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PubMed1998

Current management of pediatric atlantoaxial rotatory subluxation.

STUDY DESIGN: A retrospective clinical review of 20 children seen during a 7-year period who had atlantoaxial rotatory subluxation. OBJECTIVE: To define the effectiveness of imaging and treatment measures and to identify risk factors for recurrence, the series was reviewed to analyze cause, management, and outcome. SUMMARY OF BACKGROUND DATA: Rotatory subluxation of the atlantoaxial complex remains a poorly understood entity. Despite many reports in the literature, there is no consensus about which imaging studies should be used for diagnosis and which patients benefit from collar immobilization, traction, or surgical fusion. METHODS: Between August 1990 and April 1997, 20 children with atlantoaxial rotatory subluxation were treated. Fourteen patients (70%) were girls and six (30%) were boys (mean age, 6.4 years). All patients had torticollis and neck pain with decreased cervical motion for a mean of 11.2 days before diagnosis. Seven patients (35%) had a history of pharyngitis or otitis media, four (20%) had recently undergone head or neck surgery, and four (20%) had sustained a traumatic injury; in five patients (25%), no clear cause was determined. All patients were neurologically intact and underwent plain cervical radiographs and dynamic cervical computed tomography to document atlantoaxial rotatory subluxation. Patients were then treated with a rigid cervical collar and anti-inflammatory agents (n = 5) or with cervical traction followed by immobilization (n = 15). RESULTS: In four of the five patients in collars, reduction occurred spontaneously, whereas the fifth required cervical traction and eventual fusion for recurrence. In the 16 patients treated with traction (median, 1.8 kg), the normal atlantoaxial alignment was restored in 15 patients (94%) within a mean of 4 days. Of the 20 patients treated overall, conservative management failed in 6 (30%), and they required posterior fusion because of recurrence of the atlantoaxial rotatory subluxation or unsuccessful reduction. The major factor predicting the failure of conservative management was the duration of subluxation before initial reduction. Patients with long-standing subluxation were more likely to experience recurrence and require surgery. There were no complications noted. At follow-up, all patients who were treated conservatively remained neurologically intact with a normal atlantoaxial relation. All patients who underwent surgery remained neurologically intact and had radiographic documentation of fusion. CONCLUSION: Optimal management of atlantoaxial rotatory subluxation entails early diagnosis with plain cervical radiographs and dynamic computed tomography. Closed reduction with cervical traction followed by rigid immobilization accomplished reduction in 15 of 16 patients (94%) and was curative in 10 of 16 patients (63%). Although reduction was achieved more rapidly and effectively with traction than with a collar, there may be a role for simple immobilization without reduction in patients with a short duration of symptoms. There does not appear to be a correlation between cause of atlantoaxial rotatory subluxation, age, or sex and the likelihood of recurrence.

Atlanto-Axial Joint↗
PubMed2000

Atlantoaxial rotatory subluxation in patients with Marfan syndrome. A report of three cases.

STUDY DESIGN: This is a retrospective case series of three patients, ages 9 1/2, 13, and 20 years old, with Marfan syndrome treated for atlantoaxial rotatory subluxation. In the first two cases, acute torticollis was noted postoperatively, following pectus excavatum repair. The diagnosis was made in the third patient after she presented to the emergency room with a week-long history of unresolved neck pain following minor trauma. OBJECTIVE: To report and discuss the courses and clinical sequelae of atlantoaxial subluxation in patients with Marfan syndrome. SUMMARY OF BACKGROUND DATA: Radiographic analysis of patients with Marfan syndrome has shown that increased atlantoaxial translation, larger odontoid height, and basilar impression are more prevalent in this population compared to age-matched controls. Despite these findings, there are sparse data on injuries secondary to cervical spine instability or abnormalities in this population. To the authors' best knowledge, no report of atlantoaxial rotatory subluxation in patients with Marfan syndrome exists in the literature. METHODS: Case records of rotatory instability of the atlanto-axial level were reviewed and are presented in the following report. RESULTS: The first two patients described in this report were noted to have "cock robin" posturing of their necks following pectus excavatum repairs. The first patient's subluxation was partially reduced with halo traction, and he subsequently underwent posterior spinal fusion of C1-C2 with internal fixation. The patient was well aligned postoperatively, and had no neurologic deficits. The second patient's subluxation reduced after 20 days of halter and traction; he was immobilized in a collar following discharge and reduction was maintained. The third patient's subluxation failed to reduce with halo traction; further imaging studies revealed odontoid prominence in the foramen magnum. She underwent posterior spinal fusion, occiput to C3, with satisfactory result. CONCLUSIONS: The cervical bony and ligamentous abnormalities seen in patients with Marfan syndrome may slightly increase their risk for atlantoaxial rotatory instability. Special attention to intubation and positioning, both intraoperatively and postoperatively, may be necessary in patients with Marfan syndrome. Additionally, rotatory subluxation should be included in the differential diagnosis for Marfan patients with neck pain after injury.

