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Treatment of spasmodic torticollis through spasm control and muscle reeducation: A case study
BEHAVIOR ] HERAPY 14, 178--184 (1983)
Treatment of Spasmodic Torticollis Through Spasm Control and Muscle Reeducation" A Case Study GARY E. JONES STEFAN R. MASSONG MARION F. BUCKLEY University of Southern Mississippi This case study illustrates the use of EMG feedback of muscle spasm activity in a case of spasmodic torticollis. The patient was 36-year-old female with an 8 month history of severe torticollis. A variety of medical and pharmacological interventions failed to reduce symptomatology. The patient was seen for 5 baseline and 42 training sessions. Training involved visual feedback of physiological tracings of fast-rectified and integrated EMG activity recorded over the spastic sternocleidomastoid. Training was a shaping procedure involving progressively less head support and more difficult muscle control. Counts of spasm activity and observer ratings of videotaped behavior indicate that the training produced clear improvements in head control within and across sessions. Follow-up self-report data taken at 1 year indicate continuation of improvement.
Spasmodic torticollis is a disturbance of the muscle groups of the upper back, neck, and head in which voluntary control of head movement and support is lost. Unilateral muscle spasms of the neck, in conjunction with atrophy or hypertrophy of the contralateral muscles, cause the head to deviate to one side. The result is distorted head posture, reduced mobility, and/or considerable pain. (See Martin [1981] for additional discussion of etiology and treatment.) Focusing upon the patient's own descriptions that torticollis "almost always emphasized the loss of voluntary control" (Cleeland, 1973, p. 1241), behaviorally oriented researchers have been optimistic that voluntary control of the spastic muscle groups could be regained through relearning techniques enhanced with the aid of auditory or visual feedback. Cleeland utilized EMG feedback training and aversive shock in the treatment of ten cases of torticollis. Also, Brudny (Brudny, Grynbaum, & Korein, 1974; Brudny, Korein, Grynbaum, Friedmann, Weinstein, Sachs-Frankel, & Belandres, 1976) has reported encouraging results by Requests for reprints should be sent to Gary E. Jones, Department of Psychology, Southern Station Box 9196, University of Southern Mississippi, Hattiesburg, MS 39406-9196. 17 8 o005-7894/83/0178-01845i. 00/0 Copyright 1983by Associationfor Advancementof BehaviorTherapy All rights of reproductionin any form reserved.
SPASMODIC T O R T I C O L L I S
179
teaching the patients to relax the spastic trapezius and sternocleidomastoid muscles while contracting the contralateral muscles. This case study differs from previous reports in several ways. First, extensive baseline and training data are reported. Second, this case study collected continuous on-line recordings of spasm activity (EMG). Third, physiological data are also corroborated by behavioral ratings. Fourth, this study anticipates and fosters generalization of effects through a progression of postural changes from sitting to standing. Thus, the patient was gradually trained to exert control under conditions demanding greater muscular control.
METHOD
Subject The subject (J) was a 36-year-old female. Observation revealed a woman whose head was turned to the left at an angle of approximately sixty degrees and lowered at an angle of approximately thirty degrees. Her head was constantly in motion, evidencing severe downward jerks and rolling movements. Efforts to assume a neutral head position were unsuccessful because of strong spasms of the left sternocleidomastoid muscle. The debilitating effect of this condition was quite apparent as it was necessary for her husband to assist her as she walked. She could not easily feed herself and reported considerable pain. J reported that she had awakened approximately 8 months earlier to find a stiffness in her neck. Two weeks later, J's left sternocleidomastoid began to spasm uncontrollably and the pain increased. Physical and neurological examinations were completely within normal limits with the exception of fixed head position to the left and moderate hypertrophy of the right sternocleidomastoid. A thorough clinical evaluation indicated that she was otherwise well adjusted. Repeated medical and neurological consultations had been unable to identify specific etiology or markedly ameliorate symptoms. Of several medications prescribed, only Haldol provided some relief. The second treatment involved implantation of percutaneous dorsal column stimulating electrodes (Gildenberg, 1978) which are activated at the subject's discretion. However, this procedure was ineffective. At this point, neurosurgery was recommended; either an anterior cervical rhizotomy with section of the spinal accessory nerve (e.g., Sorenson & Hamby, 1966) or a bilateral stereotactic thalamotomy (e.g., Cooper, 1964). Before agreeing to surgery, J was referred to our clinic.
