Movernent Disorders Vol. 12, No. 4, 1997, pp.

570-575 0 1997 Movement Disorder Society

Teaching Tape for the Motor Section of the Toronto Western Spasmodic Torticollis Scale
Cynthia L. Comella, Glenn T. Stebbins, Christopher G. Goetz, Teresa A. Chmura, Susan B. Bressman, and Anthony E. Lang

Summary: The Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) assesses the character and severity of cervical dystonia (CD). We developed a teaching tape of the TWSTRS scoring for the motor symptoms of CD. The tape provides investigators with visual representations of each component of the motor section of the TWSTRS as agreed upon by three independently scoring raters. The rate of agreement for the nondichotomous components was always significant, with a Kendalls coefficient of concordance W ranging between 0.98 and 0.76 (p < 0.01 for all measures). For the two dichotomous

components, a weighted K coefficient was also significant at 0.86 for lateral shift and 0.89 for sagittal shift (p .c. 0.01 for both measures). Scale deficiencies identified by the raters were an explicit definition of midline for assessment of range of motion, the absence of a separate scoring category assessing dystonic tremor, and the specification of duration for the effect of sensory tricks. These observations should be taken into account in future revisions of the TWSTRS and in refinements of other rating scales for CD. Key Words: Spasmodic torticollis rating scale-Teaching tape.

Introduction Several different rating scales have been proposed to evaluate cervical dystonia (CD) (1-9). The FahnMarsden dystonia scale has been validated for the assessment of generalized dystonia, but only includes one item for evaluation of CD (2). The Columbia rating scale (3,4), Tsui rating scale (5-8), and Jankovic rating scale (9) are specifically designed to assess CD and have been reported as outcome measures in treatment studies of CD. These scales, however, have not been validated. Because of the relatively limited number of CD patients, studies of CD will necessarily require multicenter collaborations and a uniform measure of CD severity. The TWSTRS is a validated CD scale that has been used in previous clinical reports, captures the clinical features of CD, and includes a videotape protocol such that all patients are viewed in a standardized fashion (1 0-13). We therefore chose the TWSTRS as a model for the development of a teaching tape.
A videotape accompanies this article. Received November 15, 1996; revision received May 2, 1996; accepted August 14, 1996. Address correspondence and reprint requests to Dr. C. L. Comella at Rush-Presbyterian-St. Lukes Medical Center, 1725 West Harrison, Chicago, IL 60612, U.S.A. Presented in part at the Parkinson Study Group Symposium, Washington DC, 1995.

The overall aim of this project was to provide a visual example of the motor severity components of the TWSTRS (see the Appendix), thereby providing a tool that would allow for the consistent application of this scale by different examiners. We also wished to identify areas of ambiguity in the scale that could be addressed in future scale development for CD. The resultant TWSTRS tape provides the following: 1. Short examples of scoring for each TWSTRS severity category as agreed upon by a panel of three movement disorder specialists with extensive experience in the assessment of CD; 2. Examples of complete patient examinations with accompanying scores from the panel that can be used by viewers to assess the agreement of their scores with those of the panel and to allow preliminary interrater testing for investigators intending to use the scale in future studies of CD.

Methods
The organizational team consisted of three members (C.L.C., G.T.S., and T.A.C.) who arranged for patient participation, videotaped the patients in a standardized way, organized meetings of the rating panel, and performed data entry and statistical analysis. The rating panel consisted of three recognized move-

570

CLINICAWSCIENTIFIC NOTES
ment disorder specialists (S.B.B., C.G.G., and A.E.L.) with extensive experience evaluating CD. The panel members had not previously met or examined any of the patients included in the videotapes. The rating panel met three times. Prior to the rating sessions, the rating panel reviewed the components of the TWSTRS motor severity section and the study objectives. The panel then rated the videotape segments of the individual subsections of the TWSTRS. Subsequently, the full patient cases were rated. All ratings were performed independently.

