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108

References
1 . Wakai S , Nakamura K , Niizaki K, et al. Meningioma of the
third anterior ventricle presenting with parkinsonism. Surg
Neurol 1984;21:88-92.
2. Wright AD. Parkinsonism secondary to cerebral tumours. J
Neurol Neurosurg Psychiatr 1957;20:71-72.
3. Tolosa ES, Santamaria J . Parkinsonism and basal ganglia
infarcts. Neurology 1984;34:1516-1518.
4. Samiy E. Chronic subdural hematoma presenting a parkinsonian syndrome. J Neurosurg 1963;20:903.
5. Sandyk R, Kahn I. Parkinsonism due to subdural hematoma.
J Neurosurg 1983;58:298-299.
6. Krul JMJ, Wokke JHJ. Bilateral subdural hematoma presenting as subacute parkinsonism. Clin Neurol Neurosurg
1987 ;89:107-1 09.
7. Pau A, Brambilla M, Cossu M, Schoenhuber R, Siccardi D,
Turtas S . Parkinsonism in the presence of intracranial extracerebral haematomas. Acta Neurochir 1989;96:15S160.
8. Garcia de Yebenes J, Gervas JJ, Iglesias J , Mena MA, Martin del Rio R, Somoza E. Biochemical findings in a case of
parkinsonism secondary to brain tumor. Ann Neurol 1982;
1 1 :313-3 16.
9. Koller WC, Langston JW, Hubble JP, et al. Does a long
preclinical period occur in Parkinson's disease? Neurology
1991 ;4 1 (supp! 2):8-13.

FIG. 3. CT scanning of the head after 3 months showing the
subdural hematoma had largely resolved.

fluid. After the operation, all the patients improved
greatly. As far as we know, this is the first case report on
a patient with parkinsonism due to a subdural hematoma
in whom the parkinsonism diminished with spontaneous
remission of the subdural hematoma.
The mechanism leading to parkinsonism in patients
with a subdural hematoma is not well understood. It has
been suggested that mechanical pressure on the basal
ganglia can damage both the presynaptic dopaminergic
nigrostriatal neurons and the postsynaptic dopamine receptors (8). Nevertheless, in our experience, most patients with a subdural hematoma, even at a similar location, do not show any symptoms or signs of parkinsonism. This suggests that those rare patients with
parkinsonism due to subdural hematoma are especially
vulnerable to insults to the dopaminergic system. In preclinical PD, there is already a substantial loss of nigrostriatal neurons (9). It is likely that in these preparkinsonian subjects the dopaminergic nigrostriatal neurons cannot compensate for the insult associated with the
subdural hematoma. The hypothesis that preparkinsonian
subjects are especially liable to develop parkinsonism after a subdural hematoma remains to be proven, however.
On the basis of our case, we conclude that it is not always
necessary to operate on a patient with parkinsonism due
to a subdural hematoma, and it may be reasonable to
await spontaneous recovery.
A. T. M. Hageman
M. W. I. M. Horstink
Department of Neurology
University Hospital Nijmegen
Nijmegen, The Netherlands

Movement Disorders, Vol. 9, N o . 1 , 1994

Biballism Caused by Bilateral Infarction in the
Substantia Nigra
To the Editor:
Biballism or paraballism has rarely been reported. In
1986, Hoogstraten et al. (1) found 13 cases in the literature, 6 of them with neuropathological findings. An additional case with a pathological study was described in
1989 (2). Although the lesions responsible for ballism usually involved the subthalamic nucleus (STN) ( 3 ) , the subthalamic area was obviously spared in some cases of
hemiballism or biballism (4-6). We report a new case of
biballism with unusual magnetic resonance imaging
(MRI) findings: bilateral ischemic lesions of the substantia nigra pars reticulata (SNr). This is the first case of
biballism with an MRI study.

Case Report
A 70-year-old man with a past history of mild hypertension had a brief loss of consciousness in November
1991. Transient diplopia and persistent abnormal movement disorders appeared when he regained consciousness. Movement disorders were predominantly on the left
side. The movements involved both proximal and distal
muscles of the arms and were continuous and stereotyped. They improved within a few days after thioridazine was given by a neurologist.
On February 9, 1992, the patient felt faint for few minutes, then the flinging involuntary movements suddenly
reappeared. When he was admitted 3 days later, uncontrollable jerking, flinging, and kicking movements had
caused extensive skin abrasions, grazes, and subcutaneous hemorrhages. Both proximal and distal muscles of the
arms were involved. Restlessness was experienced in the
legs. The movements were less explosive on distal seg-

