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PubMed2004

West Nile virus meningoencephalitis in a Suri alpaca and Suffolk ewe.

The first confirmed cases of West Nile virus (WNV) in the Western Hemisphere were reported in the state of New York in 1999. Since then, the virus has spread throughout the eastern and central United States and continues to extend westward. This report describes clinical signs and microscopic lesions associated with WNV infection in a Suffolk ewe and an alpaca, 2 species in which the disease has not been reported previously. In late August 2002, a 4.5-year-old female alpaca developed an acute onset of clinical signs characterized by torticollis, hyperesthesia, ataxia, recumbency, and altered mentation. The animal died 3.5 days after the onset of clinical signs. Microscopic examination of the brain revealed a mild to moderate, diffuse, lymphoplasmacytic meningoencephalitis. In early September 2002, a 3-year-old Suffolk ewe developed a rapidly progressive illness characterized by ataxia and convulsions. The apparent duration from onset of clinical signs until death was less than 8 hours. The ewe had a moderate, diffuse, lymphoplasmacytic meningoencephalitis with focal gliosis. Reverse transcription-polymerase chain reaction assays and immunohistochemistry on the brain were positive for WNV in both animals. These cases demonstrate that WNV is capable of causing sporadic, fatal, nonsuppurative meningoencephalitis in alpacas and sheep.

Animals↗
PubMed1998

Clinical, serologic, and histopathologic characterization of experimental Borna disease in ponies.

Borna disease was originally described as an equine neurologic syndrome over 200 years ago, although the infectious etiology of the disorder was unproven until the early 20th century. Borna disease virus (BDV) was finally isolated from horses dying of the disorder, and that virus has been used to experimentally reproduce Borna disease in several species of laboratory animals. However, BDV has never been inoculated back into horses to experimentally and etiologically confirm the classic clinical, pathologic, and serologic characteristics of the disease in that species. Three ponies were intracerebrally inoculated with different amounts of BDV and were evaluated clinically, serologically, and neurohistopathologically. All 3 animals developed the clinical signs characteristically described for naturally occurring Borna disease, including ataxia, torticollis, postural unawareness, rhythmic repetitive motor activities, muscle fasciculation, and cutaneous hyperesthesia and hypoesthesia over several body surfaces. Two ponies died after rapid onset of these signs 28-30 days postinoculation. The third animal made a nearly complete clinical recovery. Seroconversion occurred only after the onset of signs and to a marked degree only in the convalescent animal. Virus was recovered postmortem from 2 of the 3 ponies, and a BDV-specific nucleic acid sequence was detectable in all 3 animals using a reverse transcription-polymerase chain reaction procedure. Gross neural lesions were absent, but histopathologically there was generalized intense mononuclear perivascular cuffing, glial nodule formation, and astrocytosis in all 3 brains. Confirming a diagnosis of Borna disease is difficult and perhaps best accomplished using a combination of the clinical, serologic, and histopathologic indicators of this unusual disease supported by positive reverse transcription-polymerase chain reaction findings.

Animals↗
PubMed2006

An unusual foreign body migrating through time and tissues.

BACKGROUND: Beside infections, foreign body incidences are amongst the most frequently encountered pathologies in pediatric otolaryngology. While inhaled foreign bodies represent an acute emergency, symptoms of ingested foreign bodies sometimes appear with some delay. Typically fishbones tend to go unnoticed in a first examination and become symptomatic by fever, odynodysphagia and torticollis. Exceptionally, foreign bodies migrate and become manifest with a considerable delay. CASE REPORT: We present a case of a young girl who presented with an unusual foreign body which migrated through the cervical tissues causing repeated cervical tumescence's before being diagnosed. CONCLUSION: Repeated cervical abscesses or tumescence's in children or young patients should alert the treating physician to seek for an underlying pathology such as unnoticed foreign bodies or malformations (e.g. cysts). Further the scarce literature on these migrating foreign bodies is discussed.

Abscess↗
PubMed2007

Craniocervical junction malformation in a child with Oromandibular-limb hypogenesis-Möbius syndrome.

We report a male child with Oromandibular-limb hypogenesis (OMLH), the main features being bilateral sixth and seventh nerve palsies, limb anomalies and hypoplasia of the tongue. Additional features were shortness of the neck associated with torticollis. Radiographs of the cervical spine were non-contributory, but 3D computed tomography (CT) scanning of this area identified: a) congenital hypoplasia of the atlas; b) the simultaneous development of occiput-atlas malformation/developmental defect. To our knowledge, this is the first clinical report assessing the cervico-cranium malformation in a child with OMLH-Möbius syndrome.

