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PubMed2001

[Therapeutic outcome of spasmodic torticollis].

We investigated 117 patients with spasmodic torticollis who had visited us to seek for appropriate treatment in these 14 years. They were 71 men and 46 women, aged 44 +/- 14 (mean +/- SD) years, and suffered from this disorder during 4 +/- 5 years, maximum 26 years. Involuntary abnormal head positions, not only torticollis but also laterocollis and antero- or retrocollis, were contained in this study. Most of them were torticollis due to idiopathic focal dystonia. One or more courses of alcoholization therapy was accomplished in 82 patients who wished to be done. This therapy course consisted of about ten times totally of 99% ethanol injection to the motor point of two most hypertonic neck muscles, either side of the sternocleidomastoideus and the opposite side of the splenius in most cases, repeated every 2 or 3 weeks. One patient received as many as 98 times of this injection and resolved completely. Training to reinforce antagonistic muscles was also instructed. Twenty-one patients (26%) were resolved completely after this treatment. Fifty-four patients (66%) were ameliorated and satisfied partially, but 18 of them relapsed in 1 to 4 years after the treatment and were obliged to repeat one more course of this treatment. On the other hand, in five patients their torticollis improved under certain drug therapy alone. Sixteen patients (14%) gave up to continue the treatment within two months, and 14 patients (12%) dropped out before starting the therapy. This alcoholization therapy resulted in amelioration of torticollis in about 90% of the patients with a long effective period. Nevertheless, this alcohol injection is painful, and requires 5 to 6 months to be completed. In 2 patients who had already received many times of this injection, sudden hoarseness occurred one day immediately after the alcohol injection to the sternocleidomastoideus. This complication was presumably brought about by the unexpected infiltration of alcohol to the laryngeal area, located posterior to that muscle. They recovered in two months, but careful attention should be paid to the adverse effects. If botulinum toxin be available also in our country, we will be able to have another choice of therapy and the treatment of this disorder will become easier.

Adult↗
PubMed1991

Acquired torticollis in children.

Acquired torticollis in children is a symptom that may be due to a number of underlying causes, some of which are severe and life threatening. Musculoskeletal, ophthalmologic, infectious, neurologic, and neoplastic conditions may present early with only torticollis. Because torticollis is a symptom, the broad spectrum of possible diagnoses requires a thorough and methodical workup. A wide variety of tests may be necessary, as well as specialty consultation. No matter how common or rare the disorder, it must be considered when evaluating a child with acquired torticollis. The first step in evaluation is always a careful and complete physical examination. An outline of the disorders associated with torticollis is presented, as are illustrative case histories.

Bone Diseases↗
PubMed1980

[Surgical treatment of congenital muscular torticollis--long term follow-up study of total resection procedure (Mikulicz) and histological examination of resected sternomastoid muscles (author's transl].

Total resection procedure (Mikulicz) was performed in 27 patients under 10 years of age with typical congenital muscular torticollis. Postoperative clinical assessment was carried out in 20 of these 27 patients. Resected sternomastoid muscles were studied histologically in 14 other patients. The operative procedures and histological findings were compared in a discussion of the relative values of conservative and operative treatment. Neck contracture due to torticollis improved immediately after operation and no recidivations followed. Skeletal deformity of the face and spinal column completely subsided within two or three years after the operation. Plagiocephaly remained and seemed not to be directly related to the torticollis. There were no patients with scar formation such as disfiguration or keratosis. Subcutaneous adhesion probably of the platysma with the deeper layer was palpable in three patients, but did not affect the clinical course. Accessory nerve injury during operation occurred in one patients, causing slight weakness of the trapezius muscle. On the other hand, disappearance of the sternomastoid muscle relief was cosmetically acceptable subjectively and objectively. The resected sternomastoid muscle in 14 patients with typical congenital muscular torticollis were studied histologically in specimens sectioned longitudinally. Hematoxylin-eosin, Masson's and van Gieson's stains were used and the distribution of the fibrosis throughout the muscles was examined. Fibrous changes were detected in almost the whole length and breadth. The changes were similar in all operated patients, although the ages were different. The findings by naked eye examination were usually less than those by histological examination. This follow-up study confirmed that total resection procedure gives good clinical results of congenital muscular torticollis. Histological studies suggested the need for removal of the fibrous changes involving the whole muscular tissue, as such changes can lead to recidivations.

Child↗
PubMed1994

Botulinum A toxin treatment in spasmodic torticollis: report of 56 patients.

