Journal of Neurology, Neurosurgery, and Psychiatry, 1976, 39, 823-828

Gelastic, quiritarian, and cursive epilepsyA clinicopathological appraisal

P. K. SETHI AND T. SURYA RAO

From the Departments ofNeurology and Neurosurgical Centre, Command Hospital, Lucknow, India

SYNOPSIS A patient is reported with episodes of epileptic laughter, crying, and running occurringalone or in combination. He was found to have a discrete, well-circumscribed tumour of the lefttemporal lobe. The neurology of these epileptic events is discussed in relation to the pathologicallesion.

Laughter as an epileptic phenomenon is extremelyuncommon and equally so is episodic running.The combination ofthese two forms ofepilepsy-'gelastic' (laughter) and 'cursive' (running)-inthe same individual is rare. There are only twoearlier reports, of in all, three cases (Sisler et al.,1953; Chen and Forster, 1973). All these casesresulted from diffuse cerebral disorder. Westudied an individual with such episodic eventsof laughter, running and also crying occurringalone or in combination, associated with a dis-crete neoplastic lesion in the left temporal lobe.This case permits appraisal of cerebral localiza-tion of 'centres' for the physical expression ofemotions. Crying as an ictal event has notreceived much attention in the literature. We cointhe word 'quiritarian' epilepsy (quiritare: Latin:to cry/scream) to indicate this phenomenon.

CASE REPORT

A 33 year old infantry soldier was evacuated to ourneurological centre with a history of episodic seizuresfor one and a half years. He was observed to have fourto six seizures a day, lasting 0.5-1.0 minute. In someof these episodes he would burst into crying, while inothers into laughter and sometimes there was acurious mixture of both. The laughter did not have aninfectious quality. He was later observed to have, inaddition, episodes of running. There was no con-

Address for correspondence: Major P. K. Sethi, Command Hospital,Central Command, Lucknow 226002, India.

(Accepted 21 April 1976.)

vulsive movement and he never fell unconscious tothe ground. Sometimes these episodes could beinduced by hyperventilation, but not invariably. Oneepisode induced by hyperventilation has been docu-mented by movie and serial still photographs(Figs. 1-4). He suddenly burst into laughter, withsqueezing of the eyelids (Fig. 1) and, at the same timerubbing his upper abdomen, ran to the other end ofthe room near the door (Fig. 2), then turned and randiagonally across with an expression of fear on his face(Fig. 3), followed immediately by a frozen expressionof defiance, keeping both hands in the position ofboxing, standing against the wall as if ready to fight(Fig. 4). The whole episode lasted for about 90 sec-onds. Postictally for about 30 seconds he would brushaside any attempts to take his pulse, very arrogantly.After that, he was his normal self with no memoryfor the whole episode.There was no history of trauma or any febrile

episode in the past and no history of epilepsy in thefamily. Systemic survey and neurological examina-tion were within normal limits except for a rightcentral type of facial asymmetry. Electroencephalog-raphy showed generalized spike discharges duringseizures induced by hyperventilation with an epilepto-genic focus in the left temporal lobe. Routinelaboratory investigations and radiography of theskull did not reveal any abnormalities. Left carotidangiography showed changes consistent with arelatively avascular space occupying lesion in theanterior and middle temporal area (Fig. 5a, b).

OPERATIVE FINDINGS At left temporal craniotomy hewas found to have a light yellowish-brown, well-circumscribed, partly amorphous and partly granulartumour, located in the middle and inferior temporalgyri, with compressed oedematous brain acting as a

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r,

FIGS. 1 -4 Serial photographs of the patient in one of the epileptic episodes induced by hyperventilation.

FIG. 5 (a) Left carotid angiography anteroposterior anid (b) lateral views.

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Gelastic, quiritarian, and cursive epilepsy

false capsule. The mass extended subcorticallyinferiorly and medially towards the uncus. Thetumour was excised en masse. Its total size was4 x 4 x 3 cm. It extended up to the pole just short of2 cm, and up to the vein of Labbe. The extent of thetumour in the brain as seen by the operating surgeonis represented diagrammatically in Fig. 6. Histo-pathological examination revealed it to be an astro-cytoma grade I. The patient made an uneventfulrecovery from the operation and has been asympto-matic on anticonvulsant therapy for the last sixmonths.

