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Acute torticollis in an adolescent: case report and MRI study.

STUDY DESIGN: A case report is presented. OBJECTIVES: To describe a typical case of acute adolescent torticollis in which a disc lesion was detected with magnetic resonance imaging. SUMMARY OF BACKGROUND DATA: Acute torticollis is attributed to atlantoaxial rotary fixation of unknown etiology. The current view is that the lesion is caused by synovial fold entrapment in the C1-C2 interspace. METHODS: In a 15-year-old male adolescent, magnetic resonance imaging was performed a few hours after the onset of torticollis, and 3 weeks after resolution of symptoms.RESULTS Increased signal intensity compatible with a fluid collection was seen in the right uncovertebral region at C2-C3. This lesion was probably linked to a sudden disruption of the disc collagen fibers, and had caused excessive lateral pressure, pushing C2 toward the left. Magnetic resonance imaging at 3 weeks was unremarkable. CONCLUSIONS: The authors think that the observed disruption was a sudden and abnormal instance of a normal and, usually, very slow process of cleft formation with extension into the fibrocartilaginous core in the uncovertebral region. It is felt that this lesion may be a frequent cause of torticollis in adolescents.

Acute Disease↗

Postural laterality in torticollis and torsion dystonia.

Tests of postural rotational laterality were carried out in 63 patients suffering from torticollis (53) or torsion dystonia with torticollis (10) and in 33 normal subjects. There was a correlation between the predominant direction of postural functions and the direction of the rotatory component of torticollis. Torticollis and torsion dystonia may be due to the release of a postural rotational laterality in pre-disposed subjects and not to a presumed asymmetric lesion of the brain-stem.

Adolescent↗

Effect of prolonged neck muscle vibration on lateral head tilt in severe spasmodic torticollis.

UNLABELLED: Short term vibration of the dorsal neck muscles (10-35 s) is known to induce involuntary movements of the head in patients with spasmodic torticollis. To investigate whether neck muscle vibration might serve as a therapeutic tool when applied for a longer time interval, we compared a vibration interval of 5 seconds with a 15 minute interval in a patient with spasmodic torticollis with an extreme head tilt to the right shoulder. Head position was recorded with a two camera optoelectronic motion analyzer in six different test conditions. Vibration regularly induced a rapid change of head position that was markedly closer to a normal, upright posture. After 5 seconds of vibration, head position very quickly returned to the initial position within seconds. During the 15 minute interval, head position remained elevated. After terminating vibration in this condition, the corrected head position remained stable at first and then decreased slowly within minutes to the initial tilted position. CONCLUSIONS: (1) In this patient, muscle vibration was the specific sensory input that induced lengthening of the dystonic neck muscles. Neither haptic stimulation nor transcutaneous electrical stimulation had more than a marginal effect. (2) The marked difference in the change of head position after short and prolonged stimulation supports the hypothesis that spasmodic torticollis might result from a disturbance of the central processing of the afferent input conveying head position information-at least in those patients who are sensitive to sensory stimulation in the neck region. (3) Long term neck muscle vibration may provide a convenient non-invasive method for treating spasmodic torticollis at the central level by influencing the neural control of head on trunk position.

Female↗

Spinal cord stimulation in the treatment of spasmodic torticollis.

This report presents our observations in 63 patients undergoing chronic spinal cord stimulation for treatment of spasmodic torticollis. In this series there were 23 patients (36.5%) who demonstrated marked improvement, characterized by no evidence of torticollis, full range of motility of the head and neck and no pain. Moderate improvement was found in 20 patients (31.8%) who showed minimal residual torticollis, but had full motility and no pain. There were 5 patients (7.9%) considered mildly improved who demonstrated decrease in their torticollic position, spasms and pain, but retained some element of torticollis and/or some limitation of motility. Correlations were made demonstrating the effect on the results of age, sex, electrode array, the configuration of the applied field and the parameters of stimulation.

Adult↗

Double-blind, placebo-controlled trial of botulinum toxin injections for the treatment of spasmodic torticollis.

We enrolled 55 patients in a double-blind, placebo-controlled, parallel design study of the effectiveness of botulinum toxin (Botox) injections for the treatment of spasmodic torticollis. Patients received a standard series of injections, either placebo or Botox. We determined the sites of injection and dose per muscle by the nature of head deviation. Compared with placebo, Botox produced statistically significant improvement in the severity of torticollis, disability, pain, and degree of head turning. There were no serious side effects. During the double-blind phase, 61% of patients injected with Botox improved; 74% of patients subsequently improved during a later open phase at a higher dose of Botox. Direction of head turning, severity of torticollis, and presence or absence of jerky movements did not significantly influence the response rate. We conclude that Botox is a valuable treatment for spasmodic torticollis.