Botulinum A toxin injection is the most recent and effective treatment of various movement disorders especially focal dystonia. Spasmodic torticollis is one focal dystonia which responds poorly to both medication and surgery. Botulinum A toxin injection has been adopted as a treatment procedure at the Faculty of Medicine Siriraj Hospital, Mahidol University, Bangkok, Thailand since 1989 (before the American Food and Drug Administration approval) as a research protocal for Thai patients. This report is the first ever study of this treatment for Thai patients with spasmodic torticollis. Fifty six spasmodic torticollis patients who had been treated with botulinum A toxin injection at the Movement Disorder Clinic, Siriraj Hospital were analysed. Thirty six patients were male and the male to female ratio was 1.8:1. Most of the patients (76.79 per cent) were aged between 20-49 years and half of them were from Bangkok. Twelve patients (21.43 per cent) were classified as simple torticollis, 35 patients (62.5 per cent) were combined torticollis, 7 patients (12.5 per cent) were retrocollis, and 2 patients (3.57 per cent) were lateral collis. Three patients had generalised dystonia and 2 patients had segmental dystonia. Duration of suffering in each patient ranged from 1 month to 25 years with the mean duration of 3.70 (S.D. 5.09) years. Only four patients (7.14 per cent) refused botulinum A toxin injection due to their mild symptoms. The remaining 52 patients were given botulinum A toxin injection of 30-120 international units into the most overactive group of muscles which were responsible for abnormal neck posture (mainly sternocleidomastoid and splenius capitis). Eight patients (15.38 per cent) were lost to follow-up.(ABSTRACT TRUNCATED AT 250 WORDS)

Adolescent↗
PubMed1993

[Neuro-ophthalmologic and posturographic studies of patients with idiopathic torticollis].

The pathophysiology of idiopathic spasmodic torticollis is still unknown. In addition to genetic and other factors, one of the possible causes under discussion is a disturbance of the vestibular system. In order to analyse vestibular influences we investigated 40 patients with idiopathic torticollis using electronystagmography, fundus photography, measurement of the subjective visual vertical and posturography. The results of the neuroophthalmological investigations and the measurement of the visual vertical were within the normal range. 53% of the patients examined showed a slight central vestibular preponderance in the electronystagmography, whereas 38% demonstrated a slight pathological monocular torsion in the fundus photography. Posturographically, there were some indications of a slightly decreased gain in visual contribution to the postural control process. None of the results could be correlated to the extent of torticollis in the individual case. We propose that these results do not confirm the hypothesis of primary vestibular lesion in the pathogenesis of torticollis. A possible explanation of the results described could be a common basic lesion which causes the torticollis on the one hand and also affects the central vestibular system on the other hand.

Adult↗
PubMedDate not supplied

Congenital muscular torticollis: sequela of intrauterine or perinatal compartment syndrome.

The etiology of congenital muscular torticollis remains a mystery despite intensive investigation. Magnetic resonance imaging (MRI) scans of 10 infants with this condition showed signals in the sternocleidomastoid muscle similar to those observed in the forearm and leg after compartment syndrome. Cadaver dissections and injection studies defined the sternocleidomastoid muscle compartment. Injection studies and pressure measurements performed at the time of bipolar release in three patients with congenital muscular torticollis confirmed the existence of this compartment in vivo. Clinical review of 48 children with congenital muscular torticollis showed a relation between birth position and the side affected by the contracture. Because of the association of congenital muscular torticollis with other intrauterine positioning disorders, we postulate that head positioning in utero can selectively injure the sternocleidomastoid muscle, leading to development of a compartment syndrome. Congenital muscular torticollis may represent the sequela of an intrauterine or perinatal compartment syndrome.

Child, Preschool↗
PubMed1993

Cervical spine subluxation associated with congenital muscular torticollis and craniofacial asymmetry.

The relationship between craniofacial asymmetry, congenital muscular torticollis, and cervical spine subluxation was examined in a study of 30 children who presented to our Craniofacial Program from 1987 through 1990. Twenty-six of the 30 patients had craniofacial asymmetry and muscular torticollis without true suture synostosis documented by head and neck CT scans. These 26 patients had positional skull molding with consistent flattening of the contralateral occipitoparietal region and the ipsilateral fronto-orbital region relative to the side of the torticollis. Thirteen of the 26 patients also were found to have a C1-C2 subluxation. C1 was rotated forward of C2 on the side contralateral to the muscular torticollis in 12 of 13 patients. None of the patients with subluxation had neurologic deficits or required spinal stabilization. Ophthalmologic evaluations demonstrated amblyopia (4 patients) and horizontal strabismus (1 patient), both thought to be coincidental, with no evidence of nystagmus in any case. Seven of the 26 patients required surgical therapy for their neck muscle tightness, while the remainder responded to physiotherapy. Only 2 of the 26 patients underwent cranio-orbital reshaping for correction of their upper face asymmetry. Recognition of cervical subluxation in patients with congenital muscular torticollis may help to explain residual head-neck posturing problems even after successful neck muscle therapy.

Birth Injuries↗
PubMed1996

Torticollis secondary to ocular pathology.