571

Videotape Preparation A total of 200 CD patients were videotaped by using the TWSTRS videotape protocol (11). A release for videotape publication was obtained from each patient. Segments of these tapes were selected by the organizing committee to encompass the range of severity for each of the 12 components of the TWSTRS. The exception was the duration category, in which patient examples of scares of 0, 1, and 2 (the mildest examples) were not obtained. A total of 20 videotape segments were viewed for each of 10 rating categories: rotation, laterocollis, anterocollis, retrocollis, shoulder elevation, effect of sensory tricks, shoulder elevation, duration, range of motion, and time in midline. For sagittal and lateral shift, 10 video segments were included because only presence (score 1) or absence (score 0) were scored. For the total TWSTRS scoring, 10 patients were videotaped and the entire TWSTRS videotape protocol was shown to the rating panel. All videotape segments and videotape total TWSTRS were randomized and presented to the rating panel for scoring. Videotape Segments for Individual TWSTRS Components The video segments of the individual components of the TWSTRS were selected as fOllOW5:
1. For the segments demonstrating rotation, laterocollis, anterocollis, retrocollis, lateral shift, sagittal shift, shoulder elevation, and range of motion, patients were filmed in the seated position and instructed to allow their head to move into the spontaneous posture. Patients were filmed in the frontal and the lateral views. 2. For the duration component, patients were filmed both while seated and during the performance of a battery of activating tasks: fast finger taps, counting, and walking. The duration component in the TWSTRS was designed to be evaluated over the course of an entire examination. For development of videotape, this was not possible. Therefore, the investigators all agreed to measure the duration component over a 60-s interval. For this rating, the. maximal excursion of the head during both

rest and activation and the percent time with maximal, submaximal, and absent head posturing were assessed. 3. The range-of-motion component measures the ability of patients to move the head in each of three axes: rotation, lateral tilting, and flexiodextension. During the filming for this section, patients were filmed in the frontal and the lateral views. Patients were instructed to rotate their head as far as possible along the longitudinal axis to the right and left. They were then instructed to tilt their head to the right and to the left, bringing the ear as close to the shoulder as possible on each side without elevating the shoulder. Patients were than asked to bend their head forward so that the chin touched the chest, and then backward. These maneuvers were performed without the aid of a sensory trick. 4. The videotape segments used to assess the effect of the sensory trick were obtained by asking patients to demonstrate their trick. Patients without a known sensory trick were asked to touch the side of the face, the back of the head, and the chin and attempt to move to the midline. Each sensory trick was performed twice. Although a duration of benefit from sensory tricks was not specified in the original TWSTRS, our rating panel agreed prior to the rating sessions to assess the effect of the trick over a 10-s interval. 5. The time-in-midline component of the TWSTRS measured the time that patients could maintain a frontal head posture. Seated patients were and asked to bring their head to the midline with prompting by the examiner. Patients were filmed with instruction to maintain the forward position, and the period of time up to 1 min that this forward posture could be maintained within 10" of the midline without the use of sensory tricks was assessed. This maneuver was repeated twice and the average score for each rater was entered as the score for this category. Examples from each category were selected for inclusion in the teaching tape if all of the rating investigators scored identically or if two of the three investigators on the rating panel scored identically and the third investigator scored only 1 point differently from the other two.

Videotape for Total TWSTRS Motor Examination Scores Ten patients were videotaped by using the entire TWSTRS videotape protocol. The protocol was not modified and included all instructions for determining motor severity, including positional changes. The rating panel viewed the videotape in its entirety and rated the complete TWSTRS for each patient. As described in the original publication of the scale, the total TWSTRS mo-

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C. L. COMELLA. ET AL.
7) were selected for demonstration on the TWSTRS videotape.

tor severity score was the summation of all of the components, and the duration score was weighted twice. The two complete patient videotapes with the highest concordance representing mild and moderate-severe CD were included in the teaching videotape for selfevaluation.