Analysis of Variance↗

Diagnosis and surgical treatment of spasmodic torticollis of 11th nerve origin.

Of 22 patients with spasmodic torticollis, 7 were treated by microsurgical decompression of the 11th nerve. In these patients, there was an intermittent horizontal torticollis characterized by aggravation of the symptoms when in a resting posture, presenting with a striking contrast to the torticollis of extrapyramidal origin that was alleviated while in the resting posture and aggravated by postural stress. A tight neurovascular contact was observed at the C1 level, occurring between the principal 11th nerve and the vertebral or posterior inferior cerebellar artery. Nerve decompression was achieved in 2 by transposing the compressing artery and in 5 by sectioning at C1 or C2 the branching root of the 11th nerve that had caused the tight cross contact by locking the nerve trunk to the dura mater. The symptoms had improved after an interval of 1 to 4 weeks. After an average follow-up of 3 years, full or satisfactory relief had been obtained in 5 and some improvement had occurred in 2 patients. Possible neural mechanisms related to torticollis of 11th nerve origin are discussed.

Accessory Nerve↗

Congenital torticollis: evaluation by fine-needle aspiration biopsy.

Fine-needle aspiration biopsy, used extensively for evaluating head and neck masses in adults, also provides an excellent minimally invasive means of evaluating infants with neck masses and torticollis. Three cases of torticollis involving infants are presented. In each case, fine-needle aspiration revealed a benign fibrous lesion, ruling out malignancy. The remaining cytologic differential diagnosis between infantile fibromatosis, fibromatosis colli, and calcifying aponeurotic fibrosis is discussed, with mention of the possible causes of and treatment for congenital torticollis. We conclude that fine-needle aspiration provides a fast and accurate diagnosis of neck masses in infants who have congenital torticollis, thereby avoiding surgical procedures in this very young age group.

Biopsy, Needle↗

Selective peripheral denervation for the treatment of intractable spasmodic torticollis: experience with 168 patients at the Mayo Clinic.

OBJECT: Selective peripheral denervation is currently the primary surgical treatment for intractable cervical dystonia. The authors assessed preoperative factors to determine which, if any, correlated with outcomes in patients with torticollis who had undergone this procedure. METHODS: The records of 168 consecutive patients who had undergone selective peripheral denervation for cervical dystonia between 1988 and 1996 at the Mayo Clinic were reviewed. There were 89 women (53%) and 79 men (47%) with a mean age of 53.4 years. Selection of muscles for denervation was based on the patient's clinical presentation and electromyography mapping results. The most common torticollis vectors were rotational in 141 patients (84%) and laterocollis in 59 (35%). Seventy patients (42%) presented with combined vectors. The technique used to remedy both conditions involved denervation of the ipsilateral posterior cervical paraspinal and splenius capitis muscles. Denervation of the sternocleidomastoid muscle was performed on the contralateral side for rotational torticollis and on the ipsilateral side for laterocollis. A rigorous physical therapy program followed surgery. At the 3-month postoperative evaluation, 125 patients (77%) of the 162 who were available for follow up had moderate to excellent improvement in their head position, and pain was moderately to markedly improved in 131 patients (81%). The long-term follow up lasted a mean of 3.4 years and was undertaken in 130 patients. The original level of moderate to excellent improvement in head position and pain was retained in at least 71 patients (70%). Outcome was not predicted by preoperative head position, severity of abnormal posture of head, symptom duration, presence of tremor or phasic dystonic movements, or failure to respond to botulinum toxin treatment. Five patients recovered from postoperative complications including one myocardial infarction, one pulmonary embolism, and three respiratory failures. Three patients suffered from persistent C-2 distribution dysesthesias and three from slight shoulder weakness; one had a wound infection, and one died of respiratory arrest. CONCLUSIONS: Selective peripheral denervation is an effective method of achieving lasting improvement of dystonia in most patients with intractable torticollis.

Adolescent↗

Magnetic resonance imaging in the evaluation of infantile torticollis.

This retrospective study assessed the use of magnetic resonance imaging (MRI) in 58 infants with infantile torticollis. Eighteen patients had nonmuscular causes of torticollis (group 1); MRI identified lesions in 16 patients. Of 40 patients with a diagnosis of congenital muscular torticollis (group 2), 28 had a normal MRI. Five patients had asymmetry of the sternocleidomastoid without noticeable signal changes. Seven patients showd evidence of fibromatosis colli. Asymmetry of the posterolateral skull consistent with plagiocephaly was common. Magnetic resonance imaging did not alter treatment of group 2. Findings of compartment syndrome of sternocleidomastoid were inconsistent. Magnetic resonance imaging is not recommended for asymptomatic patients with infantile torticollis.