Abnormalities, Multiple↗
PubMed2002

Simple phonic tic in multiple sclerosis.

Movement disorders occurring in association with multiple sclerosis (MS) are rare. Among them paroxysmal dystonia is the most common, although chorea, ballism, palatal myoclonia, spasmodic torticollis, writer's cramp and generalized dystonia have been reported. We describe a 34-year old woman with MS who developed simple phonic tic characterized by throat-clearing sounds. Magnetic resonance imaging showed demyelinating lesions involving the thalamus and basal ganglia. This is the first report of tic disorder occurring as a manifestation of MS.

Adult↗
PubMed1975

Motor effects of stimulation and ablation of the caudate nucleus of the monkey.

Electric stimulation of the caudate nucleus of the lightly anesthetized monkey was undertaken to demonstrate contralateral extremity movement. Contralateral forelimb flexion was observed in 75 percent of stimulations. The caudate nuclei were subsequently ablated, and 40 percent showed abnormal movements, mainly ipsilateral torticollis and ipsilateral eye deviation. The findings are consistent with previous studies using the caudate nucleus of the cat.

Animals↗
PubMed1985

Effect of alcohol on dystonia.

An intravenous infusion of a 250-ml, 10% ethanol solution decreased dystonic scores in five of seven patients with spasmodic torticollis, but had no effect in patients with Meige syndrome, tardive dystonia, or generalized torsion dystonia. Alcohol may temporarily decrease some forms of dystonia.

Aged↗
PubMed1991

Cervical dystonia: clinical findings and associated movement disorders.

We studied 300 patients, 61% women, with mean age 49.7 years and mean duration of dystonia 7.8 years, to determine the demographic and clinical characteristics of cervical dystonia (CD) and its relationships to other movement disorders. Torticollis was present in 82%, laterocollis in 42%, retrocollis in 29%, and anterocollis in 25%; however, the majority (66%) had a combination of these abnormal postures. Scoliosis was present in 39%, local pain reported by 68%, and 32% had evidence of secondary cervical radiculopathy. In addition to CD, 16% of patients had oral dystonia, 12% mandibular dystonia, 10% hand/arm dystonia, and 10% had blepharospasm. Tremor was noted in 71% of patients; head-neck tremor was present in 60%, and tremor in other body regions was present in 32%. A family history of a movement disorder was present in 44% of the CD patients. Tardive dystonia was the cause in 6%; 11% had posttraumatic dystonia. Anticholinergic drugs provided moderate improvement in 33% of patients, but local intramuscular botulinum toxin injections relieved CD, local pain, or both in over 90% of all treated patients.

Adolescent↗
PubMed1993

Retinal disease masquerading as spasmus nutans.

Two children with congenital stationary night blindness were originally diagnosed as having spasmus nutans. Both children had the typical features of spasmus nutans including asymmetric nystagmus, head shaking, and torticollis. The diagnosis of congenital stationary night blindness was established only after each child underwent electroretinography. The nystagmus associated with retinal disease can mimic many of the features of spasmus nutans. Children suspected of having spasmus nutans should undergo complete ophthalmologic examination and electroretinography if they are myopic.

Child, Preschool↗
PubMed1996

Botulinum toxin versus trihexyphenidyl in cervical dystonia: a prospective, randomized, double-blind controlled trial.

BACKGROUND: Botulinum toxin type A (BTA) is replacing trihexyphenidyl as the treatment of choice for idiopathic cervical dystonia (ICD), but there has never been a direct comparative study. METHODS: This trial compares the effectiveness of BTA with that of trihexyphenidyl in a prospective, randomized, double-blind design. Sixty-six consecutive patients with ICD were randomized to treatment with trihexyphenidyl tablets plus placebo injection or placebo tablets plus BTA injections. Tablets were administered daily according to a fixed schedule. Dysport or saline was injected under EMG guidance at study entry and again after 8 weeks. Patients were assessed for efficacy at baseline and after 12 weeks by different clinical rating scales. RESULTS: Sixty-four patients completed the study, 32 in each group. Mean dose of BTA was 292 mouse units (first session) and 262 mouse units (second session). Mean dose of trihexyphenidyl was 16.25 mg. The changes on the Disability section of the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS-Disability) (primary outcome), Tsui Scale, and the General Health Perception Subscale were significantly in favor of BTA. More patients treated with BTA had an improvement of at least three points on the TWSTRS-Disability (14 versus 6) and on the Tsui Scale (23 versus 12). Adverse effects were significantly less frequent in the BTA group. CONCLUSION: BTA is significantly more effective in the treatment of ICD, with less adverse effects.

