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PubMed1988

A recessive form of congenital contractures and torticollis associated with malignant hyperthermia.

Two families are presented, each with two affected sibs, all four of whom seem to have a newly described and specific form of congenital contractures (arthrogryposis). The affected subjects have congenital torticollis, dysmorphic, asymmetrical, myopathic facial features, and progressive scoliosis. Two sibs had cleft palate. Malignant hyperthermia has occurred in two of the patients.

Abnormalities, Multiple↗
PubMed1990

Botulinum toxin treatment in spasmodic torticollis.

Botulinum toxin A was administered to 19 patients in a double-blind placebo controlled trial. Toxin was more effective than placebo for improving both head position and pain which was measured by an objective rating scale and videofilm assessments. Following the controlled trial, treatment with botulinum toxin was continued in an open fashion. A total of 60 patients with torticollis received toxin in a total of 117 treatment periods. The mean follow up period was 8.4 months. In 39 patients with pain there was benefit in 77% of treatment periods. Some improvement in neck posture occurred in 83% of the treatment periods with a mean duration of 12 weeks. Side effects were frequent with dysphagia being the most common (28% of treatment periods). Botulinum toxin is an effective treatment for toticollis but treatment should be initiated with doses at the lower end of the range used in this study (400-600 mouse units).

Adult↗
PubMed1991

Cervico-ocular function in patients with spasmodic torticollis.

The cervico-ocular (COR) and active and passive vestibulo-ocular reflexes (VOR) were measured in seven patients with spasmodic torticollis (ST) and six normal controls. The COR was found to be weak or absent in both groups. The VOR gain was similar in the two groups but five patients had a significant asymmetry of the response. There was no evidence of abnormal cervico-vestibular interaction during active head rotation. The study suggests that the VOR asymmetry frequently found in ST cannot be explained on the basis of an abnormal cervical input.

Adult↗
PubMed1991

A double blind trial of botulinum toxin "A" in torticollis, with one year follow up.

A double blind placebo controlled crossover trial was performed of botulinum toxin "A" in 20 patients with spasmodic torticollis. There was a statistically significant benefit for those treated with toxin; 12 on toxin improved objectively, compared with four on placebo (p less than 0.04). After a follow up period of one year, 16 still seemed to benefit from repeated toxin injections. The main side effect was dysphagia, which appeared to be dose related in individual patients.

Adult↗
PubMed1998

Further studies using higher doses of botulinum toxin type F for torticollis resistant to botulinum toxin type A.

OBJECTIVE: A previous study of botulinum toxin type F (BTX-F) treatment for torticollis had shown a dose of 520 MU to be effective, but for a much shorter duration than is usual with botulinum toxin type A (BTX-A). The objective was to assess the effect of a higher dose of BTX-F. METHODS: Four of the previously treated patients, plus an additional patient, were treated with a higher dose of 780 MU BTX-F. All were secondary nonresponders to BTX-A due to neutralising antibodies. A test injection of 40 MU BTX-F was also given into the extensor digitorum brevis muscle (EDB), to examine the time course of the biological effect of the toxin electrophysiologically. Patients were followed up at two, four, eight, and 12 weeks. RESULTS: All patients reported subjective improvement lasting from seven to 11 (mean 8.6) weeks accompanied by a significant reduction in mean clinical severity scores at two weeks. Four patients had pain which was substantially reduced. The electrophysiological studies confirmed biological sensitivity to the toxin in all patients, showing a significant change beginning at two weeks and returning to baseline at 12 weeks. The time course of this effect paralleled roughly that of the clinical response. The four patients who had previously received 520 MU BTX-F reported that the response was better and longer in duration with 780 MU. Dysphagia was more common than reported with the lower dose. CONCLUSION: Better results are possible with higher doses of BTX-F but the duration of benefit is still shorter than with BTX-A, seemingly due to a shorter duration of neuromuscular junction blockade.

Adult↗
PubMed2001

Social phobia in spasmodic torticollis.

OBJECTIVES: To study the prevalence of psychiatric comorbidity assessed by the use of a structured clinical interview in a large, representative sample of patients with spasmodic torticollis (ST) and to test the hypothesis that social phobia would be highly prevalent. METHODS: In a consecutive cohort of 116 patients with ST treated with botulinum toxin overall psychiatric comorbidity was studied prospectively with the structured clinical interview (SCID) for DSM-IV axis I disorders. Physical disability and psychosocial variables were also assessed with standardised self rating questionnaires. RESULTS: 41.3% of the subjects met DSM-IV clinical criteria A-G for current social phobia as the primary psychiatric diagnosis. This figure rose to 56% including secondary and tertiary psychiatric diagnosis. There was no correlation between severity of disease (Tsui score, severity of pain, body image dissatisfaction score) and psychiatric comorbidity. The only significant predictor of psychiatric comorbidity was depressive coping behaviour (logistic regression analysis, p < 0.01; OR=10.8). Compared with a representative sample of the general adult population, in the patients with ST the prevalence of clinically relevant social phobia is 10-fold, of mood disorders 2.4-fold, and of lifetime psychiatric comorbidity 2.6-fold increased. CONCLUSIONS: A particularly high prevalence of social phobia was found in the cohort of patients with ST. The finding of a high prevalence of social phobia and depressive coping behaviour as the main predictor of psychiatric comorbidity may make a subgroup of patients with ST particularly amenable to specific psychotherapeutic interventions.

