J Neurot (1987) 234:124-125

Journal of

Neurology
© Springer-Verlag1987

Hemichorea-hemiballism caused by arteriovenous malformations
in the putamen
A . Tamaoka ~, M. Sakuta t , and H. Yamada 2

Departments of 1Nettrologyand 2Neurosurgerj, Japanese Red Cross Medical Center~ 4-1-22, Hiroo, Shibnya, Tokyo i50, Japan

Sirs: Involuntary movements have rarely been reported in association with arteriovenous malformations [2, 4-7] and venous angiomas [I]. We have recently experienced a patient with
arteriovenous malformations mainly located in the putamen,
who gradually developed hemichorea-hemiballism. This is the
first reported case of hemichorea-hemibatlism caused by an
arteriovenous malformation.
A 66-year-old woman had been in good health until 1.5
years before her admission, when she had noticed transient
weakness of her left leg that interrupted her gait. She had also
noticed gradually developing choreiform and intermittently
ballistic movements of her left arm. One year later, she had
noticed dysarthria and irregular uncontrollable jerky movements of her left lower extremity, which were more marked in

proximal than distal muscles. During the next several months,
the movements became constant and more violent. Psychological stress or resting posture made the symptoms worse. There
were no episodes of sudden worsening or previous history of
loss of consciousness, headaches, seizures or other focal symptoms.
On admission, the general physical findings were normal.
Neurological examination showed the patient to be alert with
slight dysarthria. There were marked choreiform movements
of the entire left side including the face, with greatest
amplitude and frequency in the leg. Ballistic movements of
the left arm and leg were intermittently observed. These
movements disappeared during her sleep and worsened by the
psychological stress or resting posture. Muscular strength and

1~. 1. Computed tomographie scan showing a micronodular high-density area ranging from the right putamen to the insula (A) and contrast enhancement around the area (A')

Offprint requests to: A. Tamaoka

125
tendon reflexes were normal. The plantar responses were
flexor bilaterally. Sensation, cerebellar function and autonomic nervous system were normal. The pulses over the skull
and neck were symmetrical, without bruits.
The results of routine laboratory tests were normal. A
computed tomographic scan showed a micronodular high-density area ranging from the right putamen to the insula and
intravenously injected contrast medium enhanced its surrounding area (Fig. 1). Cerebral angiography demonstrated
no abnormalities but magnetic resonance imaging (long spin
echo image) disclosed a high signal area corresponding to the
enhanced lesion in the computed tomographic scan. These
findings indicated vascular malformations mainly located in
the right putamen without haemorrhage or infarction.
Surgical approach via a right fronto-temporal flap was carried out and the vascular malformations were excised. Histopathological examination revealed the characteristic pictures
of arteriovenous malformations. Postoperatively, the involuntary movements of hemichorea-hemiballism type became
markedly relieved.
There have been ten reported cases of involuntary movements secondary to vascular malformations [1, 2, 4-7]. None
presented with choreiform or ballistic movements, except for
the case of venous angioma in the caudate nucleus reported by
Burke et al. [1].
The movement disorder in our patient did not develop suddenly, as would be expected from a haemorrhage or infarction. The slowly progressive course of the symptoms and the
dramatic relief of involuntary movements after the excision
suggest that the involuntary movements were caused by a di-

rect mass effect of the arteriovenous malformations on the
surrounding neural parenchyma, possibly the globus pallidus.
The hemichorea-hemiballism could have been secondary to
the mass effect of the arteriovenous malformation in the putamen reducing the inhibitory outflow from the putamen to the
globus pallidus [3].

References
1. Burke L, Berenberg RA, Kim KS (1984) Choreoballismus: a nonhemorrhagic complication of venous angiomas. Surg Neurol 21:
245-248
2. Diamond MS, Huang YP, Yahr MD (1982) Sudden onset of involuntary movement disorders with arteriovenous malformations
of the basal ganglia. Mt Sinai J Med (NY) 49: 438--442
3. Johnson WG, Fahn S (1977) Treatment of vascular hemiballism
and hemichorea. Neurology 27: 634-636
4. Lobo-Antunes J, Yahr MD, Hilal SK (1974) Extrapyramidal dysfunction with cerebral arteriovenous malformations. J Neurol
Neurosurg Psychiatry 37 :259-268
5. Paterson JH, McKissock W (1956) A clinical survey of intracranial
angiomas with special reference to their mode of progression and
surgical treatment: a report of 110 cases. Brain 79:233-266
6. Podlas H, Lipschitz R, Allen ML (1979) Selective arterial embolization of the brain in an inoperable arteriovenous malformation in
the thalamus. A case report. S Aft Med J 55 : 1000-1002
7. Shaw KM, Hunter KR, Stern GM (1972) Medical treatment of
spasmodic torticollis. Lancet 1 : 1399

Received July 4, 1986