[Epilepsy with conditioned reflex seizures].
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Conditioning, Classical↗
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Seizure characteristics in chromosome 20 benign familial neonatal convulsions.
We studied a kindred of 69 affected individuals with the autosomal dominant epileptic syndrome of benign familial neonatal convulsions, linked to chromosome 20. Forty-two percent had their seizure onset on day 3, while remission took place in 68% during the first 6 weeks. Seizures were brief and the phenotype was of a mixed seizure type, starting with tonic posture, ocular symptoms, apnea, and other autonomic features. The seizure often progressed to clonic movements and motor automatisms. The postictal state was brief, and interictally the neonates looked well. The ictal EEG pattern with generalized suppression of amplitude on onset may be relatively unique. Neurocognitive outcome was usually normal, but the risk for subsequent epilepsy was 16%. Most of the later epilepsy was generalized tonic or tonic-clonic, and some seizures were provoked, raising the possibility of an unusual form of reflex epilepsy.
Chromosomes, Human, Pair 20↗
Reading epilepsy: report of five new cases and further considerations on the pathophysiology.
Five new cases of reading epilepsy (RE) are reported. This is an epilepsy syndrome belonging to the group of idiopathic localization-related epilepsies. They all have some interesting features which contribute to the understanding of the pathomechanism and nosology of this specific type of reflex epilepsy. In our first patient the precipitating effect of texts in unknown languages depended upon phonematic intricacy. With our second case, changes of script within the text (Latin to Greek) increased the precipitating effect. The visual aura experiences reported in vague terms by some patients with RE may represent ictal dyslexia. For case three RE had been misdiagnosed as phobic neurosis. Even if a patient has a history of development dyslexia, and ictal dyslexia is a feature of the seizures, the onset of RE is not during primary school age but at puberty. Our fourth patient's manifestation factor of (late-onset) RE was a change of scriptural environment (Kyrillic to Latin) . Unilateral myocloni were observed with bilateral spike wave discharge in RE, and carbamazepine possibly increased the epileptic response in RE. With the co-occurrence of RE and juvenile myoclonic epilepsy in case five, the clinical features of both syndromes remained separate. All five patients responded well to treatment with valproic acid, and all confirmed that the syndrome has no tendency to deteriorate in long-term follow-up.
Adolescent↗
Startle-induced epileptic seizures.
The present study was undertaken in order to describe 16 more patients suffering from startle-induced epileptic seizures and to clarify further the possible therapeutic activity of benzodiazepines in this rare reflex epilepsy. The interictal and ictal electroclinical data of 16 epileptic children or adolescents have been detailed. A CT-scan was performed in 10 patients; six of them showed an atrophy of the mesial surface of one or both hemispheres ("mesial hypodensity"). Benzodiazepine was associated with the previous antiepileptic treatment in the 16 ineffectively treated patients. Clonazepam was administered in three patients; one of them was completely unresponsive and two became seizure-free for a mean of 16.5 months but complained of drowsiness or ataxia. Clobazam was administered in 13 patients; 15.4% of them were completely unresponsive, 23.1% experienced drug resistance, and 61.5% obtained a good control (91.5% reduction of the reflex seizures) for a mean of 22.75 months. In spite of a possible loss of therapeutic activity, the appearance of very few unfavorable side effects and the presence of favorable side effects ("psychomotor arousal") make clobazam therapy important in the treatment of patients suffering from startle epilepsy.
Adolescent↗
Flunarizine--effective add-on therapy in reading epilepsy.
Since it was first described in 1956, reading epilepsy, an uncommon disorder, has engendered great interest among neurologists, the reason being that it is probably one of the most complex of the sensory-evoked reflex epilepsies and one in which the mechanism of seizure production is as yet unresolved. Unfortunately, it often defies the standard medications useful in the treatment of other forms of epilepsy. A patient with typical reading epilepsy, preceded by jaw-jerking, is described in this report. An additional feature of this case is the presence of a stutter from early childhood. Therapy with phenytoin, carbamazepine and sodium valproate failed to provide satisfactory control. Flunarizine, as add-on therapy to sodium valproate, produced an excellent therapeutic response.
