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 »  Introduction
 »  Case report
 »  Discussion
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Year : 2000  |  Volume : 48  |  Issue : 4  |  Page : 388-90

Pathological laughter as heralding manifestation of left middle cerebral artery territory infarct : case report and review of literature.


Department of Neurology, Institute of Medical Sciences, Banaras Hindu University, Varanasi, 221005, India.

Correspondence Address:
Department of Neurology, Institute of Medical Sciences, Banaras Hindu University, Varanasi, 221005, India.

  »  Abstract

Clinical, radiological and pathological studies in patients with stroke, presenting with pathological laughter as heralding manifestation, have shown lesions in the internal capsule and thalamus, basal ganglion, hypothalamus and ventral pons. In this report a patient with similar manifestation and having a cortical infarct in the territory supplied by superior division of middle cerebral artery is being presented. Our case suggests possible influence of dominant cerebral hemisphere, especially of Broca's area, on the motor control of laughter.

How to cite this article:
Garg R K, Misra S, Verma R. Pathological laughter as heralding manifestation of left middle cerebral artery territory infarct : case report and review of literature. Neurol India 2000;48:388


How to cite this URL:
Garg R K, Misra S, Verma R. Pathological laughter as heralding manifestation of left middle cerebral artery territory infarct : case report and review of literature. Neurol India [serial online] 2000 [cited 2020 Aug 3];48:388. Available from: http://www.neurologyindia.com/text.asp?2000/48/4/388/1492




   »   Introduction Top

Clinical, radiological and pathological studies of patients with pathological laughter following cerebrovascular events have shown lesions in the internal capsule and thalamus, basal ganglion, hypothalamus and ventral pons.[1],[2],[3],[4],[5] We report a patient with cerebral infarct involving Broca's area, who had pathological laughter as heralding manifestation of the stroke. Relevant literature has been reviewed to provide current knowledge about possible neural control of the laughter, along with review of other neurological disorders associated with pathological laughter.

   »   Case report Top

A 50 year old, right-handed and normotensive woman, without any preceding history of neurological or psychiatric disorder, suddenly experienced an attack of laughter. The laughter started abruptly without any apparent external provoking factor. It was inappropriate to the situation, and she was unable to control it. The laughter lasted approximately 15 minutes and then stopped abruptly. Immediately after, the patient noticed inability to speak and weakness of right side of the body.
General examination three days later revealed an irregular pulse with heart rate of 90/min and blood pressure of 110/76 mmHg. Neurological examination revealed that the patient was conscious and cooperative with intact comprehension. She had marked expressive aphasia, and was able to utter only few monosyllables. Writing could not be tested because of weakness. All the cranial nerves were normal except right supranuclear facial palsy. Jaw jerk was not elicitable. Fundus examination was normal. Patient had right hemiparesis (power 2/5 in upper lumbs and 4/5 in lower limbs). [Right] plantar was extensor. Reflexes were brisker on right side and normal on left side. Sensory examination was normal. Cardiac examination revealed presence of right ventricular hypertrophy and mitral valve stenosis, possibly of rheumatic origin. Results of all haematological and urine parameters were normal. Electrocardiogram revealed findings suggestive of atrial fibrillation and right ventricular hypertrophy. Transthoracic echocardiography revealed moderate mitral stenosis (valve area 1.5 cm2 with dilatation of left atrium. Blood clot in the left atrium was not visible. Electroencephalography was normal. A plain computed tomography of brain revealed a cortical infarct in the territory of superior division of left middle cerebral artery [Figure. 1].
Patient was treated with digoxin, oral anticoagulants and aspirin (320 mg/day) along with advice for physiotherapy and was discharged after 7 days. Follow-up after 1 month showed marked improvement in weakness of left upper limb. However, the speech remained unaltered. Recurrence of laughter episode did not occur during follow-up.