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ments, where writhing movements were observed. The
ballistic movements were poorly patterned. The trajectory of the arms started with abduction or adduction.
Jerking and writhing movements of the face, tongue, and
neck greatly impaired eating and talking. Walking was
impaired by both involuntary movements and cerebellar
ataxia. Abnormal movements were either triggered or increased by voluntary action, talking, or emotion, but the
patient retained some control over voluntary movements.
The involuntary movements were not continuously
present during wakefulness, and they disappeared during
sleep except during 72 h after onset. Although these abnormal movements caused complete insomnia during 3
days, consciousness remained clear. Other physical and
neurological examinations were normal except for a
marked hypotonicity, predominating on the left side. The
family history was negative.
Laboratory studies were within normal ranges except
for an increase in CPK to 1,500 IU/L. Twenty-four hours
after haloperidol treatment (15 mg daily orally) was
started, the abnormal movements appeared to be less explosive and less violent. After a week of haloperidol treatment, most of the involuntary movements had disappeared, but they started again when haloperidol was reduced to 12 mg. The patient was able to talk. No
intellectual impairment was found (Mini-Mental State
Exam, 30/30). The electroencephalogram was normal.
Computed tomography (CT) scans with and without contrast showed only cortical atrophy. One week after biballism occurred, MRI images of the brain and midbrain
were obtained on a 0.5-T system, with T1-weighted images in axial and coronal planes and T2-weighted images
in the coronal plane. Slice thickness was 3 mm. T1weighted images, in both axial and coronal planes,
showed bilateral low-signal intensity in the midbrain just
below the red nucleus, behind the CNS cerebri (Fig. l),
and in the anterior part of substantia nigra, the left more
anterior than the right (Fig. 2). These 5-mm-wide signals
appeared with abnormal high intensity on T2-weighted
images. There were no abnormalities in the upper midbrain, striatum, thalamic, or subthalamic areas or around
the third ventricle. There were no supratentorial abnormalities except brain atrophy. The MRI study suggested
ischemic damage to the midbrain relevant to an occlusion
of one or two short branches of the initial segment of the
posterior cerebral artery. This MRI study was consistent
with bilateral infarcts in the SNr. Single-photon-emission
computed tomography (SPECT), using HmPAOtechnetium-99, failed to reveal asymmetric blood flow in
subcortical and cortical areas. The outcome was favorable during the following 10 months, and the daily doses
of haloperidol were reduced to 6 mg. The abnormal movements did not reappear but mild ataxia was still observed.

109

FIG. 1. TI-weighted image shows hypointensities behind the
crus cerebri.

pearance of skin lesions secondary to forceful contact
with the bed is also classical in ballism. Grimacing and
torsion of the trunk have also been mentioned in many
cases of biballism (1,3,4). The clinical features in our case
differed from other cases, however, in that there were
few movements at rest and ballistic movements only appeared when triggered by speech, intentional movements,

Discussion
Ballism is defined by clinical criteria. The association
of stereotyped, violent, proximal movements with flinging gestures, as observed in our patient, is Characteristic
of ballism, as in previous reports (1,3,7). This description
differentiates ballistic movements from chorea. The ap-

FIG. 2. T1-weighted image shows hypointensities in the anterior
part of the substantia nigra.

Movement Disorders, Vol. 9 , No. 1 , 1994

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o r emotion. In the case of biballism described by
Hoogstraten et al. (l), in which abnormal movements
were continuously present during wakefulness, a decrease in the symptoms was also observed at rest. Remission of abnormal movements was obtained in our patient within a week of dopamine antagonist (haloperidol)
administration, although mild abnormal movements reappeared when the dose was reduced too soon to 12 mg. A
favorable response with haloperidol has been previously
reported in only 2 of 14 cases of biballism in the literature
(l), while hemiballism is usually controlled by dopamine
antagonists. The dramatic effect of haloperidol (see videotape) may be relevant to hypotonicity reduction. The
first episode of left hemiballism in November 1991 suggested a right midbrain infarct. The occurrence of a symmetrical left midbrain infarct could have triggered both
right ballism and left hemiballism reappearance.
The CT scan was normal in our case. Two other cases
of biballism with CT scans have been reported. In the first
case (4), bilateral hemorrhagic lesions were observed in
the striatum but the midbrain was normal. In the second
case (l), the CT scan was normal, although a small infarction, especially in the midbrain, could not be excluded.
In this first MRI study of a case of biballism, the thalamic and subthalamic regions appeared normal, but an
unambiguous bilateral infarct was detected in the anterior
part of the substantia nigra (SN). The possibility of a very
small infarct in the STN or functional inhibition of the
STN nucleus or damage to STN connections cannot,
however, be ruled out. Ballism, also called corpus L u y s
syndrome (8), has historically been the only hyperkinetic
movement disorder with a well-established anatomical localization (9). However, some cases of hemiballism or
biballism were related to diffuse lesions of the basal ganglia, including the pallidum, putamen, and caudate nucleus as well as the STN (3,4). In two cases of hemiballism, careful postmortem examination confirmed that the
STN was spared (5,6), suggesting that another etiology
may also be possible.
The evidence implicating the STN and its targets in the
MGP and SNr is strong, however (9-1 1). Crossman et al.
(12) and Robertson et al. (13) have demonstrated in primates that inhibition of the glutamatergic pathway in the
STN or its target in the MGP produces a hyperkinetic
movement disorder similar to ballism.
Segal et al. (14) reported a case of hemiballism with a
lesion of the STN and secondary neuroaxonal degeneration in the homolateral SNr. In the 1950s, Meyers et al.
obtained alleviation of rigidity in five parkinsonian patients by performing a lesion in the medial part of the
substantia nigra during a stereotactic procedure (15). The
lesions of our patient were placed similarly to those Meyers et al. employed to treat parkinsonian tremor and rigidity (15).
These data suggest that the bilateral lesions of the SNr
may be the cause of biballism in our patient.
Acknowledgment: We owe special thanks to Professor
A. R. Crossman and Dr. Merle Ruberg for valuable comments. We are grateful to Regis Valdes for his assistance
in videotape production. This work was partly supported