Adult↗
PubMed1985

W. W. Keen: America's pioneer neurological surgeon.

The contributions of William Williams Keen to the development of neurological surgery were greater than those of any other American surgeon in the last quarter of the 19th century. His close association with S. Weir Mitchell, the father of American neurology, spanned more than 50 years. Keen's early publications included topics of neuroanatomical and neurophysiological interest. He was the first surgeon in America to remove a meningeal tumor and effect a cure and the first in America to stimulate the brain at operation. He made contributions to the surgical treatment of hydrocephalus, craniostenosis, torticollis, trigeminal neuralgia, and nervous system trauma. Keen's surgical texts provided an important foundation for Cushing, Frazier, and those that followed. Showered with honors as America's dean of surgery, Keen lived to see the many specialties evolve.

History, 19th Century↗
PubMed2004

Primary dystonia is more responsive than secondary dystonia to pallidal interventions: outcome after pallidotomy or pallidal deep brain stimulation.

OBJECTIVE: The response of patients with dystonia to pallidal procedures is not well understood. In this study, we assessed the postoperative outcome of patients with primary and secondary dystonia undergoing pallidotomy or pallidal deep brain stimulation. METHODS: Fifteen patients with dystonia had pallidal surgery (lesions or deep brain stimulation). These included nine patients with primary dystonia (generalized and cervical dystonias) and six with secondary dystonia (generalized, segmental, and hemidystonias). There were nine male patients and six female patients. The mean age at onset was 21 years for primary dystonia and 18 years for secondary dystonia. The primary outcome measure was a Global Outcome Scale score for dystonia at 6 months after surgery. Other outcome measures were the Burke-Fahn-Marsden Dystonia Rating Scale and Toronto Western Spasmodic Torticollis Rating Scale scores. RESULTS: The mean Global Outcome Scale score at 6 months for patients with primary dystonia was 3 (improvement in both movement disorder and function). In contrast, patients with secondary dystonia had a mean score of 0.83 (mild or no improvement in movement disorder with no functional improvement). All patients with primary dystonia had normal brains by magnetic resonance imaging, whereas five of six patients with secondary dystonia had basal ganglia abnormalities on their magnetic resonance imaging scans. CONCLUSION: This study indicates that primary dystonia responds much better than secondary dystonia to pallidal procedures. We could not distinguish a difference in efficacy between pallidotomy and pallidal deep brain stimulation. The presence of basal ganglia abnormalities on the preoperative magnetic resonance imaging scan is an indicator of a lesser response to pallidal interventions for dystonia.

Adolescent↗
PubMed2003

Neuropathological observation of rabbits (Oryctolagus cuniculus) affected with raccoon roundworm (Baylisascaris procyonis) larva migrans in Japan.

Larvae of the raccoon roundworm, Baylisascaris procyonis (B. procyonis) are a known cause of cerebrospinal larva migrans in animals and humans. The present paper described details of the central nervous lesion in the rabbits (Oryctolagus cuniculus) affected with B. procyonis larva migrans in Japan. Clinically affected animals showed neurological signs including circling, torticollis, tremor of head, or ataxic gait. The most characteristic pathological alterations were large malacic lesions associated with an activated astroglial proliferation which was seen at the corpus medullare in the cerebellum including the cerebellar peduncle. Moreover, focal malacic lesions with perivascular cuffing and infiltration by lymphocytes and heterophiles were scattered everywhere throughout the brain. In these lesions or normal-appearing areas away from obvious lesions, ascarid larvae, about a maximum 65-75 micro m in diameter, were recognized. Other prominent features were minute lesions (we call them migration tract-like lesions) composed of lymphocytes, hemosiderin-laden macrophages and reactive astrocytes scattering throughout the cerebrum. In this study, we demonstrated ascarid larvae in only eight out of 23 animals diagnosed as B. procyonis larva migrans. Since it is not always possible to detect the larvae, the possibility of B. procyonis larva migrans must be given serious consideration to the characteristic lesions described above.

Animals↗
PubMed1983

The idiopathic dystonias. A note on their orthopaedic presentation.

Eight patients suffering from various forms of idiopathic dystonia are described whose initial referral was for an orthopaedic opinion. The diagnoses of these patients, who were seen over a two-year period, comprised dystonia musculorum deformans, dystonia of the foot, spasmodic torticollis and occupational cramps. Although various musculoskeletal sequelae often occur, the primary underlying neurological cause of these unusual conditions is emphasised.

