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PubMed1991

Simultaneous infantile spasms and partial seizures.

We report 11 infants with infantile spasms who had partial seizures that occurred concurrently with the infantile spasms. We studied this phenomenon with time-locked video electroencephalography. The partial seizures began before the infantile spasms and continued after one or more infantile spasms in seven patients. An infantile spasm occurred at the beginning of the partial seizure in five patients. (One patient had both seizure types simultaneously.) The clinical descriptions of these combinations of seizures were confusing, since they did not match known seizure types. The genesis of these seizures may involve an interaction between processes of focal cortical epileptogenesis (partial seizures) and brain-stem synchronization (infantile spasms).

Electroencephalography↗
PubMed1992

Clinical imitators of infantile spasms.

We report 53 infants who by clinical history were thought to have infantile spasms but who video-electroencephalograms showed were having other episodes that closely mimicked infantile spasms. Nine patients had other types of seizures. Forty-five patients had episodic symptoms that were not seizures: 11 patients had spasticity, four had gastroesophageal reflux, and the other patients had nonepileptic myoclonus, including 19 patients with benign neonatal sleep myoclonus. Three patients had more than one type of symptom. Infantile spasms imitators occurred in neurologically normal or abnormal infants, in patients with normal or abnormal interictal electroencephalograms, and in patients who also had previous or current infantile spasms. Differentiation of these episodes from infantile spasms prevented the initiation or continuation of anticonvulsant treatment appropriate for infantile spasms but inappropriate for these other behaviors.

Cerebral Cortex↗
PubMed1993

Posterior fossa abnormalities in children with infantile spasms.

In order to explore possible pathophysiologic involvement of the brain stem in infantile spasms, we retrospectively compared clinical and electroencephalographic (EEG) features of 14 children with infantile spasms who had gross posterior fossa abnormalities on neuroimaging studies with 84 children with infantile spasms who had either normal neuroimaging (n = 19) or supratentorial abnormalities (n = 65). Children with posterior fossa abnormalities how lower mean initial and follow-up developmental quotients compared to those with normal imaging or supratentorial abnormalities alone. Age of onset of infantile spasms, latency to treatment, response to steroids, and follow-up EEG pattern were not significantly different among the three groups. Six children (6%) had Dandy-Walker cysts, an association rarely reported with infantile spasms. We conclude that the presence of posterior fossa abnormalities in patients with infantile spasms portends a relatively poor developmental outcome.

Age of Onset↗
PubMed1994

The treatment of infantile spasms by child neurologists.

Infantile spasms is a refractory seizure disorder for which a number of different treatment regimens are available. No information is available on which treatment regimens are most widely used and which would be of practical importance in designing clinical trials to determine efficacy of new treatments. We proceeded to gather data on the most commonly employed methods of treating infantile spasms. A survey was mailed in 1991 to all junior, active, and emeritus members of the Child Neurology Society asking details about the treatment of infantile spasms. Telephone follow-up on a random sample of nonresponders was made. The total response rate was 58.3%. Most respondents who treat infantile spasms use corticotropin (ACTH) as their drug of first choice (88%). The most frequently used dosage was 40 IU per day, and the most frequent duration of treatment was 1 to 2 months. The most frequently reported side effects of ACTH or oral corticosteroid treatment were cushingism, behavior changes or irritability, hypertension, topical infections, and systemic infections. Of those not using ACTH, valproic acid was the next most commonly employed agent, followed by oral corticosteroids. The results were similar for all respondents regardless of age, sex, type of practice, number of cases of infantile spasms seen, location (United States or abroad) or whether the survey was completed by mail or telephone. These data suggest that there is a relative uniformity among child neurologists in the management of infantile spasms despite the publication of many alternative treatment strategies.

Adrenal Cortex Hormones↗
PubMed1980

Pathophysiology and treatment of coronary arterial spasm.

Spasm of a large coronary artery occurs most often from midnight to early morning and least often in the afternoon, due to the circadian variation of the tone of the large coronary artery in most patients with variant angina. Alpha adrenergic stimulation induces coronary spasm most easily in the early morning and least easily in the afternoon. Coronary spasm is due to strong contraction of coronary vascular smooth muscle cells which is triggered by an increase of intracellular calcium ions; hyperventilation plus TRIS-buffer infusion induces coronary spasm by decreasing hydrogen ions which antagonize the action of calcium ions. Administration of nitroglycerin promptly relieves the acute attack of coronary spasm, and calcium antagonists such as diltiazem, nifedipine and verapamil, which block the entry of calcium ions into coronary vascular smooth muscle cells and dilate large coronary arteries, prevent the occurrence of coronary spasm.