Equipment Spasm activity and general muscle activity were sampled by placing 3 Beckman miniature silver-silver chloride electrodes directly over the muscle most involved in spasm activity. This was always either the sternocleidomastoid or the trapezius muscles. Muscles were palpated and electrodes were placed along the plane of the muscle and centered over
180
JONES, MASSONG, AND BUCKLEY
the area of greatest spasm involvement. This was done since replicable absolute EMG level was less important than clear recordings of spasm activity. Electrodes were secured with adhesive collars. EMG activity was preamplified with a Cyborg J-33 and then fed to a BL-900 EMG processor. The rectified and averaged signal from the BL-900 meter display was sampled and recorded on a two-channel Hewlett-Packard physiograph. EMG activity was recorded continuously during all baseline and training periods. The subject and recording equipment were located in the same room.
Design After five initial baseline sessions, treatment was carried out in a design involving three phases spanning a total of 42 treatment sessions. Phase I (16 days) involved visual feedback of EMG activity while seated, Phase II (15 days) involved identical feedback with head unsupported by the chair, and Phase III (11 days) involved feedback while standing erect (head unsupported).
Procedure During baseline, the patient sat upright in a recliner chair with her head supported by the chair. She was instructed to simply sit as quietly as possible. These sessions were 15 min in length and run on 5 separate days. Once a reasonably stable baseline was achieved, feedback procedures were begun. Individual sessions composing each treatment phase were identical and involved an initial 5 rain baseline followed by a 15 rain training interval, Treatment sessions were scheduled twice per week. J was permitted to watch the rectified, fast-integrated EMG trace being recorded on the physiograph. Although this was initially somewhat difficult due to her head movement, she preferred being able to watch her tonic as well as phasic EMG activity. During feedback training, J was initially asked to relax the spasming muscle while contracting the contralateral sternocleidomastoid. This same strategy was continued throughout all three phases.
RESULTS Treatment success was evaluated through continuous recordings of EMG and spasm activity in addition to behavioral ratings of videotaped segments made before and after treatment. Data were sampled by dividing the periods (baseline or treatment) into thirds. One minute samples of physiological activity were then quantified from the last minute of each third. Therefore, for the 15 min pretreatment baseline days and the 15 min treatment periods, data were quantified during the 5th, 10th, and 15th minutes. The 5 min daily baselines were sampled by quantifying the 1st, 3rd, and 5th minutes. Prior to data analysis, a template was constructed which enabled the determination of criterion spasm activity. EMG activity below criterion reflected either basal tension levels or involved spasm activity which did not produce noticeable head movement. EMG activity
SPASMODIC TORTICOLLIS
60~
Initial Baseline
181
Biofeedback
e--e
EMG
o-o
Baseline
Sitting: Head Supported
Sitting: Head Unsupported
Standing
Erect
3s|
.~ 25
~ 2o 15
0
. o . . . . . . . . . 1 3 5 7 9
, 11
13
, , , , . . . , . . . oV . . . . . , • o i = 15 17 1 9 21 2 3 2 5 2 7 29 31 33 35 37 Sessions
in
39
41
43
45
47
Days
FIG. 1. Baseline and treatment counts of number of seconds per minute which showed muscle spasm activity that exceeded criterion levels (produced observable head movement).
