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PubMed1981

Human leukocyte anitgen in torticollis and other idiopathic dystonic syndromes.

Two groups totaling 67 patients with idiopathic focal, segmental, and generalized dystonia, including torticollis, were compared with normal controls to determine whether there was a difference in the frequency of A, B, and C locus human leukocyte (HLA) antigens. The results indicated no statistically significant deviations in HLA antigen frequencies between the patients and the normal controls. Thirteen of the patients with idiopathic torsion dystonia were compared with normal controls for DR locus antigens. A trend of increased DR3 antigens observed in the patients may be significant. HLA genotyping of parents and children in nine families was also studied to determine if an HLA-linked factor could be related to the dystonic syndrome in the children. The results were indeterminate, suggesting that further family studies are required to resolve this issue.

Adult↗
PubMed1991

Septic arthritis of the C1-C2 lateral facet joint and torticollis: pseudo-Grisel's syndrome.

We present the case of a 76-year-old man who experienced the sudden development of fever, rightsided neck pain and stiffness, and torticollis. A soft tissue mass was noted on the right side of his neck, but his head was tilted to the left. Computed tomography scans (with reformatted sagittal and coronal images) of the patient's cervical spine revealed destructive changes of the right lateral masses of C1 and C2 and the clivus, and a well-delineated peridontoid soft tissue mass (confirmed by magnetic resonance imaging). After the second episode of right-sided hemiparesis, he underwent transoral surgical exploration, with anterior decompression and odontoidectomy. Histologic examination of the surgical material revealed granulation tissue, fibrosis, and chronic inflammation, consistent with abscess formation with invasion and compression of the spinal cord and bone. This case suggests that nonreducible rotational head tilt to the side opposite the side of lateral mass collapse should raise the suspicion of a possible infection.

Aged↗
PubMedDate not supplied

Botulinum a toxin for spasmodic torticollis: multiple vs single injection points per muscle.

Eighty-six injections in 49 patients with adult onset spasmodic torticollis were evaluated for efficacy with respect to single point per muscle versus multiple point per muscle injection techniques. Parameters of the syndrome assessed were pain, posture deformity, range of cervical motion, disfigurement, cervical muscle hypertrophy, activity limitation, and degree of involuntary movement. The multiple point per muscle injection strategy appeared superior to the single injection per muscle technique with respect to pain (p less than 0.002, chi-square), posture deformity (p less than 0.001), range of motion (p less than 0.001), and improvement in activity endurance (p less than 0.001). No significant differences were noted with respect to cervical muscle hypertrophy or degree of involuntary movements, although the injections were considered beneficial in both groups to these syndrome components.

Adult↗
PubMed2003

Posttraumatic painful torticollis.

The development of abnormal posturing of the neck or shoulder after local injury has been termed posttraumatic cervical dystonia (PTCD). Certain features seem to distinguish a unique subgroup of patients with this disorder from those with features more akin to typical idiopathic cervical dystonia, such as onset and maximum disability that occurs very quickly after injury, severe pain and a fixed abnormal posture. In an attempt to clarify the nature of this syndrome further, we evaluated 16 such patients (8 men, 8 women). Motor vehicle accident and work-related injuries were common precipitants, with posturing usually developing shortly after trauma, and little progression occurring after the first week. A characteristic, painful, fixed head tilt and shoulder elevation were present in all but one patient, who had a painless elevated shoulder and painful contralateral shoulder depression, as well as nondermatomal sensory loss in 14 patients. Additional abnormalities included dystonic posturing in a limb (2 patients) or jaw (1 patient), limb tremor (3 patients) and "give-way" limb weakness (8 patients). The tremor and the jaw dystonia demonstrated features suggestive of a psychogenic movement disorder, most commonly distractibility. Litigation or compensation was present in all 16 patients. Intravenous sodium amytal improved the posture, pain or both in 13 of 13 patients; in 7 of 13 the sensory deficit either markedly improved or normalized. General anesthesia demonstrated full range of motion in all 5 patients assessed. Psychological evaluations suggested that psychological conflict, stress, or both were being expressed via somatic channels in 11 of 12 tested patients. Our results suggest an important role of psychological factors in the etiology or maintenance of abnormal posture, pain and associated disability of these patients. The role of central factors triggered in psychologically vulnerable individuals after physical trauma is discussed. We propose that the disorder be referred to as "posttraumatic painful torticollis" rather than characterize it as a form of dystonia until further information on its pathogenesis is forthcoming.

