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PubMed1999

Posturographic findings in patients with idiopathic cervical dystonia before and after local injections with botulinum toxin.

In this posturographic study, whole-body postural control was impaired in more than 75% of patients with idiopathic cervical dystonia (ICD) before local injections with botulinum toxin type A (BTX-A) and the impairment was independent of the direction of the torticollis. Six weeks after therapy with BTX-A, the number of pathological posturographic parameters had decreased by almost 30%, and the improvement reached statistical significance for sway path, sway area and anteroposterior sway during stance on foam with eyes closed. From a pathophysiological point of view, this improvement may be explained - at least in part - by a reduction of abnormal proprioceptive input from the neck. Accordingly, it seems possible that neck proprioceptive input plays a role in whole-body postural control in ICD patients, even though previous studies suggested that the neck input is relatively ignored in these patients.

Adult↗
PubMed1999

Unilateral pallidal stimulation in cervical dystonia.

Cervical dystonia (spasmodic torticollis) is a focal dystonia of the cervical region. Various treatment modalities have been performed with variable success rates. We present a 42-year-old woman complaining of involuntary head rotation for the last 3 years. Different medical treatments had been used for 3 years. Botulinum toxin injections resulted in temporary and moderate improvement for periods of 3-4 months. Pallidal stimulation was performed using a quadripolar electrode and a battery-operated programmable pulse generator. We conclude that a unilateral pallidal lesion or stimulation is an effective method of treatment in focal dystonia. The target must be the pallidum contralateral to the contracted sternocloidomastoid muscle. Deep brain stimulation is superior to lesioning because of the capability of manipulating the stimulation parameters which can modify the pallidotomy effect.

Adult↗
PubMed2001

Outcome of selective peripheral denervation for cervical dystonia.

30 patients with cervical dystonia were treated by selective peripheral denervation (SPD) (Bertrand procedures). Follow-up 12-75 (median 26) months after surgery showed strong group improvements in the clinical state (Toronto Western Spasmodic Torticollis Rating Scale) and in performance of ADL, impairment, lifestyle and degree of incapacity (rating scales used by Australia's Department of Veterans' Affairs). SPD can give long-term benefits important clinically, in the home and in society.

Activities of Daily Living↗
PubMed1997

Spinal cord stimulation: a quarter century of development and investigation. A review of its development and effectiveness in 1,336 cases.

The past two and a half decades have seen the development of a spinal cord stimulator from the early 2-electrode fixed system to the present multielectrode computerized systems. During these 25 years, spinal cord stimulation has been studied in the treatment of motor disorders. The effectiveness was studied in 1,336 cases, including cerebral palsy (456), dystonia (173), torticollis (90), multiple sclerosis (130), spinocerebellar degeneration (71), spinal cord injury (303) and posttraumatic brain injury (113). It has become increasingly evident that the maximum therapeutic effect is achieved by virtue of the applied field variables of the spinal cord level stimulated, the field configuration, its polarity, and the frequency of the stimulation. These observations have led to investigational corollaries of the therapeutic specificity of the applied field, the neurophysiologic mechanisms of these fields and the underlying abnormal neurophysiologic substrate, which may indeed be secondary to abnormalities in the nerve impulse itself.

Cerebral Palsy↗
PubMed1981

Treatment of patients with degenerative diseases of the central nervous system by electrical stimulation of the spinal cord.

Patients with dystonia, spinocerebellar and cerebellar ataxia and spasmodic torticollis have a reasonable chance of being significantly aided in their control of motor function and neurogenic bladder by electrical stimulation of the cervical or thoracic spinal cord. This mode of therapy has the advantages that it is not destructive of neurological tissue, effects can be varied by altering the intensity and rate of the stimulus and preliminary testing with externalization of the electrodes is predictive of the effects of chronic stimulation.

Amyotrophic Lateral Sclerosis↗
PubMed1997

The enigma of lambdoid positional molding.

One hundred and twenty-one consecutive patients presenting to the Children's National Medical Center with lambdoid positional molding (LPM) were reviewed. Clinical features included unilateral occipital flattening and alopecia and forward displacement of the ipsilateral ear, forehead, and maxilla. Head tilt and tightness of the ipsilateral sternocleidomastoid muscle were common. An unexplained preponderance of LPM was found in males (74%) and on the right side (72%), both findings statistically significant (p < 0.001). Importantly, a variety of other abnormalities were seen with LPM: torticollis (41%), large head circumference (40), excess extra-axial cerebrospinal fluid (35), developmental delay (19), and other CNS abnormalities (20%). Systemic problems affecting the mobility were also common. Only 3 patients had craniosynostosis, and only 2 with LPM required surgery for severe cosmetic deformities. An apparent increase in the incidence of LPM was attributed to current recommendations to keep infants supine to decrease the risk of sudden infant death syndrome, overutilization of infant carriers similar to cradleboards of earlier cultures, and neonatal medical problems resulting in relative immobility. No evidence was found to support the concept that LPM causes compressive brain pathology; thus, surgical treatment is not required for such fears. Further, the sequelae of underlying CNS and systemic problems associated with LPM would not be corrected by opening unfused sutures, but could even be misinterpreted as complications of surgery.