Adaptation, Psychological↗
PubMed2003

Acupuncture for acute torticollis: a pilot study.

The use of subjective end-points such as VAS pain scales in studies of acupuncture for chronic neck pain have resulted in equivocal results. This study introduces an objective parameter as the primary end-point for the assessment of acupuncture in patients with acute torticollis (stiff neck). Eighteen patients underwent a single 20-minute treatment session, with needling of two acupuncture points Hou Hsi (SI-3) and Luo Zhen (M-UE-24), on the side ipsilateral to the predominantly involved side of the neck. Measuring the angle of lateral head rotation using a simple compass and protractor, a mean improvement of 52.9% was found, more so among those presenting earlier (< 24 hours) as opposed to later (> 72 hours; p = 0.034). The use of objective parameters, as seen in acupuncture research of the gastrointestinal and respiratory tracts, should be incorporated into studies of acute and chronic neck pain. The use of sham needle points and placebo needles is problematic since both may elicit physiological responses.

Acupuncture Points↗
PubMedDate not supplied

Attachment representation and cortisol response to the adult attachment interview in idiopathic spasmodic torticollis.

OBJECTIVE: The study investigates individual differences in the mental representation of attachment and their impact on the cortisol response to psychosocial stress in idiopathic spasmodic torticollis (IST). It was hypothesized (a) that in IST insecure attachment is more prevalent than in a non-clinical control group and (b) that subjects with dismissing attachment respond with higher physiological arousal to a specific stimulus activating the attachment behavioural system than subjects with secure attachment. METHOD: 20 patients with IST and 20 healthy controls matched for age and sex underwent the Adult Attachment Interview, an hour-long, semiclinical interview on attachment experiences. During the interview salivary cortisol levels were monitored. The subjects' mental state with regard to attachment was classified using the attachment Q-sort method. Anxiety and depression were measured as potential covariates of the adrenocortical stress response. RESULTS: Compared to the non-clinical group, dismissing attachment was strongly overrepresented in IST. In IST, but not in the healthy control group, dismissing attachment correlated with an elevated cortisol response to the interview. CONCLUSION: In clinical, but not in non-clinical samples dismissing attachment may be associated with increased vulnerability to psychosocial stress. The factors contributing to this interaction are not yet fully elucidated.

Adult↗
PubMed2001

Peripheral neurotomy for torticollis: a new approach.

Most of spasmodic torticollis is classifical as cervical dystonia and the treatment of choice is chemical or surgical denervation. There are two major procedures for surgical denervation, intradural ventral rhizotomy and extradural peripheral neurotomy (Bertrand procedure). Both have advantages and disadvantages. The authors have modified these procedures to minimize the complications, with unilateral intradural ventral rhizotomy of C1 and C2, extradural denervation of the C3-C6 posterior rami, and contralateral peripheral sectioning of the branches of the spinal accessory nerve to the sternocleidomastoid muscle. 30 patients underwent this modified operation (Group A) and the results were compared with those in a matched control group of 20 patients who underwent the traditional Bertrand procedure (Group B). Only one patient in Group A showed a sensory deficit in the C2 area, while all the patients in Group B had C2 sensory disturbance. Pre- and postoperative rating scores did not differ between the two groups. The intraoperative blood loss was significantly smaller in Group A. Compared with the traditional Bertrand's operation, our procedure involves fewer complications and significantly less intraoperative blood loss.

Accessory Nerve↗
PubMed2005

Congenital muscular torticollis in children.

AIM: The aim of this study was to evaluate the data of our patients treated for congenital muscular torticollis (CMT) in the period between 1990 and 2004. Here we report our clinical experience with CMT and review the literature. PATIENTS AND METHODS: We retrospectively evaluated the data of our patients in terms of age, sex, clinical presentation, additional deformities, localization of the lesion, history of previous treatment attempts, diagnostic tests, additional abnormalities, findings at operation and surgical procedures. RESULTS: The mean age of the patients who were operated for CMT (25 females, 27 males) was 4.3 years (range: 5 months to 16 years). Most of the lesions were seen on the left aspect of the neck. There was only one case with bilateral CMT. Seventeen out of 52 patients with CMT (32%) were diagnosed in the newborn period. The most encountered complaint at application was restriction of neck motion (57%). Associated complaints such as head tilt (53%), fascial asymmetry (34%), deformity of the skull (9.6%) were seen. Sternocleidomastoid tumor accounted for only 11% of the patients' complaints. All patients in this series were treated by surgical intervention. Apart from one recurrence no other postoperative complication was observed during the follow-up period. CONCLUSIONS: Patients whose pathology does not resolve after 12 months of physical therapy or who develop facial asymmetry or plagiocephaly during the follow-up period should be operated on in order to achieve the best cosmetic result. In delayed cases additional surgery may be needed for the best cosmetic and functional result.

