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PubMed1997

Diagnosis and management of posterior plagiocephaly.

OBJECTIVE: The management of infants with posterior plagiocephaly has been controversial both because of widely differing estimates in the literature of the relative frequencies of true lambdoidal synostosis vs positional molding and because of divergent approaches to treating this problem in different institutions. Based on our experience, we hypothesized that the vast majority of children with posterior plagiocephaly did not have true synostosis and that the cosmetic impairment in such patients could be effectively treated with nonsurgical modalities. METHODS: Between 1992 and 1995, we prospectively applied in 71 infants a consistent management philosophy for these malformations that has incorporated a detailed evaluation of sutural anatomy as the basis for a physiologic approach to treatment. This approach has been directed at distinguishing true synostosis from deformational plagiocephaly and at avoiding surgery for patients with deformational abnormalities by using a combination of nonsurgical modalities to restore normal cranial growth dynamics. All children first underwent skull radiographs to determine whether the lambdoidal sutures were patent. In equivocal cases, computed tomography was also performed. Patients without true synostosis were enrolled on a course of positional therapy. In patients that did not improve after 2 to 3 months, a custom-fitted orthoplastic molding helmet was applied to facilitate passive skull recontouring. RESULTS: Forty children had patent sutures based on skull radiographs, and 29 others, in whom the radiographs were equivocal, had open sutures based on computed tomography, thus establishing the diagnosis of deformational plagiocephaly in 69. Predisposing factors for this deformity included a strong positioning preference during early infancy (n = 67), torticollis (n = 10), prematurity (n = 6), and developmental delay (n = 2). Only two patients had true lambdoidal synostosis; in each case, this was associated with synostosis of the posterior sagittal suture and was managed effectively with cranial reconstructive surgery. Thirty-five patients with deformational plagiocephaly had a dramatic improvement in their cranial contour with positional therapy alone; 34 patients failed to improve and were treated with molding helmets. All but five children, each of whom was more than 6 months old at initial intervention (P < .025), developed a normal or nearly normal head shape with these measures. CONCLUSION: The vast majority of children with posterior plagiocephaly do not have true synostosis and can be effectively managed by nonsurgical means. The impact of positional preference on the development of this process is discussed.

Craniofacial Abnormalities↗

PubMed2004

Internal carotid artery aneurysm with life-threatening hemorrhages in a pediatric patient: endovascular treatment options.

PURPOSE: To describe the management of a pediatric patient with recurrent, life-threatening nasopharyngeal hemorrhages due to a pseudoaneurysm of the internal carotid artery (ICA), most likely caused by deep neck infection following peritonsillar abscess. CASE REPORT: An 11-year-old boy presented with lymphadenopathy of the neck, torticollis, and difficulty swallowing that had persisted for some weeks. After sneezing, the patient suffered massive recurrent nasopharyngeal hemorrhages leading to anemia. Computed tomography identified a 2 x 2-cm hematoma in the left parapharyngeal space; angiography revealed a saccular aneurysm (14.1 x 8.2 mm) of the extracranial ICA adjacent to the skull base, without ongoing hemorrhage. A bare Wallstent was initially implanted, but total exclusion was achieved only after secondary placement of a Symbiot covered stent within the bare stent. CONCLUSIONS: This case demonstrates the utility of an endovascular approach to an extracranial ICA pseudoaneurysm in a pediatric patient.

Aneurysm, False↗

PubMed1985

[Treatment of Meige disease with a GABA receptor agonist].

The spontaneous occurrnce of blepharospasm and dystonic movements in face muscles, particularly those of the perioral and mandibular regions, has been named Meige's disease. Other dystonic features as spasmodic torticollis, dysphagia, spasmodic dysphonia and segmental dystonia of the limbs may, eventually, be present in the same patient. There is very little knowledge about the pathology of this disease. Many hypotheses concerning the pathophysiology of this entity have been put forward, most of them correlating the clinical response to several drugs with known action on the neurotransmitter system of the brain. There are some evidences that it may exist a dopaminergic preponderance in the disease. In the nigro-striatal pathway, one of the retrograde loops in the feed-back control of dopamine synthesis by nigral neurons is dependent on GABA. Increasing GABA activity through GABA agonists that cross the blood-brain barrier could result in a decreased dopaminergic action in the nigro-striatal pathway and, thus, ameliorate the dystonic symptoms which might have been produced by its increased function. We have used baclofen, a GABA-agonist drug, to treat five patients with Meige's disease, in a single-blinded trial. These were four females and one male, with age ranging from 50 to 63 years. The drug was started at 20mg/day, being increased by 10mg each three days reaching a maximum dose of 70mg/day. One of the patients showed marked improvement of blepharospasm and orofacial dystonia and a second patient had a moderate improvement in the same symptoms. Another patient showed moderate improvement of limb dystonia, but had no benefit in the facial movements.(ABSTRACT TRUNCATED AT 250 WORDS)