Child↗
PubMed1985

Diagnostic value of velocity-step responses.

For the nystagmic (after-)response in each direction it was evaluated in 618 patients whether the gain (initial velocity) and the time constant were normal, significantly low or high, thus constituting a specific 'response pattern'. In a number of diagnostic categories, 65% of the response patterns were 'typical', i.e., they were found in significantly high frequency. Symmetrical response patterns were predominant. Short time constant(s) and/or low gain values in either or both directions were found in labyrinthine defects. High gain values were found in otosclerosis after footplate surgery, in multiple sclerosis, hyperventilation syndrome, posterior fossa tumour and spasmodic torticollis. In those categories, except otosclerosis and posterior fossa tumour, also significantly high values of the time constant were found, as well as in the category of patients with exposed tympanic cavity and dizziness. A significant directional preponderance indicating the defective side was found for the low-frequency gain (i.e., the product of initial velocity and time constant) in 48% of the patients with unilateral canal paralysis. It is concluded that the velocity-step test is a powerful diagnostic tool in vestibular examination.

Brain Neoplasms↗
PubMedDate not supplied

Fine needle aspiration diagnosis of fibromatosis colli. A report of three cases.

BACKGROUND: Fibromatosis colli, a common cause of congenital muscular torticollis, should be differentiated from other neck masses in infants. Invasive diagnostic and therapeutic measures should be avoided. CASES: Three infants under the age of 2 months presented with neck masses--a clinical suspicion of malignancy, lymphadenopathy and teratoma. The cytologic findings included dyshesive multinucleated skeletal muscle fragments showing degenerative and atrophic changes within a background of scattered reactive fibroblasts. CONCLUSION: Fine needle aspiration biopsy is a safe and rapid method of providing a confirmatory diagnosis of neck masses in infants.

Biopsy, Needle↗
PubMed1986

Fibrodysplasia ossificans progressiva presenting as osteomyelitis-like syndrome.

An 8-year-old boy presenting with fever, neck pain, and torticollis was eventually diagnosed as having fibrodysplasia ossificans progressiva (FOP). The initial bone scan was interpreted as being suggestive of vertebral osteomyelitis. Subsequent computerized tomographic studies (CT scan), however, demonstrated paravertebral soft-tissue calcification adjacent to, but not involving, intact cervical vertebrae. The acute process of FOP was thereby distinguished from that of osteomyelitis by use of CT scan.

Cervical Vertebrae↗
PubMed2006

Mandibular asymmetry and breastfeeding problems: experience from 11 cases.

The authors review the experiences of 11 mother-infant pairs who had breastfeeding problems related to the infants' mandibular asymmetry. Lower jaw asymmetry is an early identifiable sign of torticollis, and a possible contributor to latch difficulties, nipple pain, and poor milk transfer. Pediatricians and lactation consultants should look for signs of lower jaw asymmetry combined with a preference for turning the head to one side in newborns who present with breastfeeding difficulties. By recognizing these anatomic irregularities early in the course of breastfeeding, pediatric health care providers will be able to treat breastfeeding problems promptly and proactively. They will be able to avoid unnecessary hospital admissions for dehydration symptoms or a septic workup and will intervene quickly with the recommendation of breast pumping and complementary feeding if milk transfer is not occurring.

Adult↗
PubMed2003

Visual performance after congenital nystagmus surgery using extended hang back recession of the four horizontal rectus muscles.

