew; B T Adornato; D D Duane; D D Dykstra; S A Factor; J M Massey; M F Brin; J Jankovic; R L Rodnitzky; C Singer; M R Swenson; D Tarsy; J J Murray; M Koller; J D Wallace Journal: Neurology Date: 1997-09 Impact factor: 9.910
Authors: Sara C LaHue; Kathleen Albers; Samuel Goldman; Raymond Y Lo; Zhuqin Gu; Amethyst Leimpeter; Robin Fross; Kathleen Comyns; Connie Marras; Annelie de Kleijn; Robin Smit; Maya Katz; Laurie J Ozelius; Susan Bressman; Rachel Saunders-Pullman; Cynthia Comella; Jeffrey Klingman; Lorene M Nelson; Stephen K Van Den Eeden; Caroline M Tanner Journal: Mov Disord Date: 2019-11-27 Impact factor: 10.338
Authors: Cynthia L Comella; Susan H Fox; Kailash P Bhatia; Joel S Perlmutter; Hyder A Jinnah; Mateusz Zurowski; William M McDonald; Laura Marsh; Ami R Rosen; Tracy Waliczek; Laura J Wright; Wendy R Galpern; Glenn T Stebbins Journal: Mov Disord Clin Pract Date: 2015-04-06
Authors: Cynthia L Comella; Joel S Perlmutter; Hyder A Jinnah; Tracy A Waliczek; Ami R Rosen; Wendy R Galpern; Charles A Adler; Richard L Barbano; Stewart A Factor; Christopher G Goetz; Joseph Jankovic; Stephen G Reich; Ramon L Rodriguez; William L Severt; Mateusz Zurowski; Susan H Fox; Glenn T Stebbins Journal: Mov Disord Date: 2016-03-12 Impact factor: 10.338
Authors: Eric S Molho; Mark Stacy; Patrick Gillard; David Charles; Charles H Adler; Joseph Jankovic; Marc Schwartz; Mitchell F Brin Journal: Mov Disord Clin Pract Date: 2015-12-16
Authors: Joseph Jankovic; Daniel Truong; Atul T Patel; Allison Brashear; Marian Evatt; Roman G Rubio; Chad K Oh; Daniel Snyder; Gill Shears; Cynthia Comella Journal: Mov Disord Clin Pract Date: 2018-04-26
Authors: Elizabeth Cisneros; Jeanne P Vu; Ha Yeon Lee; Qiyu Chen; Casey N Benadof; Zheng Zhang; Emily A Pettitt; Subhagya K Joshi; Richard L Barbano; Joseph Jankovic; Hyder A Jinnah; Joel S Perlmutter; Brian D Berman; Abhimanyu Mahajan; Christopher G Goetz; Glenn T Stebbins; Cynthia L Comella; David A Peterson Journal: Tremor Other Hyperkinet Mov (N Y) Date: 2021-06-23