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At least 505 records · Page 28Linked to original sources
PubMed2005

Periorbital pain associated with hemifacial spasms that responded to botulinum toxin treatment and microvascular decompression surgery: a case report.

Periorbital pain is unusual in patients with blepharospasms. We report a patient with hemifacial spasms who presented with severe ipsilateral periorbital aching pain. After treatment with botulinum toxin type A, the left hemifacial spasms and left periorbital pain improved temporarily. Microvascular decompression surgery was then performed for the facial spasms. The patient's left hemifacial spasms disappeared immediately after the operation, and his left periorbital pain was also totally resolved.

Aged↗
PubMed2004

Leptomeningeal angiomatosis with infantile spasms.

We describe a 7-month-old female with leptomeningeal angiomatosis who developed infantile spasms. She did not manifest facial nevus or ocular choroidal angioma. Leptomeningeal angiomatosis is characterized by venous angiomas of leptomeninges and usually accompanied by facial nevus, a condition known as Sturge-Weber syndrome. In Sturge-Weber syndrome, leptomeningeal angiomas can cause infantile spasms but much less frequently than in other neurocutaneous syndromes, such as tuberous sclerosis. This patient is the first reported case of leptomeningeal angiomatosis without facial nevus who developed infantile spasms. Leptomeningeal angiomas should be taken into consideration as a cause of infantile spasms, even in the absence of facial nevus. We suggest that this case is clinically within the spectrum of Sturge-Weber syndrome, and that the embryologic origin of this case is similar to that of Sturge-Weber syndrome.

Angiomatosis↗
PubMed2007

Brain malformation and infantile spasms in a SCAD deficiency patient.

This report presents a case of short-chain acyl-coenzyme A (CoA) dehydrogenase deficiency with a previously unreported presentation with brain malformations and infantile spasms. This female infant developed repeated tonic clonic seizures at the age of 3(1/2) months. She subsequently developed West syndrome at the age of 4 months. Her electroencephalogram disclosed hypsarrhythmia, and video-electroencephalographic monitoring confirmed the presence of infantile spasms. Magnetic resonance imaging revealed a small midline frontal meningocele, abnormal cortical gyration, and partial agenesis of the corpus callosum consistent with neuronal migrational disorder. Metabolic evaluation indicated ethylmalonic acidemia. Muscle biopsy with enzymatic assay of short-chain acyl-coenzyme A revealed low enzymatic activity confirming the diagnosis of short-chain acyl-coenzyme A dehydrogenase deficiency. To our knowledge, this is the first report of the coexistence of short-chain acyl-coenzyme A dehydrogenase deficiency, infantile spasms, and brain malformation. We conclude that short-chain acyl-coenzyme A dehydrogenase deficiency should be considered in the differential diagnosis of gyral abnormality, corpus callosal hypoplasia, and infantile spasms.

Agenesis of Corpus Callosum↗
PubMed2003

Wavefront analysis in eyes with accommodative spasm.

PURPOSE: To investigate changes of spherical aberration in eyes with accommodative spasm. DESIGN: Case reports. METHODS: Four eyes of two patients with accommodative spasm were studied. Ocular spherical aberration was measured with a Hartmann-Shack wavefront aberrometer for the central 4-mm zone. RESULTS: The root mean square values of spherical aberration were negative (-0.086 +/- 0.026 microm root mean square). However, when the spasm of accommodation was cured or treated with a cycloplegic agent, the value shifted toward positivity (0.025 +/- 0.004 microm root mean square). CONCLUSIONS: Excessive accommodative tone is manifested objectively by negative spherical aberration in eyes with accommodative spasm.

Accommodation, Ocular↗
PubMed1994

High-dose, long-duration versus low-dose, short-duration corticotropin therapy for infantile spasms.