exceeding criterion was associated with clear tonic or phasic spasm activity. Each minute of sampled EMG activity was then broken into 60 l-sec blocks and spasm activity which exceeded criterion activity determined for each block. The number of blocks where activity exceeded criterion was counted for each minute and yielded an index of activity which varied from 0 (no blocks over criterion) to 60 (all blocks showing greater than criterion activity). This index was then averaged for the 3 min samples in each period (baseline or treatment) to provide a single score per period per day. Fig. 1 shows the number of l-sec blocks per minute which contained criterion spasm activity for all days and phases of treatment. The figure reveals that there were wide differences in spasm severity and frequency across days. During the five pretreatment sessions, criterion spasms occurred from 13 sec/min to 32 sec/min. Baseline activity shows spasms were evident an average of 24 sec out of each minute. Presession daily baselines are also contrasted against daily training effects in Fig. 1. Training during Phase I was carried out with the patient seated in a chair which partially supported her head. Although the headrest prevented extension of the head and neck, neck and head flexion and rotation were uninhibited. J was asked to watch the pen excursions and was given a full explanation of the relationship between pen excursion and spasm activity. Although presession baselines show considerable variability across training days (range from 0 to 28 sec/min), the reduction
182
JONES, MASSONG, AND BUCKLEY
in the incidence of criterion spasms was very rapid during training. Reductions from baseline activity were seen during training on all but one day (presession baseline = 13.6 sec/min versus 2.3 during training [range = 0 to 8.7]). Inspection of raw EMG data showed that spasm frequency reduced quickly over the first 2 or 3 rain of each training session. It should be remembered that data were sampled from the 5th, 10th, and 15th minutes of training and averaged over the three samples. Therefore, the points plotted in Fig. 1 reflect spasticity levels beginning after 4 rain of training. During Phase II, the patient was asked to sit on the front of the chair with head and thorax unsupported. Spasm rate continued to decline during feedback (training = 1.4 sec/min), and there was a clear reduction in presession spasm activity across training days, which suggests that control generalized to her behavior outside of formal feedback training sessions (average Phase II presession baseline spasm level = 5.3 sec/min). Although it was anticipated that spasticity would worsen when J was asked to control while standing, it was felt that this situation was more representative of normal behavior, and therefore, the final I I sessions were run while she stood erect, facing the physiograph. There was an initial loss of control and worsening of spasticity (average spasm rates of 14 sec/min) followed by a return to low levels at least during training periods (average = 3.6 per rain). Feedback training was terminated somewhat prematurely due to (1) J's satisfaction with improvement, (2) difficulty in transportation from a remote rural area, and (3) the primary therapist's departure. In order to objectively determine whether the patient benefited significantly in terms of her ability to control her head position, 15 doctoral level clinical students were asked to observe videotaped recordings of initial and termination interviews. Observers were asked to rate: (1) amount of head movement, (2) regularity of movement, (3) degree of "head-tilt," (4) presence of spasms, (5) how noticeable the spasms were, and (6) the level of social disruption the spasms would likely produce. All ratings were made on 7-point Likert-type scales (Table 1), and raters were blind to which interview was rated. One-way repeated measure ANOVAs showed highly significant pre-topost differences on all six ratings (p < .001). Ratings suggest that during the initial interview, head movements/spasms were very noticeable, almost continuous, and very socially debilitating. Ratings during the termination session showed some noticeable head movement and "head tilt" remained. The movements noted in the termination session were qualitatively quite different--slow "rolling" movements rather than sharp head thrusts. Follow-up evaluation was collected 1 year after termination of treatment and the patient and spouse reported that progress had continued since termination. She reportedly has few muscle spasms but does continue to show some slow movement of her head. J continues to be happy with her progress. Finally, over treatment, J spontaneously reported (1) increased head
SPASMODIC TORTICOLLIS
183
TABLE 1 RATINGS OF HEAD MOVEMENT, SPASM ACTIVITY, AND DEGREE OF DISABILITY
Behavior rating A m o u n t of h e a d m o v e m e n t Regularity of movement D e g r e e o f h e a d tilt P r e s e n c e of s p a s m s How noticeable Social inhibitions
TermiInitial nation interview interview
Scale 7 = Extreme 7 = Continuous 7 = Extreme 7 = Frequent 7 = Very 7 = Severe
F (1,14)
1 = None 1 = None
5.3 5.9
2.6 2.7
97.4** 75.7**
1 1 1 1
5.5 4.4 6.9 6.0
3.0 2.1 2.7 2.6
49.6** 19.3"* 349.6** 137.9"*
= = = =
Vertical Absent Not None
** All d i f f e r e n c e s p < .001.
motility (she was able to drive her automobile once again), (2) increased stamina (she began to work again at home), (3) improved self-esteem (she felt more comfortable in the presence of others, especially strangers), and (4) decreased pain.