Adult↗
PubMed1982

Femoral artery spasm in children: catheter size is the principal cause.

The incidence of femoral artery spasm and subsequent thrombosis complicating angiography is greater in children than adults. This study was designed to assess femoral artery spasm and to determine its causes in children. In 100 unselected, consecutive infants and children undergoing left heart angiocardiography by the Seldinger technique, angiography of the femoral artery was performed to include the puncture site after completion of angiocardiography. Arterial spasm was quantified and, with mathematical analysis, related to various clinical and angiographic factors. Some degree of arterial spasm was noted in 62% of all cases; it was more frequent and severe in neonates. Although it was more intense in the infant with a small femoral artery, analysis of the data indicates that patient age, weight, length of catheterization, size of femoral artery, and catheter size have only minimal relative importance in the occurrence and severity of arterial spasm. The most important factor in spasm is the relative size of catheter to artery.

Adolescent↗
PubMed1981

Percutaneous radiofrequency facial nerve coagulation in the management of facial spasm.

The authors have developed a fluoroscopy-assisted technique of percutaneous radiofrequency facial nerve coagulation at or near the stylomastoid foramen for management of facial spasm. The details of the procedure and the operative results in the initial series of 27 cases of facial spasm are described. The series included six men and 21 women, aged from 16 to 73 years. Twenty-four patients had classical, intractable, persistent hemifacial spasm, one had an unusual bilateral facial spasm, and one a postparalytic facial spasm. The age of onset varied from 16 years to 70 years. The duration of the symptoms ranged from 3 months to 11 years. Ten patients had undergone previous blunt-needle compression of the facial nerve from one to 10 times. The operative results in the 27 patients have so far been excellent. The longest follow-up has been 1 1/2 years. Although partial facial weakness was present in 60% of the cases, it invariably disappeared within 1 to 4 months. Only three patients experienced recurrence of spasm. In two of them, recurrence followed low-temperature coagulation. The procedure is simple, easy to perform, and extremely effective. It can be performed under local anesthesia in the outpatient clinic.

Adolescent↗
PubMed2005

Hemifacial spasm in a patient with Marfan syndrome and Chiari I malformation. Case report.

Marfan syndrome, hemifacial spasm, and Chiari malformation are all relatively rare and seemingly separate entities. Marfan syndrome is caused by a defect in the gene that encodes fibrillin and leads to weakness of the artery wall. Hemifacial spasm results from compression of the facial nerve by an abnormal artery. Chiari malformation is characterized by a small posterior fossa. The authors report the case of a patient with Marfan syndrome who presented with hemifacial spasm and was also found to have a Chiari malformation Type I. The patient's Chiari malformation and hemifacial spasm were successfully treated by performing suboccipital and microvascular decompression surgeries, respectively. The pathophysiological characteristics of Marfan syndrome, hemifacial spasm, and Chiari malformation are discussed, and the authors propose a link between these conditions in this patient. The authors hypothesize that the patient's Marfan syndrome contributed to the abnormal shape of his vertebral artery and that, given the lack of space in his crowded posterior fossa due to the Chiari malformation, the artery caused compression of his facial nerve, resulting in hemifacial spasm.

Adult↗
PubMed1996

Increased basal tone and hyperresponsiveness to acetylcholine and ergonovine in spasm related coronary arteries in patients with variant angina--basal coronary artery tone in patients with variant angina.