Fifty patients in whom the diagnosis of infantile spasms had recently been made and who had hypsarrhythmic electroencephalographic findings were randomly assigned to receive either high- or low-dose therapy with corticotropin (adrenocorticotropic hormone; ACTH). Twenty-six patients receiving the high-dose therapy were treated as follows: 150 U/m2 per day for 3 weeks, 80 U/m2 per day for 2 weeks, 80 U/m2 every other day for 3 weeks, and 50 U/m2 per day every other day for 1 week, with the dosage then tapered to zero during a 3-week period. The 24 patients assigned to the low-dose therapy group received 20 to 30 U/day for 2 to 6 weeks; the dosage was then tapered to zero during a 1-week period. Population characteristics (cryptogenic vs symptomatic, treatment lag, and age at start of treatment) of the two groups were similar. Response, defined as cessation of spasms and disappearance of hypsarrhythmia, was determined objectively by serial prolonged video and polygraphic monitoring studies. Of the 26 patients treated with the high-dose therapy, 13 (50%) responded; of the 24 patients treated with the low-dose therapy, 14 (58%) responded (p value not significant). No significant difference in the relapse rate between the two groups was observed. The side effects seen in both treatment groups were similar, except that hypertension occurred more frequently in the high-dose group. These results indicate that there is no major difference in the effectiveness of these two regimens in the treatment of infantile spasms with respect to spasm cessation and improvement in the patients' electroencephalographic findings.

Adrenocorticotropic Hormone↗
PubMed1982

The value of neuroradiology in infantile spasms.

The results of neuroradiologic studies of 71 children with infantile spasms treated with ACTH were correlated with the developmental outcome at follow-up (mean 62 months, range 14 to 207 months). Fifty-two (73%) patients had an abnormal NRS on initial evaluation; 49% had cerebral atrophy, 18% had congenital anomalies, and 6% had hydrocephalus. Twenty patients were normal on initial clinical evaluation. At follow-up only the eight (40%) with normal NRS were normal. Twelve (60%) who had unexpected abnormalities on NRS were retarded at follow-up. CAT scanning is necessary to predict the developmental outcome in developmentally normal children with infantile spasm. Eight of nine patients with normal NRS in the early treatment group were developmentally normal at presentation and follow-up. Ten patients who were developmentally normal before spasms began, and had normal NRS but were in the late treatment group, were retarded at initial evaluation and follow-up. This finding suggests that early treatment of children who have a normal NRS and normal development at onset of spasm prevents mental retardation.

Adrenocorticotropic Hormone↗
PubMed2000

Hemifacial spasm caused by a contralateral vertebral artery: case report.

BACKGROUND: Hemifacial spasm is usually caused by compression of the facial nerve by ipsilateral blood vessels. Compression of the facial nerve root exit zone by a contralateral tortuous vertebral artery is very rare. METHODS: This 68-year-old woman presented with left-sided hemifacial spasm and was found to have compression of the left facial nerve by the tortuous vertebrobasilar artery, as revealed by magnetic resonance imaging and magnetic resonance angiography. Retromastoid craniectomy demonstrated compression of the left facial nerve root exit zone by the distal portion of the right vertebral artery. The vertebrobasilar junction and both vertebral arteries were moved laterally from the facial nerve and a muscle implant was interposed between the brainstem and the right vertebral artery. RESULTS: The patient has remained free of hemifacial spasm for a follow-up period of 27 months. CONCLUSIONS: Compression of the facial nerve by the contralateral tortuous vertebral artery may produce hemifacial spasm. A transposed large vessel can be secured by a sling technique or by interposing a soft implant between the brainstem and the vertebral artery.

Aged↗
PubMed2001

Fusiform aneurysm of the vertebral artery presenting hemifacial spasm treated by intravascular embolization: case report.

