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PubMed1997

Quantitative electromyography-guided botulinum toxin treatment of cervical dystonia.

The purpose of this study was to investigate the clinical and electromyographic effect of turn/amplitude analysis (TAA)-guided botulinum toxin administration in patients with cervical dystonia. Involuntary electromyographic activity was recorded from both sternocleidomastoidei, both splenii capites, and both trapezii muscles of 13 torticollis patients, aged 34-73 years, before and after botulinum toxin A (Dysport) application. Dystonic muscles were selected for the injection if mean turns/s exceeded a level of 200. Four weeks after treatment with a mean dose of 223 mu/subject, clinical improvement was observed in 12 patients (92%) and only one patient reported no effect. Electromyographic improvement could be observed in 10 patients (77%). Both turns/s and the amplitude/turn decreased by 27% on the average after treatment. The electromyographic toxin effect showed a good correlation with the clinical toxin effect (r = 0.6). No dose dependency of the changes in turn/amplitude parameters could be observed. We found TAA a valuable modality for targeting and selecting dystonic muscles and for assessing the therapeutic benefit of the toxin.

Adult↗
PubMed1994

Relation between vertical facial asymmetry and postural changes of the spine and ancillary muscles.

Some patients presenting with head tilt maintain binocular vision by the adaptation of the spinal position. It is suggested that in cases of hyper eye facial asymmetry, where one eye is positioned higher than the other, the head will tilt to equalize the ocular heights in order to maintain binocularity during concentrated visual tasks. This phenomenon is a subdivision of ocular torticollis. The continuous head tilt causes adaptations by the spine and its ancillary muscles. Early treatment by small vertical prisms can alleviate stress discomfort and prevent it in the future.

Adaptation, Physiological↗
PubMed1998

Applications of endoscopic surgery in pediatric patients.

Although the advent of endoscopic technology is expanding the fields of reconstructive and aesthetic surgery in adults, there have been few reports of the use of this technology in the pediatric population. Because of their minimally invasive nature, yet wide range of exposure, endoscopic techniques have much appeal for this age group. Here we present our experience with endoscopic pediatric plastic surgery. From February of 1995 to August of 1997, 104 patients underwent 139 procedures utilizing 5- and 10-mm endoscopes. There were 58 male and 46 female patients. The mean age at surgery was 5.6 years (range, 3 weeks to 19 years). The most common type of procedures performed were insertion of tissue expanders (n = 34), excision of benign head and neck masses (n = 27), torticollis release (n = 20), excision of vascular lesions (n = 13), and miscellaneous procedures, (n = 10). There were 26 complications in 139 procedures (19 percent). Seventeen (65 percent) were in the tissue expander group. The rest were scattered among the groups with other diagnoses. Although there did not appear to be a specific type of complication associated with endoscopy, 77 percent occurred in the first 2 months of our study. This suggests a relatively steep technical learning curve. These results demonstrate that endoscopic techniques are eminently applicable in the pediatric population, providing the benefits of small and remote incisional wounds, with complication rates that are comparable with those of conventional open surgical treatment.

Adolescent↗
PubMed1992

Benign tumors of the cervical spine.

Benign tumors of the cervical spine are relatively infrequent but have a number of common characteristics that aid in the evaluation and treatment of these lesions. The tumors are most common in the first and second decades of life, presenting as pain, neck stiffness and torticollis. In approximately 70%, the lesions are visible on plain roentgenograms and the remainder are well visualized on bone scan and computed tomographic scan. The majority are present in the posterior elements and may be treated adequately with excisional biopsy by curettage. Stage 3 lesions are best treated by marginal excisional techniques and may require adjunctive techniques such as embolization or radiation therapy. The location of the lesion and extent of excision determine the necessity for fusion.

Adolescent↗
PubMed1993

Nonrheumatoid cranial settling.

Cranial settling is a condition in which erosive changes of the atlantal lateral masses result in downward telescoping of the atlas onto the axis body, anterior displacement of the atlantal posterior arch, and subsequent ventral and dorsal cervicomedullary compression. Cranial settling is described in conjunction with rheumatoid basilar invagination and atlanto-axial instability, possibly representing the most life-threatening abnormality associated with rheumatoid arthritis. The authors describe a case of symptomatic cranial settling in a nonrheumatoid patient with chronic, severe, spasmodic torticollis resulting in erosive changes in the occipito-atlanto-axial complex.

Atlanto-Axial Joint↗
PubMed2003

Atlantoaxial rotatory fixation secondary to tuberculosis of occiput: a case report.