In patients with variant angina, previous data have been inconclusive as to whether basal coronary artery tone is elevated at the spastic and non-spastic sites. Thus, the purpose of this study was to assess the basal coronary artery tone and the responsiveness to acetylcholine (Ach) and ergonovine (Erg) in patients with variant angina. We compared the basal coronary artery tone and the constrictive responses to Ach and Erg between 31 patients (Group 1) with variant angina in whom spasm was provoked by the low doses of Ach (intracoronary 20 micrograms) or Erg(intravenous 50 micrograms) and 35 patients (Group 2) provoked by higher doses of Ach (intracoronary 100 micrograms) or Erg (intravenous cumulative dose of 350 micrograms), and 26 control subjects. Patients with variant angina in whom spasm was provoked by low doses of Ach or Erg, had a higher incidence of mixed disease, multi-vessel spasm and higher disease activity. The basal coronary artery tone at the spastic and nonspastic sites of spasm related artery was significantly more elevated in Group 1 than that in Group 2 (44 +/- 17 vs 14 +/- 11% and 26 +/- 14 vs 16 +/- 10% respectively, P < 0.05), but not in the nonspasm related artery, The magnitudes of vasoconstrictive responses to Ach and Erg at the nonspastic sites were also greater in Group 1 than those in Group 2 and the control groups (Ach; 40 +/- 20 vs 26 +/- 11, 27 +/- 12%: Erg; 37 +/- 18 vs 12 +/- 8, 13 +/- 10% respectively, P < 0.05). However, the basal coronary artery tone was not elevated at the spastic and nonspastic sites in Group 2 compared to the in control subjects. These findings suggest that the basal coronary artery tone is increased in patients with variant angina with higher disease activity at the spastic sites and nonspastic sites of the spasm-related artery, and this may be related to the occurrence of coronary artery spasm.

Acetylcholine↗
PubMed2006

Hemifacial spasm: case report.

A 53 year old lady with diabetes mellitus presented with right hemi-facial spasm (HFS). Brain MRI Scan revealed extensive pan-sinusitis and mild bilateral mastoiditis. She responded well to intravenous ceftriaxone therapy and the hemifacial spasm resolved. The symptoms of hemifacial spasm and pain over the right cheek and peri-orbital area recurred after three weeks. She was admitted to hospital for Functional Endoscopic Sinus Surgery (FESS); following findings on repeat para-nasal sinus CT-Scan. Several reviews over six month's period revealed complete resolution of hemi facial spasm symptoms save for mild intermittent right blepharospasm; particularly on exposure to wind. This is a very rare cause of hemifacial spasm and clinicians should be on the look out for infective/inflammatory aetiology of hemifacial spasm; particularly in patients who present with recent onset HFS and have features of infection and or inflammation in the cranium.

Female↗
PubMed1999

Vigabatrin in infantile spasms: preliminary result.

PURPOSE: To evaluate the efficacy of vigabatrin in the treatment of infantile spasms in Thai children. PATIENTS & METHOD: From March 1996 to May 1998, patients aged under 2 years presenting with infantile spasms at Ramathibodi Hospital were initiated with vigabatrin 35-50 mg/kg/day in two-divided doses. The dosage was escalated by 25 mg/kg weekly until spasms ceased or the maximum dose of 130 mg/kg was reached. RESULTS: There were 20 patients enrolled. The ages ranged from 3 to 23 months (mean 7.6 months). They were categorized as 4 cryptogenic and 16 symptomatic. Infantile spasms were completely controlled in 12 patients (60%). Six patients (30%) had at least 50 per cent reduction of seizure frequency. There were 2 patients whose seizure frequencies and severity were not altered. Only one patient whose infantile spasms partially responded to vigabatrin developed orofacial dyskinesis which disappeared after discontinuation of vigabatrin. Five patients had their vision evaluated which was unremarkable. Based on parental global evaluation, there was an increase in alertness, cheerfulness and interaction to the environment and stimulation in 8 out of 15 patients who were still taking vigabatrin and responded to treatment. CONCLUSION: Vigabatrin is effective for infantile spasms. A long-term follow-up of these patients is necessary to evaluate its efficacy and side-effects.

Anticonvulsants↗
PubMed2000

[Hemifacial spasm due to a compression of the facial nerve by a fusiform aneurysm of the vertebral artery: case report].

