Posttraumatic Torticollis Daniel D. Walter L.
Truong, MD; Richard Dubinsky, MD; Neal Hermanowicz, MD; Olson, MD; Bruce Silverman, DO; William C. Koller, MD, PhD
\s=b\ We report six cases of torticollis precipitated by neck trauma. The dystonia began 1 to 4 days after the trauma and differed clinically from idiopathic torticollis by marked limitation of range of motion, lack of improvement after sleep ("honeymoon period"), and absence of geste antagonistique. Worsening with action was not present; nor was there improvement with support as seen with idiopathic torticollis. Onset of pain immediately after
the trauma and marked spasms of the paracervical muscles were other predominant features. Anticholinergic therapy was without benefit; however, some improvement occurred with botulinum toxin injection. It is concluded that torticollis can be caused by peripheral trauma and that it has unique clinical characteristics. (Arch Neurol. 1991;48:221-223)
rPorticollis is a focal dystonia characterized by twisting contraction of
the neck muscles, resulting in forced turning of the head. An etiologic factor can rarely be identified for most cases of focal dystonia, although many sec¬ ondary causes have been described.1 Trauma, either to the basal ganglia structures2 or peripherally to somatic regions, has been proposed as a cause of dystonia.3-4 We report cases of tor¬ ticollis that abruptly followed trauma Accepted for publication August 20, 1990. From the Departments of Neurology, University of California at Irvine (Dr Truong), University of Kansas City (Kan) (Drs Dubinsky and Koller), University of Michigan, Ann Arbor (Dr Hermanowicz, and Providence Hospital, Detroit, Mich (Dr Silverman), and the Toledo Institute of Neurology, Maumee, Ohio (Dr Olson). Reprint requests to Department of Neurology, Irvine Medical Center, University of California at Irvine, 101 City Dr S, Orange, CA 92668 (Dr Truong).
neck, and describe the resultant clinical features.
The findings of the remainder of his neu¬ rologic examination were normal. Treat¬
REPORT OF CASES
improvement of neck posture, muscle hy¬ pertrophy, and pain. Case 2.—A 47-year-old man was carrying a long 45-kg (100-lb) bar, when he lost his grip and tried to catch it with his right hand. He heard a "pop" and "crack" from
to the
Case 1.—A 39-year-old man was injured by a blow to the right side of his neck with a steel plate without injury to his head or
loss of consciousness. He continued his work despite the severe pain in his neck, shoulder, and throat. On awakening the next morning, his head turned to the right. He was thought to have a whiplash injury producing a temporary "wryneck." How¬ ever, the disorder persisted without im¬ provement. For 6 months he received diaz¬ epam, which did not provide relief. Cervical roentgenograms, including odontoid views, were normal. A myelogram showed pro¬ truding disks at the C-4-C-5 and C-5-C-6 levels, and the patient underwent laminectomy and arthrodesis. The procedure, how¬ ever, was without benefit. Subsequent trials of anticholinergic drugs and other medica¬ tions were ineffective. When examined 4 years after the acci¬ dent, the patient presented the same fea¬ tures of torticollis that had developed the day after his injury. His chin turned to the right, with his head tilted to the left and shifted anteriorly; his left shoulder was el¬ evated. Torticollis was present with or without head support; it worsened during standing and walking. The constant devia¬ tion of the patient's head did not allow movement past the midline, and he could not touch his right shoulder with his chin. There was severe pain in the region of the hypertrophie neck muscles that were in¬ volved in the turned neck posture (primari¬ ly, the right splenus capitis and the left sternocleidomastoid). The patient had not found any maneuvers or tricks to lessen the severity of the torticollis, nor was there re¬ lief when he rested his head against the wall. The neck posture returned to normal during sleep. On awakening each morning) he also reported a period of approximately 20 minutes when his torticollis was less se-
Downloaded From: http://archneur.jamanetwork.com/ by a Western University User on 06/10/2015
ment with botulinum toxin led to marked
the left side of his neck and shoulder and felt immediate pain in the upper part of his shoulder and the lateral aspect of his neck, with numbness throughout his left arm. The next day he awoke with his head pull¬ ing toward the right. The right splenius
capitis, right upper trapezius, right sternocleidomastoid, and left scalene muscles con¬
tributed prominently to the pulling of his neck. He was unable to turn more than 30° to either the right or the left. He had not found any trick maneuvers to lessen the torticollis. His condition did not improve during sleep. His only other neurologic ab¬ normality was a preexisting horizontal
diplopia. When the patient was seated, there was a tendency for his head to pull to the right, with his chin slightly elevated. Intermit¬ tently, there were tonic contractions lasting several seconds, which involved the left
platysma, the right and left sternocleidomastoid, and the left trapezius muscles. Otherwise, the findings of the rest of his neurologic examination were normal. Cer¬ vical roentgenograms, including odontoid views; a computed tomographic (CT) scan of the head; and a cervical myelogram were normal. The patient was started on a course of trihexyphenidyl hydrochloride, without improvement. He declined further treat¬ ment.