DISCUSSION

The pathophysiology of emotional disorder hasalways fascinated neurologists. Focal lesions arethe more interesting as they permit certain in-sights and surmises into the anatomical substratesof these emotions. Despite the arguments againstlocalization, especially where disturbed behaviouris concerned, this approach has certain practicaladvantages. Moreover, the recent interest insplit-brain functions has renewed interest in theexercise of 'localization'. It is admitted thathigher cerebral functions are complex and theiranatomical substrate is not as simple as a reflexarc, but this does not mean that they need be

regarded as less bound to structures than the'lowest' functions.Yakovlev (1948) in a masterly article on

'motility, behaviour, and the brain' has con-vincingly deduced on a teleological basis thatthere are three levels of biological functions in theneuraxis. He ascribed these levels of totalneuronal functions to ento-, meso- and ecto-pallium subserving spheres of motility, servingvisceral, expressive and affective functions,respectively. The temporal lobe is in an uniqueposition as it embraces all three systems con-cerned with these three spheres of motility. In thispresentation we are predominantly concernedwith the second and third spheres-that is,expression and affect. The second sphere,biologically and morphologically in the middle,is the 'sphere ofmotility ofthe outward expressionof internal states' (Williams, 1968). The internalstates of pleasure or happiness, frustration, painor grief, fear or terror, may respectively beexteriorized as smiling or laughter, crying orsobbing, attempt at flight (running) or fight.Although these three systems are independentperipherally, centrally they are intimately relatedand interdependent (Williams, 1968). The tem-poral cortex is, therefore, in an unique positionfor an epileptic event there to be exteriorized in

FIG. 6 Diagrammatic representation oftheextent of the tumour (hatched areas).

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one or more of these three spheres. Therefore, theoccurrence of epileptic laughter, crying, andrunning are easily explained in this patient with adiscrete temporal lobe lesion.

Involuntary (pathological) laughter (or for thatmatter crying) may occur as a release phenom-enon due to a destructive lesion, as in pseudo-bulbar palsy, or as an epileptic event (gelasticepilepsy). In distinguishing between them, epi-leptic laughter is usually a short-lived paroxysmof laughter, with the patient having completeamnesia for the whole event. A patient with'release' laughter, on the other hand, has longerperiods of laughter (actually peals of laughter)and the patient is not only aware of it but, attimes, actually embarrassed about his disorder.This laughter may be provoked by the slighteststimulation, while provocation of epilepticlaughter may require techniques normally re-quired for provoking epilepsy, such as hyper-ventilation. The patient with released laughtermay have the corresponding emotional com-ponent-that is, a feeling of happiness or joy,while it is not known whether a patient sufferingfrom true gelastic epilepsy also feels elated or notas there is total amnesia for the episode.The question arises whether the temporal lobe

is the exclusive site for such epileptic events. A fewcases have been reported with temporal lobetumour (Daly and Mulder, 1957) and severalwith temporal or frontotemporal electroence-phalographic foci (Ironside, 1956; Druckman andChao, 1957; Daly and Mulder, 1957; Weil et al.,1958; Fukuyama et al., 1959; Lehtinen andKivalo, 1965; Roger et al., 1967; Zecchini andCecotto, 1967). Other reports have describedlesions of the basal ganglia, hypothalamic region,and in the wall of the third ventricle (Dott, 1938;Ironside, 1956; List et al., 1958; Gumpert et al.,1970; Gascon and Lombroso, 1971). Somehowthe hypothalamus has come to be regarded as ofspecial importance (Masserman, 1941). Martin(1950) called it a 'laughter centre'. The trend in thisdirection has been such that in some cases ofgelastic epilepsy with a temporal lobe lesion, in-direct pressure on the hypothalamus was impli-cated as a cause of epilepsy due to the proximityof the infero-medial surface of the temporal lobesto the hypothalamus. The same arguments mightwell be used in the reverse direction to explain atleast some of the cases of gelastic epilepsy

originally attributed to a hypothalamic lesion.Careful study of some of the other reported