DISCUSSION The positive findings of this systematic case study echo the recommendations made by Cleeland (1973) and Brudny that "whenever a patient with disorders of voluntary movement . . . fails to respond adequately to extended conventional rehabilitation procedures, a trial of sensory feedback is warranted" (Brudny et al., 1976, p. 61). Seemingly unable to benefit from traditional medical and neurological interventions, this study's subject responded quickly to EMG feedback. Feedback was provided by watching the physiograph write-out of electromyogram activity. Furthermore, the training effects appear to have generalized to nontraining behavior since presession baselines showed progressive decreases over the first three phases of training. The graded progression of training through several postures implemented in this study appeared to foster the acquisition of muscle control, facilitate generalization from one stage to the next, and increase muscle control under conditions of increased need for motor coordination. Ratings of videotaped interviews suggest that the reductions in spasm activity were associated with improvements in overt head control. Ratings of amount of head movement, regularity of movement, degree of "head tilt," spasms, noticeability of spasms, and level of social disruption were significant both clinically and statistically. The patient was not able to gain full control by a complete cessation of movement; however, her control after treatment showed very slow rolling motions but no uncontrolled spasm activity. She was able to hold her head erect with head oriented normally. Her self-report at follow-up (12 months) indicates that her condition stabilized at a much improved level. The patient reported
184
JONES,
MASSONG,
AND
BUCKLEY
s o m e head m o v e m e n t but very l o w f r e q u e n c y o f s p a s m s . She also indicated that her control had c o n t i n u e d to i m p r o v e s o m e w h a t o v e r the level attained at the end o f treatment.
REFERENCES Brudny, J., Grynbaum, B. B., & Korein, J. Spasmodic torticollis: Treatment by feedback display of the EMG. Archives of Physical and Medical Rehabilitation, 1974, 55, 403408. Brudny, J., Korein, J., Grynbaum, B. B., Friedmann, L. W., Weinstein, S., Sachs-Frankel, G., & Belandres, P.V. EMG feedback therapy: Review of treatment of 114 patients. Archives of Physical and Medical Rehabilitation, 1976, 57, 55--61. Cleeland, C.S. Behavioral techniques in the modification of spasmodic torticollis. Neurology, 1973, 23, 1241-1247. Cooper, I.S. Effect of thalamic lesions upon torticollis. New England Journal of Medicine, 1964, 270, 967. Gildenberg, P. L. Treatment of spasmodic torticollis by dorsal column stimulation. Applied Neurophysiology, 1978, 41, 113-121. Martin, P.R. Spasmodic torticollis: Investigations and treatment using EMG feedback training. Behavior Therapy, 1981, 12, 247-262. Sorenson, B. F., & Hamby, W.B. Spasmodic torticollis: Results in 71 surgically treated patients. Neurology, 1%6, 16, 867-878. RECEIVED: December 17, 1981 FINAL ACCEPTANCE:June 25, 1982 U . S , Postal Service statement of ownership, management, and circulation required by 39 U . S . C . 3685 of: BEHAVIOR THERAPY Published in 5 issues (January, March, June, September, and November) by the Association for A d v a n c e m e n t of Behavior Therapy, 420 Lexington Avenue, N e w York, N e w York 10170. Editor: Alan E, Kazdin, Ph.D., University of Pittsburgh School of Medicine, Western Psychiatric Institute and Clinic, 3811 O ' H a r a Street, Pittsburgh, P A 15213. Managing Editor: Rex Forehand, University of Georgia, Athens, G A 30601. O w n e d by the Association for Advancement of Behavior Therapy, 420 Lexington Avenue, N e w York, N e w York 10170. K n o w n bondholders, mortgagees, and other security holders owning or holding I percent or more of total amount of bonds, mortgages, or other securities: none. The purpose, function and nonprofit status of this organization and the e x e m p t status for Federal income tax p u r p o s e s have not changed during the preceding 12 months.
EXTENT AND NATURE OF CIRCULATION A. TOTAL NO. COPIES pRINTED (Net press run) B. PAID CIRCULATION | . SALES THROUGH DEALERS AND CARRIERS, STREET VENDORS AND COUNTER SALES 2, MAIL SUBSCRIPTIONS C. TOTAL PAID CIRCULATION (Sum of IOBI and 1082) D. FREE DISTRIBUTION BY MAIL, CARRIER OR OTHER MEANS SAMPLES, COMPLIMENTARY, AND OTHER FREE COPIES
AVERAGE NO. COPIES EACH ISSUE DURING PRECEDING 12 MONTHS
ACTUAL NO. COPIES OF SINGLE ISSUE PUBLISHED NEAREST TO FILING DATE
4240
4100
0
0
3119
3074
3119
3074
55
55
E. TOTAL DISTRIBUTION (Sum of C and D)
3174
3129
F. COPIES NOT DISTRIBUTED 1. OFFICE USE, LEFT OVER, UNACCOUNTED, SPOILED AFTER PRINTING
1066
971
0
0
4240
4100
2. RETURNS FROM NEWS AGENTS G. TOTAL ( Sutn orE, FI and 2--should equal net press
run shown in A)
Treatment of Spasmodic Torticollis Through Spasm Control and Muscle Reeducation" A Case Study GARY E. JONES STEFAN R. MASSONG MARION F. BUCKLEY University of Southern Mississippi This case study illustrates the use of EMG feedback of muscle spasm activity in a case of spasmodic torticollis. The patient was 36-year-old female with an 8 month history of severe torticollis. A variety of medical and pharmacological interventions failed to reduce symptomatology. The patient was seen for 5 baseline and 42 training sessions. Training involved visual feedback of physiological tracings of fast-rectified and integrated EMG activity recorded over the spastic sternocleidomastoid. Training was a shaping procedure involving progressively less head support and more difficult muscle control. Counts of spasm activity and observer ratings of videotaped behavior indicate that the training produced clear improvements in head control within and across sessions. Follow-up self-report data taken at 1 year indicate continuation of improvement.