Discussion
The creation of a uniform clinical rating scale for the assessment of CD has been hampered by the independent development of several different rating scales (1-1 1). In contrast to Parkinsons disease, in which a single scalethe Unified Parkinsons Disease Rating Scale (UPDRS) (1 6-1 8)-has been validated and widely accepted, dystonia investigators have often devised individual rating scales or used those scales developed at their institutions. This diversity in measuring CD severity precludes the comparison or combination of data among different investigators. The aim of this study was not to promote the TWSTRS as the definitive rating scale for CD, but rather to provide a tool that could be used consistently across examiners. Our TWSTRS teaching tape provides visual examples of the different severities for each category of the TWSTRS motor severity scale as independently agreed upon by the rating panel. This panel consisted of members with recognized expertise in assessing dystonia. Each member of the panel has had extensive experience using different rating scales, including the Columbia scale (3,4), and the Fahn-Marsden scale (2). One member of the panel (A.E.L.) was one of the initial developers of the TWSTRS (10,ll). The members of the rating panel demonstrated a high degree of agreement for scoring the individual components of the TWSTRS over a wide range of severity. The TWSTRS tape is a tool that can be used for different purposes. Clinical investigators with experience in rating CD can compare their methods of evaluation with the rating panel. The tape provides specific visual examples of each component as agreed upon by investigators experienced in the evaluation of CD. Practitioners
TABLE 2. Individual rater scores and Kendall coeficient oj concordance ,for 10 complete TWSTRS motor severitv scores
Patient no. Rater 1
19 22 26 23

Data Analysis
Kendalls coefficient of concordance W was used to assess agreement among the three raters for each of the items of the TWSTRS, with the exception of sagittal and lateral shift. Agreement among raters for these two categories in which only presence and absence was assessed used a weighted K statistic (14). Agreement between rating investigators for the complete TWSTRS examination of a single patient was assessed by using the Kendall s coefficient of concordance W. For the two full TWSTRS cases presented in the teaching tape, 95% confidence intervals were calculated to allow comparisons of self-raters with the rating panel (15).

Results
A total of 220 videotape segments encompassing the sections of the TWSTRS motor section were rated, and 84% of the segments met the criteria for inclusion in the videotape. The rates of agreement for all individual components of the TWSTRS were statistically significant with Kendalls range 0.98-0.76 (all p < 0.01) and weighted K range 0.86-0.89 (all p < 0.01). The interrater agreement was highest for rotation, anterocollis, and retrocollis and lowest for lateral shift (Table 1). The interrater agreement for the total TWSTRS score for the 10 complete patient examination segments was also significant (Kendall = 0.85, p < 0.01) (Table 2). The two cases with the highest concordance (cases 2 and
TABLE 1. Interrater agreement for individual components of TWSTRS motor severity scale
TWSTRS exam item
~

W
0.98 0.84 0.95 0.94 K-0.86 K-0.89 0.77 0.76 0.90 0.81 0.79

X2 55.65 48.09 54.19 53.71

df 19

P
<0.001 <O.OOl <0.001 <0.001

Rater 2
21 21 25 22 16 17

Rater 3 18
22 24 21 13

Rotation Laterocollis Anterocollis Retrocollis Lateral shift Sagittal shift Duration Effect of sensory tricks Shoulder elevation Range of motion Time in midline

1
2 3 4 5 6 7

19
19 19

NA NA
43.89 43.10 51.38 46.08 44.91

NA NA
19 19 19 19 19

<0.01 <0.001
<0.001 <0.01 <O.OOl 10.001

18
20 17 20 22 22

18
19 17 23 21

18
19 21 25
~~

8
9 10

<0.001

TWSTRS, Toronto Western SpdsmOdic Torticollis Rating Scale,

TWSTRS, Toronto Western Spasmodic Torticollis Rating Scale. W = 0.85, xz (9) = 22.97, p < 0.01.