Adult↗
Epilepsy or paroxysmal kinesigenic choreoathetosis?
Epileptic seizures induced by sudden movement and paroxysmal kinesigenic choreoathetosis (PKC) have often been confused in the past, owing to the close similarity of the attacks, the equally good response to anticonvulsants, and the frequent occurrence of epilepsy and PKC in the same family, or even in the same patient. The pathophysiology of PKC is still unclear and its relationship with epilepsy open to discussion. The sparing of consciousness and the lack of postictal phenomena are constant features of PKC, thus differentiating this syndrome from epilepsy. We report the case of an 8-year-old boy with frequent brief tonic attacks, without loss of consciousness, triggered by sudden movement. The neurologic examination, EEG and MRI did not help to differentiate between epilepsy and PKC. Only the occurrence of a longer seizure with clouding of consciousness and the recording of the postictal abnormalities on the EEG supported a diagnosis of reflex epilepsy induced by movement.
Anticonvulsants↗
Interleukin-1alpha in the brain is induced by audiogenic seizure.
We examined the expression of the sleep-inducing cytokine interleukin-1alpha (IL-1alpha) in the brains of audiogenic seizure-susceptible mice subsequent to the induction of sound-induced seizure. Animal models of epilepsy often require lesioning or trauma that may nonspecifically alter IL-1alpha expression. To avoid this, we employed the Frings mouse strain; a model of auditory-evoked reflex epilepsy. Frings mice were exposed to a high-intensity sound stimulus to induce a tonic extension seizure, and the expression of IL-1alpha transcripts in different brain regions was measured thereafter. Compared to control animals, IL-1alpha transcripts were elevated 6 to 8 h postseizure in the hypothalamus, but not hippocampus, by a dexamethasone-sensitive pathway. Similar results were obtained from the genetically distinct DBA/2J audiogenic seizure-susceptible mouse strain. These findings demonstrate that the expression of IL-1alpha is altered following generalized seizure activity, induced by noninvasive sensory stimulation, in a brain-region-specific manner.
Acoustic Stimulation↗
[Video electroencephalography in seizure disorders of children].
A total of 225 video-EEG examinations were performed in 179 children with various types of paroxysmal events, including 41 children aged 1-24 months of life and 138 patients aged 3-18 years. The studies were repeated in 25 patients; 80 one-hour recordings, 115 two-hour and 30 three-hour recordings were made. In selected cases EEG stimulating methods were employed, as well as tests provoking psychogenic events. In younger children video-EEG allowed confirming and more precise characterization of seizures in 19 of 41 patients (46.3%), whereas the corresponding number in older children was 17.4% (24 of 138 patients). In two children whose epilepsy was treated surgically, video-EEG was a significant element of preoperative diagnostic management. The use of the method allowed for diagnosing epileptic syndromes, Landau-Kleffner syndrome, reflex epilepsy, photogenic epilepsy and infant myoclonus epilepsy in 11 children. Video-EEG made it possible to rule out epilepsy in 3 of 41 younger children (7.3%) and in 24 of 138 older patients (17.4%). In 10 of 138 patients aged 12-17 years (7.3%), the method facilitated the diagnosis of psychic epilepsy, what allowed for initiation of an appropriate management without the administration of anti-epileptic drugs. In approximately one third of patients, video-EEG evaluation was helpful in selecting appropriate therapy.
Adolescent↗
[Eponyms and epilepsy (history of Eastern civilizations)].