Baclofen↗

PubMed1992

Idiopathic dystonia. Clinical profile of 76 Brazilian patients.

Dystonia may be classified by age of onset (childhood, adolescence, adult onset), body distribution of the abnormal movements (focal, segmental, unilateral, multifocal and generalized) and etiology (idiopathic and symptomatic). We studied 76 patients with idiopathic dystonia among 122 cases of dystonic syndrome (62.3% of the total). There were 48 female and 28 male patients. Adult-onset focal dystonia was the most frequent feature (37 patients). The onset of generalized dystonia was more frequently seen under the age of 20, whereas focal and segmental dystonia usually started over this age. Postural tremor of the hands was observed in 19.7% of the patients. Spasmodic torticollis was the most prevalent form of dystonia overall. Except for writer's cramp, which occurred more frequently in males, and generalized dystonia, which was equally divided between sexes, all other forms were more frequent in females. Our data suggest that differences in racial origin, social and economical status and environmental factors do not account for a different manifestation in dystonia pattern.

Adolescent↗

PubMed2000

Pharmacotherapy with botulinum toxin: harnessing nature's most potent neurotoxin.

Botulinum toxin (BTX), a potent biologic neurotoxin, commonly is associated with lethal outbreaks of food poisoning; however, it also plays a role as a therapeutic agent. Since the 1970s physicians have investigated BTX therapy in patients with neurologic disorders. The number of applications greatly expanded over the years to include certain focal dystonias (blepharospasm, torticollis, laryngeal dystonias, writer's cramp), strabismus, and a wide variety of other indications (gastrointestinal disorders, cosmetic wrinkle correction, spasticity, hyperhidrosis). BTX's safety and efficacy are reviewed.

Anti-Dyskinesia Agents↗

PubMed1994

Asterion region synostosis.

Posterior plagiocephaly historically has been associated with synostosis of the lambdoid suture. The incidence, diagnosis, and modes of treatment for stenosis of the lambdoid suture remain controversial. Commonly, the lambdoid suture is found to be open both on radiographic examination and at the time of surgery. The study reports on nine patients who presented with unilateral posterior plagiocephaly and who were found to have open lambdoid sutures, but a stenosed region of the asterion. The area of involvement included the distal-most lambdoid suture, the parietomastoid, occipitomastoid, and proximal squamosal sutures. Positional molding or torticollis was ruled out in all patients. All the patients showed progressive involvement of the skull base, including anterior shifts of the ipsilateral ear, compensatory ipsilateral frontal bossing and malar protrusion. Stenosis of the asterion was diagnosed with three-dimensional computed tomography scans, corroborated at the time of surgery and confirmed histologically. Surgical correction involved resection of the affected asterion and reconstruction using a bandeau-technique, barrel staves of the occipital bone and bone graft transposition. This approach provided excellent esthetic results in all patients.

Cranial Sutures↗

PubMedDate not supplied

A case of aspergillosis in a broiler breeder flock.

A case of aspergillosis in a broiler breeder flock having respiratory and nervous system problems caused by Aspergillus fumigatus and Aspergillus niger is documented. Dyspnea, hyperpnea, blindness, torticollis, lack of equilibrium, and stunting were observed clinically. On postmortem examination of the affected birds, white to yellow caseous nodules were observed on lungs, thoracic air sacs, eyes, and cerebellum. Histopathologic examination of lungs and cerebellum revealed classic granulomatous inflammation and cerebellar lesions, necrotic meningoencephalitis, respectively. No lesions were noted in the cerebrum histopathologically. Aspergillus hyphae were observed in stained sections prepared from lesioned organs. Fungal spores and branched septate hyphae were observed in direct microscopy. Aspergillus fumigatus and A. niger were isolated from the inoculations prepared from the suspensions of organs showing lesions.