Case 3.—A 27-year-old woman fell, strik¬ ing her head and neck against the open door
of a dishwasher. There was no loss of con¬ sciousness. The patient experienced imme¬ diate pain and muscle spasm. Her neck started to pull to the right within 2 days of the accident. Cervical roentgenograms and a CT scan of the patient's head and neck were normal. On examination 10 months
Clinical Characteristics of Posttraumatic Torticollis* Anti¬
Onset of
Patient
No. /Age,
y/Sex
Family History
1/39/M
No
Onset
Torticollis,
of Pain Immediate
d 1
With
of
ActionInduced
Sleep
Motion
Worsening
Rest
Yes
En bloc
Yes
No
Improve
"Honeymoon Effect" Yes
Range
Improve
MRI,
cholin¬
With
CT, and Myelography
Effect
RSO
No
No
No
No No
No
No
No
No
Herniated disk at
ergic
C-4-C-5, condition
persisted postoperatively 2/47/M
3/27/F 4/19/M 5/28/F 6/48/F
No
Immediate
No
Immediate
No No
En bloc En bloc
Yes No
No No
Normal Normal Normal Normal
No
Immediate
No
Yes
En bloc
No
No
No
Immediate
No
No No
En bloc
No
No
No
No
En bloc
Stenosis at C-5-C-I 2 y later
MRI indicates
magnetic
resonance
imaging; CT, computed tomography; and RSD, reflex sympathetic dystrophy.
after the accident, she had pure laterocollis right, with her head tilted 40° from the midline and marked elevation of her right shoulder. She was unable to move her head out of this position by more than 10°. She turned en bloc. There was no "honey¬ moon period" in the morning. According to her mother, the condition persisted during sleep. The patient has not found any tricks that would alleviate her laterocollis. Elec¬ tromyography showed continuous cocontraction of the right sternocleidomastoid, right scalenus médius, and right trapezius muscles. No cocontraction was noted in the corresponding left-sided muscles. Treat¬ ment with trihexyphenidyl and clonazepam was ineffective. Botulinum toxin injection into the above-mentioned muscles resulted in mild improvement. Case 4.—At the age of 19 years, the patient ran into a partially opened garage door with enough force to knock himself 3 m (10 ft) backward. He "saw stars" but did not lose consciousness. He had neck pain immediately after the accident. That same day, he developed torticollis to the right, with his head tilted to the left. He did not have nausea, dizziness, or vomiting after the accident. Cervical spine roentgeno¬ grams, including odontoid views; skull roentgenograms and a pneumoencephalogram were normal. A CT scan of the head performed 35 years later was also normal. A multilevel posterior cervical rhizotomy performed in 1985 resulted in partial, tem¬ porary relief. The patient has not had any benefit from anticholinergic medications or clonazepam. Examination 38 years after the accident revealed torticollis with tor¬ sion to the right at 30°, tilting of the head to the left at 35 degrees, and moderately se¬ vere left shoulder elevation. Deviation oc¬ curred constantly. There were no trick movements that would alleviate the tor¬ ticollis. His range of motion was limited to within 15° of the primary head position. He denied any honeymoon effect in the morn¬ ing. He turned his body en bloc, with little mobility of the neck. According to his wife, his neck improved during sleep. Electro¬ myographic studies revealed continuous cocontraction of the left sternocleidomastoid, left scalenus médius, left levator scapulae, left trapezius, left deep retrocollis, and bito the
lateral splenus capiti muscles. Botulin in¬ jection into the above-mentioned muscles resulted in moderate improvement. Case 5.—A 28-year-old woman was in¬
volved in two car accidents. In the first ac¬ cident, her car was hit from the back as she was about to turn left. She heard a "snap" in her neck. She had the feeling of a stiff neck. At the emergency department, noth¬ ing was found in the cervical roentgeno¬ grams, and the patient was treated with cyclobenzaprine hydrochloride as well as a cervical collar. On the way home, her car struck the back of a van that pulled in front of her. She was seen again in the emergency department, and a second series of cervical roentgenograms did not demonstrate any abnormality. Because of her medication, the patient was drowsy for the first 3 days. On the fourth day, she noticed some pulling in the left side of her neck, and the previous stiffness increased. When examined by us, her neck was pulled fully to the left. She was