Adolescent↗
PubMed2003

Lateral atlantoaxial joint arthrography in atlantoaxial rotatory fixation.

There has been much debate about the pathogenesis of atlantoaxial rotatory fixation. Intraarticular abnormality has not been well documented thus far. This is the first case of chronic atlantoaxial rotatory fixation in which atlantoaxial joints were examined by consecutive arthrography. A 7-year-old girl was diagnosed with atlantoaxial rotatory fixation on a three-dimensional CT scan. Arthrography of the lateral atlantoaxial joints indicated a rupture of the joint capsule on the dislocated side at first, followed by a successful repair after 5 weeks' immobilization with a halo apparatus. Because torticollis recurred after taking off the halo vest, we performed surgery in which severe adhesion of the cartilage surface of facet joint was noted on the undislocated side and release of the adhesion was needed to mobilize the atlas. Atlantoaxial arthrodesis by screw fixation facilitated a solid fusion of the segment.

Arthrography↗
PubMed2004

Herniation of a calcified cervical disc into the foramen transversarium in an 8-year-old child.

STUDY DESIGN: Herniation of a calcified C3-C4 disc into the left foramen transversarium in a child is reported. OBJECTIVE: To discuss the natural history and management of a calcified disc herniation into the foramen transversarium. SUMMARY OF BACKGROUND DATA: Cervical disc calcification in children usually follows a benign course. Herniation of the calcified disc into the spinal canal has already been described. However, herniation into the foramen transversarium has never been reported. METHODS: An 8-year-old girl presented with progressive neck pain and torticollis. Her neurologic examination was normal. She was treated using a head halter traction, analgesics, and muscle relaxants for 3 days, followed by the use of a soft cervical collar for 2 weeks. RESULTS: Computed tomography scan showed a calcified C3-C4 disc with herniation into the C3 left foramen transversarium. Her symptoms subsided and she rapidly regained full range of motion of her neck after 3 days of conservative treatment. Magnetic resonance angiography done after 2 months did not show any residual compression of the left vertebral artery. After 3 months, the herniation had completely disappeared, whereas only a small central calcification remained in the C3-C4 disc space. CONCLUSION: The natural history of cervical disc calcification is usually benign. A computed tomography scan can be performed for patients in whom a calcified disc herniation is suspected on the plain films. In this case of herniation into the foramen transversarium, magnetic resonance angiography was useful to evaluate the integrity of the vertebral arteries. Spontaneous resolution of the herniated disc and return to normal function can be expected with conservative treatment.

Calcinosis↗
PubMed2005

Hemivertebra resection in the cervical spine.

STUDY DESIGN: Retrospective study with clinical and radiologic evaluation of hemivertebra resection in children with congenital cervical scoliosis. OBJECTIVE: Assessment of hemivertebra resection in the treatment of congenital cervical scoliosis. SUMMARY OF BACKGROUND DATA: To our knowledge, this is the first report on hemivertebra resection in the midcervical spine. METHODS: Three patients with torticollis due to a cervical hemivertebra were operated on by hemivertebra resection and fusion of the adjacent vertebrae. Resection was performed by a posterior-anterior (-posterior) approach. Mean age at time of surgery was 9 years 3 months. They were retrospectively studied with a mean follow-up of 4.8 years. RESULTS: Mean segmental Cobb angle at the hemivertebra was 29 degrees before surgery, 5 degrees after surgery, and 6 degrees at latest follow-up. Head tilt improved from 17 degrees before surgery to 1 degrees after surgery, and 3 degrees at latest follow-up. Bony fusion was achieved in all cases. There was weakness of the left deltoid muscle in 1 case due to C5 root compression by a facet screw, which resolved completely after revision with change of the screw. CONCLUSIONS: Resection of a cervical hemivertebra may be an option in the treatment of congenital cervical scoliosis. A good correction of the local deformity and a complete correction of head tilt are achieved. By early operation in young children, the development of severe secondary deformities in the upper thoracic spine can be avoided.