Spasmodic torticollis is a disturbance of the muscle groups of the upper back, neck, and head in which voluntary control of head movement and support is lost. Unilateral muscle spasms of the neck, in conjunction with atrophy or hypertrophy of the contralateral muscles, cause the head to deviate to one side. The result is distorted head posture, reduced mobility, and/or considerable pain. (See Martin [1981] for additional discussion of etiology and treatment.) Focusing upon the patient's own descriptions that torticollis "almost always emphasized the loss of voluntary control" (Cleeland, 1973, p. 1241), behaviorally oriented researchers have been optimistic that voluntary control of the spastic muscle groups could be regained through relearning techniques enhanced with the aid of auditory or visual feedback. Cleeland utilized EMG feedback training and aversive shock in the treatment of ten cases of torticollis. Also, Brudny (Brudny, Grynbaum, & Korein, 1974; Brudny, Korein, Grynbaum, Friedmann, Weinstein, Sachs-Frankel, & Belandres, 1976) has reported encouraging results by Requests for reprints should be sent to Gary E. Jones, Department of Psychology, Southern Station Box 9196, University of Southern Mississippi, Hattiesburg, MS 39406-9196. 17 8 o005-7894/83/0178-01845i. 00/0 Copyright 1983by Associationfor Advancementof BehaviorTherapy All rights of reproductionin any form reserved.
SPASMODIC T O R T I C O L L I S
179
teaching the patients to relax the spastic trapezius and sternocleidomastoid muscles while contracting the contralateral muscles. This case study differs from previous reports in several ways. First, extensive baseline and training data are reported. Second, this case study collected continuous on-line recordings of spasm activity (EMG). Third, physiological data are also corroborated by behavioral ratings. Fourth, this study anticipates and fosters generalization of effects through a progression of postural changes from sitting to standing. Thus, the patient was gradually trained to exert control under conditions demanding greater muscular control.
METHOD
Subject The subject (J) was a 36-year-old female. Observation revealed a woman whose head was turned to the left at an angle of approximately sixty degrees and lowered at an angle of approximately thirty degrees. Her head was constantly in motion, evidencing severe downward jerks and rolling movements. Efforts to assume a neutral head position were unsuccessful because of strong spasms of the left sternocleidomastoid muscle. The debilitating effect of this condition was quite apparent as it was necessary for her husband to assist her as she walked. She could not easily feed herself and reported considerable pain. J reported that she had awakened approximately 8 months earlier to find a stiffness in her neck. Two weeks later, J's left sternocleidomastoid began to spasm uncontrollably and the pain increased. Physical and neurological examinations were completely within normal limits with the exception of fixed head position to the left and moderate hypertrophy of the right sternocleidomastoid. A thorough clinical evaluation indicated that she was otherwise well adjusted. Repeated medical and neurological consultations had been unable to identify specific etiology or markedly ameliorate symptoms. Of several medications prescribed, only Haldol provided some relief. The second treatment involved implantation of percutaneous dorsal column stimulating electrodes (Gildenberg, 1978) which are activated at the subject's discretion. However, this procedure was ineffective. At this point, neurosurgery was recommended; either an anterior cervical rhizotomy with section of the spinal accessory nerve (e.g., Sorenson & Hamby, 1966) or a bilateral stereotactic thalamotomy (e.g., Cooper, 1964). Before agreeing to surgery, J was referred to our clinic.
