Cumhur Ertekin
0zgiil Ekmekgi
Ne~e ~elebisoy

Le fou tire prodromique
Received: 9 July 1996
Received in revised form:
12 November 1996
Accepted: 10 December 1996

Sirs: "Fou rire prodromique" was
first described by F6r6 in 1903 as an
attack of uncontrollable laughter,
which begins abruptly and is followed after several hours by hemiplegia [2]. This interesting type of
stroke onset has rarely been reported
[4--7]. On the other hand, pathological laughter and crying, caused by
bilateral and or diffuse lesions, is
widely recognized as a symptom occurfing in various brain disorders
such as vascular pseudobulbar palsy,
amyotrophic lateral sclerosis and
multiple sclerosis [3]. We have had
the opportunity to examine and follow up three stroke patients with fou
fire prodromique.
Case 1: A 57-year-old man with a
history of hypertension and diabetes
mellitus was admitted to hospital because of a fight-sided weakness,
which followed an attack of unmotivated laughter, lasting about an hour.
Far from feeling happy he was anxious about what was going on at that
time. On admission, he was conscious and fully oriented. There was a
mild fight hemiparesis without speech
disorder. The plantar response was
extensor on the right side. The sensory examination was normal and the
cranial nerves were intact. M R I revealed an acute left pontine infarct.
The hemiparesis on the fight side recovered completely in 3 days. Uncontrollable attacks of laughter lasting 15-20 rain were repeated a few
times in that period. Two weeks after
the onset of the stroke, typical pathological laughter and crying attacks

developed, which he could not resist
and which were independent of his
emotional status. There was no clinical sign of depression. A year later,
he was admitted to the neurology
clinic for the second time because of
a left-sided hemiparesis, again with
fou rire prodromique. This time there
was a left hemiparesis with bilateral
extensor plantar responses. On MRI,
a subacute infarct at the head of the
caudate nucleus in addition to a left
pontine old infarct was detected.
During the follow-up period, features
of suprabulbar palsy were manifested
with continuing attacks of pathological laughter and crying.
Case 2: A 67-year-old man with a
history of hypertension was admitted
to hospital because of a right-sided
weakness, preceded by an attack of
irresistible laughter which lasted
nearly an hour. Neurological examination revealed a right hemiparesis
with an extensor plantar response on
the right side. There were no other
deficits. A few days later dysphagia
in addition to attacks of forced
laughter and crying developed. He
found the attacks very disturbing as
they were completely unmotivated
and recurred several times a day. MRI,
which was performed on the 1st day
and repeated a week later, revealed no
pathology. When the patient was seen
a month later he was entirely normal.
Case 3: A 85-year-old woman
with a history of hypertension was
admitted to hospital because of a
right-sided weakness that developed
after an attack of laughter which had
lasted about 15 min and was unmotivated. On examination, there was
right hemiparesis with a mild sensory aphasia. The remainder of the
examination was normal. CT performed on the 1st day was normal.
MRI could not be performed. The
hemiparesis began to recover after
the 2nd day and the attack of laughter never appeared again. The sensory dysphasia continued when she
was discharged a week later.

In our three patients an attack of
unmotivated laughter was the initial
manifestation. Other neurological
signs appeared a few hours after the
laughter attack had ceased. In contrast to previous reports, after the
acute attack the prodromal laughter
in cases 1 and 2 was seen to be
transformed into classical pathological laughter and crying. Though previous reports mention a grave clinical picture with death of the patients
shortly after the stroke [5, 7], our patients' clinical findings were mild
and a good recovery was observed
in a short time, especially in cases
1 and 2. However, in both cases
pathological laughter and crying was
observed after an interval of a few
days or weeks, so they were patients
in whom "prodromal laughter"
seemed to give way to "following
hyperemotionalism" [ 1]. Therefore
the prodromal laughter and the
pathological laughter and crying
seem to have a common mechanism.
Poeck [5] proposed that, in fou fire
prodromique there would be a loss of
the tonic control of laughter whereas
in pathological laughter and crying,
loss of the phasic control could be
the underlying mechanism. Our observations suggest that rather than
being due to different mechanisms
these two types of neurological signs
seem to be a continuum. The exact
mechanism of pathological laughter
and crying is not clear and merits
further study.