Adolescent↗
PubMed2004

Update on botulinum toxin use in facial plastic and head and neck surgery.

PURPOSE OF REVIEW: The purpose of this review is to discuss current trends in the medicinal use of botulinum toxin in head and neck and facial plastic surgery. The basic science of botulinum toxin is presented along with a comparison of the subtypes currently available on the market. Site-specific applications of botulinum toxin in the head and neck region are presented. RECENT FINDINGS: The use of botulinum toxin continues to expand, both in volume and in number of applications. The main application of botulinum toxin in facial plastic surgery is in the effacement of dynamic or hyperkinetic facial lines. The granting of US Food and Drug Administration approval for the use of Botulinum Toxin type A in the treatment of glabella lines marks a major milestone for the more widespread usage of this product in cosmetic settings. Additional cosmetic applications include crow's feet, marionette lines, and platysma banding. Noncosmetic applications in the head and neck include dystonias (including torticollis), facial and generalized muscle spasms, migraine headaches, hyperhidrosis, spasmodic dysphonia, sialorrhea, gustatory sweating, and involuntary movement disorders. SUMMARY: The use of botulinum toxin in the treatment of hyperkinetic conditions and disorders of excessive salivation is well established and enjoys an excellent safety profile. The cosmetic use of botulinum toxin continues to expand. The chronic use of botulinum toxin specifically for cosmetic purposes is worthy of additional study.

Botulinum Toxins↗
PubMedDate not supplied

Tetrabenazine treatment in movement disorders.

Tetrabenazine (TBZ) is a catecholamine depletor used for the treatment of a variety of movement disorders. The purpose of this study was to assess the efficacy of TBZ in a retrospective chart review in 3 tertiary care movement disorders centers over long-term treatment. Of 150 patients to whom TBZ was prescribed, 118 were followed up and assessed using the Clinical Global Impression of Change (CGIC), (-3 to +3), a composite grade from a patient and caregiver scale over variable periods. The patients had a variety of hyperkinetic movement disorders including dystonia (generalized and focal: axial, Meige syndrome, torticollis, blepharospasm, bruxism), Huntington disease (HD) or other choreas, tardive dyskinesia (TD) or akathisia, and Tourette syndrome. Mean patient age was 48.8 +/- 18.7 years; 48 were men (40.7%) with a mean disease duration of 93 months. The mean follow-up time was 22 months and the mean TBZ dose was 76.2 +/- 22.5 mg/d (median 75 mg, range 25-175 mg/d). The mean CGIC score was +1 (mild improvement). The group of patients who scored +3 on the CGIC (very good improvement) represented 18.6% (n = 22) of all patients. They had HD or other types of chorea 7.6% (n = 9), facial dystonia/dyskinesia (n = 7, 5.9%), 1 with TD, 2 with trunk dystonia, 2 with Tourette syndrome, and 1 with tardive akathisia. This group had the longest treatment duration and received a mean TBZ dose of 70.5 mg/d (median 75 mg/d) for a mean of 25.4 +/- 21.3 months. The report concludes that TBZ is a moderately effective treatment of a large variety of hyperkinetic movement disorders, with excellent effects in a subgroup with chorea and facial dystonia/dyskinesias.

Adult↗
PubMedDate not supplied

Management of cerebral palsy with botulinum-A toxin: preliminary investigation.

Use of intramuscular botulinum-A toxin (Botox) to produce neuromuscular blockade has been effective in treating certain ocular and facial muscular imbalances as well as spasmodic torticollis. In this preliminary open study, the effectiveness of intramuscularly injected Botox on the muscular imbalances of cerebral palsy was assessed in 27 pediatric patients. Each patient had "dynamic deformities" unresponsive to other treatment, and operation was the only other realistic alternative. The dose of Botox was calculated on a unit/body weight basis. In ambulatory patients, clinical changes in gait were assessed by a physician's rating scale. Reduction in spasticity became apparent in 12-72 h after injection; the effect of Botox after target threshold was reached lasted 3-6 months. No major side effects occurred. Botox may prove a useful adjuvant in conservative management of the spasticity of cerebral palsy. Successful management with these injections may allow delay of surgical intervention until the child is older and at less risk of possible complications, including the need for repeated surgical procedures.