Child↗
PubMed1987

Observations and analysis of results in 131 cases of spasmodic torticollis after selective denervation.

In 131 patients treated exclusively by selective denervation during the past 10 years, all or almost all the abnormal movements of spasmodic torticollis were suppressed in 115 (88%) while preserving posture and mobility. This approach was also used in certain forms of adult-onset dystonia. An appreciable amount of abnormal movements remained in the other patients, either due to residual innervation or because of limitation of denervation necessary to preserve normal movements (laterocollis) or neck stability (retrocollis). A medio-lateral approach to the posterior cervical region in the sitting position using stimulation under light anaesthesia is recommended.

Humans↗
PubMed1985

Bipolar spinal cord stimulation for spasmodic torticollis.

Four years' experience with electrical stimulation of the cervical cord is reported for 18 patients suffering from spasmodic torticollis. Permanent 1,100-Hz stimulation of the C2-C4 level resulted in a marked improvement in 50% of the patients, in a satisfactory result in 27.8% and in an unsatisfactory result in 22.2%. Muscular tension and related pain were reduced within 8 days to 4 months postoperatively. A measurable improvement of head posture rendered objective by a mechanoelectrical measuring device could only be observed after a continuous stimulation over 8-12 months. The procedure is nondestructive and reversible, complications consisted only in those related to the stimulation system itself.

Adult↗
PubMed1978

Combined stereotactic and peripheral surgical approach for spasmodic torticollis.

Thalamotomy with or without pallidotomy and peripheral denervation, if necessary, was performed in 14 cases of spasmodic torticollis or other late dystonias. Of 4 bilateral procedures, 2 had a good result, in 1 there was little change and in another the patient remained with a pseudobulbar syndrome, the only complication in this group. 1 patient only required peripheral denervation with a good result. Of the 9 patients who underwent unilateral thalamotomy, with or without pallidotomy, the result was excellent in 6 and good in 2 others, but in 4 of these 8 patients peripheral denervation was also performed.

Globus Pallidus↗
PubMed1985

Nitrous oxide ameliorates spasmodic torticollis.

In a case of therapy-resistant spasmodic torticollis administration of nitrous oxide (N2O) resulted in a dramatic transient amelioration of the dystonic movements. We suggest that manipulations of the opioid system may be useful in the therapy of this condition. Furthermore, our results with N2O may provide insight into the pathogenesis of the disease.

Dopamine↗
PubMed1992

Botulinum toxin as a treatment for blepharospasm, spasmodic torticollis and hemifacial spasm.

Fifty-two patients affected by focal dystonia or hemifacial spasm were treated with repeated injections of botulinum toxin. A clinical improvement was observed in all patients with blepharospasm; clinical benefit had a mean duration of 10 weeks. Clinical results were less impressive, but also favorable in patients affected by spasmodic torticollis and by hemifacial spasm. In the latter, the incidence of drug-induced paresis was much higher than that observed in patients with blepharospasm, even though the doses of toxin injected were significantly lower.

Adult↗
PubMedDate not supplied

Spasmodic torticollis and phobic neurosis.

Psychopathology is reported to be common in patients with spasmodic torticollis (ST). We describe two ST patients with a coexisting phobic neurosis. In the first patient the phobic neurosis appeared independent of ST. In the second patient the phobia could be directly attributed to the development of essential tremor which may be an antecedent to the onset of ST. The two phobias were successfully eliminated with paradoxical intention. The elimination of the phobia had no effect on ST. Care should be taken in assuming that the presence of psychopathology is causally relevant to the development of ST.

Adult↗
PubMed1979

The theory and therapy of hyperkineses (torticollis).

The paper presents the interaction approach in psychosomatics, as developed by the author. In this context the physical proceedings, which are oversome by the categories of the structure of space, differ from the psychic ones which are not of space but of time. The psyche itself is understood as an intentional experience, wherein the affects and unconscious proceedings are essential. Language has an important function. The psychosomatic disease is understood as a presentative symbolic event and as compensation for the loss of the cognitive ability to symbolize in the field of language. The problem of symbolization is illustrated by a case of spasmodic torticollis. The results of an examination of 81 patients are given, 23 of whom were treated psychoanalytically. The object relation, the regression to the pregenital level, the ego-regression in the form of the omnipotence of motion, and the inability for developing an own self are presented. An alternative technique of treatment is pointed out. Finally, the results of an examination of 26 cases of tic, including a case of generalized tic, are described.

Adult↗