References
1. Allman P, Hope RA, Fairbum CG
(1990) Emotionalism following brain
damage: a complex phenomenon. Postgrad Med J 66:818-821
2. F6r6 MC (1903) Le fou fire prodromique.
Rev Neurol (Paris) 11 : 353-358
3. Ironside R (1956) Disorders of laughter
due to brain lesions. Brain 79:589-609
4. Martin JP (1950) Fits of laughter (sham
mirth) in organic cerebral disease. Brain
70:453-464

272

5. Poeck K (1985) Pathological laughter
and crying. In: Vinken PJ, Bruyn GW,
Klawans HV (eds) Handbook of clinical
neurology. Elsevier, Amsterdam, pp
219-225
6. Swash M (1972) Released involuntary
laughter after temporal lobe infarction.
J Neurol Neurosurg Psychiatry 35: 108113
7. Wali GM (1993) "Fou fire prodromique"
heralding a brainstem stroke. J Neurol
Neurosurg Psychiatry 56:209-210
C. Ertekin (1~)
Department of Clinical Neurophysiology,
Ege University Faculty of Medicine,
Bomova, TR-35100 Izmir, Turkey
Tel.: +232 3880980,
Fax: +232 3422141
O. Ekmekqi 9N. ~elebisoy
Department of Neurology,
Ege University Faculty of Medicine,
Bomova, Izmir, Turkey

Jos~ M. Borrfis
Francisco Garcia Salazar
Francisco Grandas

tactic procedure for aspiration of a
colloid cyst of the third ventricle.
The surgical procedure caused a
small haemorrhage in the upper ventromedial part of the cerebral peduncle. This case illustrates an unusual
complication of stereotactic surgery
and highlights the role of midbrain
structures in the pathophysiology of
certain types of tremors.
A 64-year-old man, without previous relevant medical history, was admitted to hospital because of a brief
episode of loss of consciousness and
a minor head injury. On examination
the patient was inattentive, had an
unsteady gait and bladder incontinence. No other abnormal neurological findings were observed. Cranial
CT showed a hyperdense lesion at
the level of the third ventricle with
marked obstructive hydrocephalus.
MRI disclosed a nodular high-intensity signal in the third ventricle on
Tl-weighted and a hypointense lesion on T2-weighted images, consistent with a colloid cyst (Fig. 1).
The hydrocephalus was treated by
the insertion of a ventriculo-peritoneal shunt with improvement of
the patient's neurological status. In a

second stage, puncture and aspiration
of the intraventricular colloid cyst
was attempted by means of a stereotactic transfrontal approach under local anaesthesia, using a Leksell
stereotactic frame. The target coordinates were previously determined
from CT images of the lesion. After
three attempts to puncture the colloid
cyst with a Sedan's needle, the patient began to complain of diplopia
and the surgical procedure was
stopped. On examination an incomplete left oculomotor palsy, consisting of mydriasis not reacting to light
and paresis of adduction, was evident.
Postoperative cranial CT revealed, in addition to the intraventricular colloid cyst, a small haemorrhagic lesion in the rostral midbrain,
involving the ventromedial part of
the left cerebral peduncle at the level
of mesencephalon-diencephalon
junction (Fig. 2).
Three weeks after surgery a
tremor appeared in the right limbs,
more marked in the arm. It had resting, postural and kinetic components,
and exhibited medium to large amplitude and a frequency of around 4
Hz. The tremor became more coarse

Fig. 1 Brain MRI; T2-weighted image
showing a rounded hypointense lesion in
the third ventricle, consistent with a colloid cyst

Fig.2 Postoperative brain CT showing
a small haemorrhagic lesion in the ventromedial part of the left cerebral peduncle

Oculomotor palsy
and contralateral tremor
(Benedikt's syndrome)
following a stereotactic
procedure
Received: 13 September 1996
Accepted: 29 November 1996

Sirs: Benedikt's syndrome is a classical brain stem syndrome characterized by the association of unilateral
third nerve palsy and tremor in the
contralateral, often paretic, limbs [2].
This syndrome has been attributed to
lesions in the midbrain, involving the
ipsilateral red nucleus, substantia nigra, oculomotor fibres and the cerebral peduncle [12].
We describe the clinical and neuroimaging findings in a patient who
developed tremor with crossed oculomotor paralysis following a stereo-