We report a series of 15 children, six male and nine female, of average age 20 months, seen at a paediatric orthopaedic clinic with torticollis. Orthopaedic examination revealed a normal range of neck movement in all cases but in seven there was palpable tightness in the absence of true shortening or contracture of the sternomastoid muscle. The patients were prospectively referred for ocular examination. In five of the 15 an ocular cause for the torticollis was detected with underaction of the superior oblique muscle in three, paresis of the lateral rectus muscle in one and nystagmus in one. Another two patients were found to have an abnormal ocular examination which was thought to be unrelated to their torticollis. Three of the patients with ocular torticollis required extra-ocular muscle surgery to abolish the head tilt and one of these had a tight sternomastoid muscle. Two of the non-ocular group had surgical release of the sternomastoid muscle; in the rest, the condition either resolved with physiotherapy or required no active treatment. We recommend that all patients with torticollis and no clear orthopaedic cause are referred for ocular assessment since it is not possible clinically to distinguish ocular from non-ocular causes.

Adolescent↗
PubMedDate not supplied

Torticollis: a long-term follow-up study.

To achieve better guidelines for the future management of torticollis, this study analyzed surgical and nonsurgical management of 253 torticollis patients who were treated in this hospital from 1971 to 1993. Of those, 37 cases received operation only, 78 cases were operated after failed physical therapy, and 138 cases were treated only at the Rehabilitation Department. If free neck movement was considered to be the primary goal of treatment, most parents were satisfied with the results. However, if facial and skull deformities were the serious sequelae of torticollis, then only less than half of the surgical and nonsurgical groups of patients were graded as normal. Further, 10.9% of physical therapy group and 7% of the surgical patients need further operation to release the fibrotic bundle which limited their neck movement. Therefore, it is suggested that torticollis treatment should include early interventions such as adjusting sleep position, careful planning of physical therapy and/or operation and a long term follow-up period as essential for better management of torticollis.

Child↗
PubMedDate not supplied

Torticollis and hip dislocation.

Reports in the literature suggest that there is an association between two childhood disorders: torticollis, an easily recognized clinical deformity, and developmental dislocation or dysplasia of the hip, an occult disorder. The identification of the obvious disease, torticollis, may prompt a search for the occult disease, developmental dislocation of the hip. If the association of these two disorders is common, it may be justified to expend resources to diagnose the occult disorder in all cases in which the more obviously noticed disorder is recognized. The reported association varies between 2 and 29%. We retrospectively reviewed 70 patients with the diagnosis of congenital muscular torticollis to determine the incidence of hip dislocation or subluxation in these patients. Fifty-four patients had radiographs of their hips. Forty-one patients were available for follow-up at an average of age 3+4 years. Six patients were noted to have hip subluxation or dislocation, all at presentation. Of these, four had been referred for diagnosed hip disease, whereas two were referred for torticollis, and the hip disease was then diagnosed by the pediatric orthopaedist. No patient had abnormal radiographs or physical findings at follow-up. We conclude that the rate of hip disease in those with torticollis is approximately 8% and is lower than the 20% often quoted.

Child↗
PubMed1992

The sit-up test: an alternate clinical test for evaluating pediatric torticollis.

The sit-up test, a new evaluation method for differentiating between ocular and orthopaedic torticollis, was evaluated against the monocular occlusion test, using results of three-step testing for standardization. The study group consisted of 31 patients with torticollis between the ages of 4 and 12.5 years. Subjects were selected based only on their ability to cooperate with three-step testing. Three-step testing identified 27 of the 31 patients as having ocular torticollis, with the remaining 4 having an orthopaedic etiology. Sit-up testing correctly identified all 27 ocular torticollis patients, with no false positives or false negatives. Monocular occlusion testing detected at best 22 (81.4%) of the ocular torticollis patients, with no false positives.

Child↗
PubMed1991

Posttraumatic torticollis.

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.

Adult↗
PubMed1990

Inflammatory torticollis in children.

Acute torticollis is commonly seen in the pediatric emergency department. It often results from an inflammatory process that irritates the cervical muscles, nerves, or vertebrae. Posturing of the head occurs with unilateral spasm of the sternocleidomastoid muscle such that the child will position the head with the occiput rotated to the affected side and the chin rotated to the contralateral side. We recently treated 26 children who presented to the emergency department with acute nontraumatic torticollis. The most common causes were upper respiratory infection, sinusitis, otomastoiditis, cervical adenitis, and retropharyngeal abscess or cellulitis. Four patients had subluxation of the atlantoaxial joint as a result of the inflammatory process. Children with acute torticollis need careful evaluation for either overt or occult otolaryngologic infections. Computed tomography and magnetic resonance imaging are helpful in determining the cause of the acute torticollis and in ruling out rotatory subluxation of the atlantoaxial joint.