Female↗

[Electromyography monitoring in the treatment of torticollis with botulinum toxin].

UNLABELLED: The new method of treatment chronic torticollis is injection into affected muscles botulinum's toxin. Some physicians in diagnostic procedure, treatment and monitoring use needle electromyography (nEMG). The aim of our study was the estimation of the value of nEMG in chronic torticollis treatment. We examined 34 patients with chronic torticollis 4 weeks after botulinum's toxin injection into affected muscles. We have been looking for denervations signs in affected muscles. We found denervations signs in 26 patients. In both muscles sterno-cleido-mastoideus and trapezius we found denervations signs in 13 patients, only in sterno-cleido-mastoideus muscles in 9 patients and in 4 patients only in trapezius muscles. 17 patients from this group improved. The overall rate of improvement in this group was 65%. In 8 patient we didn't find any denervation signs but the rate of improvement in this group was extremely high--87%. CONCLUSION: EMG examination doesn't play deciding role in the monitoring of torticollis treatment with botulinum toxin.

Adult↗

[Spasmodic torticollis and vertebral hemangioma].

INTRODUCTION: Spasmodic torticollis in young patients should give rise to a clinical suspicion that this is secondary to another primary disorder. Therefore a series of diagnostic tests should be carried out before it is labelled as idiopathic. CLINICAL CASE: The patient was a thirty year old man who had had difficulty in writing with his right hand since childhood. At the age of 20 years he was diagnosed as having writer's cramp and idiopathic spasmodic torticollis. On general physical examination no abnormalities were found. On neurological examination he had: absence of reflexes of both arms, limited but painless rotation of the neck towards the left and hypertrophy of the left trapezius muscle. Laboratory, neurophysiological and neuroimaging investigations seeking a secondary cause for the torticollis were all normal. There were no Keyser-Fleischer rings. Chest X-ray showed, dorsal scoliosis with convexity to the left. CAT and MR of the spine showed a hemangioma in the body of T1. On arteriography of the supra-aortic and vertebral trunks a hemangioma was found at T1 which received contrast material via a branch of the right thyro-bi-cervico-scapular trunk. Various treatments were tried (diazepam, Botox, Dysport, tetrabenazine, baclofen, etc.) with no improvement. A definite diagnosis of secondary torticollis could not be made since the hemangioma was supplied by a very narrow vascular pedicle, so embolization was contraindicated. CONCLUSION: Cervical spinal cord alterations may cause focal dystonia due to increased excitability of the spinal motor neurone, due to dysfunction of the disinhibitory descending reciprocal paths.

Adult↗

Recurrent torticollis secondary to Langerhans cell histiocytosis: a case report.

Torticollis is a common clinical sign encountered by pediatricians and orthopaedic surgeons in a wide spectrum of childhood conditions ranging from benign to life-threatening. We report the case of a child with recurrent torticollis caused by Langerhans Cell Histiocytosis (LCH). The patient was a 1-year-old boy with recurrent torticollis, followed by a painless swelling over the right temporal bone. The diagnosis was confirmed by an open biopsy of the calvarial lesion. As LCH is a very rare cause of torticollis it was not considered in the initial differential by the primary care physicians and the diagnosis was delayed about 4 months. The patient received chemotherapy with steroids and etoposide for 52 weeks. He showed complete regression of the sign and imaging tests at the end of treatment were normal. No relapse of symptoms occurred during a follow-up period of 2 years. The rarity of this disease as well as the site and form of presentation are emphasised to alert physicians for an early diagnostic evaluation, which is important to prevent neurological lesions and other late complications.

Adrenal Cortex Hormones↗

[Local alcoholisation treatment of spasmodic torticollis].

Idiopathic spasmodic torticollis is a type of focal dystonia. Major muscles which rotate the neck are M. sternocleidomastoideus (SCM) and M. splenius capitis (Spl) on both sides. In torticollis patients, its clinical characteristics could be understood as a vectorial summing up of tonus in bilateral SCM and Spl at rest. There is not any curative treatment for dystonia yet. A variety of medications and many types of surgical interventions have been tried without consistent or satisfactory results. In recent days local injection of botulinum-A toxin has shown to be effective in weakening focal dystonias. We used pure ethanol for local injection. Fourteen patients aged between 20-77 years (mean 48.9) were treated by alcoholisation. Disease duration ranged from 5 months to 12 years (mean 4.9 years). All had torticollis alone or had segmental dystonia containing spasmodic torticollis. Patients were recorded electromyographically using surface electrodes to make sure which neck muscles were hypertonic, and were rated before and after treatment according to the stages (0; normal-5; most severe). On the bases of these recordings the two most active muscles were selected for injection. Into the motor point of these muscles 1 ml of 1% lidocaine, and then the same dose of 99% ethanol were injected. This procedure was repeated on the mean ten times (6-14 times) every other week. The number of times of injection was decided in each case. Using the paired Student t test, there was a significant (p less than 0.01) improvement of the stage for the patients after injection, with a mean of 3.7 before treatment and 2.3 after treatment.(ABSTRACT TRUNCATED AT 250 WORDS)

