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J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.51.1.146 on 1 January 1988. Downloaded from Journal of Neurology, Neurosurgery, and Psychiatry 1988;51:146-157 investigate the mesial frontal zone and the Three spontaneous seizures were recorded, Letters anterior part of the cingulate gyrus. Infre- each had the same pattern: diffuse flattening quent spikes occurred in the amygdala and and no in the Orbital frontal epilepsy: a case report paroxysmal discharge sometimes in the mesial frontal cortex. explored sites in the frontal or temporal Sir: Although an orbital frontal origin for A complex partial seizures has been sometimes 12 suggested, few cases have been completely documented.1 2 We report a patient in 23 whom electroclinical correlations were obtained by electroencephalography (EEG) and stereo-EEG recordings. The disap- 3.4 I-w VI,-#a+r - pearance of seizures after a surgical resection limited to the orbital frontal cortex confirmed the localisation of the epileptic 4.5 o I\A-.ViJV "fJ4 WOO focus. A 29 year old male had begun to experi- B ence seizures when 10 years old. The aetiology was unknown. Neurological examination was normal. The seizure pat- terns did not change during the next 19 23 ., years. They were characterised by staring, r by sudden and incomplete loss of contact -- - -. , A- followed by semi-purposeful automatisms, 3-4 v by thrashing movements if he was held, by the shouting of incoherent words and some- times by laughter. Deviation of the head and eyes to either side seemed to mimic natural Protected by copyright. movements and were accompanied by an expression of bewilderment. The seizures were brief (lasting about 30 seconds) and ended suddenly. Most remained partial. Some secondary generalised seizures occurred during sleep, usually when the patient had forgotten his medication. After the seizures, the patient claimed awareness but, in fact, he had an amnesia for the events around the attacks. They occurred in groups of 3 to 10 per day. Sometimes he remained seizure-free for 1 or 2 weeks. All therapeutic trials were unsuccessful and this refractory epilepsy prevented the patient from finding employment in spite of a professional edu- 1, cation. All the routine EEG recordings showed a normal background cerebral activity. Theta http://jnnp.bmj.com/ rhythms and sharp-waves were noted over the right fronto-temporal region (F8-T4) and they increased during sleep. The only alteration marking the beginning of a seizure was a sudden attenuation of back- C, ground activity quickly followed by move- ment artefacts rendering the recording 12 ls uninterpretable. Computed tomography, bilateral carotid angiography and ventricu- is on September 25, 2021 by guest. lography were normal. Fig Stereo-EEG recordling: there are five electrodes with eight contacts each. Contact No I The hypothesis of a right fronto-temporal is located on the tip. Electrode A. right posterior part of the gyrus rectus and orbital gyri; epileptic focus guided the choice of struc- electrode B. right middle frontal gvrus; electrode C. right mesial part of frontal pole and tures for stereo-EEG. Two electrodes (10 gvrus rectus: electrode C': leti mesial part of the frontal pole and gvrus rectus, electrode D: plots) were implanted in the right temporal miiesi'al pairt of the superior frontal gyrus and genu ofgyrus cinguli. Bipolar montage is lobe to explore the middle gyrus, amygdala perfrrmed between contiguous contacts. The discharge starts on the channel 2-3 of electrode and hippocampus. Two electrodes were C' (gvrus rectus) and 4-S of electrode A (posterior part of the intern orbital gvrus), then located in the right frontal lobe to involves the whole electrode A and the gvrus rectus part of the electrode C. 146 J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.51.1.146 on 1 January 1988. Downloaded from Letters 147 zones. We concluded that the choice of between the extent of resection and the type Intermittent pyramidal claudication as structures for exploration had been in error. of seizure were specified. Ludwig et al' presenting and sole symptom in multiple Interictal positron computed tomography presented four patients with seizures or sclerosis showed a zone of relative hypometabolism probably orbital frontal origin: one patient in the right frontal lobe but the degree of had been operated by orbital frontal Sir: Upper-motor neuron gait disturbance, depression was not statistically significant. resection which was followed by relative only appearing on strenuous exercise and Three months later a second stereo-EEG improvement. A temporal and orbital relieved by rest, is a rare variant of neu- exploration was performed. Having elimi- frontal resection was performed in a second rogenic intermittent claudication and has nated a location for the epileptic focus in the case but follow up was not reported. Our been known as "intermittent claudication of temporal lobe, the mesial frontal region and case seems to be the only one of orbital