Movement Disorders, Vol. 9 , N o . l , 1994

by a grant from the Department of Research and Technology (DRED).

Legends to Videotape
Segment 1. Biballism is present before treatment.
Segment 2. Some amelioration of movement disorders
is seen after introduction of haloperidol.
Segment 3. Much improvement is seen 1 week after
introduction of haloperidol.
Segment 4. One month later.
D. Caparros-Lefebvre
D e p a r t m e n t of Neurology

J. F. Deleume
N. Bradai
D e p a r t m e n t of Neuroradiology

H. Petit
D e p a r t m e n t of N e u r o l o g y
CHRU Lille
Lille, France

References
1. Hoogstraten MC, Lakke JPWF, Zwarts MJ. Bilateral bal-

lism: a rare syndrome. Review of the literature and presentation of a case. J Neurol 1986;233:25-29.
2. Masucci EF, Saini N, Kurtzke JF. Bilateral ballism in multiple sclerosis. Neurology 1989;39:1641-1642.
3. Buruma OJS, Lakke JPWF. Ballism. In: Vinken PJ, Bruyn
GW, Klawans HL, eds. Handbook ofclinical neurology. 5th
ed. Amsterdam: Elsevier Science, 1986:369-380.
4. Lodder J, Baard WC. Paraballism caused by bilateral hemorrhagic infarction in basal ganglia. Neurotogy 1981 ;31:48&
486.
5. Schwarz GA, Barrows LJ. Hemiballism without involvement of Luys body. Arch Neurol 1960;2:420434.
6. Reyes MG, Stevens ET. Hemiballismus from cerebellar metastases. J Neurol Neurosurg Psychiatry 1989;52:684.
7. Salomez JL, Francois M, Petit H. HCmiballisme au cours
d’une maladie de Behcet. Larc Med 1982;10:869-870.
8. Jakob A. Arteriosklerotische muskelstarre mit hinzutretendem hemiballismus. In: Foerster 0, Wilmanns K, eds. Die
extrapyramidalen erkrankungen. Berlin: Springer-Verlag,
1923:183-225.
9. Albin RL, Young AB, Penney JB. The functional anatomy of
basal ganglia disorders. Trends in Neuro Sciences 1989;12:
366375.
10. Alexander GE, Crutcher MD. Functional architecture of
basal ganglia circuits: neural substrates of parallel processing. Trends in Neuro Sciences 1990;13:266271.
1 1 . De Long MR. Primate models of movement disorders of
basal ganglia origin. Trends in Neuro Sciences 1990;13:281285.
12. Crossman AR, Sambrook MA, Jackson A. Experimental
hemiballismus in the baboon produced by injection of a
gamma-aminobutyric acid antagonist into the basal ganglia.
Neurosci Lett 1980;20:369-372.
13. Robertson RG, Farmery SM, Sambrook MA, Crossman AR.
Dyskinesia in the primate following injection of an excitatory amino acid antagonist into the medial segment of the globus pallidus. Brain Res 1989;476:317-322.
14. Segal R, Sroka H, Sandbank U, Kott E. Hemiballismus with
lesion of the subthalamic nucleus and neuroaxonal degeneration of the homolateral substantia nigra. Ann Neurol 1977;
2: 169-172.
15. Meyers R, Fry WJ, Fry FJ, Dreyer LL, Schultz DF, Noyes
RF. Early experiences with ultrasonic irradiation of the pal-

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lidofugal and nigral complexes in hyperkinetic and hypertonic disorders. J Neurosurg 1959;16:32-54.