Adolescent↗
PubMed1986

Electrophoresis and immunoblot of cerebrospinal fluid proteins in spasmodic torticollis.

Protein patterns of cerebrospinal fluid (CSF) from patients with spasmodic torticollis (ST) were investigated to determine whether abnormalities previously reported could be detected and further identified. CSF was collected from 12 patients with ST and 6 normal controls. The CSF proteins were analyzed using sodium dodecyl sulfate (SDS) polyacrylamide gel electrophoresis and silver staining. In 11 of the 12 patients with ST, a CSF protein pattern was observed which differed from that in the controls. The identity of the abnormal proteins was ascertained by blotting and immunostaining with specific antisera to IgG and ceruloplasmin (Cp). CSF from 2 of 12 patients had distinct bands staining for IgG and 7 had abnormal immunostaining for Cp.

Adult↗
PubMed1988

Blink reflex studies in focal dystonias: enhanced excitability of brainstem interneurons in cranial dystonia and spasmodic torticollis.

We have studied the orbicularis oculi reflex to paired stimuli in patients with various forms of focal dystonia and in normal controls. In normals, the conditioning stimulus (CS) facilitated the test stimulus (TS) early response (R1), but markedly inhibited the TS polysynaptic late response (R2). In all types of dystonias studied the CS facilitated the TSR1 as in normals. However, in patients with blepharospasm (alone or associated with oromandibular dystonia), spasmodic torticollis, or spasmodic dysphonia, it inhibited the TSR2 significantly less than that of the controls, with marked enhancement of the recovery curve of the late response. The TSR2 recovery curve of patients with focal arm dystonia was normal. These results are indicative of increased brainstem interneuron excitability in the various dystonias mediated by the cranial nerves, but not in focal arm dystonias such as dystonic writer's cramp. This abnormality might be caused by an abnormal input possibly from the basal ganglia upon these brainstem cells. Our results also suggest that a similar pathophysiology underlies the various focal dystonias of the head and neck.

Blepharospasm↗
PubMed1994

Late presentation of congenital muscular torticollis: use of MR imaging and CT scan in diagnosis.

A 17-year old boy presented with a 10-year history of progressive head tilt to the right. Bilateral posterolateral cervical pain was mild and he was fully functional. The right sternocleidomastoid (SCM) muscle was prominent without rotation of the head to the left. The SCM had a cord-like consistency on palpation. Magnetic resonance (MR) and computed tomography (CT) scan imaging of the neck musculature suggested fibrous tissue within the substance of the muscle. This was histopathologically confirmed when the right SCM was surgically explored and resected. Congenital muscular torticollis is usually seen in newborns, infants, and children but may also present in adolescence and young adulthood. It should be included in the differential diagnosis of cervical dystonia as one of the nondystonic causes of abnormal head posture. Combined use of MR and CT scan of neck muscles may be of help in the diagnosis.

Adolescent↗
PubMed1994

Abnormalities of somatosensory evoked potentials in spasmodic torticollis.

This study examined the N20 and N30 waves of somatosensory evoked potentials (SEPs) from median nerve stimulation at three different stimulation rates (1, 3, and 6 Hz) in patients with idiopathic spasmodic torticollis (ST). The data were compared with those collected from a group of patients affected by Parkinson's disease (PD) and normal age-matched subjects. N30 amplitude was significantly decreased in both groups of patients with respect to the controls. The decrease was larger in patients with ST. The N20 wave remained stable. The latencies of both waves were unchanged. When the stimulus rate was increased, the N30 amplitude decreased significantly, with a similar trend observed in both patients and controls. The isolated abnormalities of the N30 wave in both ST and PD support the hypothesis of a common physiopathogenetic mechanism that involves the basal ganglia or their connections with the supplementary motor area.

Adult↗
PubMed1995

Epidemiology and outcome of cervical dystonia (spasmodic torticollis) in Rochester, Minnesota.

The natural history of cervical dystonia (spasmodic torticollis) was investigated in a population-based study in Rochester, Minnesota. Eleven new cases were identified with onset during the 20-year period 1960-1979. The overall incidence rate was 1.2 per 100,000 person-years (95% confidence interval 0.5-1.9) with a female:male ratio of age-adjusted incidence rates of 3.6:1. A unitary etiology was not apparent: injury antedated onset in four of the 11 patients, whereas six had documented thyroid disease and four had diabetes. A family history of movement disorder was recorded for only one subject. Only one of the cases would have been classified as moderate in severity; the others were mild. In follow-up through 1993, progressive disability was noted in only two patients, and two others went into remission. Three cases of intracranial aneurysm were confirmed, two of which produced fatal subarachnoid hemorrahage. A third death was due to amyotrophic lateral sclerosis.

