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PubMed2006

Developmental outcome in benign myoclonic epilepsy in infancy and reflex myoclonic epilepsy in infancy: a literature review and six new cases.

Benign myoclonic epilepsy in infancy is a rare syndrome with just over 100 cases reported since the first syndromic description by Dravet and Bureau [Dravet, C., Bureau, M., 1981. The benign myoclonic epilepsy of infancy. Rev. Elecroencephalogr. Neurophysiol. Clin. 11, 438-444]. This includes 23 infants with reflex myoclonic epilepsy whose inclusion in the wider syndrome remains debatable. We have reviewed the literature and present data from six further cases. Prognosis in respect of long term seizure freedom is good with sodium valproate being the most effective medication. However, the cognitive outcome is much less certain with cognitive problems present in one-third of children who have long term follow up. The cognitive outcome in reflex myoclonic epilepsy of infancy is normal in all reported cases. The term benign may be appropriately used to describe the myoclonic seizures but must be used cautiously when counselling families about cognitive outcome. The clinical heterogeneity within this syndrome suggests that there may be a variety of genetic mechanisms that underlie the presentation. Clinicians should distinguish the syndrome of reflex myoclonic epilepsy in infancy from benign myoclonic epilepsy of infancy and all patients should continue developmental follow up for several years after diagnosis.

Child Development↗
PubMed2006

Figuring out drawing-induced epilepsy.

Rare reflex epilepsies are a clinical curiosity but may also give insight into neurocognitive processes. The authors present a patient whose seizures were precipitated by drawing but not by writing. This dissociation complements previous reports on selective impairment of writing. EEG revealed a focus in the right frontal lobe with parietal spread. This is in accord with current cognitive and neural network models of drawing.

Adult↗
PubMed2004

Evidence for augmented brainstem activated forebrain seizures in Wistar Audiogenic Rats subjected to transauricular electroshock.

Previous work from our laboratory has shown that naïve Wistar Audiogenic Rats (WARs), a genetic model of reflex epilepsy in which seizures are induced by high-intensity sound stimulation (120 dB SPL), are seizure-prone to a variety of pro-convulsive stimuli (e.g., transauricular electroshock, pentylenetetrazole and pilocarpine). On the other hand, repetitive acoustic stimulation of WARs causes a slow recruitment of limbic structures, known as audiogenic kindling, changing seizure expression to include behavior characteristic of temporal-lobe epilepsy. Thus, our hypothesis is that WARs have facilitated acoustic-limbic projections when compared to Wistar controls. Wistar controls (n = 9) and WARs (n = 9) underwent EEG electrode implants in the cortex-Cx, amygdaloid complex-AMY and inferior colliculus-IC and received one low current transauricular electrical stimulus (ES) daily, for three consecutive days, with intensities of 10, 20 and 30 mA, respectively. The video-electroencephalographic activity was recorded 1 min before and 4 min after ES. Our results confirm previously described data indicating a greater susceptibility of WARs to seizure. However, low current ES (e.g., 20 mA) triggered epileptiform activity in the AMY only after epileptiform EEG was visible in the Cx and IC electrode leads. The AMY after-discharge continued even though no evident epileptiform activity was present in the Cx. In conclusion, our results add electrophysiological data to previously published behavioral evidence of WAR enhanced susceptibility to ES seizures and, also, support the hypothesis that the acoustic-limbic circuitry is facilitated even in unkindled WARs.

Acoustic Stimulation↗
PubMed2006

Orgasm-induced seizures: a study of six patients.

PURPOSE: Reflex seizures are known as the epileptic seizures triggered by some specific stimuli in sensitive patients. They are often classified according to the stimuli that trigger them rather than by the type of the seizure. Epileptic seizures induced by sexual orgasm are very rare in the literature. METHODS: We report six patients with different epileptic syndromes who experienced seizures after sexual intercourse and orgasm. RESULTS: All patients are women whose epileptogenic focus was in the right temporal in four and left frontal in one patient. One patient had pure generalized reflex epilepsy. CONCLUSIONS: Seizures induced by orgasm are very rare, with female and right hemisphere dominance requiring complex mechanisms to occur.