We report a rare case of symptomatic hemifacial spasm caused by a fusiform vertebral artery aneurysm and by a branch of the anterior inferior cerebellar artery compressing the facial nerve at the root exit zone (REZ). A 71-year-old female had an 11-year history of right hemifacial spasm. MRIs demonstrated an aneurysm compressing the facial nerve at the REZ. Angiography disclosed a fusiform aneurysm of the right vertebral artery at the origin of the posterior inferior cerebellar artery. After the vertebral aneurysm was clipped distal to the origin of the posterior inferior cerebellar artery, a branch of the right anterior inferior cerebellar artery was also observed compressing the facial nerve at the REZ. Both the clipped aneurysm and the branch of the anterior inferior cerebellar artery were mobilized away from the REZ of the facial nerve, and a prosthesis was inserted between the branch of the anterior inferior cerebellar artery and the brain stem to keep the aneurysm away from its original position. The patient's hemifacial spasm immediately disappeared without any neurological deficits just after the surgery. Hemifacial spasm, especially caused by an aneurysm, is quite rare. In a review of the literature, we found only 4 cases of symptomatic hemifacial spasm caused by an aneurysm of the vertebral artery. This case is the first reported case of hemifacial spasm caused by both a fusiform vertebral artery aneurysm and a branch of the anterior inferior cerebellar artery compressing the facial nerve at the REZ.

Aged↗
PubMed1999

Clinical and video-polygraphic features of epileptic spasms in adults with cortical migration disorder.

The International Classification of Epileptic Syndromes considers epileptic spasms to be typical seizures of West syndrome. Literature reports show that spasms are present in epileptic syndromes other than West syndrome but there are few data on their characteristics in adults. We describe ictal, clinical and video-polygraphic findings in three patients (aged 21, 32 and 57 years) with epileptic spasms and with diffuse (case 2), focal right fronto-parietal (case 1) and bi-opercular (case 3) pachygyria. Spasms had been present since the ages of 1 month, 11 and 27 years respectively. Only one patient is mentally retarded. Two of our patients (cases 2 and 3) have partial seizures. Ictal polygraphic studies showed a positive, diffuse, high amplitude slow wave activity during spasms, with superimposed fast activity, followed by a diffuse flattening in all cases with a typical muscle pattern. Epileptic spasms, as typically described in West syndrome, can maintain the same semeiological and electroencephalographic features during adulthood in certain patients with cortical dysplasia.

Adult↗
PubMedDate not supplied

[Levodopa in hemifacial spasm. A therapeutic alternative].

OBJECTIVE: After reviewing the existing literature on the subject, a therapeutic, symptomatic alternative in the treatment of hemifacial spasm is proposed. MATERIAL AND METHOD: Three patients, all with a 5-year history of hemifacial spasm, were given one 25/250-mg tablet of Levodopa/carbidopa every 12 h. In a cross-over study, the patients were their own controls. The initial treatment phase lasted 1 month, at the end of which treatment was suspended. For an equal amount of time, the patients were given a placebo, after which they were again given Levodopa. RESULTS: After a 2-week period of treatment with Levodopa, the symptomatic aspect of the spasm disappeared in the patients. Upon replacing the drug with the placebo, the spasm reappeared. The patients have since been receiving the treatment with Levodopa, one tablet every 12 h, for a period of 4 years and the reduction of the hemifacial spasm has oscillated at 75%. CONCLUSION: We suggest the use of Levodopa/carbidopa in the treatment of hemifacial spasm as a therapeutic alternative. This treatment is within the patients' economic reach and more importantly allows them to once again lead normal lives.

Aged↗
PubMed2002

Clinical and electrographic features of epileptic spasms persisting beyond the second year of life.

PURPOSE: Few reports detailing the electroclinical features of epileptic spasms persisting beyond infancy have been published. We sought to characterize this unique population further. METHODS: We retrospectively reviewed the clinical and video-EEG data on 26 patients (4-17 years; mean, 93 months) with a confirmed diagnosis of epileptic spasms and who were evaluated at our tertiary referral center between 1993 and 2000. RESULTS: In half of our cases, epileptic spasms were associated with disorders of neuronal migration, severe perinatal asphyxia, and genetic anomalies. Interictal EEGs showed generalized slowing in the majority of patients, and a slow-wave transient followed by an attenuation of the background amplitude was the most common ictal EEG pattern associated with an epileptic spasm (19 cases). Other seizure types (number of cases in parentheses) included tonic seizures with or without a preceding spasm (13), partial seizures (11), myoclonic seizures (11), generalized tonic-clonic seizures (six), atypical absence seizures (two), and atonic seizures (one). Cases with a more organized EEG background (especially with frequencies > or =7 Hz) were more likely to have better cognition. Continued disorganization of the EEG background and persistence of hypsarrhythmia were associated with poor developmental outcome. CONCLUSIONS: Patients with epileptic spasms persisting beyond age 2 years constitute a truly refractory population, one that should be better recognized by clinicians. Interestingly, although many therapies resulted in a >50% reduction in seizures, neither neurocognitive function nor quality of life was substantially improved with intervention. The interictal EEG background is the most helpful in predicting neurologic outcome.