Air Sacs↗

PubMedDate not supplied

Pathology of listerial encephalitis in chickens in Japan.

Neural signs (torticollis, drowsiness) and mortality were observed in five chickens of a native chicken flock (reared for meat) that included 450 male birds on a farm that had 2300 native chickens and 1120 layers. Histologic lesions were observed in the medulla oblongata, optic lobe, cerebellum, and spinal cord of the affected birds. The lesions, which were most severe in the medulla oblongata, were massive abscesses with rarefaction (demyelination and malacia) of the parenchyma with gram-positive bacteria. The degenerative and necrotic areas were characterized by fibrin thrombosis, hemorrhages, and congestion in the blood vessels. Immunohistochemically, the bacteria positive for L. monocytogenes antigen were observed in the medulla oblongata, cerebellum, and spinal cord. Ultrastructurally, the small rod-shaped and thin-cell-walled bacteria were observed in the parenchyma of the medulla oblongata. Listeria monocytogenes (serotype 4b) was isolated from the medulla oblongata and spinal cord. The pathogenesis of listerial encephalitis in chickens was discussed.

Animals↗

PubMed2006

Otitis interna (labyrinthitis) associated with Salmonella enterica arizonae in turkey poults.

Otitis interna was diagnosed in five 9-to-21-day-old turkey poults with clinical signs of paralysis, opisthotonus, torticollis, blindness, and increased mortality. Gross and microscopic lesions in the poults included omphalitis, typhlitis, hepatitis, meningoencephalitis, ophthalmitis, neuritis and ganglionitis of the vestibulocochlear nerve, and otitis interna. Salmonella enterica arizonae was isolated from the brains, eyes, intestines, yolk sacs, and livers of poults. Birds with otitis interna also had meningoencephalitis. It is most likely that the S. enterica arizonae infection spread from the brain to the internal ears through the vestibulocochlear nerve. This is the first documentation of otitis interna caused by bacteria in an avian species.

Animals↗

PubMed2006

Encephalomalacia with Enterococcus durans infection in the brain stem and cerebral hemisphere in chicks in Japan.

Fourteen chicks, 7-12 days old, that died or had neurological signs (depression, paralysis, torticollis, incoordination, and rolling) were investigated pathologically and microbiologically. Variably sized foci of malacia were present within the parenchyma of the brain stem (cerebral peduncle, optic lobe, and medulla oblongata) and the cerebral hemisphere. Capillary thrombosis with congestion and hemorrhage was frequently observed within the malacia lesions. Gram-positive cocci were recognized in the blood capillary of malacia lesions. Bacteriologically, Enterococcus durans was isolated from the brain, liver, kidney, lung, and spleen. Detection of gram-positive cocci within the blood vessels in the malacia lesions observed in these cases may more strongly suggest the pathogenic role of E. durans on the malacia lesions in the brain stem and cerebral hemisphere.

Animals↗

PubMed2002

Baylisascaris procyonis (Nematoda: Ascarididae) larva migrans in free-ranging wildlife in Orange County, California.

Baylisascaris procyonis is well recognized as a cause of visceral (VLM), ocular, and neural (NLM) larva migrans in birds and mammals, including man. A study of the prevalence of larva migrans in free-ranging wildlife associated with raccoon latrines as well as a retrospective study of wildlife mortalities with neurological disease was conducted in 2000 in Orange County, California. Eighty-seven birds of 18 species and 64 mammals of 8 species were found to have NLM or VLM or both. NLM clinical signs included convulsions, torticollis, opisthotonus, head-tilts, circling, ataxia, paralysis, and visual defects. NLM lesions were characterized by focally disseminated, frequently linear, "tracklike" areas of parenchymal degeneration with varying degrees of astrocytosis in the white matter of the cerebrum, cerebellum, brain stem, and spinal cord. Larvae were rarely found in these lesions but were rather isolated in the brain-spinal cord parenchyma. At least 1 larva was isolated by digestion from each case of NLM and identified as Baylisascaris sp., most likely B. procyonis. VLM lesions consisted of granulomatous reactions surrounding intact or degenerative larvae in the parenchyma of the liver, kidney, diaphragm, and, occasionally, the lymph nodes. This report broadens the range of species of wild birds and mammals that have been found to be susceptible to larva migrans caused by B. procyonis and reaffirms the importance of raccoon latrine sites as contaminative foci for wildlife.