STUDY DESIGN: A case of nontraumatic rotatory fixation of the atlantoaxial joint associated with tuberculosis of the occipital bone in an adult. OBJECTIVES: To report a rare case of atlantoaxial rotatory subluxation associated with tuberculosis of the occipital bone in an adult and to discuss the mechanism of fixation. SUMMARY OF THE BACKGROUND DATA: Atlantoaxial rotatory fixation in adults is rare and has been reported due to variety of causes. To the authors' best knowledge no case has been reported secondary to tuberculosis of the skull bone. METHODS: A 20-year-old male presented with resistant torticollis with a duration of 5 months. RESULTS: The patient had type 1 atlantoaxial rotatory fixation secondary to tuberculosis of the occipital bone. The subluxation was partially reduced by conservative means, and healing of the occiput lesion was achieved. Thereafter, the patient had no restriction of cervical spine motion and had no reoccurrence of subluxation at a follow-up of one and a half years. CONCLUSIONS: Effusion in the atlantoaxial joint secondary to infection in the occiput due to close proximity with the joint led to the laxity of ligaments and contributed to the subluxation.

Adult↗
PubMed2005

Osteoid osteomas and osteoblastomas of the occipitocervical junction.

STUDY DESIGN: We describe our surgical experience to remove osteoid osteomas and osteoblastomas of the occipitocervical junction. In this location, vertebral artery vicinity requires special consideration. OBJECTIVES: We illustrate our surgical approaches to remove lesions confidently, while minimizing bone resection to preserve stability. SUMMARY OF BACKGROUND DATA: Up until now, osteoid osteomas and osteoblastomas of the occipitocervical junction are reported as case reports. Several treatment methods have been described to treat osteoid osteomas. Nevertheless, surgery is the treatment of choice for lesions located in the C0-C2 region. METHODS: A retrospective review of 7 patients, including 5 men and 2 women, with a mean age of 21.0 years (range 3.0-38.0) was conducted. Clinical outcomes were evaluated immediately and after a mean follow-up of 27.6 months. RESULTS: There were 6 and 1 patients who underwent surgery with the anterolateral and posterolateral approaches, respectively. In osteoid osteomas, the nidus was removed, and the peripheral condensation was drilled up to normal bone (n = 4) or partially resected (n = 2). One osteoblastoma was removed extensively up to soft tissues. No osteo-arthrodesis was performed. Before surgery, all patients complained of pain, 3 presented with neck stiffness, and 2 with a torticollis. Immediately after surgery, all complaints disappeared. One patient underwent repeat surgery 15 months later for a recurrence. At the end of the follow-up, all patients were symptom-free, and partially resected peripheral condensations were stable on computerized tomography. CONCLUSIONS: Removal of osteoid osteomas and osteoblastomas of the occipitocervical junction is safe and efficient. Stability is preserved if more than half the joints are preserved with a proper surgical approach that minimizes bone resection.

Adolescent↗
PubMed1996

Fracture of the occipital condyle.

We report the case of a patient with occipital condyle fracture who was neurologically intact. The patient was treated conservatively with a neck collar, and the symptoms resolved in 3 months. This diagnosis can be suspected in high-velocity road traffic accidents, in a patient with neck pain, disproportionate torticollis, or lower cranial nerve symptoms. Skull radiography and cervical spine films can be normal. The diagnosis is best made by high-resolution computed tomographic (CT) scanning with sagittal and coronal reconstruction.

Adolescent↗
PubMedDate not supplied

Orthopaedic aspects of prune belly syndrome.

Prune belly syndrome is a relatively uncommon disorder that is characterized by intrauterine urinary obstruction associated with cryptorchisdism, oligohydramnios, and orthopaedic deformations. The oligohydramnios is believed to produce limited intrauterine space, which in turn leads to fetal compression and the resultant deformities. The deformities observed in our patients were developmental dislocation of the hip (DDH), clubfeet, metatarsus adductus, vertical talus, and congenital muscular torticollis. These deformations should be treated aggressively because children with prune belly syndrome may be expected to have a relatively normal life if their renal function is good. The one exception is that as infants these children are very susceptible to pulmonary infections because of their inability to cough. Therefore, treatment of the hip and other deformations should be delayed until the children are old enough to be able to clear pulmonary secretions easily.

Adolescent↗
PubMed2006

Sandifer Syndrome: a continuing problem of misdiagnosis.