The history of eponyms for epilepsy in the lands of the Eastern globe present the portrait of the attitudes of both the laymen and skilled people towards the disease and patient, as well as to the Nature itself. As opposed to the West which during the Middle ages changed its concepts of epilepsy as the organic brain disease for the sublime 'alchemic' position, the people of the East were more prone to consider from the beginning of their civilization till the XIX century that epilepsy is the consequence of the evanescent spiritual and extracorporal forces which by themselves were out of their reach. As compared to the western civilization, the historical resources are, often as a consequence of a linguistic barriers, more scarce-as consequently is the number of eponyms, but are nevertheless picturesque. The medical science from Babylonian period presumed that epileptic manifestations are the consequence of the demonic or ill spiritual actions. There existed an attitude that at the beginning of an epileptic attack the patient was possessed by a demon (the Akkadic, i.e., Babylonian verb "sibtu" denoting epilepsy, had the meaning "to seize" or "to be obsessed"); at the end of the clonic phase the demon departed from the body. Different demons were responsible for different forms of epilepsy such as nocturnal and children epilepsy, absence epilepsy and pure convulsions, simple and complex automatisms, and gelastic epilepsy. Thus, the doctors from the period of Babylon aside from making primordial classification of epilepsies, knew about their clinical picture (prodromal symptoms and aura, Jackson's epilepsy. Todd's paralysis), postictal phenomena and intericatl emotional instability; provocative factors were also known (sleep deprivation, emotions, as well as alcohol, albeit in a negative sense-as a cure for epilepsy). There is no doubt than in the period of Babylon the clinical picture of serial fits and its progress to status epilepticus were clearly recognized and considered as life threatening events. Persian history of epilepsy, except from the 6th century Zoroastrian "Avesta" document, lacks the written or spoken medical heritage untill the 7th century A.D. and the Arabic conquest of the entire Moslem world. On the other hand, Islamic medicine should be freed from the simple prejudice that the Moslem authors were only the translators of Greek medicine; contrary to such a view, their work contains a high degree of individuality. Although Mohammed introduced a lot of novelty into medicine, Khoran and the Sayings do not explicitly refer to epilepsy. Of importance is to notice that Moslem medicine did not have demons in the "repertoire" of direct causes of epilepsy. The causes and the cures of epilepsy were more magic-mystical and occult in nature, which is reminiscent of the European, as well as Serbian Middle age attitudes. Avicenna recognized difference between children and adult epilepsy. He considered insomnia and afternoon siesta as well as intensive sounds and light to be a provocative factors, whereby we see that at least empirically he knew of sleep (deprivation), startle and reflex epilepsy. The XIII century invasion of Mongols brought about the recession in Moslem Medicine; it recovered only in the XVIII century under the strong influence of European medicine handed over to us through Jewish doctors of various nationalities. The story of the China history of epilepsy has its debut approximately in the 8th century B. C. Medical texts from this period name epilepsy "Dian" and "Xian" which meant "the falling sickness" and "convulsions", respectively. Chinese medical terminology often interchangeably used the words "mania", "madness" and "psychosis" for "epilepsy" which, aside from a prominent language barrier, brings additional confusion. Although Chinese documents gave the first description of the grand mal epileptic attack already in the 8th century B. C. (ABSTRACT TRUNCATED)
China↗
Photosensitive epilepsy in Papio papio as a model for drug studies.
Photically-induced epilepsy in Papio papio provides a convenient model for assessing the efficacy and the acute neurological toxicity of new anticonvulsant drugs prior to clinical trial. Its drug sensitivity is broadly comparable to that of generalized seizures in man. It also shares some specific properties with syndromes of reflex epilepsy in man.
Animals↗
[Seizure-inducing factors in the patients with childhood epilepsy].