Adolescent↗
PubMed1968

Translocation of sugar and tritiated water in squash plants.

When (14)C-sugar and THO were simultaneously introduced through a cut side vein or flap of a squash leaf (Cucurbita melopepo, Bailey cv. torticollis) concurrent translocation of (14)C-sugars, T-photosynthates and THO with parallel, almost flat, gradients was observed in the petiole for periods of 1 to 3 hr. Parallel translocation gradients were not observed when (14)C was introduced as (14)CO(2) and T by painting a leaf with THO. Autoradiography of frozen sections to locate the tissues in which THO was moving was unsuccessful. Steam-girdling blocked the movement of (14)C and T when (14)C-glucose and THO were introduced simultaneously by the flap-feeding technique. If THO moved as liquid water in the phloem along with the (14)C-sugars, as blockage by steam girdling suggests, then solution flow of sugar cannot be excluded as a mechanism of translocation.

Journal Article↗
PubMed1973

Effect of petiole anoxia on Phloem transport in squash.

Translocation of (14)C-labeled assimilates in Early Prolific Straightneck squash (Cucurbita melopepo torticollis Bailey) through a 15-centimeter oxygen-deficient zone of the petiole was studied as a function of varying periods of anaerobiosis (N(2) atmosphere). Initiation of anaerobic conditions caused an immediate and rapid decline in translocation to about 35 to 45% of the pretreatment rate within 30 to 40 minutes. This inhibition response (first inhibition response) was transient, however, and full recovery to the pretreatment rate occurred during the ensuing 60 to 90 minutes. Following this adaptation response to anaerobic conditions, translocation continued unimpaired for extended periods of time, approaching, and in some cases exceeding, 24 hours. The second inhibition response was permanent and could not be reversed by supplying air during a subsequent 20-hour period.

Journal Article↗
PubMed1996

Arthrogryposis multiplex congenita, AD 1156.

A case of arthrogryposis multiplex congenita in an eight-year-old girl was recounted by Thomas of Monmouth in a mid twelfth-century English hagiographic narrative, The Life and Miracles of St William of Norwich. The child had deformities of both hands and both feet at birth, and she developed torticollis and probably had some degree of hypotonia. She needed total care, her family took her to the tomb of St William in the cathedral at Norwich. This visit produced some sort of improvement in her health. Her parents, seeking a miracle, were satisfied that one had occurred.

Arthrogryposis↗
PubMed1987

Spinal cord stimulation and motor disorders.

Spinal cord stimulation has been investigated by us during the past 10 years in the treatment of various disorders of the motor system. The effectiveness was studied in 735 cases, including cerebral palsy (212), dystonia (129), torticollis (66), spinal cord injury (169), and degenerative diseases (159). Our results indicate that in properly selected patients, stimulating the spinal neural axis is therapeutically effective in the majority of the cases treated. Our data demonstrate that the level stimulated, the pattern and the polarity of the applied field, and the frequency of the stimulation are critical to achieve a satisfactory therapeutic result and must be individualized in each patient.

Adolescent↗
PubMed1982

Essential tremor and buccolinguofacial dyskinesias.

Essential tremor (ET) has been described in association with major dyskinesias, such as dystonia musculorum deformans, torticollis, and dystonic writer's cramp. In a series of 104 patients affected by ET, the incidence of buccolinguofacial dyskinesias (BLFD) has been investigated, including a comparison with another dyskinesia involving the facial muscles--mimic muscle tremor. The association of ET and BLFD may be casual; however, on the other hand, it might suggest the existence of a subgroup of patients suffering from a non-specific sign of motor control impairment, such as ET, in which other non-specific disorders of voluntary movement, such as BLFD, can gradually develop, particularly late in life.

Adolescent↗
PubMed1981

Superficial fibrous tumors in childhood.