unable to move her neck from this position. She would turn her body en bloc when she needed to turn to either side. She was not aware of any trick that would straighten her neck. She denied any honeymoon period in the morning. She also had pain in her left shoulder. Holding her head in a lateral po¬ sition decreased her pain. Her laterocollis persisted during sleep. There was marked cocontraction of the left sternocleidomas¬ toid and left scalenus médius muscles. Cer¬ vical spine roentgenograms, including odontoid views and those obtained during extension and flexion, odontoid view and CT scans of the spine did not reveal any abnor¬ mality. Botulinum toxin injection into the left sternocleidomastoid and scalenus mé¬ dius muscles resulted in moderate improve¬ ment. Case 6.—A
48-year-old woman developed
torticollis after an accident in which she hit the top of her head against the roof of her car. Pain in her neck developed 1 day after the accident. This area became increasingly painful, and the patient had increasing dif¬ ficulty in turning her neck from side to side. She did not have any tricks that would al¬ leviate her torticollis. A bone scan, a mag¬ netic resonance imaging scan, and a roent¬ genogram of the cervical spine obtained shortly after the accident were normal.
Downloaded From: http://archneur.jamanetwork.com/ by a Western University User on 06/10/2015
Another magnetic
resonance
performed 2 years
imaging scan
later showed a stenosis at the C-5-C-6 level. The patient developed pain in her right arm; the pain started from the middle of the back of her head and ra¬ diated to the fourth and fifth fingers. She described a feeling of tingling, numbness, and coldness in her right hand. She felt coldness in her left foot. The findings of electromyography performed a few months after the accident were normal. The patient was hospitalized twice for excessive pain. A cervical myelogram obtained 1 year after the accident showed extensive degenerative arthritis throughout the neck and lumbar spine, with mild encroachment of the sub¬ arachnoid space and flattening of the spinal cord. Cervical CT showed narrowing of the anteroposterior diameter of the spinal ca¬ nal at the C-5-C-6 level. The patient's med¬ ical history was remarkable for hypothy¬ roidism and an L-4-L-5 disk operation 10 years previously. Neurologic examination revealed weakness of flexion and extension of the lower part of the left leg and the left ankle. The right shoulder, right arm, and left leg were cold and slightly cyanotic. Deep tendon reflexes were brisk bilaterally but more pronounced in the lower part of the left leg. Toes were down going. Pinprick sensation was decreased in the lower part of the left leg. Pronator drift was observed in the right arm. The patient's head tilted forward and to the left, and her chin was pointed to the right. Range of motion, which was decreased to 30° on each side, did not improve with lying down. The condition persisted during sleep. There was mild prominence of the right splenus capitis and left sternocleidomastoid muscles. The diag¬ nosis of posttraumatic torticollis, cervical stenosis, and reflex sympathetic dystrophy was made. Anticholinergic medication had no benefit. However, treatment with botu¬ linum toxin was helpful. COMMENT
Suchowersky and Calne5 described a of torticollis following trauma to the neck. The patient involved devel¬ oped torticollis 2 weeks after the ini¬ tial event owing to atlantoaxial dislo¬ cation. None of our patients had subcase
luxation as evidenced by cervical spine roentgenograms, including odontoid views, or by cervical CT. Brin et al3 mentioned four patients with neck and shoulder injury who developed tor¬ ticollis 12 hours to 8 weeks after the injury, and suggested that dystonia
precipitated by peripheral injury may represent the clinical
response to sen¬ sory perturbation on the extrapyrami¬ dal motor pathways via axonal sprout¬
ing. Jankovic and Van der Linden6 de¬ scribed 17 patients with peripheral trauma prior to the onset of dystonia; one
of them had torticollis. The pre¬
ceding injuries occurred
1
day
to 10