   »   Discussion Top

Normal laughter is a coordinated motor function involving facial and respiratory muscles that is triggered by a specific, usually mirthful stimulus and accompanied by mood elevation. Pathological laughter may be defined as 'involuntary laughter unaccompanied by mirth that is either unmotivated or disproportionately motivated by a trivial stimulus'. Pathological laughter has usually been observed in four main clinical situations: (a) in patients with gelastic seizures; (b) in patients with pseudo-bulbar palsy; (c) in patients with sub-cortical and brainstem infarctions and (d) in patients with tumours in and around the upper brainstem.[7],[8],[9]
In patients with 'gelastic seizures', the laughter is inappropriate, stereotyped and not precipitated by either a specific or non-specific stimulus. The laughter can either be an isolated manifestation, or can be a component of complex partial seizures with loss of memory, automatism and motor symptoms. The duration of the laughter is usually short, but when associated with complex partial seizures it can be more prolonged. Patients with gelastic seizures often have other seizure types. Although ictal laughter may respond to antiepileptic drugs, these seizures are frequently difficult to control. In some patients gelastic seizures and ictal crying have both been present together. Though hypothalamic lesions are clearly associated with gelastic seizures, it has never been proved that the hypothalamus or temporal lobes are the site of origin of gelastic seizures. Attempts to remove the hypothalamic lesion or the temporal lobe have failed to control seizure. Because of the associated cognitive impairment, it has been suggested that the occurrence of gelastic seizures may result from a more general dysgenetic process affecting the limbic system. In addition to gelastic epilepsy associated with hypothalamic hamartomas gelastic seizures have also been associated with diseases of temporal lobe and mesial frontal lobe.[7],[8],[9],[10]
It has been hypothesized that laughter is possibly produced by a medullary effector centre ('laughter centre'). Medullary effector centre for laughing is subject to tonic inhibition from cerebral cortex and limbic system via an 'integrator' located in or around the hypothalamus. It has been further suggested that volitional (cortical) and emotional (limbic) influences interact with the 'laughter centre' in the lower brainstem through distinct pathways, allowing for disease to dissociate laughter from stimulus, mood, or both. Thus, diseases of upper brainstem are likely to produce pathological laughter through a supranuclear disconnection, eliminating cortical as well as limbic inhibitory influences on laughter centre.[7],[9],[11] For example, distortion of upper brainstem by extrinsic or intrinsic tumours of brainstem like clival chordoma and pontine glioma are prominent causes of pathological laughter.[12],[13] Cerebellum has also been shown to exert inhibitory influence on the descending supranuclear pathways concerned with laughter. Even a unilateral cerebellar lesion can cause pathological laughter.[14]
Exact cortical area responsible for laughter is not precisely known. Arroyo et al,[8] in electrical stimulation studies, in patients with gelastic epilepsy demonstrated possible role of cingulate and basal temporal cortex in laughter and mirth in humans. They suggested that anterior cingulate region was involved in the motor act of laughter, while the basal temporal cortex is involved in processing of laughter's emotional content in man. Perhaps emotions are subjected to the inhibitory influence of the frontal cortex as illustrated by the fact that diseases of frontal lobe produce emotional lability. House et al[15] observed that emotionalism is common after unilateral stroke and is found especially in patients with left frontal and temporal lesions. Swash[4] also reported a case of sustained involuntary laughter with accompanying alteration of mood, due to infarction of the inferior and lateral aspects of the left temporal lobe. The present case is unique, as a transient fit of laughter as the presenting symptom of left cortical infarction has not been reported. In previous reports,[2],[16] vascular involvement of thalamus and left internal capsule (posterior limb) in pathological laughter have been observed. Ceccaldi and Milandre[16] suggested that, possibly, concomitant involvement of the internal capsule in patients with thalamic infarction played an important role in the production of laughter. This case, in addition to hemiparesis also had motor aphasia. Thus possibly, motor expression of emotions (e.g. laughter) is also under influence of Broca's area. In an unusual case, Bronstein and Mendez[17] reported having observed several episodes of crying spells as a manifestation of transient ischaemic attack. Other associated neurological findings were suggestive of involvement of right cerebral hemisphere. However, since patient under report had only one attack of laughter at the onset of stroke, the laughter episode could be a manifestation of seizure activity as seizures are a common heralding event of cortical embolisation.
In conclusion, our case supports the hypothesis that unilateral lesions in the brain can cause pathological laughter and further suggests that possibly locomotor and motor speech areas of left cerebral hemisphere have a strong influence on the production of laughter.