Age of Onset↗
PubMed1997

Torticollis due to disinhibition of the vestibulo-collic reflex in a patient with Steele-Richardson-Olszewski syndrome.

A patient with the clinical picture of Steele-Richardson-Olszewski syndrome and an unusual intermittent neck twisting is reported. He had virtually no voluntary ocular movements and only very slow, low-amplitude voluntary head movements. However, in response to optokinetic or vestibular stimulation, he developed full eye deviations in the direction of the slow phase of the expected nystagmus. No quick phases were observed, and the deviation outlasted the duration of the vestibular stimuli because of defective saccades. The head also turned fully during these stimuli, quicker than on attempted voluntary movements, and remained deviated similarly to the eyes. This suggests that the neck deviations in this patient were due to a disinhibited vestibulo-collic reflex and a disturbed head position resetting mechanism. Neck electromyographic responses in response to whole-body rotation indicated that the vestibulocollic reflex responsible for the torticollis in this patient had a short latency of approximately 30 ms.

Amygdala↗
PubMed1997

Hand tremor in patients with spasmodic torticollis.

The occurrence of hand tremors in patients with spasmodic torticollis (ST) was investigated in 55 patients by clinical and quantitative measurements. Ten patients had first-order or second-order relatives affected with postural tremor. Although 40% of the patients had a medical history and clinical findings for mild postural and action tremor of the hands, only four had moderate tremor amplitudes. One patient had a severe tremor, including resting tremor, and two had mild voice tremor. A positive correlation was found between hand and head tremor. Patients with hand tremor were younger at the onset of ST than were those without. The mean amplitudes of postural tremor were only slightly higher than for the controls and much smaller than those found in classic essential tremor. The tremor caused only mild disabilities. The tremor frequencies were indistinguishable from physiologic tremor. Further analysis of the tremor records showed evidence for physiologic tremor mechanisms only. We conclude that slightly enhanced postural hand tremors are common in ST but have a low amplitude and are only rarely clinically relevant for ST patients. Although the present data support the notion of an enhanced risk of postural tremor in families of patients affected with ST, none of the criteria allowed the separation of the hand tremor of ST from other postural/action tremors and especially from enhanced physiologic tremor. Thus, the present data do not support the classification of hand tremor in ST as either "dystonic" or essential tremor.

Adolescent↗
PubMed1997

Teaching tape for the motor section of the Toronto Western Spasmodic Torticollis Scale.

The Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) assesses the character and severity of cervical dystonia (CD). We developed a teaching tape of the TWSTRS scoring for the motor symptoms of CD. The tape provides investigators with visual representations of each component of the motor section of the TWSTRS as agreed upon by three independently scoring raters. The rate of agreement for the nondichotomous components was always significant, with a Kendall's coefficient of concordance W ranging between 0.98 and 0.76 (p < 0.01 for all measures). For the two dichotomous components, a weighted kappa coefficient was also significant at 0.86 for lateral shift and 0.89 for sagittal shift (p < 0.01 for both measures). Scale deficiencies identified by the raters were an explicit definition of midline for assessment of range of motion, the absence of a separate scoring category assessing dystonic tremor, and the specification of duration for the effect of sensory tricks. These observations should be taken into account in future revisions of the TWSTRS and in refinements of other rating scales for CD.

Dystonia↗
PubMed1997

Perception of spatial orientation in spasmodic torticollis. Part 2: The visual vertical.

Twenty-nine patients with idiopathic spasmodic torticollis (ST) and matched normal control subjects were asked to align a target line to perceived earth vertical [visual vertical (VV)]. Settings were made against a whole-field random-dot background that was either stationary or rotating around the line of sight, and subjects performed the task upright and lying horizontally on their sides. Normal subjects were tested both head upright and after assuming a voluntary head tilt. Patients with ST set the VV close to true upright with a minimal deviation toward tilt of the head in contrast to normal subjects assuming a head tilt who set the VV in the opposite direction to the head tilt (Müller "E" effect). Settings against the spinning disk were biased in the direction of rotation similarly for both subject groups. Settings made against static or spinning disk performed when subjects lay horizontally were tilted in the direction of recumbence (Aubert "A" effect) similarly for both subject groups. When attempting to set the target line parallel to the long axis of the face, patients with head tilt set the line to upright, whereas normal subjects correctly estimated their tilts. One hypothesis offered to explain these results is that the patients referenced only their upright trunk for vertical and did not make use of neck proprioceptive or vestibular signals of head tilt so that all settings were made as if the trunk and head were upright. Alternatively, patients may have used only otolith signals as the reference for upright, and these are recalibrated in ST patients with head tilt. The pathological deviation becomes accepted as upright posture, and VVs and facial orientation are estimated as if the head were upright. In either case the findings indicate abnormal processing of the perception of visual verticality in ST patients.

