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(Stroke. 1997;28:2081-2083.)
© 1997 American Heart Association, Inc.

Articles

Fou Rire Prodromique Heralding a Left Internal Carotid Artery Occlusion

C. Carel, MD; J. F. Albucher, MD; C. Manelfe, MD; B. Guiraud-Chaumeil, MD; F. Chollet, MD

From the Fédération de Neurologie and INSERM U455 (C.C., J.F.A., B.G.-C., F.C.) and

Correspondence to Pr F. Chollet, INSERM U455 pavillon Riser, hôpital Purpan, place Baylac, 31059 Toulouse, France. E-mail chollet{at}purpan.inserm.fr

	Abstract

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Background Fou rire prodromique, described as pathological laughter preceding the onset of an apoplectic attack, is a rare phenomenon.

Case Description A 61-year-old man manifested pathological laughter before a sudden right hemiplegia. MRI showed a left lenticular and caudate nucleus infarct with involvement of the external capsule and prerolandic area. MRA revealed a left internal carotid and middle cerebral artery occlusion.

Conclusions The clinicoanatomic correlates of this phenomenon are discussed.

Key Words: carotid artery occlusion • cerebral ischemia • laughter

	Introduction

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Fou rire prodromique is a rare phenomenon, first described by Féré¹ as pathological laughter preceding the onset of an apoplectic attack. We report the case of a patient who presented a fit of laughter heralding right hemiplegia with severe speech disorder.

	Case Report

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On October 24, 1996, a 61-year-old right-handed man was admitted to the Department of Neurology 1 hour after a right hemiplegia of abrupt onset. On admission, severe right-sided hemiplegia involving the right side of the face with a right extensor plantar response was noticed. He was conscious but drowsy and presented with a left deviation of his head and eyes, severe mixed aphasia, and a right homonymous hemianopia. A CT scan without contrast injection, performed in the first hours, showed a spontaneous left middle cerebral artery hyperdensity. No other parenchymal abnormalities were detected.

We concluded that the patient had an acute left sylvian ischemia related to left middle cerebral artery occlusion, and the patient was treated with small dose of heparin. Anamnestic information was obtained from his general practitioner. He had a past history of angina pectoris, was a heavy smoker, and was treated for high blood pressure and dyslipidemia.

A dramatic improvement of the patient's condition was observed during the following 10 hours. He then exhibited right hemiparesis and mild nonfluent aphasia. His language was, at that moment, informative enough to allow him to explain that just before the stroke he had experienced a fit of pathological laughter. He was with his wife, talking about his daily activities, when a fit of laughter arose quickly. He did not understand what was happening, did not feel happy, felt very anxious, was unable to speak, and could not stop laughing. He could not find any external provoking factor. The phenomenon lasted at least 2 minutes. It stopped abruptly, and he then fell on his right side and became mute with a right hemiplegia. There was no loss of consciousness or abnormal movement. His wife confirmed the accuracy of the story. She also mentioned that the fit was absolutely "out of context" and that it appeared extremely strange and disturbing. She also confirmed that the right-sided deficit occurred when the laughter had stopped.

An electroencephalographic recording performed 24 hours after the onset of the stroke demonstrated theta activity in the left temporoparietal area with no epileptic activity.

Doppler ultrasonography showed a left internal carotid artery occlusion and a mild contralateral stenosis (30% to 40%). MR angiography confirmed the left internal carotid and middle cerebral artery occlusion.

A cerebral MRI was performed on the 11th day and revealed a unilateral left infarction involving lenticular and caudate nuclei and the anterior part of the insula. The thalamus and internal capsule were spared (Figs 1 and 2).

View larger version (73K): [in this window] [in a new window]   Figure 1. MRI shows left lenticular infarction in our patient.

View larger version (75K): [in this window] [in a new window]   Figure 2. MRI shows left lenticular and left insula infarction in our patient.

The patient was discharged and went home on the 12th day after the stroke. No recurrence of the fit of laughter was observed.

	Discussion

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We think that the fit of laughter presented was pathological laughter, ie, inappropriate and continuous behavior without any emotional disturbance.² It corresponds to the signs initially described by Féré¹ as prodromal laughing.

Another hypothesis is the occurrence of a paradoxical embolus during a period of high thoracic pressure such as during a Valsalva maneuver, but the clinical features of the laughter, felt with anxiety and no comic context, and the normality of the echocardiography do not support this rare and controversial phenomenon.