Animals↗

PubMed1994

Botulinum toxin in clinical practice.

Over recent years botulinum toxin type A has emerged as a safe and effective treatment for a number of previously refractory conditions associated with excessive muscle activity. The list of indications is expanding, but at present it is generally considered to be the treatment of choice for focal dystonias such as blepharospasm, torticollis, laryngeal dystonia, and oromandibular dystonia, as well as hemifacial spasm, strabismus, and some forms of limb spasticity. Carefully targeted intramuscular injections of a small amount of the toxin block the release of acetylcholine at the neuromuscular junction, producing a chemical denervation, with the aim of reducing excessive muscle activity without producing significant functional weakness. In some situations electrophysiological assessment and localisation of the muscles for injection is necessary. Treatment is symptomatic, with effects lasting 3 to 4 months and most patients requiring up to 4 injections per year to maintain the beneficial effect. Appropriate use of the toxin requires both an understanding of the physiological action of the potential muscles involved in each situation, together with a knowledge of the likely dose necessary to reduce muscle activity to the required level. Botulinum toxin represents a major advance in the management of these conditions, many of which responded poorly to previously available forms of therapy.

Botulinum Toxins↗

PubMed1995

Hereditary non-progressive torsion dystonia with intellectual disturbance.

Three siblings of a consanguineous parents with involuntary movements are reported. The mother had only a very slight neck tremor, without any other neurological abnormality, and the father had died. The 38-year-old son (Case 1) complained of involuntary movements at the age of 6. His involuntary movements were observed in the tongue, perioral region and upper and lower extremities: jerky movements with dystonic features. The 46-year-old elder brother (Case 2) experienced involuntary movements at the age of 18. Involuntary movements were observed in the upper extremities; he also had torticollis and tremulous movements in the neck, and jerky movements in the perioral region. They showed gait disturbance and dysarthria. The 35-year-old sister (Case 3) also experienced involuntary movements. When she was writing, her involuntary movements were obvious: dystonia and myoclonic jerks. Tremor in the neck was also seen. Their intelligence was below average. We concluded that this family had hereditary torsion dystonia, with myoclonus, and low intelligence. This condition may be associated with an autosomal recessive gene.

Adult↗

PubMed1989

[Surgical treatment of cervical spondylotic radiculomyelopathy with abnormal involuntary neck movements. Report of three cases].

Cervical spondylotic myelopathy usually arises in patients in their late 40s or early 50s, most frequently at the C5/6 and C6/7 levels. Recently, excellent results have been attained with microsurgery in cases of cervical spondylosis. On the other hand, treatment of cervical spondylotic myelopathy in patients with athetoid dystonic cerebral palsy entails several problems. The authors report three cases of such troublesome myelopathy. A 34-year-old male with severe athetoid movement showed cervical spondylotic myelopathy. Myelography and magnetic resonance (MR) imaging demonstrated compression of the spinal cord through the C3-C5 levels. A 47-year-old female with athetoid dystonic cerebral palsy presented myelopathy. Myelography and MR imaging showed instability and spinal cord compression at the C5/6 level. A 34-year-old male with spasmodic torticollis showed C6 radiculopathy due to cervical disc hernia at the C5/6 level. Cervical anterior decompression with interbody fusion brought temporary improvement in all the three patients. However, such problems as slippage of Halo-vest, difficulty in eating during Halo-vest fixation, relapse of neurological deficit, were experienced. Due to postoperative cervical instability, cervical laminectomy is considered to be contraindicated in such patients. Anterior decompression with bone fusion has been reported effective, but, if athetoid dystonia continues, there is a potential for myelopathic deterioration due to spondylotic changes adjacent to the fused vertebrae.

Adult↗

PubMed1976

Unilateral dysplasia of the superior articular facet of the axis.

A case of unilateral dysplasia of the superior articular facet of the axis due to incomplete or delayed ossification is described in a child. This may result in asymmetry of the articular surfaces of the axis in adult life and be a cause of torticollis. To our knowledge, no similar case has been reported.