Age of Onset↗
PubMed1992

[Intraoperative monitoring of facial electromyographic responses during neurovascular decompressive surgery for hemifacial spasm].

UNLABELLED: In this paper, we reported our experience of intraoperative facial electromyographic recordings obtained during microvascular decompression for hemifacial spasm. MATERIALS AND METHODS: Intraoperative electromyographic recordings from the mentalis muscle during stimulation of the temporal branch of the facial nerve has been attempted in 31 patients. No muscle relaxants were used except for those before intubation. Of 31 patients, 22 were female and 9 were male. The age on admission ranged from 31 to 60 years with a mean of 54 years. RESULTS: 1. Abnormal response appeared with a latency of about 10 msec after stimulation. This response disappeared in 30 out of 31 patients at the end of operation. In 4 patients, the abnormal response disappeared prior to decompression of the nerve. 2. 30 patients in whom the abnormal response disappeared were free of spasm immediately after surgery. Hemifacial spasm has been relieved in 28 patients with a follow up period of 6 months to 2 years and 7 months. The remaining two patients had mild spasm. The one patient in whom the abnormal response did not disappear had persistent hemifacial spasm. CONCLUSION: The authors think that intraoperative facial electromyographic recording is useful to identify the blood vessel that is causing the spasm and to ensure that decompression of the nerve has been accomplished.

Adult↗
PubMedDate not supplied

Low-dose topiramate is effective in the treatment of infantile spasms.

BACKGROUND: Management of infantile spasms is difficult because current treatment regimens, including many anticonvulsants and hormones, are often ineffective. We conducted this study to determine the effective dose of topiramate (TPM) in Taiwanese children with infantile spasms. METHODS: Fourteen patients with infantile spasms were given TPM at an initial dose of 12.5 mg/d, and the dose was raised by 12.5 mg every 2 approximately 3 days. If the seizure frequency did not decrease during the initial 2 weeks, the dose was raised more rapidly. Titration continued for < or = 12 weeks. Subjects were monitored by weekly visits to undergo titration. RESULTS: The etiology of the infantile spasms included a cryptogenic group (n = 3) and a symptomatic group (n = 11). Overall, spasms in 5 patients (38%) were completely controlled. A > or = 50% reduction in spasms was observed in 11 (85%) of 13 subjects during stabilization, while one patient quit the treatment. The mean dose of TPM during stabilization was 7.35 +/- 4.9 mg/kg/d. Among these, 6 patients achieved seizure control and 3 were free of seizures at TPM doses of lower than 6 mg/kg/d. CONCLUSIONS: Seizure control was achieved with lower doses of TPM therapy than suggested in previous studies.

Adrenocorticotropic Hormone↗
PubMed1991

[A prognostic study of cryptogenic infantile spasms].

The prognosis of infantile spasms is grim when a detectable brain lesion is present. In contrast, cryptogenic infantile spasms, in which there is no identifiable brain lesion, usually run a favorable course under treatment. Few studies have focused on the outcome in children with cryptogenic infantile spasms. Among 111 pediatric patients with a history of infantile spasms hospitalized over 18 consecutive years, 23 (21%) were given a diagnosis of cryptogenic infantile spasms. Follow-up ranged from 4 to 21 years. Outcome was as follows: the IQ was above 80 in 39% of cases and above 100 in 13% of cases; 42% of patients of school age were attending school but half of these had learning disabilities; 30% of patients had severe psychiatric disorders, and 22% had developmental delay and severe epilepsia. Early factors apparently associated with a good prognosis included the mild nature of psychomotor regression, persistence of spindles on EEGs recorded during NREM sleep, and prompt improvement of clinical status and EEG recordings under treatment. Conversely, severe regression, focalized EEG anomalies, failure of development to resume promptly after initiation of therapy, recurrence of spasms and hypsarrhythmia at discontinuation of treatment, and onset before 5 months or after 11 years of age were associated with a poor prognosis.

Adrenocorticotropic Hormone↗