cases of gelastic epilepsy associated with a lesionat another site shows that there was involvementof the temporal lobe as well. Two examples maybe quoted. In the case reported by Wood et al.(1958), gelastic epilepsy occurred in associationwith a neurofibroma of the trigeminal nervewhich had also deformed the inferomedial aspectof the left temporal lobe. In a case of gelasticepilepsy reported by Weil et al. (1958), a papillomaofthe third ventricle had extended to the temporallobe. Furthermore, a careful study of many ofthe reported cases of laughter occurring with ahypothalamic disorder and some ofthose of basalganglia and midline structures, such as the thirdventricle, reveals as has been rightly pointed outby Swash (1972) that the laughter in these casesis more like the pseudo-bulbar type. Anotherargument in support of this contention is thatone generally regards epilepsy as a corticalphenomenon. The hypothalamus is only one ofa series of stations concerned with control ofemotional feeling and behaviour (Alpers, 1940).Therefore, we feel that the role of the hypo-thalamus in the genesis of gelastic epilepsy is over-emphasized in the literature. At the same time, ithas to be conceded that our explanations do notaccount for all the reported cases of gelasticepilepsy. It seems that other structures in thelimbic system may also at times give rise to similarphenomena, but the temporal lobe, because of itsunique biological and morphological endow-ments, is in an especially unfavourable positionfor these epileptic events.With regard to lateralization, the left temporal

lobe seems to be a favoured site. Poeck and Pilleri(1963) found a left-sided lesion in six cases andright-sided lesion in four cases. The tumour in thepresent case was also on the left side.

In laughter as well as in crying, in addition to aplay of specific facial muscles, a series of respira-tory arrests and accelerations occur. Two facio-respiratory pathways have been postulated.Neural mechanisms involved in emotions likelaughter and crying seem to have a higher controlwith its integrating mechanisms and a 'finalexecutive pathway' for the corresponding emo-tion. The latter is dependent on neural mech-anisms arising in close association withtelencephalic and diencephalic centres concerned

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with respiration (Haymaker, 1969). The highermechanism has already been discussed briefly.The whole concept fits well with the concepts ofPapez (1937) and MacLean (1955). In theirscheme, the temporal lobe acts as higher integra-ting cortex within the limbic apparatus and isclosely connected through this system withhypothalamic cingulate, and brain stem path-ways. Theoretically, a strategically placed lesionanywhere in these pathways may give rise toinvoluntary laughter and crying. This may occuras a release phenomenon (removal of inhibition)or as an epileptic discharge (facilitation) as in thepresent case. Released involuntary laughter isgenerally due to bilateral lesions, though, rarely,a unilateral lesion may give rise to it (Swash,1972).Crying as an epileptic phenomenon has

received little attention in the literature. Mostauthors dealing with temporal lobe epilepsyemphasize that emotions occurring as warning ofminor attacks are unpleasant (Penfield, 1955).Thus crying as compared with laughter would beexpected to be a more common epileptic event. Incrying, as in laughter, a series of respiratoryaccelerations and arrests occurs in addition to adifferent play of facial muscles. The neuro-anatomical substrates of 'final executive path-ways', although akin to that of laughter, cannotbe the same as the total outward expression is oftwo entirely different moods. Not only do specificfacial muscles related to a specific emotion comeinto play, but the pattern of respiratory accelera-tion and arrests may also be different, to accountfor such things as sobbing. In addition, the neuralcells responsible for highest controls must neces-sarily be distinct, though closely related anatomi-cally to explain why a specific emotion evokes aspecific facial expression. In spite of this, theremay be some common neuronal mechanismssharing a number of synaptic pathways in themidbrain and brain stem, or interconnections ofthese two sets of pathways, or both. This wouldexplain why some normal people at times exhibitincongruity of emotional expression, such asfeeling so happy that they burst into tears andvice versa, or laughing themselves into crying.This might explain some of the episodes in whichthe patient reported here laughed or others inwhich he cried or in which there was a curiousmixture of laughing and crying.

Only a few of the reported cases of running(cursive) epilepsy have had an associated patho-logical study (Lennox, 1960; Chen and Forster,1973). Chen and Forster (1973) reviewing theavailable EEG findings in all reported cases con-cluded that a temporal lobe focus is essential forthe genesis of running epilepsy. We concur com-pletely with their conclusions because of theunique position of the temporal lobe to be the siteof these epileptic discharges. Running may beregarded as a dynamic physical expression of aninternal state of fear.The inferior and medial portion ofthe temporal

lobe has come to be regarded as a seat ofemotions.In this case the tumour also extended inferiorlyand medially. The precise location of laughter,crying, and running individually in the temporallobe is uncertain. The clinical information in ourcase is not sufficient for such precise localizationbut the role of the temporal lobe in producingthese epileptic discharges is clearly demonstrated.

We are grateful to Col A. K. Ghose, Officer Commanding,and the Director General, Armed,Forces Medical Services,for permission to publish this paper, and to Sister MaryRoss, MA, BEd, for advice on Latin etymology.

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