We evaluated the seizure-inducing factors in 264 patients with childhood epilepsy. One hundred and thirty-six patients (51.5%) exhibited some kind of inducing factors. Non-specific inducing factors were commonly recognized, such as "febrile state" (29.2%), "fatigue after exercise" (15.2%), "sleep disturbance" (9.1%), "psychic stress" (8.3%) and "emotional change" (6.8%). Specific inducing factors, which related to reflex epilepsy, was recognized in only 7 patients. "Febrile state" was dominant in young children, but the other nonspecific inducing factors were dominant in adolescent. Incidences and kinds of inducing factors also related to the epileptic syndromes an seizure prognosis. The patients with symptomatic partial epilepsies (64.3%) and grand mal on awakening (85.7%) had significantly more inducing factors, and the patients with refractory seizures had a higher incidence and more variable kinds of inducing factors than the patients with well-controlled seizures. These results suggest that seizure-inducing factors are recognized in children as well as in adults, and the clarification of inducing factors is important to the management of epilepsy, particularly of intractable epilepsy.
Adolescent↗
Experimental models of multifactorial epilepsies: the EL mouse and mice susceptible to audiogenic seizures.
This chapter reviews two well-characterized mouse epilepsy models with a multifactorial etiology, the epileptic EL mouse and mice susceptible to audiogenic seizures (AGS). Multifactorial disorders are quantitative traits where the action of more than one gene together with environmental factors contributes to the disease phenotype. The EL (epilepsy) mouse has been studied extensively as a genetic model for idiopathic complex partial seizures in humans. EL seizures are associated with an intense hippocampal gliosis in the absence of obvious neuronal loss and an elevated calcium-dependent release of aspartate that is present both before and after seizure onset. The inheritance of epilepsy is complex and several seizure frequency quantitative trait loci (QTL) have been mapped. Much of this genetic complexity may arise from the influence of environmental factors, including the seizure testing procedure, seizure history, and age. AGS, which are violent sound-induced convulsions, are considered a genetic model for generalized brainstem or reflex epilepsies. AGS susceptibility can arise as an inherited trait in some mouse strains or can be induced in genetically resistant strains from environmental factors (e.g., prior acoustic stimulation). AGS susceptibility and long-term potentiation (LTP) may also share common mechanisms. Several Asp genes have been mapped that influence AGS susceptibility. The expression of one of these can be modified by genomic imprinting and another has been identified as the X-linked 5-HT2e serotonin receptor. The genetic dissection of convulsive behavior in EL and AGS susceptible mice could help identify candidate genes for human multifactorial epilepsies.
Animals↗
Effects of drugs modifying brain levels of catecholamines on photically induced epilepsy in Papio papio.
The behavioral and electrographic effects of DL-amphetamine, disulfiram, FLA-63, and propranolol on the photically induced epilepsy of the Senegalese baboon were evaluated. Amphetamine produced somatic hypokinesia with enhancement of eye movements, diminution of spontaneous paroxysmal activity, and little change in photosensitivity. The acute effects of disulfiram and FLA-63 were poor, but the latter was lethal in the days following administration. Propranolol showed no consistent effects. Participation of catecholamine processes in this type of experimental reflex epilepsy are discussed.
Amphetamine↗
Hyperthermic seizures: an animal model for hot-water epilepsy.
Freely ambulant wistar adult rats of both sexes when exposed to a hot water jet on the head (50 degrees C - 55 degrees C) for a period of 8-10 minutes, manifested seizure activity similar to the ones noted in 'hot-water epilepsy' (HWE) in humans. Depth electrode recording from the hippocampus revealed seizure discharges during the ictus lasting from 34 seconds to three minutes, followed by low voltage indeterminate activity and a quiescent resting phase. Seizure initiation was noted to be critically dependent on the rectal temperature of 41.5 degrees C and regional hippocampal temperature of 37 degrees C. There appeared to be no clear evidence for kindling phenomenon. Intervention of hyperthermia by cooling the body after the ictus prevented subsequent occurrence of spontaneous seizures. Pathological study of the brain revealed ischaemic changes in specific topographic areas like Sommer's sector in hippocampus, layer 4 and 5 neurons of the cerebral cortex and reticular neurons in the brain stem- a pathological feature reminiscent of the human epileptic brain. Seizure initiation by hyperthermic stimulation with hot water poured over the head, the progression and the EEG recording the seizure activity in these rats appears to resemble the HWE in human subjects and could thus serve as the first animal model for this form of "reflex' epilepsy. This has given new insight into the understanding of human HWE. Our preliminary observations in humans has suggested that HWE is a type of hyperthermic seizure similar to febrile convulsion but differs from it with respect to stimulus and rate of rise in temperature in a susceptible individual.