the spinal cord". More appropriately, how- the cingulate gyrus, we explored the frontal frontal epilepsy in which a resection ever, the syndrome may be called Intermit- orbital region and the frontal pole. Five restricted to the orbital gyrus has been tent Pyramidal Claudication (IPC). It electrodes were inserted through the frontal reported and followed by cessation of manifests itself as a feeling of discomfort, lobes (fig). Frequent interictal spikes and seizures. Similar localisation of inter-ictal indolent heaviness and weakness in the legs spike-waves were recorded consistently in spikes and ictal discharge combined with the with a spastic gait-pattern, after a period of the frontal orbital region without extension lack of spreading to other structures led us walking. Upper-motor neuron signs and to any ipsilateral cortical zone, but with to choose a restricted resection. symptoms may appear on exertion and dis- occasional propagation to the contralateral We conclude that no clear-cut clinical appear on rest. During rest or mild exercise, orbital frontal region. Many paroxysmal pattern is diagnostic of a frontal origin of patients are typically free of complaints and discharges unaccompanied by clinical man- complex partial seizures. Moreover, the symptoms. IPC is generally thought to be ifestations were found to occur in a very discharges are rarely limited to the orbital due to transient ischaemia of the spinal restricted zone during sleep. Electroclinical frontal zone; more often they spread to cord, secondary, for example, to arte- seizures were always characterised by repeti- other frontal or temporal regions. When riosclerosis, (syphilitic) arteritis, spinal cord tive spike discharges located only in the such an origin is suspected stereo-EEG can compression or vascular malformations.`' right orbital frontal region. Elsewhere be of real diagnostic value in delimiting the Multiple sclerosis is usually not mentioned flattening was observed as previously (fig). epileptic focus.7 as a cause. However, in the pertinent litera- Because irritative and epileptic foci were A ROUGIER ture on multiple sclerosis, IPC is mentioned restricted to the same orbital frontal region, P LOISEAU as "a common presenting symptom, whichProtected by copyright. we performed a cortical resection of the Department of Neurology and Neurosurgers, usually quickly progresses into persistent orbital gyri, gyrus rectus and area sub- H6pital Pellegrin 33076, paresis".'6 Remarkably, informative case callosa. The patient has now been seizure Bordeaux CUdex, reports or reviews on this subject are scarce. free for 18 months since the operation. His- France This prompted us to present the case histor- tological examination of the excised brain References ies of two patients with IPC which proved to showed no abnormalities. be the initial and sole symptom of multiple Complex partial seizures may originate in Ludwig B, Ajmone Marsan C, Van Buren J. sclerosis for 6 and 41 years. the frontal lobe3" and to distinguish their Cerebral seizures of probable orbito-frontal origin. Epilepsia 1975;16:141-58. Patient I. Five years before admission, this origin from those of temporal lobe origin 2 Tharp BR. Orbito-frontal seizure. A unique 52 year old construction worker noticed the patterns of the discharge spread seem to electroencephalographic and clinical weakness, clumsiness and a heavy feeling in be more useful than the characteristics of the syndrome. Epilepsiaz 1972;13:627-42. both legs, predominantly on the right, after automatisms themselves.' 5 Geier et al3 have 3 Geier S, Bancaud J, Talairach J, Bonis A, walking or bicycling for about 45 minutes. pointed out the high frequency of motor Szikla G. Enjelvin M. The seizures of frontal Then he was likely to stumble and fall. The manifestations:deviations of the head and lobe epilepsy. A study of clinical manifest- symptoms always disappeared after sitting eyes (86, 4%/), clonic and or tonic man- ations. Neurologj 1977;27:951-8. or standing still for about 4 Williamson PD. Spencer DD, Spencer SS, 15 minutes. Dur- ifestations (77, 3%), and phonatory man- Novelly RA. Mattson RH. Complex partial ing rest or mild exercise he was asymp- ifestations (86%). Topographic localisation seizures of frontal lobe origin. Ann Neurol tomatic. Time delay to onset of the of frontal lobe epilepsy by routine EEG is 1 985;18:497-504. symptoms gradually decreased. One year http://jnnp.bmj.com/ often difficult.6 For these reasons 5 Brey R. Laxer KD. Type 1 2 complex partial later, he noticed numbness and clumsiness nasoethmoidal and supra-orbital electrodes, seizures: no correlation with surgical of his right arm, on using it extensively for electrocorticography and sometimes stereo- outcome. Epilepsia 1985:26:657-60. over an hour. These symptoms disappeared EEG have been used.6 6 Quesney LF. Seizures of frontal lobe origin. In: after a 30 minute rest. Two vears after onset, Usually, when an orbital frontal epileptic Pedley TA, Meldrum BS.
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