Bilateral Akinesia in Drug-Induced Parkinsonism
After a Subthalamic Lesion
To the Editor:
A contralateral limb reduction of all of the major motor
parkinsonian signs, including akinesia, may ensue unilateral lesioning of the subthalamic nucleus (STN) either in
animal models (1,2) or in human parkinsonism (3). The
case of a woman rendered parkinsonian as a result of the
treatment of her hemiballismus with tetrabenazine illustrates the counterpart of these situations.
A 70-year-old right-handed woman suddenly developed
hemiballismus of her left extremities in October 1991. She
could not walk, feed, dress, or wash herself unaided. She
had had hypertension but no history of previous cerebrovascular events. On hospital admission, examination revealed severe left choreoballistic movements with no sensory loss, motor deficits, or pyramidal signs. The rest of
the neurologic and general medical examinations was normal. Computed tomography (CT) scan showed mild cortical atrophy and a small right subthalamic haematoma
visible only on a single 5-mm slice (Fig. 1). During the
next 4 months, treatment with dopaminergic antagonists
(haloperidol, up to 6 mg/day; tiapride, up to 400 mg/day;
clorpromacine, up to 200 mg/day) alone and in different
combinations was ineffective, although withdrawal of
them led to some further deterioration. She had also tried
valproic acid (up to 1,500 mg/day), clonazepam (up to 20
mg/day), and endovenous sulpiride, (100 mg) without

FIG. 1. Brain CT scan without contrast showing mild cortical
atrophy and a small subthalamic haematoma (arrow).

111

benefit. Decubitus ulcers appeared on her left limbs in
spite of limb protection because of repeated traumatism
due to violent involuntary movements. By month 5, tetrabenazine (50 mg t.i.d.1 resulted in a marked improvement in her choreoballistic movements. However, over
the ensuing weeks, while taking tetrabenazine as her only
medication, she developed a severe parkinsonism. On examination at the end of month 5, she had marked hypomimia and axial rigidity, and was unable to walk unaided
because of instability. Her four limbs were equally affected by marked bradykinesia, whereas rest tremor and
marked rigidity were seen only on the right extremities.
As tetrabenazine was slowly tapered off over 3 weeks,
hemiballistic movements reappeared and parkinsonism
improved.
The primate model of parkinsonism postulates that motor disturbances of Parkinson’s disease (PD) are a result
of increased thalamic inhibition due to excessive excitatory drive from the STN to the internal division of the
globus pallidus (GPi) (4). Thus, recent reports on alleviation of all major motor parkinsonian signs, including akinesia, by unilateral lesioning of the STN (1,2) not only
seem to provide a practical support to a theoretical model
(4), but they might also encourage future surgical or pharmacological inactivation of the STN for the amelioration
of PD ( 1 3 ) .
Our patient had hemiballismus and a hematoma localized in the STN area. This lesion apparently prevented
the development of contralateral tremor and rigidity, thus
supporting the proposed role of excessive STN activity in
the pathophysiology of the parkinsonian signs. However,
the presence of akinesia on both sides indicates that a
STN lesion alone would not necessarily result in a disappearance of all major motor parkinsonian signs.
It could be argued that in spite of the ensuing ballism,
the CT scan cannot confirm the STN as the unique site of
the lesion in our patient. Other surrounding structures
connected to the STN could be damaged. Particularly,
the lesion may have encroached on the ventroposterolatera1 nucleus of the thalamus or damaged the pallidal efferents passing through the fasciculus lenticularis. Either
of these would be an explanation as to why there was no
rigidity and tremor in the opposite limb. The crucial
point, however, is that she developed akinesia in the
limbs contralateral to the lesion when given tetrabenazine.
Central to the theory of basal ganglia functioning in
parkinsonism is the postulate that dopamine deficiency of
nigral origin may lead to overactivity of the GPi (4). Thus,
it seems likely that lesions that interrupt some striosubthalamopallidal pathways more or less reestablish the
normal functioning on the pallidofugal pathways to the
thalamus. It is known that in reserpinized monkeys, reserpine bilaterally diminishes the pallidal neuronal activity (6). Tetrabenazine, a reserpine-like drug, could act
over the pallidal movement-related cells, which have a
partially bilateral representation (7), in such a way that a
unilateral STN lesion alone could not compensate for the
possible bilateral interactions at the level of both pallidum
caused by the drug.
We think that the case discussed could contribute to
further illustrate the mechanisms of akinesia in humans.

Movement Disorders, Vol. 9, No. 1 , 1994