months before the onset of the dysto¬ nia. Sheehy and Marsden7 noted that three cases in their series of 60 pa¬ tients had a traumatic preceding event. They did not report the time re¬ lationship between the injuries and the occurrence of torticollis. In our pa¬ tients, the torticollis began within 1 to 4 days of the traumatic events. The immediate onset of pain after the trauma and preceding the torticollis is of interest. Our cases did not reveal any pathology, despite intensive diag¬ nostic studies. All patients were healthy prior to the incidents. Three patients heard cracking in their necks at the moment of the injuries. Fur¬ thermore, in contrast to previous re¬ ports, there was no history of familial neurologic disorders in our cases. In idiopathic torticollis, there is cocontraction of synergistic muscles, such as the sternocleidomastoid and the con-
tralateral splenus capitis, resulting in twisting of the head. Patients with idiopathic torticollis are generally able to turn their heads easily in the same direction of the dystonic posture, although turning to the opposite side may be difficult. In our six cases, there was marked decrease in range of mo¬ tion, and the patients were unable to turn their heads to either side. This relative fixation of the neck maybe a diagnostic clue for trauma-induced torticollis (Table). Although not re¬ ported by Suchowersky and Calne,5 this fixation was also seen in their pa¬ tient (0. Suchowersky, MD, oral com¬ munication, November 1989). This diagnostic clue, however, has also been seen by one of us (D.D.T.) in psychogenic torticollis. Psychogenic movement disorders often betray themselves through frequent change of symptoms, inconsistency over time, or incongruency with the classical dystonia.8 In contrast, in our patients, the symptoms were unchanged and consistent over time. Another diagnos¬ tic clue is the report by four of our six patients that their torticollis persisted during sleep, in contrast to idiopathic torticollis, which often disappears. The clinical picture also differed from idiopathic torticollis in that a honey¬ moon period was not seen in our pa¬ tients. Although this is not always present, patients with idiopathic tor¬ ticollis frequently have a short episode of improvement each morning on awakening. The majority of our pa-
tients with trauma-induced torticollis did not improve with rest or support. But they also did not worsen with ac¬ tion, as is seen with idiopathic torticol¬ lis. The above-mentioned features may favor the classification of "nondystonic" torticollis in our patients. None of them responded to anticholinergics. Owing to the short follow-up of our cases (except cases 1 and 4), no state¬ ment can be made about remission. Jankovic and Van der Linden6 ana¬ lyzed 17 patients with dystonia occur¬ ring after trauma. Possible predispos¬ ing factors include exposure to neuroleptics, personal and family history of dystonia, essential tremor, prematu¬ rity, or developmental delay.6 These predisposing factors were lacking in our cases, which may imply direct ef¬ fect of the peripheral trauma. As with other acquired disorders, an aggres¬ sive search for cause should be initi¬ ated. In conclusion, the clinical picture of torticollis induced by peripheral trauma can be differentiated from its dystonic counterpart by the following features: decreased range of neck mo¬ tions; persistence during sleep and head support; symptoms unchanged with action; and lack of family history, honeymoon period, and geste antago-
nistique.
The authors want to express their appreciation Young, MD, who referred two of the pa¬ tients in this series to Dr Truong, and to Kathy Mistura, RN, and Karen Haglund for their dedi¬ cated assistance.
to Anne
References 1. Calne DB, Lang AE. Secondary dystonia. In: Fahn S, Marsden CD, Calne D, eds. Dystonia 2. New York, NY: Raven Press; 1988:9-33. 2. Brett EM, Hoare RD. Progressive hemi-dystonia due to focal basal ganglia lesion after mild head trauma. J Neurol Neurosurg Psychiatry.
1981;44:460.
3. Brin MF, Fahn S, Bressman SB, Burke RE. Dystonia precipitated by peripheral trauma. Neurology. 1986;36(suppl 1):119. 4. Schott GD. The relationship of peripheral trauma and pain to dystonia. J Neurol Neurosurg Psychiatry. 1985;48:698-701. 5. Suchowersky 0, Calne DB. Non-dystonic
Downloaded From: http://archneur.jamanetwork.com/ by a Western University User on 06/10/2015
of torticollis. Adv Neurol. 1988;50:501-508. 6. Jankovic J, Van der Linden C. Dystonia and tremor induced by peripheral trauma: predisposing factors. J Neurol Neurosurg Psychiatry. cause
1988;51:1512-1519.