PURPOSE: To evaluate the results of surgical correction of congenital nystagmus based on the use of extensive recessions of the four horizontal rectus muscles. The outcome of this procedure in terms of visual performance of the patients has been analyzed in follow-ups of at least 1 year. METHODS: Surgery was performed on 42 patients. An extended retroequatorial recession of the four horizontal rectus muscles was performed between 13 and 15 mm from the muscle insertion with hang back sutures in every case. Preoperatively, all cases showed either a horizontal pendular (4 cases) or jerk nystagmus (38 cases). RESULTS: After surgery, all cases showed full preservation of ductions in all gaze positions. Torticollis decreased in all cases. Binocular best-corrected vision improved in 19 (45.2%) patients and in the remaining 23 (54.8%) it remained unchanged. Consecutive exotropia was observed postoperatively in 7 patients. A second surgical procedure was performed in 9 cases (21.4%) and a botulinum injection in 3 cases (7%). All cases of diplopia resolved. Patient satisfaction was high in 38 cases (96%). CONCLUSIONS: Extended recession of horizontal rectus muscles using hang back sutures offers a good option for the surgical correction of sensorial and neuromuscular nystagmus, providing improvement in binocular best-corrected vision in 45% of the patients (21.4% more than 0.2 logMar units), a high satisfaction rate, and few associated complications. In patients with low vision this can be considered a success that may improve quality of life.

Adolescent↗
PubMed2005

Safety, effectiveness, and duration of effect of BOTOX after switching from Dysport for blepharospasm, cervical dystonia, and hemifacial spasm dystonia, and hemifacial spasm.

BACKGROUND: Local injection of botulinum toxin type A is first-line treatment of blepharospasm, cervical dystonia, and hemifacial spasm; however, there is uncertainty about the optimal dose of toxin for each indication as well as dose-conversion ratios that should be used when switching products in clinical practice. OBJECTIVE: The goal of this study was to compare the safety, effectiveness, and duration of clinical effect of BOTOX and Dysport and Dysportdagger after drug switching (Dysport to BOTOX) among patients with movement disorders. METHODS: A total of 48 patients diagnosed with blepharospasm (n = 27), cervical dystonia (n = 12), or hemifacial spasm (n = 9) were evaluated during a single-arm, crossover-design study in which each patient was his/her own historical control using a 5:1 or 4:1 conversion ratio of Dysport to BOTOX units. Patients were assessed (using Jankovic, Visual Analog Scale, general pain scale, Toronto Western Spasmodic Torticollis Rating Scale [TWSTRS] scores) after the last injection of Dysport and the first injection of BOTOX. Moreover, each patient kept a diary during treatment to track onset and duration of therapeutic effect. RESULTS: Although BOTOX and Dysport were both clinically effective, this effect was more significant with BOTOX compared to Dysport based on TWSTRS for cervical dystonia (p = 0.012), and Jankovic scores for blepharospasm (p = 0.006). Longer duration of effect also was noted with BOTOX than with Dysport (blepharospasm, 62.2 days vs 47.4 days (p = 0.001); cervical dystonia, 64.3 days vs 44.6 days (p = 0.014); hemifacial spasm, 65.1 days vs 41.8 days (p < 0.014), respectively). Of the 48 patients, 19 experienced at least one adverse drug reaction (ADR) during Dysport treatment, with the most commonly reported ADRs being ptosis for blepharospasm and hemifacial spasm and neck weakness for cervical dystonia. No patient reported an ADR during BOTOX treatment. CONCLUSIONS: Results suggest therapeutic effectiveness is enhanced with BOTOX compared to Dysport at a dosing ratio between 5:1 and 4:1 (Dysport:BOTOX). Safety and duration of therapeutic effect also are enhanced with BOTOX. Further research is needed.

Adult↗
PubMed2005

Comparison of botulinum toxin serotypes A and B for the treatment of cervical dystonia.

OBJECTIVE: To directly compare two serotypes of botulinum toxin (BoNTA and BoNTB) in cervical dystonia (CD) using a randomized, double-blind, parallel-arm study design. METHODS: Subjects with CD who had a previous response from BoNTA were randomly assigned to BoNTA or BoNTB and evaluated in a blinded fashion at baseline, 4 weeks, 8 weeks, and 2-week intervals thereafter until loss of 80% of clinical effect or completion of 20 weeks of observation. CD severity was measured with the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS), and adverse events were assessed by structured interview. Statistical analysis included Wilcoxon rank sum test, log rank tests, and Kaplan-Meier survival curves for duration of effect. RESULTS: A total of 139 subjects (BoNTA, n = 74; BoNTB, n = 65) were randomized at 19 study sites. Improvement in TWSTRS score was found at 4 weeks after injection and did not differ between serotypes. Dysphagia and dry mouth were more frequent with BoNTB (dysphagia: BoNTA 19% vs BoNTB 48%, p = 0.0005; dry mouth (BoNTA 41% vs BoNTB 80%, p < 0.0001). In clinical responders, BoNT A had a modestly longer duration of benefit (BoNTA 14 weeks, BoNTB 12.1 weeks, p = 0.033). CONCLUSION: Both serotypes of botulinum toxin (BoNTA and BoNTB) had equivalent benefit in subjects with cervical dystonia at 4 weeks. BoNTA had fewer adverse events and a marginally longer duration of effect in subjects showing a clinical response.