Equipment Spasm activity and general muscle activity were sampled by placing 3 Beckman miniature silver-silver chloride electrodes directly over the muscle most involved in spasm activity. This was always either the sternocleidomastoid or the trapezius muscles. Muscles were palpated and electrodes were placed along the plane of the muscle and centered over
180
JONES, MASSONG, AND BUCKLEY
the area of greatest spasm involvement. This was done since replicable absolute EMG level was less important than clear recordings of spasm activity. Electrodes were secured with adhesive collars. EMG activity was preamplified with a Cyborg J-33 and then fed to a BL-900 EMG processor. The rectified and averaged signal from the BL-900 meter display was sampled and recorded on a two-channel Hewlett-Packard physiograph. EMG activity was recorded continuously during all baseline and training periods. The subject and recording equipment were located in the same room.
Design After five initial baseline sessions, treatment was carried out in a design involving three phases spanning a total of 42 treatment sessions. Phase I (16 days) involved visual feedback of EMG activity while seated, Phase II (15 days) involved identical feedback with head unsupported by the chair, and Phase III (11 days) involved feedback while standing erect (head unsupported).
Procedure During baseline, the patient sat upright in a recliner chair with her head supported by the chair. She was instructed to simply sit as quietly as possible. These sessions were 15 min in length and run on 5 separate days. Once a reasonably stable baseline was achieved, feedback procedures were begun. Individual sessions composing each treatment phase were identical and involved an initial 5 rain baseline followed by a 15 rain training interval, Treatment sessions were scheduled twice per week. J was permitted to watch the rectified, fast-integrated EMG trace being recorded on the physiograph. Although this was initially somewhat difficult due to her head movement, she preferred being able to watch her tonic as well as phasic EMG activity. During feedback training, J was initially asked to relax the spasming muscle while contracting the contralateral sternocleidomastoid. This same strategy was continued throughout all three phases.
RESULTS Treatment success was evaluated through continuous recordings of EMG and spasm activity in addition to behavioral ratings of videotaped segments made before and after treatment. Data were sampled by dividing the periods (baseline or treatment) into thirds. One minute samples of physiological activity were then quantified from the last minute of each third. Therefore, for the 15 min pretreatment baseline days and the 15 min treatment periods, data were quantified during the 5th, 10th, and 15th minutes. The 5 min daily baselines were sampled by quantifying the 1st, 3rd, and 5th minutes. Prior to data analysis, a template was constructed which enabled the determination of criterion spasm activity. EMG activity below criterion reflected either basal tension levels or involved spasm activity which did not produce noticeable head movement. EMG activity
SPASMODIC TORTICOLLIS
60~
Initial Baseline
181
Biofeedback
e--e
EMG
o-o
Baseline
Sitting: Head Supported
Sitting: Head Unsupported
Standing
Erect
3s|
.~ 25
~ 2o 15
0
. o . . . . . . . . . 1 3 5 7 9
, 11
13
, , , , . . . , . . . oV . . . . . , • o i = 15 17 1 9 21 2 3 2 5 2 7 29 31 33 35 37 Sessions
in
39
41
43
45
47
Days
FIG. 1. Baseline and treatment counts of number of seconds per minute which showed muscle spasm activity that exceeded criterion levels (produced observable head movement).