Atlanto-Axial Joint↗
PubMed2001

Muscle paralysis produced by botulinum toxin type A injection in treated torticollis patients compared with toxin naive individuals.

We sought to determine whether the response to varying doses of botulinum toxin type A (BTX-A) injected in BTX-A-treated torticollis patients differed from the same injections given in toxin-naive individuals. We have developed a technique to objectively measure muscle weakness resulting from BTX injections in humans and have validated the technique in those not previously treated with BTX. We now examine BTX-A-treated torticollis patients to see if their response to BTX-A injection is similar to that of toxin-naive individuals. We injected 11 torticollis patients who had been receiving BTX-A injections with a standard 5-mouse unit (mu) dose into one extensor digitorum brevis (EDB) muscle and a varying dose into the other EDB, measuring muscle paralysis 2 weeks after the injection. Nine of the 11 patients were clinical and electrophysiologic responders. Two patients were non-responders. In the 9 responding patients the dose response curve to increasing doses of BTX-A was very similar to that seen in toxin-naive individuals. The mean muscle paralysis from the standard 5 mu dose was also similar to that previously reported in toxin-naive individuals. Torticollis patients who continue to respond clinically to BTX-A injections demonstrate essentially the same degree of muscle paralysis from the EDB injections as do subjects who have never been exposed to BTX-A.

Adult↗
PubMed1989

Spasmodic torticollis due to a midbrain lesion in a case of multiple sclerosis.

A case of multiple sclerosis is described in which spasmodic torticollis occurred abruptly and abated after 1 year. Magnetic resonance imaging (MRI) demonstrated a lesion in the mesencephalon. Other symptoms and physical signs that developed at the same time as the spasmodic torticollis were compatible with the lesion that had not been present on MRI 18 months previously. There are very few reports of spasmodic torticollis due to an identified focal lesion; there is evidence from experimental work on animals that midbrain lesions may cause spasmodic torticollis but there has been no previous human example.

Adult↗
PubMed1982

Spasmodic torticollis: a behavioral perspective.

The literature on spasmodic torticollis is critically reviewed. The currently most popular etiological hypothesis characterizes torticollis as an extrapyramidal disorder, the symptoms of which are aggravated by stress, but there is no unequivocal evidence available to support this view. Psychological mechanisms have been suggested but not elaborated or tested in any detail. A wide range of treatments has been advocated but controlled studies have not been reported, and the problems of assessing outcome have never been tackled adequately. Behavioral treatments have been evaluated more rigorously than other approaches (particularly EMG feedback training), and the literature suggests that they benefit some patients. It is argued that psychologists have the potential for making a very significant contribution to the understanding and management of torticollis. In discussing outcome measures, the more promising techniques that have been used are summarized and a list is presented of the factors which must be considered when assessing torticollis symptoms. Directions for future research are outlined and priorities suggested.

Adult↗
PubMed1996

Refractory dystonia during propofol anaesthesia in a patient with torticollis-dystonia disorder.

PURPOSE: To report a case of refractory dystonia under propofol anaesthesia in a patient with Torticollis-Dystonia disorder. CLINICAL FEATURES: A 38-yr-old man presented for an MRI scan for investigation of a Torticollis-Dystonia disorder. There was a biphasic response to propofol with complete amelioration of the torticollis and limb dystonia initially with subsequent recurrence under deep propofol anaesthesia. Coadministration of midazolam, diazepam, and thiopentone were not successful in abolishing the recurrent dystonia. CONCLUSIONS: Propofol should preferably be avoided in patients with torticollis and dystonias. Where complete control of movements is required, it may be necessary to consider general endotracheal anaesthesia with muscle relaxants.

Adult↗
PubMed1999

Three-dimensional spiral CT scanning in children with acute torticollis.

Three-dimensional spiral CT scanning is now becoming a common investigation in children who have a history of acute torticollis. In the last year, 21 consecutive children who came to our unit with a history of acute torticollis were assessed using standard plain radiographs and a 3-dimensional spiral CT scan. Ten patients had a history of recent trauma. Spiral CT scanning revealed that 13 children had atlanto-axial rotatory subluxation (AARS). Plain radiographs had only a sensitivity of 33% and specificity of 71% in detecting AARS. Sixteen children were treated using a Halter traction. Four failed to resolve clinically and were put on a halo traction after 3-dimensional CT scanning again confirmed residual AARS. Two children remained symptomatic after halo traction, with persisting rotatory and anterior subluxation on repeat spiral CT. They both underwent a posterior in-situ fusion, with no attempt at open reduction. Plain radiography is limited in investigating acute torticollis in children. Spiral 3-dimensional CT reconstruction has an important role to play in both the investigation and management of children who present with acute torticollis.

Acute Disease↗