Adult↗
PubMed2006

CDIP-58 can measure the impact of botulinum toxin treatment in cervical dystonia.

We compared the responsiveness of the Cervical Dystonia Impact Profile (CDIP-58), Medical Outcome Study Short Form-Health Survey (SF-36), Functional Disability Questionnaire (FDQ), and Pain and Activities of Daily Living subscales of the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) in participants with cervical dystonia treated with botulinum toxin A. Subscales of CDIP-58 were more sensitive in detecting statistical and clinical change than comparable subscales of the SF-36, FDQ, and TWSTRS.

Adult↗
PubMed1985

Lisuride in dystonia.

Lisuride hydrogen maleate, 0.4 to 5 (mean, 3) mg/d, was given orally to 42 subjects with various types of dystonia. In seven of the eight patients who improved (one with segmental dystonia, one with myoclonic dystonia, two with spasmodic torticollis, two with cranial dystonia, and two with tardive dystonia), the response was confirmed by double-blind placebo substitution. No patients with a suspected structural brain lesion improved. There was no consistent pattern of response among those patients with different forms of idiopathic (primary) dystonia. Lisuride improved some patients, but had no effect on other, apparently identical, patients.

Administration, Oral↗
PubMed1988

Neuromuscular effects distant from the site of botulinum neurotoxin injection.

We assessed the severity and temporal profile of distant neuromuscular effects from a single dose (280 units) of botulinum neurotoxin injected into neck muscles for torticollis. We performed single-fiber EMG studies on the biceps brachii of six patients to measure jitter (20 pairs) and fiber density on the initial treatment day and then again, at least once more, after 2 to 12 weeks. No patient developed weakness beyond the neck muscles or decrement of the biceps response to repetitive 3-Hz nerve stimulation. Between the baseline and the last follow-up study, the average of mean MCD increased from 29 microseconds to 38 microseconds (31%). Mean fiber density increased concurrently or earlier from 1.35 to 1.79 (33%). There were no electrophysiologic signs of presynaptic blockade, even at 2 and 4 weeks. The effects we observed are compatible with stimulation of terminal sprouting by the neurotoxin, without significant presynaptic inhibition of acetylcholine release. We therefore believe that higher dosages of the neurotoxin may be used if clinically indicated.

Adult↗
PubMed1988

Pathology in brainstem regions of individuals with primary dystonia.

Examination of brains from four individuals with the clinical diagnosis of primary dystonia revealed histopathologic abnormalities in two cases. A 29-year-old man with a 15-year history of dystonia musculorum deformans (DMD) had numerous neurofibrillary tangles (NFT) and mild neuronal loss within the locus ceruleus; occasional NFT were also recognized in the substantia nigra pars compacta, pedunculopontine nucleus, and dorsal raphe nucleus. A 68-year-old man with a 35-year history of Meige syndrome had moderate-to-severe neuronal loss in several brainstem nuclei, including the substantia nigra pars compacta, locus ceruleus, raphe nuclei, and pedunculopontine nucleus. Infrequent NFT were also noted in substantia nigra. An examination of these and other brain regions in a 10-year-old boy with a 6-year history of DMD and a 50-year-old woman with a 3-year history of spasmodic torticollis did not disclose similar abnormalities.

Adult↗
PubMed1997

Botulinum toxin type B: a double-blind, placebo-controlled, safety and efficacy study in cervical dystonia.

We enrolled and treated 122 patients with idiopathic cervical dystonia in a double-blind, placebo-controlled safety and efficacy study of botulinum toxin type B (BotB). Both A-responsive and A-resistant patients were enrolled. Patients received intramuscular injections of either BotB (2,500 U, 5,000 U, or 10,000 U) or placebo. The primary outcome measure of efficacy was the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS)-Total score at 4 weeks following study drug administration. Secondary measures of efficacy were TWSTRS-Severity, -Disability, and -Pain subscale scores, and Analog Pain Assessment, Investigator Global Assessment, Patient Global Assessment, and Sickness Impact Profile scores. Duration of effect was estimated with an intent-to-treat analysis of responders. Safety measures included clinical parameters, laboratory tests, and adverse events. The primary and most of the secondary analyses indicated a statistically significant treatment effect and a dose response. BotB is safe, well tolerated, and efficacious in the treatment of cervical dystonia at the doses tested.

Adult↗