BACKGROUND: A rare case of fusiform vertebral artery aneurysm causing hemifacial spasm was successfully treated by intravascular embolization. CASE DESCRIPTION: A 53-year-old man presented with left hemifacial spasm persisting for 2 years. No other clinical symptoms were observed. Vertebral angiography revealed a fusiform aneurysm of the left vertebral artery, and thin-slice spoiled gradient-recalled acquisition magnetic resonance imaging demonstrated the aneurysm compressing the root exit zone of the left facial nerve. The parent artery was occluded together with the aneurysm by intravascular embolization with Guglielmi detachable coils (GDCs). The patient is free of left hemifacial spasm without any complication. CONCLUSION: Hemifacial spasm caused by aneurysms, especially fusiform aneurysms, is quite rare but can be treated by parent artery occlusion and coiling the aneurysm with GDCs.

Cerebral Angiography↗
PubMed1997

Three-dimensional imaging for presentation of the causative vessels in patients with hemifacial spasm and trigeminal neuralgia.

BACKGROUND: In patients with hemifacial spasm and trigeminal neuralgia, preoperative detection of the relationship between the blood vessels and the cranial nerves involved is essential. METHODS: We studied the causative vessels in 20 patients with hemifacial spasm and six patients with trigeminal neuralgia by means of magnetic resonance (MR) imaging with spoiled gradient recalled acquisition in the steady state (SPGR), MR angiography, and three-dimensional (3-D) imaging reconstructed from the data of SPGR MR imaging by the surface rendering method at a workstation. RESULTS: In all patients, the preoperative SPGR MR images demonstrated that the causative vessels compressed or were in contact with the root exit or root entry zone (REZ) of the facial or trigeminal nerve. These causative vessels were identified by inspection of the MR angiographic and 3-D images. The 3-D images provided clear information as to the anatomic relationship between the causative vessels and the REZ of these nerves. These findings were corroborated by the intraoperative findings. The symptoms were completely relieved after surgery in 18 of the patients with hemifacial spasm and in all six patients with trigeminal neuralgia. In all patients, sufficient decompression was depicted on the postoperative SPGR MR images at the causative vessels and the REZ of the nerve. CONCLUSION: SPGR MR images, MR angiography, and 3-D images are useful for the identification of the causative vessels in patients with hemifacial spasm or trigeminal neuralgia. The 3-D images are particularly useful for the simulation planning of the operative procedure.

Adult↗
PubMed1983

Infantile spasms and pertussis immunisation.

The possible roles of pertussis immunisation and of other factors in the aetiology of infantile spasms were investigated by analysis of 269 cases reported to the National Childhood Encephalopathy Study. In 34% of the cases an antecedent factor which may have caused infantile spasms was identified; the commonest of these were perinatal hypoxia (38 cases) and tuberous sclerosis (16 cases). Case-control analyses showed no significant association between infantile spasms and pertussis immunisation in the 28 days before onset. There was, however, some clustering of cases immunised with either diphtheria-tetanuspertussis or diphtheria-tetanus vaccines in the 7 days before onset. The excess compared with controls was compensated for by a corresponding deficit over the remaining period up to 28 days. It is suggested that these vaccines do not cause infantile spasms but may trigger their onset in those children in whom the disorder is destined to develop.

Acute Disease↗
PubMed1986

Treatment of facial spasm with botulinum toxin. An interim report.

Forty-eight patients were given serial injections of botulinum toxin in their eyelids for treatment of eyelid spasm during a two-year interval. Ninety-four percent obtained relief of spasm from botulinum toxin injection. The duration of the spasm-free interval as well as the incidence of ptosis and of diplopia was dose dependent. The marked increase in the incidence of these side effects with only a small increase in the duration of the spasm-free interval, when a dose of 25 units per lid was used, leads the authors to conclude that this dose is too high and should not be used. Since diplopia was most commonly caused by paresis of the inferior oblique muscle, and since blepharospasm usually can be controlled by excising the upper lid protractors, further studies are required to determine whether lower lid injection is necessary and, if it is found to be so, whether injecting only the lateral portion of the lid would be adequate.

Blepharospasm↗
PubMed1997

Blepharospasm and hemifacial spasm. Randomized trial to determine the most appropriate location for botulinum toxin injections.