A review is made of superficial fibrous tumors of childhood and differences between the pattern of fibrous tumor proliferation in children and adults are discussed. The fibrous tumors that occur specifically in childhood and infancy include recurring digital fibrous tumor, juvenile aponeurotic fibroma (cartilage analogue of fibromatosis), juvenile hyalin fibromatosis, fibrous hamartoma of infancy, congenital fibromatosis and fibromatosis colli (Torticollis). Palmoplantar fibromatosis and fibrosarcoma may appear in all ages. Dupuytren contracture, however, is not usually observed early in life. Similarly, the histiocytomas and dermatofibromas which are the most common fibrous tumors of adults are extremely rare in childhood.

Child↗
PubMed1976

Congenital deformities in two Clydesdale foals.

The deformities observed in 2 Clydesdale foals are described. Both had abnormal joint positions in the forelimbs and discrepancies in the symmetry of the vertebral column. The changes were only mild in one case but extreme in the other where it was accompanied by torticollis, scoliosis and vertebral fusion. A comparison is made with deformities described in the contracted foal syndrome and some of the developmental implications discussed.

Abnormalities, Multiple↗
PubMed1988

The effect of detomidine (Domosedan) on the maintenance of equine pregnancy and foetal development: ten cases.

Detomidine was administered throughout 10 pregnancies in eight mares. An intravenous injection of 20 micrograms/kg body weight was given weekly from Day 14 to Day 60 of gestation and thereafter every four weeks until parturition. One mare suffered torsion of the large colon and was destroyed on Day 86; the foetus was normally developed. A further mare aborted at 167 days. The remaining eight pregnancies continued to full term. One foal was delivered by caesarean section because of torticollis and, of the seven foals born spontaneously, one had bilateral upward patellar fixation at one month old. Therefore, although only six of the 10 foals developed normally, the other four cases showed no pathological similarities to suggest a common cause. Although these data were based on a small number of mares, they did not suggest that the repeated administration of detomidine had specific adverse effects on the pregnancies.

Animals↗
PubMed1966

Analysis of some central actions of nicotine injected into the cerebral ventricles of cats.

1. In unanaesthetized cats intraventricular injections of 2.5-10 mug nicotine produced blinking, narrowing of the palpebral fissures, retching, vomiting and asynchronous twitching of the ears. With larger doses (30-100 mug) the ear twitching was interrupted for a short time by laying back of the ears. Respiration first became laboured and deep, then rapid and shallow following which panting occurred. There was salivation, loud calling, micturition and defaecation. With still larger doses (300-1000 mug) there was torticollis, ataxia and blind charging sometimes followed by a clonic-tonic convulsion.2. In cats anaesthetized with chloralose only some of these effects followed the intraventricular injection of nicotine, i.e. the ear response, respiratory changes and salivation. In addition, the pinna reflex was facilitated.3. The ear response and the facilitation of the pinna reflex did not occur on perfusion of nicotine from a lateral ventricle to aqueduct. They result from an action on superficial structures in the cervical cord between C1 and C2. Applied to this region of the cord, nicotine produced the ear response within 10-60 sec, sometimes in concentrations as low as 1/100,000. Applied below C2, nicotine was ineffective.4. Transecting the cord below C2 or cutting the dorsal and ventral roots of C1, C2 and C3 bilaterally did not affect the ear response produced by topical application or by intraventricular injection of nicotine. Transection of the cord above C1 abolished it.5. Hexamethonium applied to the cervical cord between C1 and C2 inhibited the ear response and the facilitation of the pinna reflex whether produced by nicotine applied topically or injected intraventricularly.6. The salivation and the respiratory changes produced by intraventricular injections of nicotine did not occur when nicotine was perfused from a lateral ventricle to the aqueduct. They result from an action of the nicotine on structures situated superficially in the brain stem. Nicotine had no sialogogue or respiratory effect when applied to the region of the cord at which it produced an ear response, but perfused through the subarachnoid space from interpeduncular fossa to cisterna magna or injected into the subarachnoid space alongside the brain stem, it produced these effects.7. Hexamethonium perfused from interpeduncular fossa to cisterna magna inhibited the salivary secretion as well as the respiratory changes produced by nicotine similarly applied.8. The efferent pathway for the salivation is parasympathetic since it no longer occurred after cutting the chorda-lingual nerve or after intravenous atropine.9. Intravenous injections of nicotine also produced the ear response with facilitation of the pinna reflex, salivation and hyperventilation in the anaesthetized cat, but only the ear response and facilitation of the pinna reflex are central effects. The salivation and hyperventilation following intravenous injection are due to peripheral actions of nicotine.

Journal Article↗