We believe that this is a new case of fou rire prodromique heralding an ischemic stroke. Only two cases have been reported in the literature.^{3 4} The other causes reported are bleeding⁵ and tumor.⁶ In most cases, the lesion is bilateral and involves the brain stem³ or the thalami and hypothalamus. The patient described by Ceccaldi⁴ was the first with proven unilateral stroke (involving the left posterolateral thalamus and the adjacent internal capsule). Our patient's lesion was more external (Fig 1) and strictly unilateral. The lesion affected the posterior part of left internal capsule, left striatum, and insula. The genu of the left internal capsule and left thalamus were spared. No other infarction was detected on MRI.

The precise anatomy of normal laughter remains unclear, and the pathogenesis of pathological laughter is unknown. A hypothesis was proposed by Wilson in 1924.⁷ He assumed the existence of a "facio-respiratory center" located in the brain stem under the control of two bilateral routes: the first is voluntary and is composed of the pyramidal tract, and the second would be involuntarily inhibited by the first. The latter route would control the emotional activation of the fascio-respiratory center. Other authors stated that the site of this center could be the medial thalamus, subthalamus, or hypothalamus.²

According to Wilson's hypothesis, the occurrence of pathological laughter should be the consequence of bilateral lesions. Nevertheless, it appears that a unilateral lesion of the corticospinal motor tract, in charge of voluntary control on the facio-respiratory center, can deregulate this system and can provoke loss of the tonic control of laughter² without mood involvement. In our patient, we can argue that the pyramidal tract was affected unilaterally.

Considering the clinicopathological correlations of pathological laughter and crying, Poeck,² in a summary of 30 cases verified at autopsy, drew three main conclusions: (1) a single cortical lesion cannot produce pathological laughter, (2) the anterior arm and the genu of the internal capsule are always damaged, and (3) a unilateral lesion of the fibers connecting cortex and subcortex to motor nuclei of the pons and the medulla can produce these symptoms. In our case, the left pyramidal tract was affected and a lesion was detected in the posterior arm of the internal capsule. Furthermore, the anterior part of the left insula was damaged, and to our knowledge this area is not known to participate in laughter.

The precise mechanism of the fou rire prodromique is very unclear, and one can address the question of a functional or a lesional disturbance of the laughter system. Fou rire prodromique has always been described as a transient symptom. According to the literature, it lasts between a few seconds and 30 minutes. Moreover, the time course of the symptoms can vary greatly. In Féré's third case, the patient experienced several short fits of pathological laughter several weeks before the permanent hemiplegia occurred.¹ In the case described by Wali,³ the laughter lasted 30 minutes and ended abruptly, immediately followed by a locked-in syndrome. A symptomless interval of time of a few minutes was reported by Ceccaldi and Milandre⁴ between the fou rire prodromique and the permanent deficit. In contrast, in the present case the pathological laughter was followed immediately by the deficit.

We found no conclusive arguments regarding the nature of the phenomenon. One could suspect the occurrence of a gelastic seizure during the acute phase of the stroke, but in our case no epileptic features (eg, loss of consciousness, convulsion, recurrence, spikes on electroencephalography) were detected. Furthermore, in the literature no cases were followed by a generalized seizure or by any recorded epileptic feature. An ischemic mechanism can be proposed, in which case the fou rire prodromique could correspond to a transient ischemic attack, but most of the cases reported in the literature have been described in nonischemic diseases.

	Footnotes

 
Service de Neuroradiologie (C.M.), hôpital Purpan, Toulouse, France.

Received June 5, 1997; accepted July 21, 1997.

	References

Top Abstract Introduction Case Report Discussion References

 
1. Féré M.Ch. Le fou rire prodromique. Rev Neurol (Paris). 1903;11:353-358.

2. Poeck K. Pathological laughter and crying. In: Vinken PJ, Bruyn GW, Klawans HV, eds. Handbook of Clinical Neurology, Vol 1 (45). Amsterdam, Netherlands: Elsevier; 1985:219-225.

3. Wali GM. `Fou rire prodromique' heralding a brainstem stroke. J Neurol Neurosurg Psychiatry. 1993;56:209-210.[Abstract/Free Full Text]

4. Ceccaldi M, Milandre L. A transient fit of laughter as the inaugural symptom of capsular-thalamic infarction. Neurology. 1994;44:1762.

5. Martin JP. Fits of laughter (sham mirth) in organic cerebral disease. Brain. 1950;70:453-464.

6. Ceccaldi M, Donnet A, Grisoli F, Poncet M. Fou rire prodromique et tumeur prérolandique. Rev Neurol (Paris).. 1995;151:206-208.[Medline] [Order article via Infotrieve]

7. Wilson SAK. Some problems in neurology, II: pathologic laughing and crying. J Neurol Psychiatry. 1924;16:299-333.

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