exceeding criterion was associated with clear tonic or phasic spasm activity. Each minute of sampled EMG activity was then broken into 60 l-sec blocks and spasm activity which exceeded criterion activity determined for each block. The number of blocks where activity exceeded criterion was counted for each minute and yielded an index of activity which varied from 0 (no blocks over criterion) to 60 (all blocks showing greater than criterion activity). This index was then averaged for the 3 min samples in each period (baseline or treatment) to provide a single score per period per day. Fig. 1 shows the number of l-sec blocks per minute which contained criterion spasm activity for all days and phases of treatment. The figure reveals that there were wide differences in spasm severity and frequency across days. During the five pretreatment sessions, criterion spasms occurred from 13 sec/min to 32 sec/min. Baseline activity shows spasms were evident an average of 24 sec out of each minute. Presession daily baselines are also contrasted against daily training effects in Fig. 1. Training during Phase I was carried out with the patient seated in a chair which partially supported her head. Although the headrest prevented extension of the head and neck, neck and head flexion and rotation were uninhibited. J was asked to watch the pen excursions and was given a full explanation of the relationship between pen excursion and spasm activity. Although presession baselines show considerable variability across training days (range from 0 to 28 sec/min), the reduction
182
JONES, MASSONG, AND BUCKLEY
in the incidence of criterion spasms was very rapid during training. Reductions from baseline activity were seen during training on all but one day (presession baseline = 13.6 sec/min versus 2.3 during training [range = 0 to 8.7]). Inspection of raw EMG data showed that spasm frequency reduced quickly over the first 2 or 3 rain of each training session. It should be remembered that data were sampled from the 5th, 10th, and 15th minutes of training and averaged over the three samples. Therefore, the points plotted in Fig. 1 reflect spasticity levels beginning after 4 rain of training. During Phase II, the patient was asked to sit on the front of the chair with head and thorax unsupported. Spasm rate continued to decline during feedback (training = 1.4 sec/min), and there was a clear reduction in presession spasm activity across training days, which suggests that control generalized to her behavior outside of formal feedback training sessions (average Phase II presession baseline spasm level = 5.3 sec/min). Although it was anticipated that spasticity would worsen when J was asked to control while standing, it was felt that this situation was more representative of normal behavior, and therefore, the final I I sessions were run while she stood erect, facing the physiograph. There was an initial loss of control and worsening of spasticity (average spasm rates of 14 sec/min) followed by a return to low levels at least during training periods (average = 3.6 per rain). Feedback training was terminated somewhat prematurely due to (1) J's satisfaction with improvement, (2) difficulty in transportation from a remote rural area, and (3) the primary therapist's departure. In order to objectively determine whether the patient benefited significantly in terms of her ability to control her head position, 15 doctoral level clinical students were asked to observe videotaped recordings of initial and termination interviews. Observers were asked to rate: (1) amount of head movement, (2) regularity of movement, (3) degree of "head-tilt," (4) presence of spasms, (5) how noticeable the spasms were, and (6) the level of social disruption the spasms would likely produce. All ratings were made on 7-point Likert-type scales (Table 1), and raters were blind to which interview was rated. One-way repeated measure ANOVAs showed highly significant pre-topost differences on all six ratings (p < .001). Ratings suggest that during the initial interview, head movements/spasms were very noticeable, almost continuous, and very socially debilitating. Ratings during the termination session showed some noticeable head movement and "head tilt" remained. The movements noted in the termination session were qualitatively quite different--slow "rolling" movements rather than sharp head thrusts. Follow-up evaluation was collected 1 year after termination of treatment and the patient and spouse reported that progress had continued since termination. She reportedly has few muscle spasms but does continue to show some slow movement of her head. J continues to be happy with her progress. Finally, over treatment, J spontaneously reported (1) increased head
SPASMODIC TORTICOLLIS
183
TABLE 1 RATINGS OF HEAD MOVEMENT, SPASM ACTIVITY, AND DEGREE OF DISABILITY
Behavior rating A m o u n t of h e a d m o v e m e n t Regularity of movement D e g r e e o f h e a d tilt P r e s e n c e of s p a s m s How noticeable Social inhibitions
TermiInitial nation interview interview
Scale 7 = Extreme 7 = Continuous 7 = Extreme 7 = Frequent 7 = Very 7 = Severe
F (1,14)
1 = None 1 = None
5.3 5.9
2.6 2.7
97.4** 75.7**
1 1 1 1
5.5 4.4 6.9 6.0
3.0 2.1 2.7 2.6
49.6** 19.3"* 349.6** 137.9"*
= = = =
Vertical Absent Not None
** All d i f f e r e n c e s p < .001.
motility (she was able to drive her automobile once again), (2) increased stamina (she began to work again at home), (3) improved self-esteem (she felt more comfortable in the presence of others, especially strangers), and (4) decreased pain.