BACKGROUND: Sandifer Syndrome is an uncommon clinical entity characterized by gastroesophageal reflux, irritability and abnormal movements of the body and contortions of the neck. The majority of paroxysmal cases, in particular, tend to show an association with epilepsy. METHODS: The clinical, laboratory and 6-month observation results of the four patients (two boys, two girls) have been presented. RESULTS: The 6-month prospective observation/treatment of four patients aged between 2 and 14 months (mean age, 6.5 +/- 5.2 months) with a diagnosis of Sandifer Syndrome has been investigated. Due to paroxysmal extensor jerks, two of the patients were misdiagnosed with infantile spasm and they were treated accordingly. In the clinical observations of the patients, abnormal neurobehavioral attacks (irritability, crying, head/eye version, torticollis, extensor spasm and dystonic posture) 5-10 times daily were observed. In two of the patients, motor growth retardation was observed, in one patient, bronchospasm attacks were observed, and in all the patients iron deficiency anemia was observed. The electroencephalograms of the patients which were taken during the routine, sleepless and paroxysmal behaviors were normal; the gastroesophageal scintigraphies were positive in the manner of reflux. Management of the infant with gastroesophageal reflux disease, in addition to nonpharmacological interventions pharmacologic therapy, including metoclopramide HCl and Fe (6 mg/kg per day, oral) was useful for the patients, and their paroxysmal attacks decreased dramatically (0-2 attacks/day). CONCLUSION: These findings suggest that infants or children with these atypical movements should be evaluated for Sandifer Syndrome. Expensive and comprehensive neurologic examination may be unnecessary. Early diagnosis permits prompt treatment and relief of the problem. Medical management is usually successful.

Anemia, Iron-Deficiency↗
PubMed1994

Infantile systemic hyalinosis in a black infant.

A black girl was born with flexion contractures and experienced pain on movement by 1 week of age. She subsequently developed perioral papules, gingival hyperplasia, perianal nodules, torticollis, diarrhea, rectal prolapse, and inability to open her mouth. Her skin became increasingly sclerodermatous, and velvety, hyperpigmented plaques arose over bony prominences. A skin biopsy specimen showed hyaline material in the papillary dermis with lack of elastic fibers. Ultrastructural examination revealed fibrillogranular material around fibroblasts and blood vessels. This child had the clinical, histologic, and ultrastructural features of infantile systemic hyalinosis. This disorder has not been described in a black infant. Previous case reports of infantile systemic hyalinosis are reviewed and unusual features of our case are discussed.

Connective Tissue Diseases↗
PubMed2005

A pilot study of botulinum toxin A for headache in cervical dystonia.

OBJECTIVE: To evaluate the prevalence of associated headache (HA) pain with craniocervical dystonia and the therapeutic effect of BoNT-A injections on the HA component when injected for cervical dystonia. BACKGROUND: HA associated with craniocervical dystonia is a recent formally codified entity, but has not been systematically studied. METHODS: We identified 44 subjects from three movement disorder clinics who presented with craniocervical dystonia and concurrent HA pain. The subjects were injected with botulinum toxin type A (BoNT-A) and prospectively evaluated with the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS), headache diaries, Headache Impact Test (HIT-6), and Migraine Disability Assessment Scale (MIDAS), along with HA pain anatomy and adverse events, at baseline, and at 4, 8, and 12 weeks post-injection. RESULTS: As expected, all aspects of the TWSTRS robustly improved. Headache diaries and the HIT-6 also improved at 4, 8, and 12 weeks post-injection. Sections of the MIDAS improved, and adverse events were minimal. CONCLUSION: BoNT-A safely improves headache associated with craniocervical dystonia when administered for the primary condition of craniocervical dystonia.

Adult↗
PubMed2005

Traumatic atlantoaxial rotatory subluxation.

Atlantoaxial rotatory subluxation should be considered in the presentation of traumatic torticollis. This case report discusses the characteristic radiographic findings and appropriate management.

Adolescent↗
PubMed1991

Evidence for variable gene expression in a large inbred kindred with autosomal recessive spondylocostal dysostosis.

Seven members of a large inbred kindred with autosomal recessive spondylocostal dysostosis were examined clinically, radiographically, and sonographically. The subjects were three adults, one adolescent, and three children under 3 years of age. One child was the offspring of a first cousin marriage which showed quasi-dominant inheritance. Six subjects had short stature owing to widespread vertebral dyssegmentation with variable reduction in rib number and rib fusion. One subject was of normal stature, had limited vertebral dyssegmentation, an extra rib, and no rib fusion. Five subjects showed the plagiocephaly-torticollis sequence. Four of the five male subjects had inguinal herniation on one or both sides. All subjects had normal renal ultrasonography. The youngest subject died of cardiopulmonary complications and is thought to represent one extreme in the expressivity of the gene in this kindred.

Adolescent↗
PubMed1986

Observations on the innervation of the sternomastoid muscle.

Nine out of fifteen cases having division of the spinal component of the accessory nerve and the upper cervical motor roots as treatment for spasmodic torticollis had residual movements in the sternomastoid of sufficient magnitude to make further surgery necessary before the muscle was effectively paralysed. These observations imply a more complex innervation for the muscle than is to be found in most anatomical texts.