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CLINICAWSCIENTIFIC NOTES
who do not routinely use a CD rating scale or who are not familiar with the TWSTRS rating criteria may use it as an introduction to a validated rating scale to follow patient progress. The two complete patient examinations included at the end of the tape provide an opportunity for independent scoring and comparison with the score agreed upon by the rating panel. These full video cases may be particularly useful for groups of investigators who wish to reach a high degree of interrater reliability. We identified areas of deficiency in the scale that could be addressed in future scale development. First, the instructions for the range-of-motion component did not clearly define the midline and full range for each of the three axes of movements. Second, there was not a TWSTRS component to assess the presence and severity of dystonic head tremor. The rating panel agreed that a separate scoring category for this feature of CD would be a useful addition to future versions of the TWSTRS. Third, a duration for the effect of sensory trick was not included in the original TWSTRS. The investigators in this study agreed that a 10-s duration of effect would be a practical reflection of clinical experience. Another benefit of the development of this teaching tape was the visual representation of the different head postures seen in CD as agreed upon by the rating panel. For example, it might be difficult to distinguish between anterocollis and anterior sagittal shift or between lateral shift and laterocollis. The visual examples provided on the TWSTRS tape help to clarify these distinctions. Dystonic activation of different muscle groups that underlie the different head postures (20,21), and the recognition of these subtle differences, may be especially important when using selective chemodenervation with botulinum as a treatment. None of the patients showed occasional or intermittent CD signs, so the TWSTRS teaching tape does not include examples of duration scores 0, 1, or 2 . It may be that, in general, CD is a more continuous disorder without interspersed asymptomatic episodes, or that patients with only intermittent symptoms may not be referred to a Movement Disorders office. Similarly, in the complete TWSTRS cases presented, there is not an example of very mild CD. Previous CD rating scales used as outcome measures in medication and chemodenervation trials include the Tsui scale, the Jankovic scale, and the Fahn-Marsden dystonia scale. This project did not compare the TWSTRS with any of the other CD scales developed. The decision to use the TWSTRS for development of the teaching tape was based on three considerations: first, the TWSTRS has been previously validated (1 I); second, the availability of a specific videotape protocol with the TW-

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STRS ensured that all patients were viewed in a consistent manner, including during specific tasks that could activate CD (1 1); and third, in addition to the motor severity scale evaluated in this study, the TWSTRS also provides additional scales for subjective assessment of pain and disability secondary to CD. The overall goal of the TWSTRS tape was to provide a tool for uniform application of a CD rating scale that would promote collaborative research among diverse examiners. As a model of rating scale development and application, the UPDRS has replaced most other Parkinson disease scales and is now the standard for rating Parkinsons disease. The recent development of a teaching tape for the UPDRS highlights the utility of providing visual examples of the scale components in order to ensure uniform application (22). Similarly, the Unified Huntingtons Disease Rating Scale provides videotaped examples of the rating criteria (23,24). Future versions of the TWSTRS or other CD rating scales should address the deficiencies identified in this study and provide visual demonstration of scale applications in order to refine further our ability to rate CD consistently.

Legend to Videotape The videotape demonstrates examples of the rating categories for two sections of the motor severity scale for the TWSTRS: rotation and laterocollis. The complete teaching tape contains demonstrations for all the sections as well as the full videotape protocol of two test cases which can be used for individual rating comparisons to the scores given by the panel members. The complete teaching tape is available through the offices of WE MOVE (see Acknowledgements section for information).
Cynthia L. Comella Glenn T. Stebbins Christopher G. Goetz Teresa A. Chmura Rush-Presbyterian-St. Lukes Medical Center Chicago, Illinois, U.S.A.