PURPOSE: The purpose of the study is to analyze the effectiveness and side effects of botulinum toxin using four different treatment site applications to determine the most successful treatment regime with the least side effects. METHODS: In a prospective trial, 92 patients (50 blepharospasm and 42 hemifacial spasm) were assigned randomly to 1 of 4 different treatment groups (standard [S], brow [B], inner orbital [IO], or outer orbital [OO]). Each treatment group had a different pattern of injection sites in the orbicularis. A total of 285 treatments were given, and the mean follow-up time was 16.4 months. RESULTS: In the blepharospasm group, patients assigned to the standard group had a significantly longer duration of effect than for those in the brow, inner orbital, and outer orbital groups (8.1 weeks compared with 4.5, 4.2, and 3.1 weeks, respectively; P < 0.001). In the hemifacial spasm group, patients in the outer orbital group had significantly shorter duration of effect than those in standard, brow, or inner orbital group (7.2 weeks compared with 12.6, 12.8 and 10.4 weeks, respectively; P < 0.001). The four major complications of botulinum toxin treatment were epiphora, ocular irritation, ptosis, and diplopia. The inner orbital treatment produced significantly more episodes of ptosis (13% of treatments). However, the standard treatment produced the most epiphora and ocular irritation (18% of treatments). CONCLUSIONS: The position of the injection sites around the orbicularis influences the effectiveness and side effects of botulinum toxin treatment for patients with blepharospasm and hemifacial spasm. The further the treatment is away from the eyelid margin, the lower the risk of ocular side effects. The standard treatment produces the longest duration of effect in the blepharospasm group but with the most transient ocular irritation and epiphora. In the hemifacial spasm group, the brow treatment has an equally long duration of effect as that of the standard treatment with fewer side effects.

Aged↗
PubMed1988

Can barbiturate anaesthesia cure infantile spasms?

Five patients with infantile spasms and hypsarrhythmia and one with Lennox-Gastaut syndrome were treated with brief thiopentone anaesthesia as the primary treatment of infantile spasms. Thiopentone (30 mg/kg) was given intravenously and burst suppression was reached in EEG in three patients by this dose. The results were disappointing. In three patients a transient beneficial effect on spasms and hypsarrhythmia was seen, but all patients relapsed. Three other patients had anaesthesia for surgery. The spasms ceased and hypsarrhythmia disappeared dramatically, and the effect was permanent. The possible mechanisms of the therapeutic effect are discussed. It seems advisable to give anaesthesia and surgery prior to steroid treatment in any case where the both are needed.

Anesthesia↗
PubMed2002

Vigabatrin for infantile spasms.

We reviewed 20 infants receiving vigabatrin for infantile spasms. Patients were not enrolled in a formal study. All families obtained the medication abroad. Age at initiation of vigabatrin ranged from 1 to 48 months; nine infants had received prior treatment with various antiepileptic medications. Patients were begun on the lowest practical dose of 125-250 mg/day, with gradual daily increments to a target of 100 mg/kg/day, but maintained at the lowest effective dosage. Video electroencephalogram was obtained to document resolution of spasms and hypsarrhythmia. Of 20 infants, 12 responded with cessation of spasms and resolution of hypsarrhythmia, at doses of 25-135 mg/kg/day (median = 58 mg/kg/day). Partial responses were observed in six patients, whereas two had no response at 111 and 125 mg/kg/day. Additional new seizure types developed in three infants after initial response to vigabatrin. Increasing the vigabatrin did not have any clinical benefit. Vigabatrin is an effective, well-tolerated treatment for infantile spasms. The response is dose-independent, suggesting that starting at a low dose and gradually increasing, rather than beginning with an arbitrary 100 mg/kg/day dose is advantageous.

Anticonvulsants↗
PubMedDate not supplied

Infantile spasms: ictal phenomena.