Adult↗
[Reading epilepsy crises].
The authors describe two cases of "reading epilepsy" followed for nine and seven years respectively. In the first case the seizures consisted in an alexic aura followed by secondary tonic clonic convulsion; in the second case the seizures consisted in jaw clenching followed by loss of consciousness. In both patients neuroradiological studies including C.T. were negative and in both the seizures were controlled by anti-epileptic treatment. Clinical and electro encephalographic features tended to differ in the two cases and the authors review the literature on the subject. These observations led to recognise three groups of reflex epilepsy as follows: the first group includes focal secondary generalised seizures with parieto-occipital spikes and spikes and waves in parieto-occipital lobes; the second group includes seizures starting with jaw clenching corresponding with bursts of posterior sharp-waves; the third group consists of endogenous cases which can not be included in the other two groups.
Adult↗
[Paroxysmal, kinesigenic choreoathetosis. (2 cases in a family)].
Paroxysmal kinesigenic choreoathetosis is a rare disease of unknown origin, considered by some authors a reflex epilepsy, by some an extrapyramidal disorder and related recently to brainstem atrophy. Two patients, father and son, aged 52 and 22 years, are described suffering from this disease starting at age 40 and 13. In both patients the attacks consisted in slow flexion and extention movements starting in the foot and extending to the knee and the arm and were usually monolateral, but in the son some attacks were bilateral causing him to fall and involved also face muscles. In both patients neuroradiological investigations were normal. Neurophysiological studies including blink reflex, short latency somatosensory evoked potentials and brainstem acoustic evoked potentials did not show any involvement of the brainstem. In one case carbamazepine therapy, at low dosage, was tried and cured the patient.
Adult↗
Classical conditioning of kindled seizures.
As a model of reflex epilepsy, rats were classically conditioned to show paroxysmal spike activity to an auditory stimulus. A tone stimulus was paired with seizures induced by kindling. After repeated pairings, paroxysmal discharges were elicited at tone onset. The duration of the tone, proximity to the stimulation, and the number of pairings appeared to be important in the conditioning process.
Acoustic Stimulation↗
Postcallosotomy seizure outcome in hemiconvulsion-hemiatrophy-epilepsy syndrome.
Recently, three patients with hemiconvulsion-hemiplegia-epilepsy syndrome who underwent callosotomy were monitored for more than four years. All patients had atrophy of the right brain hemisphere with left hemiparesis and seizures. Two cases were probably the result of traumatic intracranial hemorrhage and one was due to an unknown cause. Wada tests were done in cases 1 and 2, which showed spared motor function of the atrophic hemisphere, supporting the choice of callosotomy instead of hemispherectomy. Patient 1 had atypical absence seizures and asymmetric generalized tonic seizures before surgery, the latter of which decreased by about 60% after callosotomy. Patient 2 had simple partial seizures of the motor type and complex partial seizures, the latter of which were also induced by touch (somatosensory-induced reflex epilepsy). This patient's complex partial seizures disappeared completely, but the simple partial seizures remained unchanged. Patient 3 had generalized tonic seizures, simple partial seizures of the sensory type and complex partial seizures. After surgery, the frequency of the generalized tonic seizures decreased more than 90%. The simple partial seizures of the sensory type remained unchanged. There were two new types of seizures after surgery, simple partial seizures of the motor type and brief generalized myoclonic jerks. All patients had significant reductions in numbers of seizures of more than 50%.
Adolescent↗