Adult↗
PubMed2004

Intractable reflex audiogenic epilepsy successfully treated by peri-insular hemispherotomy.

We report a case of an infantile hemiplegia seizure syndrome (IHSS) that presented with intractable reflex audiogenic startle epilepsy which in itself is an uncommon form of seizure disorder. Peri-insular hemispherotomy provided complete seizure control. Also of particular interest was that this syndrome resulted from an iatrogenic brain injury sustained during the course of a caesarian section. We review the different mechanisms of birth injury reported in the literature and, discuss the physiopathogenesis of the hemispheric damage in this patient. We also review the literature on "reflex epilepsy" as it applies to this case. Intractable reflex audiogenic (startle) epilepsy in IHSS submitted to hemispherotomy has not previously been reported.

Cerebral Cortex↗
PubMed2001

Gait epilepsy. A case report of gait-induced seizures.

Reflex epilepsy includes a group of epileptic syndromes in which seizures are induced by a stimulus, either simple (visual, somatosensory, olfactory, auditory) or more complex (e.g., eating, thinking, reading). We document a case of reflex epilepsy in which focal seizures are triggered exclusively by gait. The patient is a young boy whose walking was impaired by abnormal motor phenomena on the left side. These phenomena were elicited by gait and were accompanied by a distinctive ictal pattern with centro-temporal discharges. After comparing this patient with others reported in the literature, we determined that he has an unusual type of reflex epilepsy for which we coined the term "gait epilepsy." This disorder must be considered when physicians are making a differential diagnosis in patients who have symptoms that suggest paroxysmal kinesigenic dystonia (PKD) or selective epileptic gait disorder.

Cerebral Cortex↗
PubMed2005

Monozygous twin brothers discordant for photosensitive epilepsy: first report of possible visual priming in humans.

PURPOSE: The interaction of genetic predisposition and the environment in the development of epilepsy is often discussed, but, aside from some animal reflex epilepsies, little evidence supports such interaction in the development of reflex epilepsy in humans. METHODS: We describe the history of a 16-year-old boy in whom photosensitive epilepsy developed after a period of weekly exposures to high-intensity light flashes. RESULTS: Both he and his clinically unaffected monozygotic twin were found to be photosensitive. CONCLUSIONS: This case report suggests that some genetic forms of human reflex epilepsy may be elicited by repeated environmental exposure to the appropriate stimulus, similar to some of the stimulus-induced epilepsies seen in animals.

Adolescent↗
PubMed2003

An unusual case of benign reflex myoclonic epilepsy of infancy.

A previously healthy one-year-old boy, the youngest child of unrelated parents, presented with a four-week history of episodes of myoclonus triggered only by tactile stimulation to his head. There had been no loss of developmental skills. The electroencephalogram (EEG) revealed generalised polyspike wave activity both with and without clinical correlate. The infant was started on sodium valproate, which resulted in cessation of the myoclonic episodes one week after starting therapy. At subsequent follow-up (at 18 months) the infant was seizure free and a repeat EEG was normal. This case of non-progressive reflex myoclonic epilepsy of infancy triggered only by head tapping (and not by acoustic stimuli) is an extremely rare phenomenon. Reflex myoclonic epilepsy of infancy represents a distinct subtype of myoclonic epilepsy in infancy. It should be considered as an age-dependent idiopathic generalised epileptic syndrome with an apparently good prognosis.

Anticonvulsants↗
PubMed1997

[Benign reflex myoclonic epilepsy in infants].

BACKGROUND: Myoclonic epilepsy of infancy are seldom benign. CASE REPORT: A 25-month old girl developed myoclonic jerks either spontaneously either as reflex responses to auditory and tactile stimuli, such as sudden touching of the face or trunk from the age of 4 months. The jerks disappeared after valproate therapy. Neurological examination was normal with a follow-up of 9 months. CONCLUSION: This condition resembles that described in 1995 by Ricci et al. In must be differentiated from other myoclonic epilepsies of infancy, reflex epilepsies and hyperekplexia. It could be the earliest from of idiopathic generalized epilepsy.