DISCUSSION The positive findings of this systematic case study echo the recommendations made by Cleeland (1973) and Brudny that "whenever a patient with disorders of voluntary movement . . . fails to respond adequately to extended conventional rehabilitation procedures, a trial of sensory feedback is warranted" (Brudny et al., 1976, p. 61). Seemingly unable to benefit from traditional medical and neurological interventions, this study's subject responded quickly to EMG feedback. Feedback was provided by watching the physiograph write-out of electromyogram activity. Furthermore, the training effects appear to have generalized to nontraining behavior since presession baselines showed progressive decreases over the first three phases of training. The graded progression of training through several postures implemented in this study appeared to foster the acquisition of muscle control, facilitate generalization from one stage to the next, and increase muscle control under conditions of increased need for motor coordination. Ratings of videotaped interviews suggest that the reductions in spasm activity were associated with improvements in overt head control. Ratings of amount of head movement, regularity of movement, degree of "head tilt," spasms, noticeability of spasms, and level of social disruption were significant both clinically and statistically. The patient was not able to gain full control by a complete cessation of movement; however, her control after treatment showed very slow rolling motions but no uncontrolled spasm activity. She was able to hold her head erect with head oriented normally. Her self-report at follow-up (12 months) indicates that her condition stabilized at a much improved level. The patient reported
184
JONES,
MASSONG,
AND
BUCKLEY
s o m e head m o v e m e n t but very l o w f r e q u e n c y o f s p a s m s . She also indicated that her control had c o n t i n u e d to i m p r o v e s o m e w h a t o v e r the level attained at the end o f treatment.
REFERENCES Brudny, J., Grynbaum, B. B., & Korein, J. Spasmodic torticollis: Treatment by feedback display of the EMG. Archives of Physical and Medical Rehabilitation, 1974, 55, 403408. Brudny, J., Korein, J., Grynbaum, B. B., Friedmann, L. W., Weinstein, S., Sachs-Frankel, G., & Belandres, P.V. EMG feedback therapy: Review of treatment of 114 patients. Archives of Physical and Medical Rehabilitation, 1976, 57, 55--61. Cleeland, C.S. Behavioral techniques in the modification of spasmodic torticollis. Neurology, 1973, 23, 1241-1247. Cooper, I.S. Effect of thalamic lesions upon torticollis. New England Journal of Medicine, 1964, 270, 967. Gildenberg, P. L. Treatment of spasmodic torticollis by dorsal column stimulation. Applied Neurophysiology, 1978, 41, 113-121. Martin, P.R. Spasmodic torticollis: Investigations and treatment using EMG feedback training. Behavior Therapy, 1981, 12, 247-262. Sorenson, B. F., & Hamby, W.B. Spasmodic torticollis: Results in 71 surgically treated patients. Neurology, 1%6, 16, 867-878. RECEIVED: December 17, 1981 FINAL ACCEPTANCE:June 25, 1982 U . S , Postal Service statement of ownership, management, and circulation required by 39 U . S . C . 3685 of: BEHAVIOR THERAPY Published in 5 issues (January, March, June, September, and November) by the Association for A d v a n c e m e n t of Behavior Therapy, 420 Lexington Avenue, N e w York, N e w York 10170. Editor: Alan E, Kazdin, Ph.D., University of Pittsburgh School of Medicine, Western Psychiatric Institute and Clinic, 3811 O ' H a r a Street, Pittsburgh, P A 15213. Managing Editor: Rex Forehand, University of Georgia, Athens, G A 30601. O w n e d by the Association for Advancement of Behavior Therapy, 420 Lexington Avenue, N e w York, N e w York 10170. K n o w n bondholders, mortgagees, and other security holders owning or holding I percent or more of total amount of bonds, mortgages, or other securities: none. The purpose, function and nonprofit status of this organization and the e x e m p t status for Federal income tax p u r p o s e s have not changed during the preceding 12 months.
EXTENT AND NATURE OF CIRCULATION A. TOTAL NO. COPIES pRINTED (Net press run) B. PAID CIRCULATION | . SALES THROUGH DEALERS AND CARRIERS, STREET VENDORS AND COUNTER SALES 2, MAIL SUBSCRIPTIONS C. TOTAL PAID CIRCULATION (Sum of IOBI and 1082) D. FREE DISTRIBUTION BY MAIL, CARRIER OR OTHER MEANS SAMPLES, COMPLIMENTARY, AND OTHER FREE COPIES
AVERAGE NO. COPIES EACH ISSUE DURING PRECEDING 12 MONTHS
ACTUAL NO. COPIES OF SINGLE ISSUE PUBLISHED NEAREST TO FILING DATE
4240
4100
0
0
3119
3074
3119
3074
55
55
E. TOTAL DISTRIBUTION (Sum of C and D)
3174
3129
F. COPIES NOT DISTRIBUTED 1. OFFICE USE, LEFT OVER, UNACCOUNTED, SPOILED AFTER PRINTING
1066
971
0
0
4240
4100
2. RETURNS FROM NEWS AGENTS G. TOTAL ( Sutn orE, FI and 2--should equal net press
run shown in A)