Axis, Cervical Vertebra↗

PubMed1986

Sonography of neck masses in children.

The sonograms and medical records of 49 patients were reviewed to determine if there is a characteristic sonographic appearance for certain neck masses. Neck masses included: inflammatory masses (12), noninflammatory masses (23), and thyroid masses (14). Four entities including thyroglossal duct cyst, fibromatosis colli, cystic hygroma, and multiple lymphadenopathy showed characteristic sonographic appearance. Thyroglossal duct cysts were seen as midline or slightly off midline cystic masses. A sinus tract extending superiorly was nicely demonstrated in one patient. The mass in fibromatosis colli (neonatal torticollis) appeared as a well defined mass clearly within the sternocleidomastoid muscle, uniformly echogenic, but less echogenic than the normal surrounding muscle, without good through-transmission. A cystic or primarily cystic mass with linear septations was the characteristic finding of the cystic hygroma. Multiple lymphadenopathy demonstrated multiple discrete, oval, relatively hypoechoic masses along the cervical lymphatic chain. The sonographic appearance of inflammatory masses was variable, being either inhomogeneously echogenic or of mixed echogenicity. Hemangiomas were either echogenic with cystic vascular spaces or linear septations or relatively homogeneously echogenic. Intrinsic thyroid masses could be distinguished from extrinsic masses in most cases. The demonstration of calcification in a mass was useful in narrowing the differential diagnosis. Although it is not a specific finding, the presence of calcification highly suggests a neoplastic lesion, particularly neuroblastoma or teratoma. Not only can the location, extent, and internal characteristics of a mass be determined, but in certain entities, the sonographic appearance is characteristic and an accurate diagnosis can be made.

Adolescent↗

PubMedDate not supplied

[Predisposing factors of the migraine syndrome in children].

INTRODUCTION: The aim of this paper was to define characteristics which could be used to recognize groups of children at risk for migraine syndrome. MATERIAL AND METHODS: Predisposing factors of migraine syndrome were investigated by conducting a poll among 24.828 children aged 3-16, in Vojvodina, during the period between 1988-2004. RESULTS: For the purpose of defining recognizable characteristics of children with migraine syndrome, a comparison of children with migraine headaches, non-migraine headaches, and those without headaches was done among 24.828 children. Apart from direct heredity, the following factors should be accepted as predisposing factors of migraine syndrome: child's order of birth (second child), length of breast-feeding, age at which the ready-made industrial food was introduced, as well as the age at which the child started the whole-day stay (which is in inverse proportion to the risk of migraine attacks and the age of the first symptoms), particularities in behaviour pattern, family social status, atmosphere in the family, previous paroxysmal torticollis, episodic vertigo of unknown etiology, recurrent abdominal pain, and lack of cerebral hemisphere dominance. CONCLUSION: Children with migraine headaches have particularities, not clear enough for the time being, but recognition of which is necessary for early prevention of migraine syndrome in childhood.

Adolescent↗

PubMed1996

Current topics in neuroleptic-induced extrapyramidal symptoms in Japan.

This article reviews current topics in neuroleptic-induced extrapyramidal symptoms in Japan, focusing especially on the clinical features of akathisia and dystonia. Akathisia is a common side effect associated with antipsychotic drugs. It is most commonly characterized by subjective inner restlessness and objective motor signs, especially in the lower extremities. The mechanisms underlying akathisia remain unclear and controversial; however, an increase in the activity of beta-adrenergic systems relative to dopaminergic systems has been hypothesized, based on clinical therapeutic observations that beta-blocking agents are effective in this condition. A Japanese version of the Barnes Akathisia Scale has recently been established and uses a standardized videotape method for its precise evaluation. Various acute and chronic manifestations of neuroleptic-induced dystonia have been reported in Japan, including blepharospasm, difficulty in opening the eye lids, torticollis, retrocollis, oculogyric crisis, and Pisa syndrome. This review also introduces several other topics related to drug-induced extrapyramidal symptoms in Japan. These include; 1) the Drug-Induced Extra-Pyramidal Symptoms Scale (DIEPSS), which has recently been established, 2) studies on the discontinuation of anticholinergic drugs, and 3) a summary of extrapyramidal symptoms induced by drugs other than neuroleptics.

Akathisia, Drug-Induced↗

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