Susan B. Bressman Columbia Presbyterian University New York, New York, U.S.A.
Anthony E. Lang Toronto Hospital (Western Division) Toronto, Ontario, Canada

Acknowledgment: This work has been supported by an educational grant from Athena Neuroscience. We thank Teresa A. Chmura for videotaping and editing. The complete TWSTRS teaching tape can be obtained by contacting the offices of Mt. Sinai Medical Center, 1 Gustave L. Levy Place, Box 1052, New York, NY 10029, U.S.A.; phone I-800-437-MOV2.

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C. L. COMELLA, ET AL.

Appendix: The Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) (11)
A. Maximal excursion: Rate the maximum amplitude of excursion by asking the patient not to oppose the abnormal movement; the examiner may use distracting or aggravating maneuvers. When the degree of deviation is between scores, chose the higher of the two. 1. Rotation (turn: right or left) 0 None 1 Slight (<% range) (1-22) 2 Mild (/4--% range) (2345) 3 Moderate (%-3/4 range) (46-67) 4 Severe (>Y4 range) (68-90) 2 . Laterocollis (tilt: right or left) (exclude shoulder elevation) 0 None 1 Mild (1-15) 2 Moderate (16-35) 3 Severe (>35) 3. Anterocollis/retrocollis (a or b) a. Anterocollis 0 None 1 Mild downward deviation of the chin 2 Moderate downward deviation (--% possible range) 3 Severe (the chin approximates the chest) b. Retrocollis 0 None 1 Mild backward deviation of the vertex with upward deviation of the chin 2 Moderate backward deviation (--% possible range) 3 Severe (approximates full range) 4. Lateral shift (right or left) 0 Absent 1 Present 5. Sagittal shift (forward or backward) 0 Absent I Present B. Duration factor: Provide an overall score estimated through the course of the standardized examination after estimating maximal excursion (exclusive of asking the patient to allow the head to deviate maximally). Weighted x 2 . 0 None 1 Occasional deviation (<25% of the time) most often submaximal 2 Occasional deviation (<25% of the time) often maximal, or intermittent (25-50% of the time) most often submaximal

C.

D.

E.

F.

3 Intermittent (25-50% of the time) often maximal, or frequent (50-75% of the time) most often submaximal 4 Frequent (50-75% of the time) often maximal, or constant (>75% of the time) most often submaxima1 5 Constant (>75% of the time) often maximal Eftect of sensory tricks 0 Complete relief by one of more trick 1 Partial or only limited relief by tricks 2 Little or no benefit from tricks Shoulder elevatiodanterior displacement Absent Mild (<% possible range) intermittent or constant, or moderate and intermittent Moderate (/-% possible range) and constant (>75% of the time), or severe (>2/3 possible range) and intermittent Severe and constant Range o motion (without the aid of sensory tricks): If f limitation occurs in more than one plane of motion, use the individual score that is highest. 0 Able to move to the extreme opposite position 1 Able to move the head well past the midline but not to the extreme opposite position 2 Able to move the head barely past the midline 3 Able to move the head toward but not past the midline 4 Barely able to move the head beyond an abnormal posture Time (up to 60 s) that the patient is able to maintain the head within 10of the neutral position without the use of sensory tricks (the mean of two attempts) 0 >60 s 1 46-60 s 2 3145s 3 16-30 s 4 <15 s Total severity score = sum of A to F (maximum score = 35)

References
1. Weiner WJ, Lang AE. Movement disorders: a cornpreherisive survey. Mount Kisco, NY: Futura, 1989. 2. Burke RE, Fahn S , Marsden CD, Bressman SB, Moskowitz C, Friedman J. Validity and reliability of a rating scale for the primary torsion dystonids. Neurology 1985;35:73-77. 3. Fahn S . Assessment of the primary dystonias. In: Munsat TL, ed. Quant{fication of neurologic deficit. London: Butterworth, 1989: 24 1-270. 4.Greene P, Kang U, Fahn S, Brin M, Moskowitz C, Flaster E. Double-blind, placebo-controlled trial of botulinurn toxin injections for the treatment of spasmodic torticollis. Neurology 1990; 40: 1213-1 2 18. 5 . Tsui J, Eisen A, Stoessl A, Calne S, Calne DB. Double-blind study