Truong, MD; Richard Dubinsky, MD; Neal Hermanowicz, MD; Olson, MD; Bruce Silverman, DO; William C. Koller, MD, PhD
\s=b\ We report six cases of torticollis precipitated by neck trauma. The dystonia began 1 to 4 days after the trauma and differed clinically from idiopathic torticollis by marked limitation of range of motion, lack of improvement after sleep ("honeymoon period"), and absence of geste antagonistique. Worsening with action was not present; nor was there improvement with support as seen with idiopathic torticollis. Onset of pain immediately after
the trauma and marked spasms of the paracervical muscles were other predominant features. Anticholinergic therapy was without benefit; however, some improvement occurred with botulinum toxin injection. It is concluded that torticollis can be caused by peripheral trauma and that it has unique clinical characteristics. (Arch Neurol. 1991;48:221-223)
rPorticollis is a focal dystonia characterized by twisting contraction of
the neck muscles, resulting in forced turning of the head. An etiologic factor can rarely be identified for most cases of focal dystonia, although many sec¬ ondary causes have been described.1 Trauma, either to the basal ganglia structures2 or peripherally to somatic regions, has been proposed as a cause of dystonia.3-4 We report cases of tor¬ ticollis that abruptly followed trauma Accepted for publication August 20, 1990. From the Departments of Neurology, University of California at Irvine (Dr Truong), University of Kansas City (Kan) (Drs Dubinsky and Koller), University of Michigan, Ann Arbor (Dr Hermanowicz, and Providence Hospital, Detroit, Mich (Dr Silverman), and the Toledo Institute of Neurology, Maumee, Ohio (Dr Olson). Reprint requests to Department of Neurology, Irvine Medical Center, University of California at Irvine, 101 City Dr S, Orange, CA 92668 (Dr Truong).
neck, and describe the resultant clinical features.
The findings of the remainder of his neu¬ rologic examination were normal. Treat¬
REPORT OF CASES
improvement of neck posture, muscle hy¬ pertrophy, and pain. Case 2.—A 47-year-old man was carrying a long 45-kg (100-lb) bar, when he lost his grip and tried to catch it with his right hand. He heard a "pop" and "crack" from
to the
Case 1.—A 39-year-old man was injured by a blow to the right side of his neck with a steel plate without injury to his head or
loss of consciousness. He continued his work despite the severe pain in his neck, shoulder, and throat. On awakening the next morning, his head turned to the right. He was thought to have a whiplash injury producing a temporary "wryneck." How¬ ever, the disorder persisted without im¬ provement. For 6 months he received diaz¬ epam, which did not provide relief. Cervical roentgenograms, including odontoid views, were normal. A myelogram showed pro¬ truding disks at the C-4-C-5 and C-5-C-6 levels, and the patient underwent laminectomy and arthrodesis. The procedure, how¬ ever, was without benefit. Subsequent trials of anticholinergic drugs and other medica¬ tions were ineffective. When examined 4 years after the acci¬ dent, the patient presented the same fea¬ tures of torticollis that had developed the day after his injury. His chin turned to the right, with his head tilted to the left and shifted anteriorly; his left shoulder was el¬ evated. Torticollis was present with or without head support; it worsened during standing and walking. The constant devia¬ tion of the patient's head did not allow movement past the midline, and he could not touch his right shoulder with his chin. There was severe pain in the region of the hypertrophie neck muscles that were in¬ volved in the turned neck posture (primari¬ ly, the right splenus capitis and the left sternocleidomastoid). The patient had not found any maneuvers or tricks to lessen the severity of the torticollis, nor was there re¬ lief when he rested his head against the wall. The neck posture returned to normal during sleep. On awakening each morning) he also reported a period of approximately 20 minutes when his torticollis was less se-
Downloaded From: http://archneur.jamanetwork.com/ by a Western University User on 06/10/2015
ment with botulinum toxin led to marked
the left side of his neck and shoulder and felt immediate pain in the upper part of his shoulder and the lateral aspect of his neck, with numbness throughout his left arm. The next day he awoke with his head pull¬ ing toward the right. The right splenius
capitis, right upper trapezius, right sternocleidomastoid, and left scalene muscles con¬
tributed prominently to the pulling of his neck. He was unable to turn more than 30° to either the right or the left. He had not found any trick maneuvers to lessen the torticollis. His condition did not improve during sleep. His only other neurologic ab¬ normality was a preexisting horizontal
diplopia. When the patient was seated, there was a tendency for his head to pull to the right, with his chin slightly elevated. Intermit¬ tently, there were tonic contractions lasting several seconds, which involved the left
platysma, the right and left sternocleidomastoid, and the left trapezius muscles. Otherwise, the findings of the rest of his neurologic examination were normal. Cer¬ vical roentgenograms, including odontoid views; a computed tomographic (CT) scan of the head; and a cervical myelogram were normal. The patient was started on a course of trihexyphenidyl hydrochloride, without improvement. He declined further treat¬ ment.