Accessory Nerve↗
PubMed1996

Sex prevalence of focal dystonias.

The sex prevalence of idiopathic focal dystonia is reported from a data base review of all patients seen at the National Hospital of Neurology, Queen Square and King's College, London up to 1993. There was a higher prevalence of females to males in all categories of focal dystonia involving the craniocervical region. The female to male ratio for cranial dystonia was 1.92:1 (P < 0.01) and 1.6:1 (P < 0.001) for spasmodic torticollis. On the other hand, twice as many men than women had writer's cramp (M:F = 2.0:1, P < 0.01). At present, there is no clear explanation to account for this differences in the sex prevalence of different types of focal dystonia.

Dystonia↗
PubMed1998

What is the optimal dose of botulinum toxin A in the treatment of cervical dystonia? Results of a double blind, placebo controlled, dose ranging study using Dysport. German Dystonia Study Group.

OBJECTIVES: Botulinum toxin injections have become a first line therapeutic approach in cervical dystonia. Nevertheless, published dosing schedules, responder rates, and frequency of adverse events vary widely. The present prospective multicentre placebo controlled double blind dose ranging study was performed in a homogenous group of previously untreated patients with rotational torticollis to obtain objective data on dose-response relations. METHODS: Seventy five patients were randomly assigned to receive treatment with placebo or total doses of 250, 500, and 1000 Dysport units divided between one splenius capitis (0, 175, 350, 700 units) and the contralateral sternocleidomastoid (0, 75, 150, 300 units) muscle. Assessments were obtained at baseline and weeks 2, 4, and 8 after treatment and comprised a modified Tsui scale, a four point pain scale, a checklist of adverse events, global assessment of improvement, and a global rating taking into account efficacy and adverse events. At week 8 the need for retreatment was assessed and then the code was unblinded. For those still responding, there was an open follow up until retreatment to assess the duration of effect. RESULTS: Seventy nine per cent reported subjective improvement at one or more follow up visits. Decreases in the modified Tsui score were significant at week 4 for the 500 and 1000 unit groups versus placebo (p<0.05). Additionally positive dose-response relations were found for the degree of subjective improvement, duration of improvement, improvement on clinical global rating, and need for reinjection at eight weeks. A significant dose relation was also established for the number of adverse events overall and for the incidence of neck muscle weakness and voice changes. CONCLUSION: Magnitude and duration of improvement was greatest after injections of 1000 units Dysport; however, at the cost of significantly more adverse events. Therefore a lower starting dose of 500 units Dysport is recommended in patients with cervical dystonia, with upward titration at subsequent injection sessions if clinically necessary.

Adult↗
PubMed2002

Pallidal deep brain stimulation in patients with cervical dystonia and severe cervical dyskinesias with cervical myelopathy.

OBJECTIVES: Surgical treatment of complex cervical dystonia and of cervical dyskinesias associated with cervical myelopathy is challenging. In this prospective study, the long term effect of chronic pallidal stimulation in cervical dystonia and on combining the technique with spinal surgery in patients with severe cervical dyskinesias and secondary cervical myelopathy is described. METHODS: Eight patients with a history of chronic dystonia who did not achieve adequate benefit from medical treatment or botulinum toxin injection participated in the study. Five patients had complex cervical dystonia with tonic postures and phasic movements. Three patients had rapidly progressive cervical myelopathy secondary to severe cervical dyskinesias and dystonia in the context of a generalised movement disorder. Quadripolar electrodes were implanted in the posteroventral lateral globus pallidus internus with stereotactic CT and microelectrode guidance. In the three patients with secondary cervical myelopathy, spinal surgery was performed within a few weeks and included multilevel laminectomies and a four level cervical corporectomy with spinal stabilisation. RESULTS: Improvement of the movement disorder was noted early after pallidal surgery, but the full benefit could be appreciated only with a delay of several months during chronic stimulation. Three months after surgery, patients with cervical dystonia had improved by 38% in the severity score, by 54% in the disability score, and by 38% in the pain score of a modified version of the Toronto western spasmodic torticollis rating scale. At a mean follow up of 20 months, the severity score had improved by 63%, the disability score by 69%, and the pain score by 50% compared with preoperatively. There was also sustained amelioration of cervical dyskinesias in the three patients who underwent spinal surgery. Lead fractures occurred in two patients. The mean amplitude needed for chronic deep brain stimulation was 3.8 V at a mean pulse width of 210 micros, which is higher than that used for pallidal stimulation in Parkinson's disease. CONCLUSIONS: Chronic pallidal stimulation is effective for complex cervical dystonia and it is a useful adjunct in patients with cervical dyskinesias and secondary cervical myelopathy who undergo spinal surgery.

Adult↗