Age Factors↗
PubMed1988

Eating epilepsy.

The term "reflex epilepsy" denotes epilepsies characterized by a specific mode of seizure precipitation. Seventeen patients in whom the act of eating precipitated seizures are described. Interictal EEG showed generalized seizure discharges in three and focal discharges in five patients. In four patients EEG was recorded during eating. In one of these, the frequency of seizure discharge increased, but none had a clinical seizure during the recording. Eating epilepsy is predominantly a disorder of young adults, and the triggering mechanisms are extremely complex and elaborate. The role of the limbic system and amygdala in the precipitation of eating-induced seizures is discussed.

Adolescent↗
PubMed2005

Vasopressinergic hypothalamic neurons are recruited during the audiogenic seizure of WARs.

The Wistar Audiogenic Rat (WAR) is a genetic model of reflex epilepsy with seizures induced by high-intensity sound stimulation (120 dB SPL). In spite of the known neural substrates involved in WAR seizure phenotype, neuroendocrine hypothalamic neurons were never investigated. In this work, AVP immunohistochemistry in the hypothalamus and radioimmunoassay (RIA) in plasma and in hypothalamic and hypophysial tissues were performed on both controls and WARs in order to evaluate the dynamics of AVP release due to seizure induction. Susceptible animals (WARs) displayed at least tonic-clonic convulsions followed by clonic spasms, while resistant Wistar rats (R) had no convulsive behavior. Animals were sacrificed at 3 instances: basal condition (without stimulus) and at 3 and 10 min after sound stimulation. For the immunohistochemistry AVP study, brains were harvested and processed by the avidin-biotin-peroxidase detection method. Optic densitometry was used for quantifying AVP labeling in supraoptic (SON) and paraventricular (PVN) hypothalamic nuclei. SON presented higher densitometry levels (%D--relative to background) for both WARs and R when compared to PVN. Nevertheless, both nuclei presented a marked decrease, referenced to basal levels, in %D for WARs at 3 min (approximately 35%) against a discrete change for R (approximately 90%). RIA results were significantly higher in the hypophysis of WARs when compared to R rats, at 3 min. Also, at 3 min, plasma AVP in WARs (89.32 +/- 24.81 pg/mL) were higher than in R (12.01 +/- 2.39 pg/mL). We conclude, based on the AVP releasing profiles, that vasopressinergic hypothalamic neurons are recruited during the audiogenic seizure of WARs.

Acoustic Stimulation↗
PubMed2004

Epilepsy and driving in Japan.

PURPOSE: The driving regulations in Japan were amended in 2002, which lifted the absolute ban on driving by persons with epilepsy (PWE) and granted licenses to PWE after a 2-year seizure-free period. METHODS: To survey the effect of the new driving regulations, we sent questionnaires both to the driving authorities (DAs) and to doctors of the Japan Epilepsy Society (JES). RESULTS: Around 1,400 PWE legally obtained a driving license within 1 year after the amendment, licenses were rejected in 157, and 61 had the license withheld for <6 months. In most cases, the attending doctor assessed fitness for driving; 171 doctors responded to the questionnaire. One third of them commented on a positive change in attitude of PWE with respect to driving. Their main remarks included the need to shorten the seizure-free period to qualify for fitness to drive and the need for special guidelines for conditions such as rare seizure occurrence, recently diagnosed epilepsy, or reflex epilepsy. Problems of assessment identified included difficulty in deciding the time for reassessment, distress of PWE over cancellation of license, cost of the assessment, responsibility of the assessing doctors in case of seizure recurrence, and protection of privacy. They requested the DAs to promote publicity about the information and asked the JES to establish a guideline for assessing fitness to drive. CONCLUSIONS: The results highlighted the need for cooperation between the DAs and the JES for further amendment of the regulations as well as the importance of education for the public, patients, and professionals.

Accidents, Traffic↗