Case 3.—A 27-year-old woman fell, strik¬ ing her head and neck against the open door
of a dishwasher. There was no loss of con¬ sciousness. The patient experienced imme¬ diate pain and muscle spasm. Her neck started to pull to the right within 2 days of the accident. Cervical roentgenograms and a CT scan of the patient's head and neck were normal. On examination 10 months
Clinical Characteristics of Posttraumatic Torticollis* Anti¬
Onset of
Patient
No. /Age,
y/Sex
Family History
1/39/M
No
Onset
Torticollis,
of Pain Immediate
d 1
With
of
ActionInduced
Sleep
Motion
Worsening
Rest
Yes
En bloc
Yes
No
Improve
"Honeymoon Effect" Yes
Range
Improve
MRI,
cholin¬
With
CT, and Myelography
Effect
RSO
No
No
No
No No
No
No
No
No
Herniated disk at
ergic
C-4-C-5, condition
persisted postoperatively 2/47/M
3/27/F 4/19/M 5/28/F 6/48/F
No
Immediate
No
Immediate
No No
En bloc En bloc
Yes No
No No
Normal Normal Normal Normal
No
Immediate
No
Yes
En bloc
No
No
No
Immediate
No
No No
En bloc
No
No
No
No
En bloc
Stenosis at C-5-C-I 2 y later
MRI indicates
magnetic
resonance
imaging; CT, computed tomography; and RSD, reflex sympathetic dystrophy.
after the accident, she had pure laterocollis right, with her head tilted 40° from the midline and marked elevation of her right shoulder. She was unable to move her head out of this position by more than 10°. She turned en bloc. There was no "honey¬ moon period" in the morning. According to her mother, the condition persisted during sleep. The patient has not found any tricks that would alleviate her laterocollis. Elec¬ tromyography showed continuous cocontraction of the right sternocleidomastoid, right scalenus médius, and right trapezius muscles. No cocontraction was noted in the corresponding left-sided muscles. Treat¬ ment with trihexyphenidyl and clonazepam was ineffective. Botulinum toxin injection into the above-mentioned muscles resulted in mild improvement. Case 4.—At the age of 19 years, the patient ran into a partially opened garage door with enough force to knock himself 3 m (10 ft) backward. He "saw stars" but did not lose consciousness. He had neck pain immediately after the accident. That same day, he developed torticollis to the right, with his head tilted to the left. He did not have nausea, dizziness, or vomiting after the accident. Cervical spine roentgeno¬ grams, including odontoid views; skull roentgenograms and a pneumoencephalogram were normal. A CT scan of the head performed 35 years later was also normal. A multilevel posterior cervical rhizotomy performed in 1985 resulted in partial, tem¬ porary relief. The patient has not had any benefit from anticholinergic medications or clonazepam. Examination 38 years after the accident revealed torticollis with tor¬ sion to the right at 30°, tilting of the head to the left at 35 degrees, and moderately se¬ vere left shoulder elevation. Deviation oc¬ curred constantly. There were no trick movements that would alleviate the tor¬ ticollis. His range of motion was limited to within 15° of the primary head position. He denied any honeymoon effect in the morn¬ ing. He turned his body en bloc, with little mobility of the neck. According to his wife, his neck improved during sleep. Electro¬ myographic studies revealed continuous cocontraction of the left sternocleidomastoid, left scalenus médius, left levator scapulae, left trapezius, left deep retrocollis, and bito the
lateral splenus capiti muscles. Botulin in¬ jection into the above-mentioned muscles resulted in moderate improvement. Case 5.—A 28-year-old woman was in¬
volved in two car accidents. In the first ac¬ cident, her car was hit from the back as she was about to turn left. She heard a "snap" in her neck. She had the feeling of a stiff neck. At the emergency department, noth¬ ing was found in the cervical roentgeno¬ grams, and the patient was treated with cyclobenzaprine hydrochloride as well as a cervical collar. On the way home, her car struck the back of a van that pulled in front of her. She was seen again in the emergency department, and a second series of cervical roentgenograms did not demonstrate any abnormality. Because of her medication, the patient was drowsy for the first 3 days. On the fourth day, she noticed some pulling in the left side of her neck, and the previous stiffness increased. When examined by us, her neck was pulled fully to the left. She was