 

  »   References Top

1.Fere MC : Le fou rire prodromique. Rev Neurol(Paris) 1903;11 : 353-358.   Back to cited text no. 1    
2.Ceccaldi M, Poncet M, Milandre L et al : Temporary forced laughter after unilateral strokes. Eur Neurol1994; 34 : 36-39.   Back to cited text no. 2    
3.Wali GM : 'Fou rire prodromique' heralding a brainstem stroke. J Neurol Neurosurg Psychiatry1993; 56 : 209-210.   Back to cited text no. 3    
4.Swash M : Released involuntary laughter after temporal lobe infarction. J Neurol Neurosurg Psychiatry 1972; 35 : 108-113.   Back to cited text no. 4    
5.Asfora WT, DeSalles AAF, Abe M et al : Is the syndrome of pathological laughing and crying a manifestation of pseudobulbar palsy? J Neurol Neurosurg Psychiatry 1989; 52 : 523-525.   Back to cited text no. 5    
6.Carel C, Albucher JF, Manelfe C et al : 'Fou rire prodromique' heralding a left internal carotid artery occlusion. Stroke1997; 28 : 2081-2083.   Back to cited text no. 6    
7.Poeck K : Pathological laughter and crying. In: Frederiks JAM ed. Handbook of clinical neurology, Vol. 1. Clinical Neuropsychology. Amsterdam; Elsevier 1985: 219-225.   Back to cited text no. 7    
8.Arroyo S, Lesser RP, Gordon B et al : Mirth, laughter and gelastic seizures. Brain1993; 116 : 757-780.   Back to cited text no. 8    
9.Dark FL, McGrath JJ, Ron MA : Pathological laughing and crying. Aus NZ J Psychiatry 1996; 30 : 472-479.   Back to cited text no. 9    
10.Tasch E, Cendes F, Li LM et al : Hypothalamic hamartomas and gelastic epilepsy: A spectroscopic study. Neurology 1998; 51 : 1046-1050.   Back to cited text no. 10    
11.Kinnier Wilson SA : Some problems of neurology. IInd Pathological laughing and crying. J Neurol Psychopathol 1924; 4 : 299-332.   Back to cited text no. 11    
12.Shafqat S, Elkind MSV, Chiocca EA et al : Petroclivial meningioma presenting with pathological laughter. Neurology 1998; 50 : 1918-1919.   Back to cited text no. 12    
13.Matsuoka S, Yokota A, Yasukouchi H et al : Clival chordoma associated with pathological laughter. J Neurosurg1993; 79 : 428-433.   Back to cited text no. 13    
14.Doorenbos DI, Haerer AF, Payment M et al : Stimulusspecific pathological laughter: A case report with discrete unilateral localization. Neurology 1993; 43 : 229-230.   Back to cited text no. 14    
15.House A, Dennis M, Molyneux A et al : Emotionalism after stroke. BMJ1989; 298 : 991-994.   Back to cited text no. 15    
16.Ceccaldi M, Milandre L : A transient fit of laughter as the inaugural symptom of capsular-thalamic infarction. Neurology1994; 44 : 1762.   Back to cited text no. 16    
17.Bronstein YL, Mendez MF : Crying spells as symptoms of a transient ischaemic attacks. J Neurol Neurosurg Psychiatry 1999; 67 : 255.   Back to cited text no. 17    

 

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