Adult↗
PubMed1989

Diagnosis of acute atlanto-axial rotatory fixation.

We report three cases of atlanto-axial rotatory fixation in adults. Early diagnosis was made by clinical tests showing restricted head rotation in maximal neck flexion and asymmetry of the transverse processes of the atlas, confirmed by cineradiography. Early treatment by traction may obviate long-term problems of torticollis and instability.

Acute Disease↗
PubMed2000

Long-term developmental outcomes in patients with deformational plagiocephaly.

OBJECTIVES: To determine whether there was an increased rate of later developmental delay in school-aged children who presented as infants with deformational plagiocephaly without obvious signs of delay at the time of initial evaluation. METHODS: A retrospective medical record review of 254 patients evaluated at the Craniofacial Center of the Children's Hospital and Regional Medical Center in Seattle, Washington, from 1980 through 1991 was completed. Consenting patient families were interviewed via telephone to determine what, if any, special medical or educational problems had occurred for the children who had had plagiocephaly in infancy or their siblings with normal head shapes. RESULTS: A total of 181 families from the medical record review could be notified about the study and 63 families agreed to participate in a telephone interview. The sample of participants for the telephone interview was random to and representative of the group as a whole. The families reported that 25 of the 63 children (39.7%) with persistent deformational plagiocephaly had received special help in primary school including: special education assistance, physical therapy, occupational therapy, speech therapy generally through an Individual Education Plan. Only 7 of 91 siblings (7.7%), serving as controls, required similar services (chi(2) = 21.24). Delays could not be specifically anticipated at the time of the diagnosis of deformational plagiocephaly from any simple set of factors including treatment with helmet therapy, although effected males with reported uterine constraint were at the highest risk for subsequent school problems. CONCLUSIONS: Infants with deformational plagiocephaly comprise a high-risk group for developmental difficulties presenting as subtle problems of cerebral dysfunction during the school-age years. There is a need for additional research on the long-term developmental problems in infants with deformational plagiocephaly. plagiocephaly, facial asymmetry, torticollis, developmental delay.

Developmental Disabilities↗
PubMed2003

Retropharyngeal abscess in children: clinical presentation, utility of imaging, and current management.

OBJECTIVE: We sought to describe the clinical presentation of patients with retropharyngeal abscess (RPA), utility of imaging studies, and implications on management. METHODS: A retrospective chart review was performed at a tertiary-care, pediatric hospital with cases identified by a discharge diagnosis of RPA; posttraumatic RPA cases were excluded. Patients without confirmatory radiographic findings, fluoroscopy, or computed tomography (CT) were excluded. RESULTS: Sixty-four cases involving 64 patients were studied. The median age of the patients was 36 months; 48 (75%) of the 64 patients were younger than 5 years. The most common chief complaints were neck pain (38%), fever (17%), sore throat (17%), neck mass (16%), and respiratory distress or stridor (5%). In 29 children (45%), it was noted that there was limitation of neck extension, in 23 (36.5%) torticollis, and in 8 (12.5%) limitation of neck flexion. The physical examination revealed stridor with wheezing in only 1 patient (1.5%) and wheezing in 1 other (1.5%). Twenty-seven patients (42%) underwent surgery; 37 (58%) were treated with antibiotics only. Performance of a surgical procedure was significantly associated with CT scan findings. Ten (37%) of 27 patients with defined abscess on CT scan were treated with antibiotics alone. There were no treatment failures in either the antibiotic-only group or the antibiotics-plus-surgery group. CONCLUSIONS: Children with RPA present with limitation of neck movement, especially difficulty extending their neck to look up. They rarely present with respiratory distress or stridor. CT scan is useful to distinguish patients with RPA from those with retropharyngeal cellulitis. Most patients with retropharyngeal cellulitis and some with RPA can be treated successfully without surgery.

Adolescent↗
PubMed2006

Cervical spine and temporomandibular joint arthritis in a child with Kawasaki disease.

Severe neck pain, stiffness, and torticollis in a child with Kawasaki disease commonly represent aseptic meningitis or lymphadenitis. Here we present an unusual case of severe cervical spine and bilateral temporomandibular joint arthritis in a 5-year-old boy with a relapse of Kawasaki disease and coronary artery ectasia. The patient had a favorable response to a second course of intravenous immunoglobulin G and indomethacin therapy, with complete resolution of his symptoms.

Arthritis↗