unable to move her neck from this position. She would turn her body en bloc when she needed to turn to either side. She was not aware of any trick that would straighten her neck. She denied any honeymoon period in the morning. She also had pain in her left shoulder. Holding her head in a lateral po¬ sition decreased her pain. Her laterocollis persisted during sleep. There was marked cocontraction of the left sternocleidomas¬ toid and left scalenus médius muscles. Cer¬ vical spine roentgenograms, including odontoid views and those obtained during extension and flexion, odontoid view and CT scans of the spine did not reveal any abnor¬ mality. Botulinum toxin injection into the left sternocleidomastoid and scalenus mé¬ dius muscles resulted in moderate improve¬ ment. Case 6.—A
48-year-old woman developed
torticollis after an accident in which she hit the top of her head against the roof of her car. Pain in her neck developed 1 day after the accident. This area became increasingly painful, and the patient had increasing dif¬ ficulty in turning her neck from side to side. She did not have any tricks that would al¬ leviate her torticollis. A bone scan, a mag¬ netic resonance imaging scan, and a roent¬ genogram of the cervical spine obtained shortly after the accident were normal.
Downloaded From: http://archneur.jamanetwork.com/ by a Western University User on 06/10/2015
Another magnetic
resonance
performed 2 years
imaging scan
later showed a stenosis at the C-5-C-6 level. The patient developed pain in her right arm; the pain started from the middle of the back of her head and ra¬ diated to the fourth and fifth fingers. She described a feeling of tingling, numbness, and coldness in her right hand. She felt coldness in her left foot. The findings of electromyography performed a few months after the accident were normal. The patient was hospitalized twice for excessive pain. A cervical myelogram obtained 1 year after the accident showed extensive degenerative arthritis throughout the neck and lumbar spine, with mild encroachment of the sub¬ arachnoid space and flattening of the spinal cord. Cervical CT showed narrowing of the anteroposterior diameter of the spinal ca¬ nal at the C-5-C-6 level. The patient's med¬ ical history was remarkable for hypothy¬ roidism and an L-4-L-5 disk operation 10 years previously. Neurologic examination revealed weakness of flexion and extension of the lower part of the left leg and the left ankle. The right shoulder, right arm, and left leg were cold and slightly cyanotic. Deep tendon reflexes were brisk bilaterally but more pronounced in the lower part of the left leg. Toes were down going. Pinprick sensation was decreased in the lower part of the left leg. Pronator drift was observed in the right arm. The patient's head tilted forward and to the left, and her chin was pointed to the right. Range of motion, which was decreased to 30° on each side, did not improve with lying down. The condition persisted during sleep. There was mild prominence of the right splenus capitis and left sternocleidomastoid muscles. The diag¬ nosis of posttraumatic torticollis, cervical stenosis, and reflex sympathetic dystrophy was made. Anticholinergic medication had no benefit. However, treatment with botu¬ linum toxin was helpful. COMMENT
Suchowersky and Calne5 described a of torticollis following trauma to the neck. The patient involved devel¬ oped torticollis 2 weeks after the ini¬ tial event owing to atlantoaxial dislo¬ cation. None of our patients had subcase
luxation as evidenced by cervical spine roentgenograms, including odontoid views, or by cervical CT. Brin et al3 mentioned four patients with neck and shoulder injury who developed tor¬ ticollis 12 hours to 8 weeks after the injury, and suggested that dystonia
precipitated by peripheral injury may represent the clinical
response to sen¬ sory perturbation on the extrapyrami¬ dal motor pathways via axonal sprout¬
ing. Jankovic and Van der Linden6 de¬ scribed 17 patients with peripheral trauma prior to the onset of dystonia; one
of them had torticollis. The pre¬
ceding injuries occurred
1
day
to 10