Adult↗

[Spastic torticollis].

Fifty-seven cases of spastic torticollis are reported, and its etiologic factors, including stressful situations, are analyzed. Plasma serotonin measurements have added new insight into pathogenetic mechanisms of the condition. The range of "reflex" spastic torticollis has been limited on the basis of biochemical and EMG findings. Fairly high incidence of attendant extrapyramidal symptoms and their polymorphism are pointed out, and, the need for a differential approach to the treatment of spastic torticollis being emphasized, a schedule of combined treatment, incorporating eglonyl and amitriptyline, is proposed for clonic-tonic torticollis.

Amitriptyline↗

Spasmodic torticollis after closed head injury.

Spasmodic torticollis, once thought to be psychogenic, is now recognized as an extrapyramidal movement disorder. A patient with onset of classical torticollis after closed head injury and intracranial hypertension is described. The patient had no clinical or radiologic evidence of brain stem dysfunction, but brain stem auditory evoked potentials suggested pontomesencephalic conduction disturbance ipsilateral to head and neck movements. This finding accords with some previous clinical and experimental evidence that pontomesencephalic structures ipsilateral to the movements may be involved in torticollis. The observation suggests that spasmodic torticollis may develop as a sequel to trauma in head-injured patients and should be differentiated from posttraumatic seizures.

Adult↗

Familial congenital muscular torticollis: case report and review of the literature.

A family history of congenital muscular torticollis is uncommon. Only a small number of cases have been reported in the English literature. This is a report of five female children who are interrelated and have congenital muscular torticollis. Three of the children are sisters, and four are the offspring of consanguineous matings. No environmental factor could be identified that would predispose the children to congenital muscular torticollis. In this family, it is apparent that genetic factors are influencing the development of this condition. The cause of congenital muscular torticollis is unknown, but heredity may play a more important role than has previously been suspected.

Child↗

Pseudotumor of infancy and congenital muscular torticollis.

Pseudotumor of infancy presents as a discrete, firm mass in the distal sternocleidomastoid muscle in infants two to four weeks of age. Congenital muscular torticollis may develop from the resultant fibrosis in 10 to 20 percent of cases. Hip dysplasia is an associated feature of congenital muscular torticollis in approximately 10 percent of cases. Pseudotumor of infancy must be differentiated from other causes of cervical soft tissue masses. Diagnostic choices include fine-needle aspiration biopsy, cervical radiography, ultrasonography, computed tomographic scanning of the head and neck, and magnetic resonance imaging. Left untreated, congenital muscular torticollis may lead to significant craniofacial asymmetry and scoliosis. Heat, massage and passive stretching exercises are the preferred initial treatments for pseudotumor and torticollis. More than 70 percent of patients will respond to this approach. Surgery should be reserved for treatment of cases that persist past the first year of life.

Diagnosis, Differential↗

[The clinical usefulness of botulinum toxin type A for spasmodic torticollis and facial spasm].

We studied the clinical effectiveness of botulinum toxin type A for spasmodic torticollis and that for facial spasm by multicenter, non-blinded study. The freeze-dried crystalline botulinum toxin type A (AGN 191622; Allergan Inc., Irvine, CA) was injected into the hyperactive muscles and the clinical course was followed for 22 weeks. Repeated injections were done, if necessary, with an interval of 4 weeks. The toxin was highly effective in both disorders. In spasmodic torticollis, clinical severity improved in 38 (63.3%) and the global improvement was seen in 39 (65.0%) out of 60 patients. Subjective improvement was seen in 56 (93.3%). In facial spasm, 52 (92.9%) out of 56 patients improved after the treatment. Unfavorable reactions, mainly consisting of neck muscle weakness and dysphagia in torticollis and facial weakness in facial spasm, were mostly due to the excessive action of the toxin. They were usually mild and transient. No patients discontinued the trial because of side effects. Botulinum toxin injection is a very useful and safe method for the symptomatic treatment of spasmodic torticollis and facial spasm.

Adolescent↗