Adult↗
PubMed1998

Localized proton NMR spectroscopy in the striatum of patients with idiopathic spasmodic torticollis.

Single voxel proton spectroscopy was used to study brain metabolism in idiopathic spasmodic torticollis. Peak metabolite ratios in long echo time (135 ms) spectra were evaluated in the basal ganglia of 16 patients (29-65 years). Eight of them were untreated; the other eight were examined 4-6 weeks after administration of botulinum toxin type A. As compared with 60 control spectra, patients showed a significant, therapy-resistant decrease in N-acetyl-L-aspartate (NAA)/choline (Cho). NAA/creatine (Cr) and Cho/Cr were close to normal in the post-treatment group but significantly reduced in untreated patients. This result is consistent with the hypothesis that striatal Cr is reversibly elevated in chronic muscle stimulation. The right and left striatum were affected to the same extent.

Aspartic Acid↗
PubMed1986

Prenatal exposure to the fungicide dinocap causes behavioral torticollis, ballooning and cleft palate in mice, but not rats or hamsters.

The present study is an evaluation of the developmental toxicity of dinocap in three rodent species using an in vivo teratology screen. Our protocol uses postnatal viability, weight gain, and morphological and behavioral development through weaning to assess the developmental toxicity of compounds. Dinocap administered orally on days 7 to 16 of gestation to the CD-1 mouse resulted in increased postnatal mortality at 25 mg/kg/d (80% in block 1 and 40% in block 2). Many of the treated pups that died during the neonatal period were "ballooned" and had cleft palates. Although there was no treatment related mortality in the 12 mg/kg/d dosage group, 6% (14/226) of these mice and 24% (23/96) of the survivors from the 25 mg/kg/d dosage group displayed torticollis (a twisting of the neck resulting in an abnormal tilting of the head). These tilted-head mice held the head and forepart of the body tilted constantly to one side, both when resting and walking. The tilt was in either direction but was always constant for a given animal; in different mice, the angle varied considerably from almost 0 to 30 degrees. Some mice circled repeatedly in one direction in the home cage, others bobbed their heads and did back-flips, while others rolled over, always rolling in the same direction. In the hamster, developmental toxicity was seen at (100 and 200 mg/kg/d) or near (50 mg/kg/d) maternally toxic doses but no behavioral alterations were noted and none of the pups were ballooned.

Animals↗
PubMed1987

The Foerster-Dandy operation for the treatment of spasmodic torticollis.

The short-term results of the Foerster-Dandy operation in 9 patients with spasmodic torticollis had been studied retrospectively. A good result was obtained for dyskinetic movement, but less for head tilt. Neck pains improved in 4 patients, but developed in 2 other patients postoperatively. Limitation of neck motility and abduction of the arms was seen in all patients. One patient died following a cerebral sinus thrombosis. Other surgical procedures are discussed and the results of studies of anterior rhizotomy are compared. The Foerster-Dandy procedure is indicated for severe disability or pain of more than 2 years duration not responding to other treatments.

Adult↗
PubMed1987

Spasmodic torticollis: the problem of pathophysiology and assessment.

In 17 patients with idiopathic spasmodic torticollis (ST) quantitative indices for both signs (extent and direction of the head deflexions) and complaints (of deflexion, shaking and pain) were collected. In the literature deflexion in the horizontal plane is most frequently considered, but analysis of the data shows that deflexion in the coronal and sagittal planes is also important. Furthermore, it is found that especially the deflexions in the coronal and sagittal plane covariate with the patients' complaints, but not with horizontal deflexion. On the basis of these and related data, it is suggested that we are dealing with at least two subtypes of ST. Finally, the patient's neuroticism and depression scale values are within the normal range and do not show significant correlations with ST duration. The present study provides no evidence that ST is a psychogenic disorder. ST should be regarded as a central nervous system disorder of unknown aetiology.

Adult↗