months before the onset of the dysto¬ nia. Sheehy and Marsden7 noted that three cases in their series of 60 pa¬ tients had a traumatic preceding event. They did not report the time re¬ lationship between the injuries and the occurrence of torticollis. In our pa¬ tients, the torticollis began within 1 to 4 days of the traumatic events. The immediate onset of pain after the trauma and preceding the torticollis is of interest. Our cases did not reveal any pathology, despite intensive diag¬ nostic studies. All patients were healthy prior to the incidents. Three patients heard cracking in their necks at the moment of the injuries. Fur¬ thermore, in contrast to previous re¬ ports, there was no history of familial neurologic disorders in our cases. In idiopathic torticollis, there is cocontraction of synergistic muscles, such as the sternocleidomastoid and the con-
tralateral splenus capitis, resulting in twisting of the head. Patients with idiopathic torticollis are generally able to turn their heads easily in the same direction of the dystonic posture, although turning to the opposite side may be difficult. In our six cases, there was marked decrease in range of mo¬ tion, and the patients were unable to turn their heads to either side. This relative fixation of the neck maybe a diagnostic clue for trauma-induced torticollis (Table). Although not re¬ ported by Suchowersky and Calne,5 this fixation was also seen in their pa¬ tient (0. Suchowersky, MD, oral com¬ munication, November 1989). This diagnostic clue, however, has also been seen by one of us (D.D.T.) in psychogenic torticollis. Psychogenic movement disorders often betray themselves through frequent change of symptoms, inconsistency over time, or incongruency with the classical dystonia.8 In contrast, in our patients, the symptoms were unchanged and consistent over time. Another diagnos¬ tic clue is the report by four of our six patients that their torticollis persisted during sleep, in contrast to idiopathic torticollis, which often disappears. The clinical picture also differed from idiopathic torticollis in that a honey¬ moon period was not seen in our pa¬ tients. Although this is not always present, patients with idiopathic tor¬ ticollis frequently have a short episode of improvement each morning on awakening. The majority of our pa-
tients with trauma-induced torticollis did not improve with rest or support. But they also did not worsen with ac¬ tion, as is seen with idiopathic torticol¬ lis. The above-mentioned features may favor the classification of "nondystonic" torticollis in our patients. None of them responded to anticholinergics. Owing to the short follow-up of our cases (except cases 1 and 4), no state¬ ment can be made about remission. Jankovic and Van der Linden6 ana¬ lyzed 17 patients with dystonia occur¬ ring after trauma. Possible predispos¬ ing factors include exposure to neuroleptics, personal and family history of dystonia, essential tremor, prematu¬ rity, or developmental delay.6 These predisposing factors were lacking in our cases, which may imply direct ef¬ fect of the peripheral trauma. As with other acquired disorders, an aggres¬ sive search for cause should be initi¬ ated. In conclusion, the clinical picture of torticollis induced by peripheral trauma can be differentiated from its dystonic counterpart by the following features: decreased range of neck mo¬ tions; persistence during sleep and head support; symptoms unchanged with action; and lack of family history, honeymoon period, and geste antago-
nistique.
The authors want to express their appreciation Young, MD, who referred two of the pa¬ tients in this series to Dr Truong, and to Kathy Mistura, RN, and Karen Haglund for their dedi¬ cated assistance.
to Anne
References 1. Calne DB, Lang AE. Secondary dystonia. In: Fahn S, Marsden CD, Calne D, eds. Dystonia 2. New York, NY: Raven Press; 1988:9-33. 2. Brett EM, Hoare RD. Progressive hemi-dystonia due to focal basal ganglia lesion after mild head trauma. J Neurol Neurosurg Psychiatry.
1981;44:460.
3. Brin MF, Fahn S, Bressman SB, Burke RE. Dystonia precipitated by peripheral trauma. Neurology. 1986;36(suppl 1):119. 4. Schott GD. The relationship of peripheral trauma and pain to dystonia. J Neurol Neurosurg Psychiatry. 1985;48:698-701. 5. Suchowersky 0, Calne DB. Non-dystonic
Downloaded From: http://archneur.jamanetwork.com/ by a Western University User on 06/10/2015
of torticollis. Adv Neurol. 1988;50:501-508. 6. Jankovic J, Van der Linden C. Dystonia and tremor induced by peripheral trauma: predisposing factors. J Neurol Neurosurg Psychiatry. cause
1988;51:1512-1519.