Ictal phenomena were studied during three separate six-hour video/polygraphic recording sessions in 10 patients with infantile spasms; 1,079 spasms occurred. Frequency during wakefulness (7.7 spasms/hour) was greater than that during sleep (2.5 spasms/hour); 46.6% of spasms occurred in clusters. Spasms were composed of one or more of three phases: a myoclonic contraction, a tonic contraction, and/or an arrest of activity. The most common types were myoclonic-tonic (40.3%) and myoclonic alone (36.3%). When classified by postural motor phenomena, 41.6% were "flexor", 16.3% "extensor", 39.0% "mixed", and 3.1% "arrest" alone. Electrographic monitoring revealed that myoclonic contractions were associated with an initial paroxysmal event. Tonic contractions and arrests were usually associated with suppression of electroencephalographic activity with or without rhythmic activity. Knowledge of these clinical and electrographic features is important for diagnosis and evaluation of proposed treatments.

Cerebral Cortex↗
PubMed1999

Hemifacial spasm caused by a pilocytic astrocytoma of the fourth ventricle.

An 11-year-old male in whom hemifacial spasm was the presenting sign of a pilocytic astrocytoma within the fourth ventricle is reported. The child's hemifacial spasm decreased substantially after resection of the tumor. Hemifacial spasm is largely a disease of adults and only rarely is attributed to brain tumors. In contrast, this marks the fifth case of hemifacial spasm reported in a child, three of which have been attributed to tumors of the fourth ventricle.

Astrocytoma↗
PubMedDate not supplied

Infantile spasms in Down syndrome--effects of delayed anticonvulsive treatment.

To investigate the impact of treatment lag in infantile spasms (IS) on treatment response, occurrence of later epilepsy, and long-term cognition and behavior in patients with one single etiological entity, we examined 18 patients with Down syndrome (DS) and earlier IS retrospectively (follow-up period of 32-180 months with a mean of 85.1 months), and determined their history and present condition, in terms of previously mentioned items. There was a statistically significant correlation between treatment lag and lag to cessation of spasms (R=0.55, P=0.02), developmental quotient (DQ) (R=-0.75, P=0.003), and score of autistic features (AF) (R=0.57, P=0.04). Moreover we found that the later the response to treatment of IS, the lower was the DQ (R=-0.86, P=0.001) and the higher was the score of autistic features (R=0.5, P=0.06). A long duration of spasms also determined a low DQ (R=-0.93, P<0.0001) and a high score of autistic features (R=0.66, P<0.01). All patients with persistent epilepsy (n=5) had had a treatment lag of over 2 months. Conversely, for all children treated within 2 months (n=8) spasms ceased within 3 months of treatment and none of them had later epilepsy. This group of patients with a treatment lag of less than 2 months had earlier treatment response (P=0.002), higher DQ (P=0.004) and lower score of autistic features (P=0.006). The data stress the importance of a short treatment lag in view of mental development and prevention of later epilepsy and autistic features, and raise the question of antiepileptogenic effect in this specific condition.

Adolescent↗
PubMed1988

Botulinum toxin injections in the treatment of blepharospasm, hemifacial spasm, and eyelid fasciculations.

Seventy-six patients with blepharospasm (mean age 56.9 years) received 248 injection treatments with botulinum A exotoxin (mean 3.1 treatments per patient): 87.0% of treatments led to total relief of spasms for a mean interval of 14.1 weeks. The average duration of response remained fairly constant over the first six injection series, although patients with the most severe spasms had shorter intervals than patients with less severe symptoms. Twenty patients with hemifacial spasm (mean age 56.9 years) received 44 treatments (mean 1.9 treatments per patient): In 93.1% of cases there was total relief of periocular and perioral spasms, with a mean interval of 17.4 weeks. The average duration of response for the third series of treatments was much shorter than the mean durations for the first two treatments. Side effects were always transient and included ptosis (23.3%), dry eyes (18.1%), tearing (5.5%), and strabismus (1.4%). No patient had a systemic reaction to the drug. Chronic benign eyelid fasciculations were also successfully treated in 3 patients with single treatments.

Adult↗