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of botulinum toxin in spasmodic torticollis. Lancet 1986;2245246. Tsui JKC, Fross RD, Calne S, Calne DB. Local treatment of spasmodic torticollis with botulinum toxin. Can J Neurol Sci 1987;14: 533-535. Tsui JKC, Eisen A, Mak E. Carruthers J, Scott A, Calne DB. A pilot study on the use of botulinum toxin in spasmodic torticollis. Can J Neurol Sci 1985;12:314-316. Stell R, Thompson PD, Marsden CD. Botulinum toxin in spasmodic torticollis. J Neurol Neurosurg Psychiutr?, 1988;51:920923. Jankovic J. Treatment of hyperkinetic movement disorders with tetrabenazine: a double blind crossover study. Ann Neurol 1982; 11:4147. Consky ES, Basinki A, Belle L, Ranawaya R, Lang AE. The Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS): assessment of validity and inter-rater reliability. Neurology 1990;40 (suppl 1):445. Consky ES, Lang AE. Clinical assessments of patients with cervical dystonia. In: Jankovic J, Hallett M, eds. Therapy with bofulinum toxin. New York: Marcel Dekker, 1994:211-237. Comella C, Buchman AS, Tanner CM, Brown-Toms NC, Goetz CG. Botulinum toxin injection for spasmodic torticollis: increased magnitude of benefit with electromyographic assistance. Neurology 1992;42:878-882. Dubinsky RM, Gray CS, Vetere-Overfield B, Koller WC. Electromyographic guidance of botulinum toxin treatment in cervical dystonia. CIin Neuropharmacol 1991;14:262-267. Seigel S, Castellan NJ Jr. Nonparametric statistics fo r the hehavioral sciences. 2nd ed. New York: McGraw-Hill, 1988.

575

6. 7.

8.
9. 10.

11. 12.

13. 14.

15. Hays WL. Sfatistics. 3rd ed. New York: Holt, Rinehart and Winston, 1981. 16. Fahn S, Elton RL, and members of the UPDRS Developrncnt Committee. Unified Parkinsons Disease Rating Scale. In: Fahn S, Marsden CD, Calne DB, Goldstein M, eds. Recent development in Parkinsons disease; vol 2. Florham Park, NJ: Macmillan Health Care Information, 1987:153-164. 17. Richards M, Marder K, Cote L, Mayeux R. Inter rater reliability of the Unified Parkinsons Disease Rating Scale motor examinations. Mov Disord 1994;9:89-91. 18. Martinez-Martin P, Gil-Nagel A, Garcia M, et al. Unified Parkmsons Disease Rating Scale: characteristics and structure. Mov Disord 1994;9:76-83. 19. Nonnally JC. Psychometric theory. New York: McGraw-Hill, 1978. 20. Berkovitz BKB, Moxham BJ. A textbook ~f head and neck anatomy. New York: Yearbook Medical, 1988:82-138. 21. Richmond FJR, Vidal PP. The motor system: joints and muscles of the neck. In: Peterson BW, Richmond FJ, eds. Control of head movement. New York: Oxford University Press, 1988:1-21. 22. Goetz CG, Stebbins GT, Chmura TA, Fahn S, Klawans HL, Marsden CD. Teaching tape for the motor section of the Unified Parkinsons Disease Rating Scale. Mov Disord 1995; 10:263-266. 23. Como PG. Internal validity and consistency of the Unified Huntingtons Disease Rating Scale (UHDRS) [Abstact]. Neurology 1995;45(suppl 4):A254. 24. The Huntingtons Study Group. The Unified Huntingtons Disease Rating Scale. Mov Disord 1996;ll: 136-142.

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